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1 THE RELATIONSHIP BETWEEN RESOURCES AND SENSE OF COHERENCE IN ADOLESCENT SIBLINGS OF INDIVIDUALS WITH AUTISM SPECTRUM DISORDER By LAURA OOSTERBAAN SMITH A DISSERTATION PRESENTED TO THE GRADUATE SCHOOL OF THE UNIVERSITY OF FLORIDA IN PARTUIAL FULFILLMENT OF THE REQUIREMENTS FOR THE DEGREE OF DOCTOR OF PHLOSOPHY UNIVERSITY OF FLORIDA 2012
2 2012 Laura O. Smith
3 In Memory of My Mother Virginia Helene Oosterbaan (n ee Boot)
4 ACKNOWLEDGEMENTS I would like to thank my dissertation committee for their support, fortitude, and consideration of my academic, professional, and personal goals. Dr. Jennifer Elder, my committee chair and advisor, extended me an invitation to work with her, shared her expertise without hesitation, and provided unconditional support for my academic suc cess. Her professional experience with families of persons with an Autism Spectrum Disorder and her desire to advance the science were the beginnings of this investigation. The encouragement and guidance she provided sustained me and made this accomplish ment possible. I am also grateful for Dr. Meredeth Rowe who made statistics enjoyable by her excellent teaching ability and mentored me throughout the journey. Dr. Constance Shehan provided a sociological viewpoint and raised questions that added to the study design and theoretical framework. Her feedback acknowledged and clarified my ideas, and I am grateful for her professional perspectives regarding the family. Finally, Dr. Erich Storch served as an external committee member and he extended his clinic al role as psychologist. He reminded me of the logistics related to committee members and sincerely grateful for your many contributions. There are many resources that contributed to my success and I am happy to recognize them. First, I could not have completed this degree without the financial support from the University of Florida The Merdinger & Charlotte Liberty Scholarships provided monies that allowed me time to pursue this degree full time and for that I will always be indebted to their sponsors. In addition, I would also like to thank Autism Speaks for recognizing me in their local chapter as the Scientific Ambassador, which allowed me to meet many families and get involved with fundraising efforts to advance
5 the science of autism. The International Autism Network and the Centers for Autism and Related Disorders in Tampa and Gainesville, Florida provided free recruitment assistance. Finally, the many hours of scholarly assistance from Cindy Garvan, PhD, Daniel A. Wells, PhD, and Dolores V. Boot, was greatly appreciated. My relationship with the God of my understanding and the love of my family, relatives, and friends are beyond words. The reality of obtaining a Doctor of Philosophy degree is a lifetime dream and the discipline required was beyond anything that I could have accomplished by myself. I attribute my success to the people and events that have intersected my path during this time. Many individuals co me to mind but there is always the risk of leaving someone out unintentionally. Thus, in a general way I reference my supportive words and actions. My husband, Randy, and my children Robert, Zachary, and Kaycie have made personal sacrifices that many times went unacknowledged and sustained me when I needed it most. I love you and the only way to adequately express my appreciation to you was to take this project to complet ion. Finally, I would like to thank the study participants who made this project possible. I am inspired by every family who expressed an interest in the study and grateful for the adolescent siblings who completed an extensive research packet. Your devo tion and loyalty to your sibling with an autism spectrum disorder speaks to your commitment to making this world a better place. I dedicate my career to you and families everywhere that are challenged by the unexpected.
6 TABLE OF CONTENTS page ACKNOWLEDGEMENTS ................................ ................................ ............................... 4 LIST OF TABLES ................................ ................................ ................................ ............ 8 LIST OF FIGURES ................................ ................................ ................................ .......... 9 ABSTRACT ................................ ................................ ................................ ................... 10 CHAPTER 1 INTRODUCTION ................................ ................................ ................................ .... 12 Problem ................................ ................................ ................................ .................. 12 Study Aims ................................ ................................ ................................ .............. 13 Theoretical Foundation ................................ ................................ ........................... 14 Salutogenesis Model and the Nursing Metaparadigm ................................ ............ 16 Stress, Resource, and Sense of Coherence ................................ ........................... 17 Stress ................................ ................................ ................................ ............... 17 Resources ................................ ................................ ................................ ........ 18 Sense of Coherence ................................ ................................ ......................... 21 Research Questions ................................ ................................ ............................... 22 Summary ................................ ................................ ................................ ................ 22 2 A REVIEW OF THE LITERATURE ................................ ................................ ......... 26 A utism S pectrum D isorder as an Indicator of Family Stress ................................ ... 27 Measurement of ASD Severity ................................ ................................ ......... 27 Parental Stress Related to ASD Severity ................................ ......................... 28 Siblings of Individuals with ASD ................................ ................................ ....... 32 ASD Severity ................................ ................................ ................................ .... 33 Sibling Attributes ................................ ................................ .............................. 34 Summary ................................ ................................ ................................ .......... 39 Sibling Resources ................................ ................................ ................................ ... 40 Social Support ................................ ................................ ................................ .. 41 Sibling Relationships ................................ ................................ ........................ 42 Coping ................................ ................................ ................................ .............. 45 Psychological Well Bei ng ................................ ................................ ................. 47 Summary ................................ ................................ ................................ .......... 49 Life Orientation and Coping ................................ ................................ .................... 49 S ense O f C oherence and Stress ................................ ................................ ...... 50 S ense O f C oherence and Resources ................................ ............................... 52 Summary ................................ ................................ ................................ ................ 53
7 3 METHODS ................................ ................................ ................................ .............. 54 Research Design ................................ ................................ ................................ .... 54 Sample ................................ ................................ ................................ ............. 54 Instrumentation ................................ ................................ ................................ 56 Procedure ................................ ................................ ................................ ......... 64 Statistical Analysis ................................ ................................ ............................ 66 Data scr eening ................................ ................................ ........................... 66 Missing data ................................ ................................ ............................... 66 Analytic Plan ................................ ................................ ................................ ..... 67 Scientific Rigor and Ethical Considerations ................................ ............................ 69 4 FINDINGS ................................ ................................ ................................ ............... 71 Sample Characteristics ................................ ................................ ........................... 71 Descriptive Statistics ................................ ................................ ............................... 72 Research Question One ................................ ................................ ................... 73 Resear ch Question Two ................................ ................................ ................... 73 Research Question Three ................................ ................................ ................ 74 Multivariate Analyses ................................ ................................ .............................. 75 Mediating Variable ................................ ................................ ............................ 75 Moderating Variable ................................ ................................ ......................... 76 Summary ................................ ................................ ................................ .......... 77 5 DISCUSSION ................................ ................................ ................................ ......... 87 Findings Related to Previous Research ................................ ................................ .. 87 Sibling Attributes ................................ ................................ .............................. 87 Coping Strategies ................................ ................................ ............................. 89 Relationship Networks ................................ ................................ ...................... 90 Sibling Well Being ................................ ................................ ............................ 91 Conceptual Framework ................................ ................................ ........................... 92 Stress ................................ ................................ ................................ ............... 93 Resources ................................ ................................ ................................ ........ 94 Sense of Coherence ................................ ................................ ......................... 94 Strengths, Limitations, and Directions for Future Research ................................ .... 95 Strengths ................................ ................................ ................................ .......... 95 Limi tations ................................ ................................ ................................ ........ 96 Future Direction ................................ ................................ ................................ 97 APPENDIX A QUESTIONAIRE ................................ ................................ ................................ ... 100 B INSTRUMENTS ................................ ................................ ................................ .... 101 C INFORMED CONSENT FORM ................................ ................................ ............. 110 REFERENCES ................................ ................................ ................................ ............ 119
8 LIST OF TABLES Table page 4 1 Demographic Characteristics for Sibling Family Environment ............................ 78 4 2 Ra nge, mean, standard deviation and median scores on each study instrument to sibling demographics of gender and birth rank. ............................ 80 4 3 Sibling d emographic variables are correlated to scale scores of coping, support, well being, a nd sense of coherence scores ................................ .......... 81 4 4 Re sults of Friedman Test ................................ ................................ .................. 82 4 5 Correlations for scale scores o .. 82 4 6 Hierarchical regression models are examined to test the model. Significant mediating and moderating variables are identified.. ................................ ........... 83
9 LIST OF FIGURES Figure page 1 1 Theoretical framework for study. Adapted from by Aaron Antonovsky (1979), .............................. 24 1 2 Model diagram for current study ................................ ................................ ......... 25 4 1 Path analyses and significance for mediating model. ................................ ......... 84 4 2 Beta weight and significance of predictor to outcome when predictor and mediator are regressed on SOC levels. ................................ .............................. 84 4 3 A path analyses to test for interaction (moderating) variable.. ............................ 85 4 4 Graph of sibling coping strategies on s ense o f coherence scores when autism severity is high and low ................................ ................................ .......... 86
10 Abstract of Dissertation Presented to the Graduate School of the University of Florida in Partial Fulfillment of the Requirements for the Degree of Doctor of Philosophy THE RELATIONSHIP BETWEEN RESOURCES AND SENSE OF COHERENCE IN ADOLESCENT SIBLINGS O F INDIVIDUALS WITH AUTISM SPECTRUM DISORDER By Laura Oosterbaan Smith Ma y 2012 Chair: Jennifer H. Elder Major: Nursing Science Research involving sibling dyads, particularly those in which one of the siblings has a disability, is of vital importance to health care providers because of the integral role family members play in providing, promoting and protecting the needs of their disabled constituent. Furthermore, inconsistent adjustment scores have been identified in siblings of individuals with an autism spectrum disorder. This study aimed to characterize adolescent siblings of persons with autism, Pervasive Developmental Disorder Non Specified (PDD N sense of coherence, coping strategies, relationship networks, and psychological well being from self reporting instruments. A secondary aim was to test the theoretical framework of Stress, Health & Coping: A model of Salutogenesis Parents provided consent and completed a demographic survey and the Childhood Autism Rating Scale, 2 nd Edition. Sibling assent yielded a sample of adolescent siblings (N=96). Data were obtained using: Ne twork of Relationship Questionnaire Social Provision Version; Adolescent Coping Orientation for Problem Experiences; Youth Self Report; and Sense of Coherence. Sibling demographics were
11 related to scale scores of each instrument providing rich descriptio ns of this adolescent group. ASD severity was analyzed as a predictor of sibling coherence and each sibling resource. Mediating and moderating variables were used to test the model in hierarchical regression models. Consistent with previous research, sib ling age and gender contributed 8% of the variance to the model. Females ( t = 2.88, p < .01) and older siblings ( t = 2.17, p < .05) perceived more supportive relationships with their brother/sister on the autism spectrum. There was an indirect relationship ( r = .154, p = .10) between sibling age age. This adolescent group reported the greatest amount of support came from parents, followed by friends and siblings respective ly. An indirect effect was identified and siblings with a greater number of problems had lower SOC scores regardless of ASD severity. Furthermore, sibling coping strategies interacted with ASD severity, suggesting siblings need more coping strategies to ma nage when there are high levels of ASD severity. Findings support the need for future intervention studies.
12 CHAPTER 1 INTRODUCTION Research involving sibling dyads, particularly those in which one of the siblings has a disability, is of vital impo rtance to health care providers because of the integral role family members play in providing, promoting and protecting the needs of their cognitive and emotional st ate (Kim, McHale, Crouter & Osgood, 2007; Vygotsky, 1978). These early perceptions provide operative frameworks that are used for self identification and impact sibling interactions (Branje, van Lieshout, van Aken & Haselager, 2004; Ratey, 2001). As a resu lt, the typically developing sibling acquires internal and external resources to adapt to the challenges presented by the disability. It is necessary to identify resources that influence sibling adaptation in order to facilitate their adjustment and enhanc e conditions for all members within the family environment. According to the Centers for Disease Control (CDC) 2010 statistics, 1.5 million people in the United States have an Autism Spectrum Disorder (ASD). Families of children with ASD are faced with c omplex adjustment and care demands that present significant challenges to all family members including siblings. In research comparing behavioral adjustment and relationship quality among siblings of persons with ASD to al palsy and typical siblings, siblings of a person with ASD were found to have greater difficulty adjusting (Meaden, Stoner, & Angell, 2010; Orsmond & Seltzer, 2007b; Stoneman, 2001). Problem A review of the sibling literature identifies inconsistencies among studies and these inconsistencies make it difficult to identify siblings who may be vulnerable to poor
13 adjustment outcomes. Researchers continue to investigate the family system, sibling resources, and biologic markers to explain why siblings of ind ividuals with ASD may be at greater risk for adjustment disorders. The unique challenges associated with sibling adjustment are thought to be related to hallmarks of the ASD disability which include difficulty communicating, inappropriate social behavior s, and the absence of physical disfigurement (Hastings, Kovshoff, Ward, delgi Espinosa, & Remington, 2005; Lobato & Kao, 2002; Fisman Wolf, Ellison & Freeman, 2000). There are indications that early involvement of siblings in the care of their brother/sist er with ASD has a positive impact on both sibling groups (Blacher & Begum, 2009; Hodopp & Urbano, 2007; Maynard & Martini, 2005; Orsmond, Kuo & Seltzer, 2009). Despite correlates of parental characteristics and a genetic phenotype to sibling adjustment, sp ecific sibling resources that are utilized remain unclear. Given the rising prevalence (CDC, 20 1 0), complex nature of a utism, and inconsistencies of sibling adjustment it is critically important to characterize typically developing (TD) siblings of indiv iduals with ASD. Studies have evolved from descriptive designs correlating gender, birth order and age of siblings with ASD and other disability types to exploratory designs investigating personality traits. In the proposed study, the Salutogenesis Mod el (Antonovsky, 1987) is used as the theoretical framework to guide the research. Study Aims Th is dissertation study has two aims: 1) characterize adolescent siblings of individuals with an Autism Spectrum Disorder (ASD) through a description of demographic data, coping strategies, social support psychological well being, and life orientation; 2) test the Salutogenesis Model by Antonov sky (1987) via corre l ation and
14 regression analyses to determine the association of resources on the relationship between ASD severity and sibling coherence levels. Theoretical Foundation Coping: A Salutogenesis Model was used as a theoretical framework to guide the investigation (Fig 1 1). The model has sociologic roots and has received empirical support in adolescent populations (Antonovsky & Sagy, 1986; Margalit & Eysenck, 1990). A sub structed model (Fig. 1 2) includes three conceptual relationships stress, resource, and coherence and these will be operationalized to test the model. The concept of coherence is the dependent variable and is defined as a life orientation or personal disposition. The Sense of Coherence (SOC) instrument (Antonovsky, 1987) measures the ability of a person to stay well under difficult environments. It is used in the study to relate sibling resources (coping strategies, relationship quality, and psycholog ical well being) and stress (ASD foundation. A salutogenic approach, translated from the Latin derivatives salut (happy) and genesis Through this orientation, investigators search for ways individuals effectively adapt. The approach xv). Individual cases of normal adjustment under stressful conditions are examined to determine what contributes to this disposition. According to the theory, tension, a byproduct of the stressor, is indirectly related t o
15 among individuals because there is variation in their generalized resistive resources (GRR) and psychosocial stressors, or generalized resistive deficits (GRD). These independent factors, resistive resources and resistive deficits, are combined into one concept referred to as the GRR RD (Antonovsky, 1987). Successful management of the stressor is anticipated for individuals with high SOC levels. The theory of Salutoge nesis rejects the view that people are either healthy or sick. Instead, it asserts that an individual continuum. Persons naturally migrate to the wellness end of the continuum and this movement is referred to individuals participate in the social system and the physical environment, they counteract the inconsistency related to the stress, which helps them maintain their balance. A change in their position o n the health continuum is influenced by an levels and resources. develop ment of his/her SOC levels beginning in childhood thro ugh early adulthood and then his/her SOC levels stabilize. According to the theory, when tension is created by the stressor, 1987). The salutogenic model is a coping theory and used in health promotion research. There are three major assumptions: (a) heterostasis and disorder are a usual occurrence throughout life with internal and external factors assisting the organism to achieve homogeneity and balance; (b) a per SOC levels is a measure of the ir
16 outlook of the world as ordered, predictable, and manageable; and, (c) resources not identified in the literature require a qualitative investigation (Antonovsky, 1987). When an individual understands the stressor, finds the necessary resources to manage it, and has an emotional investment in adjusting to it, his/her health can remain stable under stressful life conditions. Thus, the researcher identifies resources that continuum instead of focusing on the stressor. In this study, SOC scores will be described for adolescent siblings of individuals with ASD To examine the model, sibling coping strategies, social support and psych ological well being are correlated to AS D severity (stress) and Sense of Coherence levels. Salutogenesis Model and the Nursing Metaparadigm The TD adolescent siblings of individuals with ASD have an atypical family environment where routines, expenses, and caretaking are greater concerns in com parison to other adolescent sibling groups. These influences must be evaluated for each individual family member. The health care community is often the first place that family members of persons with ASD present for treatment and care (Twoy Connolly, & No vak, 2006). Therefore, nurses and other health professionals have a responsibility to evaluate the environment of individuals with ASD in order to promote optimal health within the family environment. The sociologic framework of Salutogenesis is appropri ate for discovering resources within the environment that can be utilized by the typically developing adolescent sibling to manage stress related to ASD. Nurses can play an instrumental role by understanding and promoting the necessary resources to provid e greater meaning, comprehension, and manageability of the stressor (ASD). T he theoretical
17 concepts used in the proposed study and their relationships are discussed in the next section. Stress, Resource and Sense of Coherence Stress It occurred to Anto novsky (1979) that not all stress was detrimental. In fact, it became clear to him that people who overcame stressful situations had different dispositions and strengths in their characters than individuals who succumbed to it. use it posited that there is physiologic evidence for successful stress management (resilience). Today, supports a paradigm shift in neurobehavioral science. Medical anthr opologists consider the psychological, sociological and biological influences on the identified stressor and assert that these influences cannot be taken out of context (Weisner, 2005; Wiley & Allen, 2009). In this study, the family environment influences typically developing siblings of individuals with ASD and this social context creates an impact and contributes to the challenges of isolating and rank (younger/older), sib ling age difference, and family size are demographic variables that will be explored in relation to study variables and reported to describe siblings of individuals with ASD. Studies indicate that in families in which one member has ASD, severity of the AS D and difficult behavior in particular, were associated with higher levels of perceived stress in parents, caregivers, and siblings (Lecavalier, Leone, & Wiltz, 2006; Davis & Carter, 2008; Schieve, Blumberg, Rice, Visser & Boyle, 2008). In addition, severa l
18 2007 ; Lewis et al., 2006). In the current study, stress is operationalized by the seve rity score of ASD measured by the Children with Autism Rating Scale 2 nd Edition (CARS 2; Schopler, Van Bourgondien & Love, 2010). T he relationship of ASD severity to each generalized resistive resources resource deficit (GRR RD) and his or her SOC levels was examined. Resources A resource is defined as a reserve supply of an adaptive quality, and/or adeptness at finding solutions to the stressor. The type of resource impacts the adolescent sibling by serving as a functional learning system that influences the process of his or her development (Moshman, 2005). Parental characteristics, family environments, and sibling variables have been investigated to differentiate siblings of individuals with ASD from other siblings (Smith & Elder 2010). Inconsistencies in the data and the limited number of studies substantiat e the need for further research. The resources in the study include adolescent sibling report of social support coping strategies, and psychological well being. Previous investigators have examined t hese variables through parental report in relation to sibling adjustment ( Gavidia Payne, 2006 ; M ugno, et al., 2007 ; Rivers & Stoneman, 2003 ). Further investigation through sibling self report c ontributes to the characterization of siblings of individuals with ASD. Psychological Well b eing Previous studies (Glasberg 2000; Kaminsky & Dewey, 2001; Orsmond et al., 2009; Pilowsky, Yirmiya, Doppelt, Gross Tsur & Shalev, 2004 ; Rivers & Stoneman, 2003; Ross & Cuskelly, 2006 ) have found correlations between sibling adjustment levels and sibling c oping style, social support, competence,
19 self concept, relationship quality, and knowledge of the ASD Adjustment was measured using the Child Behavior Checklist (CBCL; Achenbach, 199 1). Several investigators have reported h igher levels of internalizing (anxiety and depression) and externalizing (conduct disorders, aggression, and antisocial) behaviors in siblings of individuals with ASD compared to siblings of other types, although th e se findings are not consistent across studies (Kim, McHale, Crouter, & Osgood, 2007; Orsmond & Seltzer, 2007) Relationship network s Research regarding t he relationship quality of siblings has primarily focused on the interaction between the typically developing (TD) sibling and his/her brother/sister with ASD. Different instruments have been used among studies but they were all designed to measure the quality of a relationship in a usual sibling dyad and not a relationship where one member has a disabi lity. Researchers have reported that siblings of individuals with ASD have similar or more favorable relationship quality than siblings without a disabled family member. Relationship characteristics include high levels of admiration tolerance, empathy, an d selflessness and low levels of quarreling and competition (Ross & Cuskelly, 2006; Verte, Roeyer, & Buysee, 2003). Their data suggest that siblings of individual s with ASD have adequate relationship s and may have greater adaptive ability than siblings wit hout a disabled family member (Stoneman, 2001; Ferraioli & Harris, 2010). Two other studies investigated social support for siblings of persons with ASD (Rivers & Stoneman, 2003; Bayat, 2007). The adolescent literature review found empirical evidence that adolescents reported a preference for peer support over parental support ( Scholte, Engels, deKemp & Overbeek, 2007; Stice, Ragan, and
20 Randall, 2004). In consideration of this, the Network of Relationship Inventory Social Provision Version (NRI SPV; Furma n & Buhrmester, 2009) was selected for this study. It measures support, conflict and relative power between the TD adolescent sibling and one parent, brother/sister with ASD, and a friend. The support scale for each individual was correlated to ASD severi ty and sibling SOC levels and entered into the hierarchical regression model. P redic tive variables were examined for an indire ct (mediating ) or interaction ( moderating) effect using hierarchical regression to control for sibling age and gender. In additio n, each scale variable was examined in separate regression model to determine if they had an interaction with ASD severity to moderate the relationship between ASD severity and sibling SOC scores. Coping st rategy. perception of stress rela ted to the ASD determines its impact on him/her (Bishop, Reichler, Cain & Lord, 2007) and the coping strategy employed (Antonovsky, 1987; Lazarus, 1990 ; McCubbin & Patterson, 1983) For example, one sibling may report hiding his feelings to avoid disruption or conflict with in the family while another sibling may act out to gain attention ( Hutson & Alter, 2007 ; Ferraioli & Harris, 2010). Furthermore, the life course of development, including the period of adolescence, influences (Bjorkland, 2005). Adolescence, according to Erikson (1969), is the stage of identity formation. This and behaviors that contribute to his/her psychosocial adjustment (Moshman, 2005; Ratey, 2001). The Adolescent Coping Orientation for Problem Experiences (A COPE; Patterson & McCubbin, 1987) was used to measure coping strategies used by TD
21 siblings. Total scores were correl ated to the ASD severity and TD sibling SOC levels. The coping strategy total score was then tested as a mediating and moderating variable. Sense of Coherence dispositi on, and this is measured using the sense of coherence (SOC) instrument (Antonovsky, 1987). The SOC is comprised of three components meaningfulness manageability and comprehension and each component is influenced by the a nd social structure. To provide further clarity for the SOC concept, the following discussion is provided. Comprehension ability. Thus, an individual with a high level of comprehension can mak e sense of the stressor, explain it, and know what to expect. Manageability refers to the adequate access to necessary resources, thereby enabling an individual to deal with the stressor. Antonovsky (1987) reports that persons with high manageability SOC s cores look at circumstances. The final component of the SOC, meaningfulness is defined as an es not require the person to have a full understanding of the stressor; rather, he/she has enough the stressor is almost always the result of the value society places on the stressor (Antonovsky, 1987). The amount of meaning, manageability, and comprehension varies among individual s but the levels for each are intertwined; thus, they cannot be separated from
22 the total SOC score. Individual resources influence all three components but not to the same degree (Antonovsky, 1987). the SOC in the Salutogenic Model. Th e present study tests the model by statistic ally analyzing the relationships between stress, resources and SOC levels in TD adolescent siblings of individuals with ASD. Research Questions 1. Are demographic variables of age, gender, birth rank, family size, and age difference of typically developing s iblings of individuals with an autism spectrum disorder (ASD) related to their coping strategies (A COPE), social support networks (NRQ SPV), psychological well being (YSR), and life orientation (SOC) scores? 2. Do siblings of individuals with ASD perceive g reater levels of support from parents, friends, or sibling s on the autism spectrum ? 3. Do coping strategies relationship network, and psychological well being scores c orrelate with ASD symptom severity as measured on the Child Autism Rating Scale 2 nd Ed. (CARS 2) and life orientation as measured on the sense of coherence instrument (SOC) in typically developing, adolescent siblings of persons with ASD ? 4. Is the typically developing sibling resource (coping, support, or psychological well being) a media ting or moderat ing variable between the association of ASD severity and typically developing sibling SOC levels? Summary This chapter has provided a general background for the study and the research aims. The theoretical framework of Stress, Health & Cop ing: A Salutogenesis Model guides the investigation (See Figure 1 1) and is explained as it relates to a nursing meta paradigm. Conceptual and operational definitions are detailed for each major construct stress, sibling resources, and sense of coherence A brief reference to background evidence for the study hypotheses is also presented along with the research questions.
23 A review of the literature of siblings of individuals with ASD follows in the next chapter and describe s the concepts of stress psychological well being, coping, social support, and sense of coherence in prior research.
24 Figure 1 1. Theoretical framework by Aaron Antonovsky (1979) Adapted from
25 Figure 1 2 Model f or current study
26 CHAPTER 2 A REVIEW OF THE LITE RATURE The aim of this dissertation study is to characterize the typically developing (TD) adolescent sibling of individuals with Autism Spectrum Disorder (ASD). In this chapter, a review of the sibling literature of individuals with ASD is divided into three sec tions. In the first section, ASD severity and behaviors are described as operative definitions of stress in the research study. Next, sibling resources of individuals with ASD are identified in previous investigative work. The final section explains the life orientation concept and the sense of coherence (SOC) instrument (Antonovsky, 1987). The Diagnostic Statistical Manual of Mental Disorders Revised (DSM IV R) describes core features of ASD including alterations in communication, cognition, and behavi ors ( A merican P sychiatric A ssociation; APA, 2006 ) Speech and/or language impairment, problems interacting with others, and heightened sensory stimulation are symptoms of the disability exhibited in children before they reach 30 months of age. Throughou t this study, ASD will refer to individuals with a diagnosis of autism, pervasive developmental disorder not otherwise specified (PDD Families of individuals with ASD face many challenges when trying to provide optimal health care for loved ones with this chronic neuro biological, developmental disorder. N avigation of the health care system for quality health care providers, social support services, treatment options, and educational opportunities becomes a routine requirement Disabilities of any nature require much from family members but caring for individuals with ASD have been found to be particularly taxing on the family system (Gupta & Singhal, 2005; Schieve et al., 2004). Prior investigative work has identified ASD sym ptom severity to be associated with increased stress levels in unaffected
27 siblings and parents (Fisman et al., 2000; Hastings et al., 2005; McHale, Sloan, & Simeonsson, 1986; Rodrigue, Geffken, & Morgan, 1993 ). The functional capacity of individuals with ASD can vary but most have a normal lifespan and may outlive their parents. Thus, siblings of those with ASD may also be future caregivers for these individuals, and this has significant social implications. To date, little is known about the resources TD adolescent siblings have that prepare them to assume this critically important role. Thus, this research builds on previous sibling research and fills a gap in the literature. An improved understanding of the resources y to adjust to living with an individual with ASD may promote healthier, life long relationships and reduce the chronic needs of persons with ASD on society (National Institute of Health NIH, 2010; Hatfeld & Lefley, 2005). ASD as an Indicator of Family Str ess Research over the past ten years has investigated the association of ASD severity to parent and sibling characteristics (Smith & Elder, 2010). The type of spectrum diso rder and measurement contribute to differences between studies. Parental and siblin g characteristics as they relate to ASD severity in previous research are summarized in the next section Measurement of ASD Severity Investigators have used multiple instruments for measuring the severity of core symptoms (social reciprocity, behavior, and communication) in all types of ASD (Inglese, 2009). The instruments have different psychometric properties and ease of use. They may also be limited to specific age groups. The variety of instruments offers researchers and health providers a selectio n from which to choose; however, the specificity and sensitivity of the instruments vary. A reliable and valid measure of ASD
28 uses multi method techniques. These include observation and assessment of the developmental level of the child, an interview with the caregiver including a detailed developmental history, and screening for associated disorders (fragile X, mental retardation). Comparison of ASD severity between studies warrants caution because of discordance between the instruments (Inglese, 2009; Sa and Sigurdardottir, 2003). Many factors must be considered before selecting an instrument to measure ASD severity. These include the expertise of the person evaluating, the time needed to administer the instrument, the type of data analysis, and the planned utilization of findings regarding ASD severity. The Childhood Autism Rating Scale (CARS; Schopler, Reichler, DeVillis, & Daly, 1980) was developed prior to the DSM IV when sensory peculiarities and stereotypical behaviors of persons with ASD were not differentiated. Despite this, the CARS is an effective measure for identifying behaviors of ASD because it includes an assessment of non specific difficulties such as mental retardation, activity level, and language delays (Relli ni et al., 2004; Saemundson et al., 2003). The instrument was originally used to diagnose ASD through observation techniques; however, parent report has become an acceptable method by persons who have a moderate level of expertise (Schopler et al., 1980). Within the past year, a revision of the CARS, the CARS 2 (Schopler et al., 2010), has improved specificity for ASD. Following the theoretical framework of the Salutogenic model, the proposed investigation defines the stressor as ASD severity measured by the CARS 2. Parental Stress Related to ASD Severity The family environment is the context for the sibling relationship that is influenced
29 For several decades, investigations have associated parental stress levels with disability severity. Consistently, higher stress levels are found among parents of children with disabilities when compared to parents of non disabled children (Boer & Dunn, 19 92; Dyson, 1996; Lardieri et al., 2000). Furthermore, studies have shown that parents of persons with ASD have higher stress levels when compared to parents of children with other disabilities (Benson, 2006; Bishop et al., 2007; Hastings & Johnson, 2001; S chieve et al., 2007; Rao & Beidel, 2009; Tomanik, Harris, & Hawkins, 2004). These investigative findings are pertinent to sibling research because a cognitive, emotional, and behavioral response to parental stress has been found among family members (Hast ings, 2002, 2003; Hastings et al., 2005). Empirical data support elevated parental stress levels in parents of children with ASD. It remains unclear what services are warranted and under what circumstances they should be offered. Researchers investig ating the behavioral symptoms of individuals with ASD and parental stress levels have used different instruments. Thus, study methodology may lead to a spurious interpretation of findings that suggest aberrant ASD behaviors increase parent stress levels ( Hastings, 2002; Lecavalier et al., 2006). It is possible that a bidirectional relationship exists; however, unique family environments and cross sectional data make it difficult to isolate predictive factors. Herring et al. (2006) conducted one of the few longitudinal studies that examined the relationship between child behaviors and parental stress. The researchers collected data on children upon their initial ASD diagnosis and 12 months post diagnosis. Emotional and behavior problems of 123 children (age 20 51 months) with/without PDD NOS or communication delays were found to be significantly ( p < .01) correlated to
30 behavior scores at the second data collection. In a regression analysis, there were significant differences between maternal and paternal per p p < .01) impacted by behavior scores of the child. In a final analysis, the researchers reported that the emotional and behavioral problems of the children had a greater impact on parental stress levels than the presence or abs ence of PDD NOS, Social s upport Soc ial support as a study variable has been conceptualized as the number of connections outside of the immediate family or the number of services the family utilizes for treatmen t of the ASD (Benson & Karlof, 2008; Fisman et al., 2000). Social support has been found to have both a positive and negative association to ASD severity and parental stress. Furthermore, social support has been found to mediate, and/or moderate the relati onship between them. This discrepancy among study findings may be explained by the research design but it also suggests that social support may not be favorable under all conditions. For example, consider the following study. A group of researchers tes ted social support as a moderating variable between stress and negative outcomes (Dunn, Burbine, Bowers, Tantleff Duff, 2001). Negative outcomes of mothers ( n = 39) and fathers ( n =19) were operationalized as depression scores, social isolation, and spousal relationship problems. An inventory of social support was used to assess the type and amount of services parents received. An
31 interaction effect between social support and stressors was found; yet, there was no reduction in the amount of social isolation parents experienced. In addition, the study found social support moderated the relationship between stress and isolation such that parents with increased stress reported lower levels of isolation when they received higher levels of social support (Dunn et al., 2001). Coping. Several investigations examined coping in families of persons with ASD and researchers have used several theories to guide their research. The theoretical framework of Lazarus (1993) was used to ascertain what c oping strategies ( task o riented, emotion oriented, social diversion, and distraction) were predictors of parental stress in 77 caregivers of children with ASD (Lyons, Leon, Roecker Phelps, Dunleavy, 2010). Emotion based coping was the most frequent strategy used and its int eraction with ASD severity was significant ( p = .05). In a regression analysis, emotion focused coping was regressed on the pessimism scale of the dependent variable (parent stress). p = .001) indicated that parents who used emotion based coping were more pessimistic as the severity of the ASD increased. Twoy et al., (2004) examine d the differences in coping scores based on f amily demographics of gender and ethnic background using five coping strategies identified in the resilience theoretical framework (McCubbin & Patterson, 198 3). Acquiring social support, reframing, seeking spiritual support, mobilizing family to acquire and accept help, and passive appraisal were compared to normative data No gender differences were found on the five coping subscales. However, group results compared to normative data indicated lower mean scores in acquiring social support, seeking spiritual support, and passive appraisal. As expected, greater scores in mob ilizing
32 resources were found in parents with children who have ASD Cultural differences were also evident among participating subjects. While statistically greater mean scores of reframing efforts were found in Asians ( t (43) = 2.85, p = .007), Caucasian s showed higher mean scores in passive appraisal ( t (43) = 2.37, p = .022). Furthermore, subjects who spoke English as a second language relied more on spiritual support and used significantly less passive appraisal ( t (52) = 2. 87, p = .006) as a coping strategy than the English speaking group. These findings describe utilization of social support as a coping strategy. Ecological influences were correlated to the type of coping strategy employed by parents of individuals with ASD. There is empirical supp mental health, social support, and coping. There are also investigations that relate parental stress levels to compro mised parenting and sibling adjustment ( Benson and Karlof, 2008; Fisman et al., 2 000; Hastings, 2003; Macks & Reeve, 2007; Ross & Cuskelly, 2006; Verte et al., 2003). The next section reviews investigative findings related to siblings of individuals with ASD. Siblings of Individuals with ASD Early sibling research compared sibling s of children with autism to other disability types ( Gold, 1993 ; Mate, 1990 ; McHale et al., 1986; Piven et al., 1994). To date, the family composition, disability type, and sibling adjustment levels are the variables primarily examined for differentiating between sibling groups. While the majority of siblings of persons with ASD have characteristics similar to those of siblings of children with other disabilities adjustment seems to be more difficult for siblings of individuals with ASD ( Macks & Reeves, 2 007 ; Pilowsky et al., 2004 ; Verte et al., 2003; Rodrigue et al., 1993 )
33 Inconsistent research findings of sibling adjustment are explained by methodological differences and the developmental changes associated with children (Orsmond & Seltzer, 2007b). While these factors may explain differences between studies, they may also suggest that not all siblings have difficulty within their family environments. Several investigators have found data which support s adaptive characteristics withi n this sibling group that may not be found in siblings without a disabled family member (Benderix & Sivberg, 2007; Mascha & Boucher, 2006; McHale & Gamble, 1989; Stoneman, 2001). In this section, studies are summarized that explore ASD severity and attrib utes in siblings of individuals with ASD. ASD Severity their behavior and adjustment (Lobato & Kao, 2000; Ross & Cuskelly, 2006). The disruptive behavior of siblings wit h ASD may be regarded as usual until the TD child is mature (Bjorkland, 2005) and can comprehend the cognitive, social, or communicative impairments of the disorder (Glasberg, 2000). When the ASD disability goes unrecognized for prolonged periods, it may c hallenge the ability of the typically confrontational behaviors may result and lead to additional stress for the TD sibling including difficulty establishing friendsh ips (Bagenholm & Gilberg, 1991; Buhrmester, 1996). In all family environments, sibling relationship stressors present challenges that erupt for multiple reasons (Kluger, 2011; Kramer & Conger, 2009), but researchers have identified additional challenges r elated to stress when the brother and/or sister has ASD (Orsmond & Seltzer, 2007b; Stoneman, 2001). Aggressive and unpredictable behavior
34 in a brother and/or sister with ASD was reported by the TD sibling as a reason for added tension within the sibling r elationship (Benderix & Sivberg, 2007 ; Ferraioli & Harris, 2010; Mascha & Boucher, 2006 ). Therefore, sibling rivalry may be minimal or enhanced based on the characteristics of the TD sibling and the competencies or severity of the ASD. One group of re searchers hypothesized that TD siblings of persons with ASD would have greater expression of internalized behaviors (emotional components) based on genetic mechanisms and empirical support from prior investigative work (Pilowsky et al., 2004). The data di d not support significant differences in adjustment between sibling groups. A majority (87%) of siblings in the ASD group had normal adjustment scores and when these were correlated to demographic data and ASD severity, the researchers reported that older siblings described their brother or sisters with ASD more favorably ( p < .009) and adjustment scores were associated with ASD severity. These findings suggest that the competencies and behavior of the sibling with ASD impact adjustment in the TD sibling and also that older siblings seem to adjust better. Sibling Attributes Research investigating sibling attributes includes variables of age, gender, birth order, self competence, social competence, and characteristics of the family environment (e.g. parental traits, marital status, and parenting style). Early descriptive research compares siblings with other disabilities and recent exploratory research examines sibling characteristics of individuals with ASD. The study findings are presented in the following discussion. Demographics The developmental stages of children impact adjustment levels; thus, age is a covariat e in the sibling research of persons with ASD and is supported by
35 data in studies that have compared sibling age to adjustment (Glasberg, 2000) Gold (1993) and Rodrigue et al. (1993) found children who had an average age of nine years to have lower depre ssion scores than older children. Contrary to this, an investigation by Verte et al. (2003) collected data from parents who reported siblings ages 6 11 had higher internalizing behavior scores (depression) than the older sibling group (ages 12 16). In an effort to control for developmental differences, the proposed study will limit participation to adolescent siblings of individuals with ASD. Normative sibling data show that adolescents have greater conflicts than younger siblings (McHale, Updegraff, Eri kson & Crouter, 2001). In addition, depressive symptoms and behaviors are more prevalent in adolescence (APA, 2008) and these are frequently associated with normative stressors (Beardslee, Gladstone, Wright, & Cooper, 2003; Tiet et al., 2001). Moshman (2 005) describes the influence of modeling behaviors from peers rather than parents in adolescence. A confounding effect of age on the relationships analyzed in the proposed study will be examined. Self Concept In the review of the literature, few studi es were found to investigate self self concept is directly related to behavior scores (Achenbach, 1991a). These findings are consistent with Kaminsky & Dewey (2002). Anothe r investigation by Verte et al., (2003) analyzed the influence of self concept as a mediating factor of social competence in siblings of individuals with ASD. Sibling scores of persons with high functioning autism (HFA) were compared to a matched control group. Siblings of
36 children with HFA had higher scores on the subscales of honesty trustworthiness and on verbal self concept and these correlated positively to his/her self concept scores. L evels of depression, self concept and behavior were evaluate d by investigating group differences between siblings of persons with ASD and siblings who lived in families where the brother/sister did not have a disability ( Macks & Reeves 2007). The researchers hypothesized that there would be no difference in scores bet ween sibling groups. However, siblings of ASD were found to have significantly higher levels of self concept ( p < .01) more positive views of their intellect ( p < .003), more positive views of their behavior in general ( p < .003), and greater anxiety leve ls ( p < .005) than the control group. Because previous studies have found gender and number of siblings were risk factors associated with sibling behavior (McHale et al. 1986), a risk scale w as completed for each child Results of the analyses then predict ed greater challenges in psychosocial and emotional adjustment for siblings of individuals with ASD These data support the influence of demographic variables psychosocial character. A final attribute of the typically developing sibling is the broad autism phenotype (BAP). The phenotype refers to a genetic inheritance of features that resemble one or more of the characteristics of ASD but are not severe enough to be diagnosed as ASD (Mazefsky, Williams, & Minshew, 2008; Pilowsky et al., 2004). Several research teams reported that TD siblings, especially older ones, had a greater number of language and social deficits in a group comparison of siblings in families without disability (T oth, Dawson, Meltzoff, Greenson, & Fein, 2007 ; Virkud, et al., 2008; Yirmiya, Gamliel, Shaked, & Sigman, 2006; Yoder, Stone, Walden, & Malesa, 2009).
37 A recent study by Orsmond & Seltzer (2009) investigated 57 adolescents (age 12 18) siblings who had a bro ther or sister with ASD and their mothers. One of the two study aims was to examine how genetic vulnerabilities (self reported BAP factors) interacted with environment on social well being To answer the research question, two separate regression analyse s were completed. The first one used ASD behaviors as the stressor and regressed sibling depression and gender. Both were found to be significant predictors ( p < .05). A trend ( p < .10) was found when sibling BAP and behavior problems were used as interac tion terms. In the second regression analyses, a sibling life event was operationalized as the stressor and outcome variable. In siblings with high BAP scores, depression ( B = .39; p = .05) and anxiety scores ( B = .36; p = .05) were significantly predicte d. It is not clear if the expression of altered communication and social deficits in unaffected siblings is related to the environment or genetics. Thus, investigators must carefully consider a screening measure or exclusion criteria for cognitive or le arning differences when enrolling unaffected siblings of individuals with disability (Seltzer, Abbeduto, Krauss, Greenbergy & Swe, 2004). Sibling a daptation & adjustment. Sibling adaptation is a major concept in the literature and can be defined as the de velopment of physical and behavioral characteristics that allow siblings to thrive in their environment s ( Wiley & Allen 2009) The literature review on parental characteristics and sibling attributes offers explanations for poor adjustment in typically de veloping sibling s of individuals with ASD (Lewis et al., 2006; Mugno et al., 2007). Hastings 2003 ) cross sectional exploratory study c ompare d sibling adjustment scores using the Strengths and Difficulty Questionnaire (SDQ; Goodman, 1997) across
38 two gro ups of typically developing siblings who had a brother/sister with a disability (ASD and DS) and a normative sample Maternal stress levels were also correlated to the behaviors of the child with ASD. Typically developing s iblings of children with either d isabilit y type had greater overall adjustment problems on the SDQ and lower levels of prosocial (interaction) behavior than the normative sibling group. In the hierarchical regression analyses, maternal stress and behavior of the disabled were not predict ive of adjustment yet demographic variables (SES, family size, education levels) explained 35% of the variance. Sibling variables (age, gender, and birth order) accounted for a significant proportion of the variance (55%). Consistent with Kaminsky & Dewe y (2001), females were found to a djust more easily. Ross & Cuskelly (2006) found that adjustment was associated with coping skills and knowledge of ASD in a group of typically developing sibling individuals with ASD ( N = 25). Sibling participants rated recent interactions with their affected brother or sister with ASD and the coping skills they initiated. The responses were then classified into five categories. The investigation revealed that 52% of the siblings of indi viduals with ASD classified an interaction as aggressive ; 20% described it as a social problem ; 16% as syndrome specific ; 4% as concern for their sibling and 8% as unclassified Eleven children reported two incidents of aggressiveness on the Child Behavior Checklist (CBCL; Achenbach, 1991) and for them sadness was highly correlated and significant ( p < .001), while nervousness and anger were moderately correlated but insignificant ( p >.001). Twenty one children (84%) reported at least one aggressive inciden t. The researchers further analyzed coping strategies and discovered that the most common strategy was wishful thinking, followed by social withdrawal, distraction, problem
39 solving, social support, resignation, cognitive restructuring, blaming others, and self criticism. Although the investigators did not find that coping and knowledge of ASD contributed to sibling adjustment scores (measured by the mother), there were differences in adjustment scores in siblings of individuals with ASD. Normative data comp arison s showed TD sibling scores were 40% lower in adjustment, although the mean scores on behavior comparisons were similar (Ross & Cuskelly, 2006). The findings of sibling adaptation levels and sibling behaviors of persons with ASD are not yet fully un derstood. To provide greater understanding, larger study samples comparing siblings on individual factors (demographics, behaviors, and adjustment) or longitudinal data are needed. Antonovsky & Sagy's (1986) seminal work suggests that resources and psychos ocial stress have a direct relationship. Summary The behavior and severity of the ASD disability, parental characteristics, family environment, and characteristics of the TD sibling influence adaptive responses to the ASD. Previous research has identifie d higher adjustment scores in older female siblings and family environments with higher SES and education levels (Hastings, 2003; Kaminsky & Dewey, 2001). Direct measures of the TD sibling prove to be more accurate than parents repor t (Verte et al., 2003). Research within the past ten years on TD siblings has also identified a phenotype expression of mild forms of the ASD disability that requires prior screening before investigation (Pilowsky et al., 2004; Mazefsky et al., 2008; Tot h et al., 2007; Yirmiya et al, 2006). In order to manage tension created by stress, individuals utilize internal and external resources ( Antonovsky, 1987; Folkman, 2000 ; Lazarus, 1993 ; Moos, 1984 ). The proposed study will operationalize resources through examination of the TD
40 well being coping strategies, and relationship networks. A brief overview of the literature describing each of these factors is summarized in the following section. Sibling Resources There is adequate empir ical data to support the theory that the needs of a child with ASD may add a significant stressor to the family environment. Siblings of individuals with ASD are socialized into a family environment and the type and utilization of resources they attain may be heavily influenced by interacting variables within the family (Compas, Connors Smith, Saltzman, Thomsen, & Wadsworth, 2001; Singh et al., 2007). For example, parents adjust to the needs of their child with disability and may alter the type of parental guidance, discipline, and attention given to TD siblings (Powers, 2004). B iological, psychological, sociologic al and cultural factors determine the resources available to individuals (Weisner, 2005), thus influencing their life orientations (Antonovsky, 1 987). Orsmond et al. (2009) compared the relationship between the TD adolescent sibling and the TD adult siblings of individuals with ASD and found adult sibling relationships with their brother/sister with ASD to be influenced by their early environment and resources. These findings are consistent with other investigations of the TD siblings in early childhood. Researchers have found that factors contributing to the interactive nature of sibling relationships were d styles, family resources and sibling perceptions of the ASD severity (Yoder, et al., 2009; Knott, Lewis & Williams, 2007; Yirmiya et al., 2006). A first step for promoting the health and adjustment of TD siblings of ind ividuals with ASD is to identify what resources and environments contribute to positive
41 outcomes. If specific resources can be linked to favorable sibling disposition vulnerable environments may be identified. Furthermore, intervention studies can be co mpleted for an evaluation of their impact for improving interactions within sibling dyads (Knott et al., 2007; Kramer & Conger, 2009). This is vital for the unaffected sibling who may be a future caregiver for his/her brother/sister with ASD (Blacher & Beg um, 2009; Feiges & Weiss, 2004; NIH, 2009). In this section, sibling research studies related to support, relationships, coping, and psychological well being are presented. Social Support In the literature review of adolescence, social support has been measured directly and indirectly with adjustment as the outcome measure. Social networks, psychosocial assets, and appraisal of the support are common definitions of the social support construct. The variety of operational definitions measured across stud ies of child and adolescent health, including investigations reviewed for siblings of individuals with autism, makes it difficult to assess social support (Meaden, Stoner & Angell, 2010). An accurate assessment of sibling social support must consider the developmental level and psychological well being (Iervolini et al., 2002). Gupta & Singhal (2005) report that children < 12 years old recognized social support as the amount of caregiving received. In contrast, older adolescence (> age 12) define d social support as peer relationships or experiences outside the home. Consistent with this finding, a longitudinal investigation of adolescent girls (N = 456) found peer support ( r = 4.36) was greater than parental support ( r = 3.88) for all three time points (Stice, et al., 2004). This data suggests adolescent peer relationships impact social support. The research team also discovered that the adolescents who reported low levels of parental and peer support had significant levels ( p < .02) of depressi ve symptoms and this
42 finding was greater for females. posits social support and depression have re ciprocal relationship s and this finding has been supported in investigations of adolescents (Huang & Guo, 2009). The cons truct of social support in ASD research has been defined as a network of relationships and includes an appraisal of the quality of social interaction. Kaminsky & Dewey (2002) investigate d whether TD siblings (N = 90) aged 11 12 of children with ASD had dif ferences in psychosocial adjustment and loneliness when compared to siblings of children with Down syndrome (DS) and siblings of children without disabilities. The researchers found that self reported support from friends was higher among siblings across all groups, but siblings of individuals with DS or ASD reported higher levels of support from family members and teachers than the control group. Siblings of individuals with ASD and DS self reported lower loneliness scores when these were compared to pee rs of the same age No gender or interaction effects were discovered, nor was a significant difference found in scores between siblings of those with ASD and siblings of individuals with DS. The findings from this study suggest that TD siblings of individu als with ASD and DS have an adequate network of social support. Sibling R elationships The sibling relationship quality can be argued as an indicator of social support because of its significance as a psychosocial asset for all siblings within the family environment (Brody, 1998; Kim et al., 2007). Thus, it has been used as a predictor of adjustment and an outcome variable in the research on siblings of persons with ASD (Kaminsky & Dewey, 2001; Orsmond & Seltzer, 2009; Rivers & Stoneman, 2003). Kaminsk y & Dewe y ( 2001) examined the effects of dominance, affection, companionship, intimacy, admiration, and competitiveness in sibling relationships
43 among three groups (N =90). The severity of autism, SES, family size, and maternal education were compared amon g siblings. Siblings of persons with ASD, DS and without disability were age and gender matched. Although no significant differences were found among groups on familial demographic data, there were differences in the relationship scores. Siblings of chil dren with ASD and DS reported significantly ( p < .01) greater admiration for their brother/sister than siblings of children without disability and significantly ( p < .001) less quarreling and competitive behaviors. Post hoc comparisons between groups showe d siblings of children with ASD had less social interaction compared to siblings in the control group. This was not true for siblings of children with DS who had similar levels of social interaction as siblings of children without a disability. As part of a longitudinal study, Orsmond et al., (2009) compared TD adolescen t ( n = 57) and adult ( n = 142) sibling relationships by measuring their levels of involvement and affection. The aim of the study was to explore sibling relationship characterist ics and examine predictors of psychological well being coping and social support. A major finding from the study was that adolescents engaged in significantly ( p < .001) more shared activities with their siblings than adult participants did; however, measures of positive affection in the sibling relationships were not statistically significant between age groups. Consistent with previous research female siblings pa rticipated in more shared activities during adolescence with their disabled siblings but there were no gender differences in the adult group (McHale & Gamble, 1986, Orsmond et al., 2009). In addition, social support from family members and friends was fo und to be greater in adolescents ( p < .001) than in the adult group. The researchers also discovered that adult siblings who reported a positive affect in their sibling relationship also reported
44 higher levels of parental support. Depression scores as a m easure of psychological well being were examined between sibling groups and no differences were found. When comparisons among only siblings, closest siblings in age of multiple siblings, and other were made, depressive symptoms were found to be a significa nt predictor of relationship quality for siblings closest in age for both the adolescent and adult groups (Orsmond et al., 2009). Rivers & Stoneman (2003) found that f amily levels of adjustment (indicated by social support and marital status) correlated w ith the quality of the sibling relationship. The authors hypothesized that marital stress would have an unfavorable influence on the sibling relationship but would be mediated by increased levels of social support. The investigation found that marital stre ss and social support interacted to predict the quality of the sibling relationship. Interestingly, the benefits of social support were found only in families that reported positive sibling relationships. Siblings in unfavorable relationships and environme nts of marital stress remained there despite social support resources. I t is not clear for these families if social support added to the stress within the e nvironment, or if a troubled sibling relationship was the reason the family was utiliz ing social se rvices Regardless, data indicate different patterns of behavior are exhibited in favorable relationships than in unfavorable relationships (Rivers & Stoneman, 2003) Similar to other study findings (Macks & Reeve 2007; Orsmond et al., 2009) the data of sibling birth order and age span between siblings suggested older siblings had better relationships with the child with ASD and parents scores of sibling relationships were lower than siblings self reported scores
45 In order to examine the s ibling relationship in concordance with other realms of social support, the Network of Relationship Inventory Social Provision Version (NRI SPV; Furman & Buhrmester, 2009) was used. This revised instrument is similar to the Sibling Relationship Questionnai re (Buhrmester & Furman, 1990), which has been used in prior research in siblings of individuals with ASD (Fisman et al., 2000; Floyd, Purcell, Richardson, & Kupersmidt, 2009; Kaminsky & Dewey, 2001). The NRI SPV provides a matrix of relationships that can be used to compare and contrast subscales of support, power, and conflict in sibling with ASD, parent, and friend. Coping the context of any given life situation (Antonovsky, 1979; Lazarus 1993; Moos, 1984). The majority of research o n children and adolescents has focused on differences between individuals within a population in order to examine what types of coping contribute to successfu l adjustment (Blount, et al., 2008; Skinner & Zimmer Gembeck, 2007). Consistent with this approach, studies investigating coping strategies of siblings of individuals with ASD have examined coping in relationship to individual characteristics (age, gender birth order), parental influences (mental health, stress levels, marriage), and psychosocial variables (anxiety, behaviors, depression, self concept). Although empirical support indicates that the construct of coping is of vital importance when examining the effects of stress, it is unclear what type of coping strategies produce optimal health outcomes in TD siblings of individuals with ASD (Dellve, Cernerud, Lillemor, & Hallberg, 2000; Orsmond & Seltzer, 2007b; Ross & Cuskelly, 2006).
46 Orsmond & Seltzer (2009b) examined predictors of sibling relationship quality in adult and adolescent siblings of individuals with DS and ASD (N = 301). Three instruments measured the TD sibling resources and appraisals. First, an appraisal tool (designed for the study) mea sured the impact of the disability on eight areas of the of problem focused coping (active coping, planning, suppression of competing activities, and positive interpre tation/growth) and four subscales of emotion focused coping (denial, focus on and venting of emotions, behavioral disengagement, and mental disengagement). In addition, a measure of pessimism was included. No significant differences in levels of coping we re found between adolescent and adult sibling groups; however, siblings of individuals with ASD reported greater levels of pessimism than siblings of individuals with DS. Dellve et al. (2000) conducted a qualitative investigation aimed at understanding th e coping mechanisms of siblings of children with deficits in attention, motor control, inductive analysis, the researchers found that the coping process could be conceptualized in to four categories: gaining understanding, gaining independence, following a bonding responsibility, and balancing. The researchers found that requirements vs. concerns for their brother or sister with a disability. Conceptualized categories of coping are defined in a broad sense. Common responses to stressors include the mechanisms used to manage the stressor for a specific situation (cognition, perception, behavior). Researcher s are also interested in
47 the long is needed to define clearly the subtypes of coping and identify which are most effective in childhood and adolescence (Compas et al., 2001; S kinn er, Edge, Altman, and Sherwood, 2003). The Adolescent Coping Orientation for Problem Experiences (A COPE; Patterson & McCubbin, 1987) was developed to address the unique stressors of adolescence, including identity formation, assuming autonomy from p arents, and conforming to peer and social demands. The measure has been used in healthy male and female adolescents (N = 361) as a tool to evaluate the most frequently used coping strategy in difficult situations (Chapman & Mullis, 2000). The investigator s used multiple factor analyses of the 12 subscales to differentiate three types of coping strategies (problem focused, cognitive focused, and emotion focused. Emotion focused coping was the most common strategy used by both groups of 7 th through 12 th grad e students. These findings are concerning because emotion focused coping strategies are not aimed at the core problem and have been found to be associated with psychological maladjustment (Moos, 1984). Psychological Well Being Psychological well being is defined as an optimal state of health or adjustment in the proposed study. The construct of well being has been operationalized in the majority of investigations in TD siblings of individuals with ASD by scores on behavioral measures (Meaden et al., 20 10). In addition to behavior scores, psychological well being in siblings of individuals with ASD has also been defined as self competence and self concept (Macks & Reeve, 2007; Verte et al., 2003). Quality of life and stress measures have been used in fam ily adjustment and adaptation research (FAAR)
48 frameworks, but thus far parents have completed these measures because instruments suitable for children and adolescents are not available. A plethora of research in children has examined the relationships among stress, family function, and psychological events. Research investigations have revealed that these concepts are directly related. As the number of adverse life events increases, repor ted levels of depression and disharmony within the family increase: these findings are particularly salient in adolescent populations (Pike & Eley, 2009; Sholte et al., 2007 ; Tiet et al., 2001). Two global patterns of behavior are evaluated by the CBCL ( Achenbach, 1991) in many studies to operationalize adjustment and psychosocial well being in TD siblings of individuals with ASD. Internalizing (shy, anxious) and externalizing (aggressive, acting out) behaviors of unaffected siblings has been associated w ith the stress of the family environment and the severity of maladaptive behaviors in the brother or sister with ASD (Hastings, 2003; Ross & Cuskelly, 2006; Verte et al., 2003). These behaviors have been associated with marital satisfaction and birth order but findings are not consistent. two global patterns of behavior (internalizing /externalizing) and suggested that siblings may find it difficult to adapt when there a re abrupt changes in family life. In contrast, Hastings (2003) found that while siblings of individuals with ASD had lower adjustment scores than a control group, both genders had better adjustment scores if they were older than the affected brother/sister Typically developing (TD) siblings of individuals with ASD describe aggressive behaviors of their brother or sister with ASD as a factor that contributes to difficult
49 relationships and increased stress within the family environment (Benderix & Sivberg, 2007; Mascha & Boucher, 2006). Aggression in one sibling has also been linked to higher levels of externalizing behaviors in other siblings ( Natsuaki, Ge, Reiss, & Neiderhiser 2009). For example, a research design included 390 sibling pairs that were mo nozygotic and dizygotic twins, full siblings, half siblings and unrelated siblings. These groups were separated and evaluated over two waves of data collection. Aggression in one sibling was associated with externalizing behaviors in the other sibling at both time intervals despite controlling for pre existing externalizing behaviors and punitive parenting across sibling groups. These findings suggest that clinicians psychological w ell being of siblings. Summary Further research relating sibling resources is necessary based on the review of the literature for siblings of individuals with ASD. Within this population, there is empirical data to support inconsistent levels of adjustm ent despite twenty years of investigation. Social support, coping, and psychological well being are constructs that have self report img instruments with adequate psychometric properties for examining their impact on the TD sibling. The association of sib ling resources to ASD severity and life orientation or sense of coherence (SOC) will be examined to further characterize the adolescent sibling. In the final section of the literature review, a summary is provided of investigations that have used the SOC instrument in adolescent populations. Life Orientation and Coping and was operationalized by Antonovsky (1979) in the sense of coherence (SOC)
50 instrument. vel of SOC determine s his/her ability to regain composure (cope) under tension (by product of stressor). In other words, a person reporting high levels of coherence will remain healthy in the face of stressful situations. The SOC is a stable indicator of c oping influenced by resources that can vary within an individual by approximately 6% from the mean over time and still be considered unchanged (Antonovsky, 1987; Honiken et al., 2009; Kuppelomaki & Utriainen, 2003). The Salutogenesis Model (Antonovsky, 198 7) posits that individuals have resources that influence their tension management and impact their health. According to Antonovsky (1987), between stressors and remains stable once it is established. Life orientation is also a process. Thus, the SOC is dynamic between populations and during stages of development (Kuppelomaki & Utriainen, 2003; Antonovsky & Sagy, 1986). The SOC in early adolescence is characterized by unpredictability and young teens are expected to have lower levels of SOC than teens in the later years (Antonovsky, 1996; Antonovsky & Sagy, 1986). By early adulthood (age 30), Antonovsky (1987) claims a person will have established a rational and consistent way of looking at the world. The SOC concept describes the outlook of the TD adolescent sibling and it is used to test the Salutogenesis Model. The relationship of the SOC to the symptom severity of terature review identifies previous research that has correlated the SOC concept to stress and resources. SOC and Stress A longitudinal study (N= 1435) examined the stability of SOC levels over a five year period in adults aged 25 74 years old and correla ted SOC scores to health and
51 significantly greater ( p < .01) at the point of initial data collection remained higher than the mean scores at the second data collection. The investigators reported a significant ( p < .001) drop in the SOC scores over time from subjects who were older, had lower SES, and had chronic health problems. sectional study and administered SOC and behavioral health instruments to 14 15 year old eighth grade students (N= 383) from six public schools. The researchers examined the effects of SES and gender on each variable. No differences in coherence were found between the two socioeconomic groups; however boys (49%) had significantly higher SOC scores (t = 4.50; p < .001) than girls. Furthermore, a greater number of problematic health behaviors (eating disorders, drugs, and activity levels) were reported by girls of higher socioeconomic status and were significantly related to lower SOC levels. A final study looked at the moderating effects of SOC on stress and health. Nielsen & Hansson (2007) examined self reported illness and self reported symptoms as an outcome measure. In a randomized sample of urban students (N=3458), stress and SOC were measured in adolescents. Participants indicated whether they had been exposed (yes/no) to divorce, death or disease of family member, conflict, or other stressful events. The adoles cents were also queried on nine confounding variables: parental support, academic achievement, communication ability with parents and peers, well being at school, breakfast, smoking, and pubertal development. In the final analyses, two groups (none and > 1) were formed. Logistic regression of the two health outcomes ( self reported illness/self reported symptoms ) and gender was performed.
52 First order associations were included in two models. Both models included an interaction term of stress and SOC and th e second model included the confounding variables. The prevalence of poor health outcomes on both measures was higher in adolescents who reported stress and low coherence levels. Girls had a greater number of adverse health outcomes than boys and these we re significantly ( p = .01) associated in both genders to levels of SOC. When controlling for confounds, the experience of severe stress was associated with poor health outcomes ( self reported illness) but not for the SOC scores. The findings suggest that the SOC is stable in adolescents who have high stress levels and are consistent with the Salutogenic model of coping. SOC and Resources Honiken et al., (2009) published a longitudinal investigation that aimed to examine the association between SOC scores in adolescence to psychosocial indicators on subscales of the CBCL and YSR. In addition, the researchers conducted a regression analysis on the subscales and problem syndromes of the instruments to examine whether the presence of certain behaviors as indi cated by these instruments predicted SOC levels (Honiken et al., 2009). Baseline data (CBCL, YSR, and SOC instruments) years old (N = 1086) and subsequent data collect ions were undertaken when the participants reached age 12, 15, and 18 (N = 840). Findings revealed that at both the third and fourth data collection (age 15 and 18) boys SOC levels were significantly greater than girls (65.4 vs 60.8; p <.0001; and 65.4 v s. 61.7; p < .0001), but remained stable across groups over time. The predictors (CBCL and YSR) at age 18 showed a significant gender interaction on three subscales with males scoring higher than females: externalizing problems ( p = .02), attention proble ms ( p = .02), and delinquent
53 behavior ( p = .02). Furthermore, there was an inverse relationship on CBCL total scores and externalizing problem scores to SOC scores (p < .0001). Two symptom subscales of the externalizing behaviors (aggressive and destructiv e behaviors) also predicted SOC scores with maladaptive scores increasing over time. Furthermore, the p < .05) than adolescent scores using the YSR. The overall findings of the study suggest that early childhood behaviors have an effect on later behavior and SOC scores. Summary Siblings of individuals with ASD are an understudied group that has been found to be resilient in nature and to have adjustment difficulties. There are likely characteristics of the sibling groups that use different resources (coping, social support, and p sychological well being ) to thrive in the family environment. Life orientation is a construct at the center of the Salutogenic Model and is used as the dependent variable in this study through the Sense of Coherence instrument (Figure 1 1 ). In the n ext chapter, the associations of ASD severity, TD sibling resources, and sibling SOC scores are examined through a correlation framework. Additional analyses test the model and include significant relationships in hierarchical regression models to examine each resource as a mediating and moderating variable (Baron & Kenny (1986).
54 CHAPTER 3 METHOD S The majority of psychosocial studies found in the literature review compared siblings of individuals with an autism spectrum disorder (ASD) to siblings of typically developing (TD) youth or individuals with other disabilities. There is a paucity of research examin ing the associations of psychological well being, coping strategies, and relationship networks to health/wellness within a group of adolescent sibl ings of individuals with ASD. Thus, it remains un clear wh ich sibling resources may be effective to reduce tension and manage stress related to having a brother/sister with ASD. Therefore, it was the purpose of this study was to describe characteristics of adolescent, TD siblings of individuals with an ASD and examine resources that influence their coherence levels using the theoretical framework of Salutogenesis (Antonovsky, 1987) to guide the research. Research Design The study employs a descriptive, expl oratory research design to assess the characteristics and behaviors of adolescent siblings of individuals with ASD and to examine the conceptual relationships of the Salutogenesis model (Antonovsky, 1987). Although causal relationships cannot be evaluated using this methodology, the investigation provides beneficial information about these relationships and future interventions for experimental studies. Sample A purposive, convenience sample was comprised of TD adolescent siblings (aged 1 1 18 years ) of brothers/sisters diagnosed with P ervasive D evelopmental D isorder N on S pecified ( PDD NOS; herein referred to as
55 ASD) At the end of the recruitment period, 160 families were enrolled and 99 families returned the research pac ket for a 62% response rate. The study sample (N=96) was reduced because three families did not meet inclusion criteria (Appendix A). Demographic information was completed by parents and contained information about the family environment, TD adolescent s ibling, and their child with ASD. Individuals with ASD were between 2 22 years of age (mean 12.04, SD 4.5) and their diagnoses were confirmed using the parent completed Child Autism Rating Scale 2 nd Edition (CARS2; Schopler et al., 2010). Power. In order to present meaningful results a 5% risk of committing a Type I error and a 20% risk of committing a Type II error was accepted. The Bonferonni procedure (.05/k where k = 12) was used in answering the first research question to reduce the risk of Type I err or when reporting multiple significance (Warner, 2005). The was reduced to .004 in the analyses of sibling gender and birth rank to all the major variables. To test the theoretical model, a sample size (N=96) was adequate for six predictors and a large effect size (R = .58) was established (Cohen, 1988). Further increase of statistical power was obtained through a homogenous sample (Table 3 1). Recruitment. The primary recruitment strategy for siblings was through Internet based websites of national and local autism provider groups. Study notifications were post ed to chapters of the Autism Society of America The Primary Investigator (PI) volunteered as the Scientific Ambassador (community liaison) for Autism Speaks and advertisement for the study was provided on their national website. In addition, local provi ders of healthcare or social services to families with members on the autism spectrum were contacted and asked to assist with recruitment. Some providers made
56 contact with their constituents; others agreed to post IRB approved flyers in their waiting rooms Finally, two Centers for Autism and Related Disorders (CARD) and the International Autism Network (IAN) recruited study participants. Approximately 25% of the sample originated from Florida and 27 states were represented in the final sample. Despite thi s, participants were an overall homogenous group (see Table 3 1). Instrumentation Data were collected using instruments that required participants to answer questions with pen/pencil (Appendix B). Because the study was mail based, parents were encourag ed to contact the PI by telephone or email as necessary and to freely answer any questions their child had while completing the self administered questionnaires. Parents completed a demographic sheet and the CARS 2 ( Schopler et al., 2010 ). The self report ing instruments completed by the adolescent siblings included: (a) the Youth Self Report (YSR; Achenbach, 2001), (b) Adolescent Coping Orientation for Problem Experiences (A COPE; Patterson & McCubbin, 1987), (c) Network Relationship Inventory Social Provi sion Version (NRI SPV; Furman & Buhrmester, 1994), and (d) the Sense of Coherence (SOC; Antonovsky, 1987). Demographic information. A demographic form was developed to obtain information about the family environment and included listing current residents within the family household by age and gender (names omitted). Separate sections for employment, and amount of additional time they spent care giving their child with ASD. T he person completing the form identified ethnicity, diagnosis of the ASD, and whether professional treatment for the ASD was currently received. The reported length of time to complete the demographic form was five minutes.
57 Child Autism Rating S cale Th e Child Autism Rating Scale 2 nd Edition (CARS 2; Schopler et al., 2010) was used to measure ASD severity. The second edition is revised and includes two versions a standard (ST) and high functioning (HF). Parents were asked the diagnoses of the family me mber and if they reported a diagnosis of HF was mailed to them. Some parents identified their questionnaire. Thus, CARS2 ST versions were completed o n individuals with a higher functioning ASD. Both versions of the CARS 2 instruments distinguish between mild, moderate, and severe autism but raw scores rather than severity levels were used in the analysis because two versions of the instrument were use d to describe one sample. The CARS 2 can be completed in 5 10 minutes and requires minimal training for quality administration (Schopler et al., 2010). On the phone or via email, parents were advised that they would complete the CARS 2 instrument and tha t professionals used the measure for differentiating the ASD diagnoses from other disability types. Parents were reassured of their capability in completing the questionnaire and were told: 1) to read all the answers for each question before selecting the answer that best describes their child, 2) to avoid over or under severity score was a major study variable. Each instrument provided an area to write a description for each question and parents were i nstructed to use it to clarify their answers. The CARS 2 uses a Likert scale for 15 items. Behaviors are rated in comparison to an unaffected individual of similar age to their child with ASD. The score 1 denotes a normal behavior and a score of 4 depic ts a severely abnormal behavior The scores are
58 summed across four subscales to obtain a total raw score T scores can be obtained for percentile ranks (Schopler et al., 2010). The CARS is widely used and adequately differentiates levels of ASD severity. Total scores > 30 are strongly suggestive of ASD. Scores > 37 are considered moderate to severe ( Magyar & Pandolfi, 2007 ). Reliability and validity have been established for the CARS 2 standard (ST) and the CARS ha coefficients range from .43 to .81 with a median value of .69 for CARS2 ST and .53 to .88 with a median value of .79 for the CARS2 HF. In an examination of the test retest reliability of the instrument, Schopler et al. (1980) found an alpha of .88 for t he total score, although reliability was reduced in 10 of 15 adolescents assessed twice over a four year inter val Another study found the instrument to have adequate inter rater agreement with a Pearson correlation coefficient of .73 (Schopler et al., 201 0). Magyar & Pandolfi (2007) found that internal consistency coefficients fell below .80 on all four factors: social The CARS has also been examined for content and convergent validity with the DSM IV (Rellini et al., 2004) and the ADI R (Saemundsen et al., 2003). Convergence is reported as 100% between the CARS and DSM IV criteria ween the ADI R and the CARS. The CARS 2 (Schopler et al., 2010) is a revised edition and takes into account the intellectual functioning by administration of a Standard and High Functioning version. It has sensitivity levels of .79 and specificity is .58 with a false positive rate of 27% and a false 2 for the combined version was .89.
59 Youth Self R eport The Youth Self Report (YSR; Achenbach, 2001) is a measure of social competencies and beh avior problems in youth. This instrument includes many of the same items as the CBCL (Achenbach, 1991) for ages 4 18; and the teacher report form (TRF; Achenbach, 1991). It was developed to obtain 11 reports of their own competencies and prob lems in a standardized format. This measure is one component in multiple assessments necessary to accurately evaluate competencies and problems in adolescents (Achenbach 1991). The YSR is designed for a fifth grade reading level and was completed indep endently by 11 18 year old TD siblings. The YSR is comprised of 112 self report items answered by circling 0 if item is not true 1 if the item is somewhat true or sometime s true and 2 if the item is very true or often true On several items, the youth is asked to describe the problem in question. A score for every problem item is provided in addition to sum scores (raw) scores. T scores for the syndrome scales internalizing & externalizing and total problem can be reported when comparing scores from di fferent study samples. Reliability and validity have been established for the YSR (Achenbach, 1991a; Achenbach, Dumenci & Rescorla, 2002; Ivanova et al., 2007). One week test re test reliability for the problem scales was r = .68 for 11 14 year olds and r = .83 for 15 18 year olds. On the total problem score, the r was .70 for 11 14 year olds and .91 for 15 18 year olds. Over a 7 month interval, the r was .49 for problem scales in a general population of 11 14 year olds and .56 for total problems. In a sample of 15 17 year olds, 6 month stability was r = .69 for the total problem scale (Achenbach, 1991a).
60 Internal consistency has also been established for each problem scale and the scores (.95). In this study, the total problem scale was used to measure the amount a nd type of difficulty (resource deficit) that the sibling may be experiencing. It was derived from the following subscales: anxious/depressed ; withdrawn/depressed ; somatic complaints ; social problems ; thought problems ; attention problems ; rule breaking beh avior ; aggressive behavior ; and other problems Higher scores indicated a was .95 on the total problem scale and ranged from.78 to .87 on the nine subscales. The YSR has also shown good content and conver gent validity. Its design was based on the CBCL, and all but the asthma and allergy problem items were significantly correlated; thus, they were eliminated from both measures (Achenbach, 1991). Content validity for the YSR was supported in a sample of matc hed referred and non referred youths through significant discrimination of instrument items. Criterion validity was supported in the analyses after demographic factors were controlled (Achenbach, 1991). The YSR is a well established clinical tool for the measurement of behavior and adjustment in youth. In addition, the CBCL is one of the most widely used measures of child behaviors and is cited in a majority of sibling studies in the ASD research literature (Orsmond & Seltzer, 2007; Stoneman, 2001). Raw sc ores rather than T scores are recommended for use in statistical analyses and normative comparisons. This tool has multiple applications including epidemiology, screening for potential diagnoses, the measurement of intervention outcomes, and the identifica tion of problematic behaviors related to medical conditions (Ivanova et al., 2007).
61 Network of Relationship Inventory Social Provisions Version. The NRI SPV (Furman & Buhrmester, 1985) is a standardized measure used to identify roles that network member s play in meeting a variety of social needs in children, adolescents, and adults. The 38 item questionnaire is appropriate for individuals 11 years and older. addition, they were asked to identify one friend of the same sex and then estimate the length of friendship in years and months. Finally, they answered the questions in re gard to their brother or sister with ASD. The length of the sibling relationship was calculated by first taking absolute values of the difference in sibling age and then adding this value siblings were older. The adolescent sibling used the NRI SPV to rate how often the quality occurred in each relationship (one parent sibling with ASD, and same sex friend ). For example, ed their answers using a Likert scale ranging from 1 5; little or none (1), very much (2) somewhat, (3) very much (4) extremely much or (5) the most Higher scores indicated a greater amount of perceived support. Seven of the subscales ( affection, reliable alliance, enhancement of worth, intimacy, instrumental help, companionship, and nurturance of other ) were used to provide a scale score s. In this study support scores were kept separate for parent, friend and sibling in the bivariate and multivar iate analyses. Reliability and validity have been established for the subscales of this instrument, and for identifying similarities and differences among the parents, friends, and siblings in the social networks of children and adolescents (Furman & Buhr mester, 2007; Noack
62 & Buhl, 2004). Internal consistency has been established for each subscale and for each relationship (alpha ranges .59 .95). Test re test reliability data was examined in 200 adolescents (100 female) over a one year interval. Both the scale identifying support features and the scale identifying negative interactions were significantly correlated across time points (ranging from .49 to .70) and were stable in relationships with parents and in relationships with friends. There was a sig nificant effect related to relationship type [ F (5, 870) = 82.52, p < .05], with mean sub scale and scale scores varying across parents (mothers/fathers were identical), siblings, and friends (Furman & Buhrmester, 1985). Averaging the three items that make up each subscale derives scale (factor) scores on the NRI SPV and this was done because more than two items were answered. In this study, the social support scale score for parent, friend, and sibling from .84 .86. Adolescent Coping Orientation for Problem Experiences. Adolescent siblings completed the A COPE (Patterson & McCubbin, 1987) because the instrument examines the strategies adolescents engage in when managing tension or taking action to re solve a problem. This 54 item instrument is designed for persons age 11 and point Likert scale ( 1 = never; 2 = hardly, 3 = sometimes; 4 = often and 5 = most of the time ). Test retest reliability for the instrument ( r = .83) is adequate (McCubbin & Patterson, 1987). The A COPE has been used to evaluate coping behaviors in healthy populations, in ps ychiatric populations ( Recklitis & Noam 1999), in populations with
63 chronic health conditions, and cross culturally. Thus, it is considered a well established instrument that reliably measures coping behaviors in adolescents from a variety of backgrounds a nd with a variety of potential life stressors (Blount et al., 2008). Reliability and validity data have been reported for the A COPE (Blount et al., 2008; Copeland & Hess, 1995; Patterson adequate i Sen se of Coherence The sense of coherence (SOC; Antonovsky, 1987) is the core construct of a complex theoretical model of health (Salutogenesis) and was the dependent variable in position on a health/disease continuum. More specifically, SOC is an indicator of a person s ability to manage tension. The supporting theoretical framework posits that an (Figure 1 how to & Lindstrom, 2005). The 13 item self report instrument has been measured in children as young as 10 to score each question as a 1 or 7 (the extremes) on the Likert scale (Antonovsky, 1987); this tendency was not found in this study. Though the SOC does not separate items into subscales, each question is designed to express one of three components: compr ehensibility, manageability, and meaningfulness. A total score is obtained by summing the responses to the 13 items with higher scores indicating greater SOC levels. No cutoff
64 scores are established for high and low SOC levels because health has been conc eptualized to be on a continuum. Furthermore, there may be a point at which high levels of the SOC no longer protects the health of individuals, though there is a lack of evidence in this area (Antonovsky, 1993; Eriksson & Lindstrom, 2005). Reliability and validity data for the SOC is well established (Antonovsky, 1987; Eriksson & Lindstrom, 2005). Internal consistency is excellent based on SOC 13 data .92. In this study the This measure also shows acceptable test retest stability from .69 .78 (1 year) and .64 (3 years). The means of the SOC 13 have ranged from 35.39 (SD = 0.10) to 77.60 (SD = 13.80). The convergent validity for predicting health outcomes is discrepant (Eri ksson & Lindstrom, 2005). Long term health perspectives have higher validity in groups of psychiatric, post traumatic stress, and orthopaedic patients (Eriksson & Lindstrom, 2005). SOC is purported to become stable at age 10 (Antonovsky, 1987); however, da ta show that the SOC changes over time (Nilsson, 2003). The SOC instrument is thought to be structurally stable, yet the concept appears to be multi dimensional. Although there is a lack of empirical evidence to support it as a valid instrument for differ entiating health outcomes in adolescents, the SOC has been used cross culturally and has been shown to be a reliable instrument in the Antonovsky, 1993). Procedure The Institu tional Review Board of the University of Florida granted permission for the study and all recruitment materials. Initial contact with persons who responded to study advertisement was made by telephone and/or email. Potential subjects were
65 interviewed to s ee if they met inclusion criteria ( Appendix A ). Parents of families who met inclusion criteria were given an explanation of the study purpose and general guidelines to complete the study. The parent gave a verbal or written consent via phone or email befo re a research packet was mailed to them. Research packets contained the consent form, an instruction sheet, a demarcate completion by the sibling or parent. The instruments were placed in a mixed order to reduce study fatigue bias (Portney & Watkins, 2009). The time required for completing the instruments ranged from 45 60 minutes. A postage paid and a self addressed envelope to the PI were included in the research packet t o facilitate the response rate. Written instructions advised parents to read the instruments and consent form completely prior to enrolling their typical developing (TD) sibling in the study. The and this acknowledged (parent, adolescent) consent. To ensure confidentiality of the data, each research packet was assigned an identification number and upon their r eturn to the researcher, the signed consent and assent form were separated from the instruments. The researcher did not advise parents or counsel siblings based on their responses although families were encouraged within the introductory letter to engage in discussion regarding sibling perceptions upon study completion. A gift card for $20.00 to Walmart was used as a small incentive to recruit adolescents and was provided with a thank you note upon return of the study materials.
66 Statistical Analysis The theoretical framework of the Salutogenesis Model (Antonovsky, 1987) explains the relationship between human resources to stress and coherence levels using a post positivist philosophy. Data on self report instruments were entered into the Statistical Pack age for the Social Sciences (SPSS) Version 16.0 (Chicago, Illinois). Data screening The PI created a database in SPSS using a number keypad and the final dataset were re entered with an error rate of less than 1%. Missing data The sibling d ataset had random missing values (n = 85) and no systematic pattern was identified for values used in the analyses. CARS 2 instruments (n = 6) were returned to the parent because t hey evaluated the TD sibling (evidenced by low raw scores and reported in follow up communication). In addition, several other adolescent instruments (n = 5) were resent identifying uncompleted pages; all but two forms were returned to the researcher. For missing data, a code (999, 888) was entered into the raw dataset. After data explanation for absent data. A mean value for the missing question was obtained and imputed fo r random data absences, and a record of each imputation was made. Because of the low number of missing values, this method allowed the participant to remain in the analyses and did not increase the likelihood of Type II error (Fields, 2005).
67 Analytic Pl an Answering the first three research questions satisfied the primary aim of the study. Univariate and bivariate statistics were calculated to describe the sample across all data points for each model variable. Central tendency and distribution for scale s cores on each instrument were calculated. A T test to compare gender and birth order for each major variable was done. A Spearman rho statistic (correlation matrix) examined the demographic variables of sibling age, family size, and age difference to each major variable. In addition, the scale scores of each major variable were related to raw scores on the CARS 2 and the total SOC score. The Friedman test was used to compare the rank of sibling support from a parent, friend, and their brother/sister with A SD. A test of the model (Figure 1 2) satisfied the secondary aim of the research and was completed after checking assumptions. To do this, all study variables were placed in the model and outliers and multicollinearity were examined by Df beta and diagnos tics A ssumptions for multiple regression including normality, linearity, and independence of error terms were not violated. The final research question was answered in two parts. First, a mediating variable, or indirect effect, was examined for resources significant to both the predictor (ASD severity) and the dependent variable (SOC). Next, a moderating or interaction effect, of each resource (coping, support from each relationship, and well being) was analyzed. Finally, significant mediating and modera ting relationships were diagramed and reported. To examine whether the significant resource acted as a mediating variable, three hierarchical regression models were used to explain the variance of each predictor on the dependent variable beyond that attrib uted by sibling age and gender. In the first
68 model the path analysis included: ASD severity regressed on SOC levels (path a ); Path b was examined in separate models by regressing ASD severity on each resource that had a significant correlation; significan t resource variables were then regressed separately on the sibling SOC levels to create path c Standardized Beta and significance, R 2 Change, and F value were reported for each regression model. Finally, the model was tested for a moderating relationship through a hierarchical regression analysis of the interaction effect separately for each resource scale score. A centered variable for the predictor (ASD severity) was computed by subtracting the predictor variable from its mean score. A centered variable for each moderator was obtained in the same manner. An interaction term was computed by multiplying the centered predictor by the centered moderator. A hierarchical linear regression model was run to examine how much different sibling SOC levels vary in relation to each of their resources and the ASD severity score. In step one, sibling age and gender were entered and in step two the predictive centered variable was entered (path a ), followed by the moderator centered variable (path b ); and the interaction term (path c ). For each model, the interaction term was evaluated for significance. A significant F ratio of the interaction term and its clinical nature was reported for each model. A plot of the mean SOC to a high and low number of each resource and a low or high level of ASD severity was graphed to express the phenomenon In summary, descriptive statistics were completed and used to answer the first three research questions. Sibling age and gender were compared to the major study variables; a conservative approach was taken reducing the from .05 to .004 according to the Bonferroni pr ocedure. The study had adequate power to do multivariate analyses
69 for six predictors and there was a large effect size. The final research question was answered via multivariate analyses. Scientific Rigor and Ethical Considerations The study was guided by a post positivist philosophical approach and findings contribute to the science of adolescent siblings of individuals with ASD. Potential relationships between variables were found through ethical investigative methods and the study was adequately powered to statistically analyze these relationships. The integrity of the study relies on psychometrically sound instrumentation, sample size, data collection procedures, and meeting assumptions for analyses. Children and individuals with disability are vulne rable populations; thus, research investigating these groups must maintain high standards. A discussion of the methods used to maintain these standard and the study limitations are presented in this section. In this study, self report by the adolescent si bling provided a direct measure of their life orientation, coping strategies, well being, and support networks. Parents adherence to confidentially of the TD sibling answers, in a written and signed consent form, allowed siblings to provide honest answers to the instrument questions. A mail based survey permitted the adolescent sibling sufficient time and added convenience researcher and further discussion of the study materials (Portney & Watkins, 2009). The benefit of sibling participation was limited to an opportunity for self inventory through answering the instrument questionnaires. A Walmart gift card in the amount of $20 was provided to each sibling who retu rned a completed research packet. While the study procedures are not overly obtrusive and risk to participants is low, the instruments do inquire about sensitive and personal matters. Thus, several minors
70 disclosed information that could be concerning to t heir parent. To protect the anonymity of the adolescent sibling, the study responses were separated from the consent forms and kept anonymous. A snapshot evaluation offered in cross sectional, mail based research may offer screening data; however, it is i nadequate for evaluating a true risk ( Achenbach, 1991 ). Integrity of data was maintained by using an encrypted laptop and transfer of the data to the research team done through a protected server. Written and electronic data will be purged seven years aft er the dissemination of study findings. Conclusion This chapter provides the research design for the investigation. Included in this are description of the target population and each instrument. In addition, the investigative procedures are delineated including the analytical plan. Finally, ethical considerations and scientific rigor for investigating the relationships between sibling resources and severity of ASD were discussed
71 CHAPTER 4 FINDINGS Siblings have a life long relationship and research on sibling interaction consistently identifies a psychosocial impact on each family member throughout the life cycle. In this investigation, families (N = 96) participated in the study if they had a typically developing adol escent sibling with a brother/sister diagnosed with autism (n = 55), Pervasive Developmental Disorder Non Specified (PDD NOS; n = 19), or information regarding the sibli being, and social support. Multivariate analyses were used to test the health promotion model of Salutogenesis (Antonovsky, 1987) and fulfill a secondary study aim. This investigation was designed to test the following hypotheses: 1. Sibling age and gender confound the relationship of the major study variables. 2. Support from friends of typically developing siblings is greater than support from parents or from their brother/sister on the autism spectrum. 3. T here are significant correlations of scale scores on the A COPE, NRI SPV, and YSR to the ASD severity score and SOC score in TD adolescent siblings of individuals with ASD. 4. Adolescent coping strategies, network support, and psychological well being scale scores as measured on the A COPE, NRI SPV, and YSR mediate and/or moderate the relationship between severity of ASD as measured by the CARS 2 and life orientation as measured by the SOC in typically developing adolescent siblings of individuals with ASD. Sample Characteristics Descriptive data for the family environment are presented in Table 4 1. Parents reported themselves as primarily White (84%) and married (87%). The m ajority of mothers (62%) and fathers (52%) reported an education beyond high school and income levels > than $60K/year (65%). Individuals with an ASD were predominantly
72 male (n = 77) and ranged in age from 2 21 years old ( m = 12, SD = 4.5). The ASD diagno ses were categorized as autism (57%), PDD syndrome (23%). Adolescent siblings were 11 18 years old ( m = 14.0, SD = 2.03) and mostly female (57%). A majority of TD, adolescent siblings were older (59%) and 47% were first born chil dren. Family size ranged from 2 to 7 siblings ( m = 2.8, SD = 1.04); and although most siblings were 2 years apart in age, the age difference between siblings ranged from 1 to 12 years (m = 3.89, SD = 2.73) in the study. Descriptive Statistics The primary aim of the study was to characterize adolescent siblings. Descriptive data satisfied this aim and answered the first three research questions. First, we hypothesized that only the demographic variable of TD sibling age and gender would influence associat ions of the study variables. Central tendency and distribution of the major variables to each demographic variable was examined (Table 4 2). A T test to compare sibling gender and birth rank (younger/older) was completed. Other demographic variables includ ing sibling age, family size, and sibling age difference were correlated to each of the study variables (Table 4 3). The second hypothesis was that support from friends would be greater than support from their sibling on the autism spectrum or their parent A Friedman test compared mean scores on the NRI SPV matrix to answer this question (Table 4 4). Finally, we anticipated a significant correlation between scale scores on each instrument to raw scores of ASD severity and adolescent sibling SOC scores (T able 4 5) and these were examined before testing for a mediating variable. Multivariate analyses using hierarchical regression examined mediating and moderating relationships. These findings are described in this section.
73 Research Question One Central ten denc ies of sibling gender and birth order can be found in Table 4 2. These categorical variables were compared to the major study variables using a T test. Continuous variables of sibling age, family size, and age difference between siblings were correlat ed to each instrument scale score (Table 4 3). These findings are described below to answer the first research question. Female siblings perceived a significantly greater amount of support from friends ( t = 3.84, p < .001) than males. Levels of sibling s upport ( t = 2.88, p = .005) and Sense of Coherence scores ( t = 2.19, p = .03) were greater for females and neared significance. Older siblings had higher scores on the sibling support scale than siblings who were younger than their brothers/sisters with an ASD ( t = 2.17 p = .03). The age of the TD sibling was correlated only to the parent support scale of the NRI SPV. Younger siblings reported a greater amount of parent al support ( r = .22, p = 03). Family size ranged between 2 and 7 children and the majority of families had 2 siblings (n = 48). Sibling age differences were between 1 12 years (mean = 3.89; SD = 2.73) and the majority were 2 years apart in age. Sibling age was the only demograp hic variable that had a significant association to SOC levels, coping strategies, support scales, or well being. Research Question Two The investigator was interested in the amount of support between the adolescent sibling and their parent, brother/siste r with ASD, and a same sex friend. The NRI SPV provided a scale score of support for each relationship type (range 1 5) and the mean score was compared using the Friedman Test (Table 4 4). Parent support had the highest mean score ( m = 3.67, SD = .69) an d ranked greater than support f ro m a friend
74 ( m = 3.38, SD = .70) and sibling ( m = 2.93, SD = .69). There was a significant difference between ranks [ = 57.80 ( df 2), p < .001] and a large association between groups because the scores are not independen t of one another. Research Question Three The total score on each instrument was correlated to ASD severity and TD sibling SOC scores to answer the third research question (Table 4 5). Only the total problem scale of the YSR was correlated to both ASD sev erity ( r = .322 p < .01) and TD sibling SOC scores ( r = .725 p < .01). Higher levels of ASD severity were associated with greater problems in TD siblings but only 9% of the variance could be attributed. As expected, there was an inverse relationship be tween total problems to TD siblings SOC levels. TD siblings with more problems had lower SOC scores indicating they had more difficulty managing tension (SOC). The NRI SPV sibling support scale score was significantly related to TD sibling SOC scores ( r = .246, p = .02). A greater amount of sibling support was associated with greater SOC scores but it had a weak and inverse relationship to ASD severity ( r = .071, p > .05). This finding suggests that positive sibling relationships contribute to TD sibling s SOC levels and higher levels of ASD severity result in less sibling support. characterizing adolescent siblings and answered the first three research questions. The first hy pothesis was confirmed from the data, and age and gender were co varied in the multivariate analyses. In addition, older, TD siblings reported greater support from their brother/sister with ASD. The second hypothesis was not supported by the study data bec ause mean scores of parent support were greater than mean scores of friend support. Finally, the third hypothesis was partially supported because the total problem
75 scale on the YSR was related to ASD severity and TD sibling SOC scores; and sibling support correlated to TD sibling SOC levels. Multivariate analyses tested the model using total problems as a mediating variable and each resource as a moderating variable. These findings are described in the following section. Multivariate Analyses Mu ltivariate analyses of the data were necessary in order to satisfy the second aim of the study and answer the final research question. The full model was built using hierarchical regression S ib ling age and gender were entered first. These variables were significant [ F =3.828 (2, 88); p = .025] and contributed 8% of variance to the model. p = .03). Next, all the scale scores were entered. Together, they significantly contributed 50% variance to the model [ F = 16.022 (6, 82); p = < .001]. Assumptions for multiple regression analyses were examined and no violations of normality, linearity, independence, and homoscaedasticity were found. Each resource was analyzed separately as a mediating and moderating vari able and these analyses are described below. Mediating Variable To test an indirect (mediating) effect of each resource variable on the relationship between ASD severity and adolescent SOC levels, significant correlations must exist between the predictor and dependent variable, predictor and mediating variable, and the mediating variable and the dependent variable (Baron & Kenny, 1986). This was true for only one resource variable sibling total problem scores. To examine the mediating effect, three hier archical regress ion models were built (Table 4 6 ). Sibling age and gender were entered into the model first followed by the raw score of ASD severity (path a ). This variable added 6% variance to the model and an inverse relationship
76 between variables was significant suggesting that greater levels of ASD severity reduced SOC scores in TD adolescent siblings (Model 1A). ASD severity contributed 10% to the model (path b ) and significantly predicted the TD sibling total problem scale of the YSR [ F = 10.561 (1 ,90), p = .002]. TD siblings had greater problem scores when there was higher ASD severity total problem score of TD siblings significantly predicted their SOC scores (path c). The sibling total problem score added 48% va riance to the model [F = 48.553 (2,89) p = < .001] and there was a strong, inverse relationship ( r = .707) suggesting that the greater the problem score, the lower SOC levels. A mediating variable was identified (Model 1D) because when TD sibling total problem score and ASD severity were entered together, ASD severity was no longer significant ( 025, p = .74). These findings mean that ASD severity (stress) predicts TD sibling well being, which mediates the relationship between ASD symptom severity and TD sibling SOC scores. Therefore, in the presence of t otal problems, siblings are at greater risk for lower SOC levels. A diagram of this relationsh ip is depicted in Figures 4 3 and 4 4. Moderating Variable A variable is identified as being a moderator if the causal relationship between two variables changes as a result of its interaction with the predictor on the dependent variable and significant co rrelations are not necessary (Baron & Kenny, 1986). Each resource variable coping, support of parent, support of friend, support of sibling, and well being was tested separately for an interaction effect and the only significant resource was found to b e coping strategies. A moderating variable affects the direction or strength of the relationship between the independent and dependent variables within
77 a correlational framework and this is described for the coping variable. A description of the model and Beta and significan ce levels are found in Table 4 3. The path analyses ( Figure 4 2 ) as described by Barron & Kenny (1986) showed insignificant (>.05) main effects (path a and path b), and a positive interaction effect (path c). The moderating model was bui lt in hierarchical regression and sibling age and gender were p ut in the model first (Table 4 6 ). These variables contributed 8% of the variance and were significant [ F = 3.941 (2, 89), p < .001]. Next, the centered predictor, centered moderator, and int eraction term were added and the standardized B eta coefficients and significance of each path were examined ( Figure 4 3 ). The findings suggest that sibling coping strategies specify the condition under which ASD severity is related to TD sibling SOC level s. The graph ( Figure 4 4 ) depicts the difference in sibling mean SOC scores when there are few coping strategies and low levels of ASD severity compared to when there are many coping strategies and high levels of ASD severity. The finding tells us that si bling coping strategies are necessary in cases of high ASD severity in order for siblings to maintain their sense of coherence. Summary The model of Salutogenesis was supported by the data in this sibling group. These associations do not indicate a causal relationship but they support the study aims and provide future direction for experiments using clinical interventions. The final section is a discussion of the dissertation study results, its strengths, limitations, and suggestions for future direction.
78 Table 4 1. Demographic Characteristics for Sibling Family Environment Age: (n) Mean Median SD Range Father (88) Mother (96) Sibling with ASD (95) Typically developing sibling (96) 45.69 42.93 12.03 13.69 14.00 7.03 5.60 4.46 2.03 25 62 31 56 2 21 11 18 N (%) Adolescent sibling Female* Male 57 (59%) 39 (41%) Individual with ASD Female Male* 19 (20%) 77 (80%) Ethnicity White Hispanic African American Other Missing 81 (86%) 4 (4%) 2 (2%) 7 (7%) 2 (2%) Family Size/ Number of children Mean (SD) One sibling Two siblings > 3 siblings 2.82 (1.04) 48 (50%) 26 (27%) 22 (23%) Type of ASD Autism PDD NOS 55 (57%) 19 (20%) 22 (23%) Marital Status Married Divorced living with s/o Divorced not living with s/o Widow 83 (87%) 8 (8%) 4 (4%) 1 (1%) Post Secondary Education Mother Father 62 (65%) 50 (52%) Employment Mothers Fathers 62 (65%) 23 (24%) Income 91,000 or > 61,000 90,000 31,000 60,000 Below 30,000 41 (43%) 21 (22%) 26 (27%) 7 (7%) Professional Treatment for ASD Yes No 73 (76%) 23 (24%)
79 Table 4 1. Continued N (%) TD Sibling Birth Rank to ASD Older Younger 61 (63%) 35 (37%) TD Sibling Birth Order Oldest Middle Youngest 49 (51%) 21 (22%) 25 (26%) Sib Age Difference 1 year 2 year 3 year 4 7 years 8 12 years 12 (11%) 23 (22%) 21 (20%) 24 (23%) 16 (15%) p < .01
80 Table 4 2 Range, mean, standard deviation and median scores on the Sense of Coherence (SOC); Adolescent Coping Orientation for Problem Experiences (ACOPE), Youth Self Report (YSR), and the Network of Relationship Inventory Social Provision Version (NRI SPV) to sibling demographics of gender and birth rank. R ange Mean (SD) Median SOC (N=96) ACOPE (N= 94) YSR (N=96) NRI SPV Parent Support (N = 96) NRI SPV Friend Support (N=95) NRI SPV Sibling Support (N=96) Gender (n) Male (39) Female (56) 24 79 57.15 (13.24) 58.00 29 90 63.28 (13.60)* 65.00 110 183 154.31 (16.68) 155.00 94 207 160.51 (22.51) 164.00 5 114 48.82 (27.43) 45.00 6 119 38.90 (25.00) 34.50 1.72 4.78 3.62 (.60) 3.61 1.56 4.89 3.72 (.71) 3.83 1.61 4.33 3.07 (.71) 3.11 2.11 4.72*** 3.60 (.62) 3.61 1.56 4.00 2.70 (.53) 2.78 1.17 4.72** 3.11 (.76) 3.25 Birth Rank (n) Younger (34) Older (59) 32 87 60.40 (12.15) 60.50 24 90 61.02 (14.63) 61.00 117 188 158.85 (16.77) 163.00 94 207 157.42 (22.36) 157.00 5 113 42.49 (26.67) 37.00 6 119 43.18 (26.36) 38.00 1.72 4.89 3.68 (.64) 3.75 1.56 4.78 3.68 (.69) 3.83 1.94 4.72 3.32 (.73) 3.39 1.61 4.61 3.41 (.69) 3.44 1.56 4.00 2.74 (.61) 2.81 1.17 4.72* 3.06 (.73) 3.11 p value < .05 ** p value < .01 *** p value < .001
81 Table 4 3. Sibling demographic variables of age, age difference, and family size are correlated to scale scores of coping, support, well being, and sense of coherence variables. The instruments include: Adolescent Coping orientation for Problem Experiences (ACOPE), Network of Relationship Inventory Social Provision Version (NRI SPV), Youth Self Report (YSR), and Sense of Coherence (SOC) Variable 1 2 3 4 5 6 7 8 9 1) Sibling Age 2) Sibling Age Difference .15 3) Family Size .11 .16 4) Coping Strategies (A COPE) .14 .15 .09 5) Parent Support (NRI SPV) .22* .02 .09 .15 6) Friend Support (NRI SPV) .04 .03 .18 .43** .15 7) Sibling Support (NRI SPV) .18 .06 .05 .18 .39** .38** 8) Total Problem Behaviors (YSR) .00 .07 .15 .01 .02 .07 .16 9) Sense of Coherence (SOC) .15 .01 .18 .08 .12 .10 .25* .68** p < .05 ** p < .01
82 Table 4 4. Results of Friedman Test are shown. Siblings perceived higher levels of support from a parent, friend, and sibling respectively Ranks of Sibling Support Variable Mean Rank Support Parent 2.48 Support Friend 2.11 Support Sibling 1.41 Table 4 5. Correlations for scale scores on the Youth Self Report (YSR), Adolescent Coping orientation for Problem Experiences (ACOPE), and the Network of Relationship Inventory Social Provision Version (NRI SPV) to Severity of Autism Spectrum Disorder (ASD) measure d by Child Autism Rating Scale 2nd Ed. and to Sense of Coherence (SOC) scores. Significant Correlations of ASD severity and SOC scores to Major Study Variables ASD Severity CARS 2 Life Orientation SOC Coping Strategies (A COPE) .045 .080 Support (NRI SPV) Parent Friend Sibling .025 .088 .071 .122 .102 .246* Total Problem Behaviors (YSR) .322** .725*** p < .05 ** p < .01 *** p < .001
83 Table 4 6 Hierarchical regression models are examined to test the model. Significant mediating and moderating variables are identified. Data displayed in Model 1 is derived from total problem scale on the Youth Self Report (YSR). ASD severity was the raw score on the Child Autism Rating Scale 2nd Ed. (CARS 2). Coping strategi es was obtained from the scale score of the Adolescent Orientation for Problem Experiences (A COPE). Test of Model/Research Question Four Model/Predictor p value R 2 F change Mediating Model for Total Problems Model 1A (path a) ASD severity Model 1B (path b) ASD severity Model 1C (path c) Total Problems Model 1D (Indirect Effect) ASD severity Total Problem .247 .319 .71 .025 .7 0 .01** .002** *** .743 *** .06 .10 .48 .48 6.354** 10.561** 99.517*** 48.553*** Moderating Model for Coping Model 2A (path a) Centered Predictor Model 2B (path b) Centered Moderator Model 2C (path c) Interaction (severity X total problems) .19 .114 .263 .03* .251 .008** .05 .01 .07 5.107* .780 7.339** p < .05 ** p < .01 *** p < .001
84 Mediating Model Figure 4 1 Beta weights and significance for mediating model: path a) the predictor (ASD severity = raw score of Child Autism Rating Scale) to outcome (Sense of Coherence); path b) predictor (ASD severity) on mediator (total problem score on the Youth Self Report; path c) mediator (total problem) to outcome (Sense of Coherence). Mediating Model Figure 4 2 S hows change in Beta and significance of predictor to outcome when predictor and media tor are regressed on SOC levels.
85 Moderating Model for Coping Strategies Figure 4 3 A path analyses to test for interaction (moderating) variable. Beta weights and significance for the centered predictor variable (ASD severity) obtained fro m the Child Autism Rating Scale, centered moderator variable (coping strategies) obtained from the Adolescent Orientation for Problem Experiences, and interaction term obtained by multiplying centered predictor and centered moderator (path c).
86 Figure 4 4. Shows the interaction of severity of autism spectrum disorder (ASD) and scores. SOC levels are significantly reduced when ASD severity is high and coping strategies are low. A reference line shows mean SOC score of Israeli adolescent boys (Margalit & Eysenck, 1990). Mean SOC score __ Adolescents
87 CHAPTER 5 DISCUSSION with an autism spectrum disorder (ASD) by describing their resources and sense of coherence (SOC). In a secondary study aim, the model of Salutogenesis (Antonovsky, 1987) was tested. A discussion of the study findings and their relation to the published r esearch are included in this chapter. In addition, the clinical and theoretical implications related to the study findings are addressed, and recommendations for future research are provided. Finally, the implications for nursing are described. Findings Related to Previous Research Major variables from previous research of siblings of individuals with an ASD include sibling adjustment and relationship quality as well as social support and coping strategies. First, data related to sibling demographics, or attributes are described. Next, being are related to earlier research. Finally, in this section, the conceptual framework of three constructs stress, resources, and sense of coherence and data supporting them are presented. Sibling Attributes Sibling age and gender were covaried in the study because the stages of child development and sibling gender are known to influence individual perceptions of well being, support, and coping strategies (Glasberg, 2000, Giallo & Gavidia Payne, 2006; Gupta & Singhal, 2005, McHale et al., 1986; Verte et al., 2003). In this study, a cohort of adolescent siblings, 11 18 years old participated ( m = 14.0 years, SD = 2.3); age and gender c ontributed 8% of the variance to the full model.
88 It is possible that categories of early, middle, and late adolescence would provide more information although no cut off points were used to categorize the scale level variable. Based on the correlative fi ndings, older siblings held more positive views of their siblings than younger (coded 0) TD siblings ( t = 2.17, p = .03). This finding is in consistent with McHale (2001) whose research team investigated an adolescent group from two parent families and foun d younger siblings had less conflict. It has also been reported that older siblings have better relationships with the child with ASD (Hastings, 2003; Kaminsky & Dewey, 2001; Macks & Reeve, 2007; Orsmond et al., 2009). Consistent with this, the study sampl e had a greater number of siblings who were older (n = 61) and their birth rank was significantly different ( t = 2.17 p < .03) in relation to sibling support The age difference between siblings was examined; and, while 40% (n = 40) of the siblings were f our or more years apart in age, this did not correlate with the major study variables. Orsmond et al. (2009) found that siblings closest in age had greater levels of depression; therefore, creating a cutoff point and categorizing the age difference variab le may have provided more information. The majority of siblings were female (60%) and they reported higher SOC scores ( m = 63.28, SD 13.60 ) and lower total problem scores than the males ( m = 57.15, SD 13.24) They also perceived significantly (p < .001) greater amount of support from parents and friends. These findings are consistent with Kaminsky & Dewey (2001) and McHale & Gamble (1986) who described female siblings of a person with disabilities as having greater relationship quality with their parent s and sibling than the male comparison group. Several explanations for this include the fact that females may exhibit better social skills and/or they have less phenotypic expression of genetic
89 susceptibility to ASD (Constantino, et al., 2006; Seltzer, et a l., 2004). Screening for a benign autism phenotype (BAP) was not completed for study participants although TD siblings were excluded if parents reported they had a diagnosed learning disability. The number of children in the home determined family size. Fifty percent of the adolescent siblings (n = 48) reported having only their brother/sister with ASD; 25% had two siblings; and the remaining study participants (n=22) had three or more siblings. Despite this variability, sibling family size did not signi ficantly correlate to the major study variables. This finding is in contrast with Kaminsky & Dewey (2002) who reported data analyses are necessary to determine the imp act of family size on sibling resources and SOC. Coping Strategies The Salutogenic model (Antonovsky, 1987) is a coping theory and was used as the framework of this study. Total scores on the Adolescent Coping Orientation for Problem Experiences (A COPE; Patterson & McCubbin, 1987) had a significant interaction effect with ASD severity ( = .263, p < .01) and supported the theory. Therefore, TD siblings who had brothers/sisters with high levels of ASD severity (higher stress) had higher SOC levels if they utilized a greater number of coping strategies. The A COPE was not utilized in prior research investigating siblings of individuals with ASD and this makes it is difficult to compare findings related to TD sibling coping. In a qualitative investigation, Dellve et al., (2000) categorized coping strategies from strategies were related to contextual concepts of sibling requirements and sibling concerns Siblings had a
90 two most common cop ing strategies regardless of the type of sibling interaction. Finally, Orsmond (2007) found siblings had better relationships with their brother/sister on the spectrum when they engaged in problem focused rather than emotion focused coping strategies. Th ese comparative studies on coping strategies substantiate that the demands of ASD are great and provide information necessary to design future intervention studies. For example, siblings may find that they are better able to cope if they are taught to iden tify and articulate the problems encountered from having a sibling with ASD rather than just respond to them emotionally. This could have important positive implications over a lifetime of interactions with their sibling with ASD. Relationship Networks A m atrix of support from a parent, same sex friend, and their sibling with an ASD was useful for characterizing TD, adolescent siblings because the data were not consistent with other sibling groups. The reasons remain unclear but several explanations are of fered. Adolescents have previously reported greater levels of perceived support from friends than from parents (Stice, Ragan, and Randall, 2004, Noack & Buhl, 2004) and greater support from siblings than friends (Furman & Buhrmester, 1985). Consistent wi th Kaminsky & Dewey (2002), parent support scores were higher than friend support scores especially among females although only sibling support was significantly correlated ( r = .24, p = .02) to TD sibling SOC scores. Despite these associations, sibling s upport did not predict TD sibling SOC levels and this was likely because sibling gender was controlled by hierarchical regression analysis. Finally, siblings who
91 reported greater levels of sibling support also reported higher parental support ( r = .391, p < .001). These findings are consistent with Orsmond et al. (2009) who reported that adult siblings who regarded their sibling relationship as positive also reported higher levels of parental support. Because siblings are receiving greater support from parents than friends the study reveals that parents are working hard to meet the TD sibling demands and those related to ASD. Twoy, et al., (2004) found lower means scores from parents in acquiring social support, seeking spiritual support, and passive ap praisal when compared to normative data. Consistent with previous research (Smith & Elder, 2010), TD siblings are influenced by the behavior of parents ; thus, their expression of social behavior may be similar. In addition, care giving demands (Dyson, 199 skills (Gupta & Singhal, 2005) may interfere with the TD sibling social interactions outside the home. In consideration of these findings from previous research and social support from f amily members and friends greater in adolescents ( p < .001) than in the adult group, interventions mus t begin early lifetime. Participation in group sports or activities could be very beneficial for improving social skills, nurturing the sibling relationship, and developing support from friends. Sibling Well B eing Psychological well being, measured by the total prob lem scale on the Youth Self Report (Achenbach, 2001), was consistent with adolescent siblings in the general population (Kim, et al., 2007). When total problem scores were converted to T scores, 16% of the sample was noted to be at risk and 84% were not at risk. Although male siblings reported a greater number of problems, there were no significant gender
92 differences ( t = 1.836, p = .07). Consistent with prior research, severity of the ASD was significantly correlated ( r = .32, p < .01) with the TD siblin (Benson, 2007; Hastings, 2007). Of interest, this association was stronger than the relationship of ASD severity to the TD siblings SOC scores ( r = .21, p total problem scores were found to have a strong and sig nificant association ( r = .725, p Antonovsky, 1987) instrument. Furthermore, it mediated the relationship of ASD severity as measured by the Child Autism Rating Scale 2 nd Edition (CARS 2; Schopler et al., 2010) to sibling SOC levels. This finding indicates that TD adolescent siblings who have more problems are influenced to a greater degree by the ASD severity. No interaction effect was found. In summary, sibling demo graphics of age, gender, birth rank, sibling age difference, and family size were examined by analyzing each to sibling coping, support, and well being. Female siblings consistently reported significantly (p < .001) greater levels of relationship quality t han their male counterparts. Both genders utilized a greater number of coping strategies when ASD severity levels were high. The number of adolescent total problems and sibling SOC levels had a significant and inverse relationship ( r = .725, p < .001). T his finding makes theoretical sense because sibling disposition and manage tension (SOC). A further discussion of the theoretical framework follows. Conceptual Framew ork The model of Salutogenesis (Antonovsky, 1987) is recursive to the sense of coherence concept (Figure 1 1). In this study, the model was modified and the sense of
93 coherence (SOC) was the dependent variable (Figure 1 2). Three concepts stress, resourc es, and life orientation relationships. It was hypothesized that ASD severity (the stressor) was related to the be pre dicted by the severity of the stressor and sibling SOC level. Stress Previous research has compared ASD severity to parental stress, sibling adjustment, and sibling relationship quality (Davis & Carter, 2008; Fisman et al., 2000; Hastings, 2003; Orsmond, 2007). A diagnosis of Autism Spectrum Disorder by definition assumes a wide variety of symptoms in three general areas of function: communication, sensory, and social function (CDC, 2010). Lardieri, Blacher & Swanson (2000) and other researchers conclude d that behaviors associated with the disorder make it more difficult for siblings and parents to adapt. In contrast, Pilowsky et al. (2004) found no difference when comparing siblings of ASD to siblings without a disabled brother/sister. These researchers reported that 87% of siblings of individuals with ASD had no adjustment difficulty and their findings are supported by data from this study. Clearly, ASD impacts each family member and siblings are no exception. The mean score (34.54, SD = 7.84) on the Child Autism Rating Scale 2 nd Ed. (CARS 2; Schopler et al., 2010) indicates that the individual with ASD had symptoms for a moderate impairment in communication, social, and behavioral indices in the study sample. There was a significant and inverse correl ation to sibling SOC scores ( r = .213, p = .03) suggesting that higher levels of ASD severity reduced TD sibling SOC scores. ASD severity was not found to be a major predictor of sibling coherence total problems or sibling support because the raw score of ASD severity contributed only 5% of
94 variance to the TD sibling SOC score; 9% to the sibling total problem score, and 4% to sibling support scores. Thus, the majority of siblings are adapting, managing, and relating well to their brother/sister with ASD. Resources Each sibling resource was correlated to ASD severity and sibling SOC scores. (<. 05) related to ASD severity. The scores for sibling coping strategies, support from their brother/sister with ASD, and total problems had significant associations (p < .05) to their SOC scores It makes theoretical sense that resources predi (SOC); it is more difficult to explain how the ASD severity predicts sibling resources because severity remains fairly stable over time. Rather, sibling resources are more likely predicted by the ASD severity. Whil total problems and coping scores were associated a bidirectional relationship is likely, and clinically, we cannot generalize the study findings due to the cross sectional study design. Sense of Coherence nsion, measured by their SOC scores, was the dependent variable in the theoretical framework. It was significantly related to ASD severity (r = .21 p = .03), as measured on the CARS 2; the total problem score ( r = .676, p = .003) on the YSR (Achenbach, 2001) and sibling support ( r = .246, p = .02) on the NRI reported that adolescent boys had greater SOC scores than their female peers. In this study sample, girls had greater scores tha n boys ( t = 2.19, p = .03). The mean SOC score in this study sample was 60.79 ( SD = 13.72) and this finding shows that siblings
95 are managing the stress of ASD quite well. While this is reassuring data, the resources investigated in the study could account for only 50% of the variance. In summary, the conceptual framework of the Salutogenesis model guided the research and was supported by data from the study but was limited by the research design. Stress was operationalized as ASD severity and, as expected, was significantly (p < .05) related to sibling SOC scores. Although stress (ASD severity) predicted one sibling resource, total problem scores, it is likely that a bidirectional relationship exists. Strengths, Limitations, & Directions for Future Research This investigation contributed to the science of siblings of individuals with an autism spectrum disorder by describing adolescent coherence levels, coping strate gies, relationship networks, and well being. In addition, the associations between stress, sibling resources and coherence were explored. Theoretical underpinnings provided a meaningful data analyses although additional sibling resources that correlate w ith sibling SOC levels need to be identified in future research. A discussion of these follows. Strengths Few studies have investigated adolescent siblings of individuals with an ASD and rich data was obtained in a direct self report methodology that has been shown to be more accurate than parent report (Verte et al., 2003). A large homogenous group throughout the United States completed mail based surveys providing siblings sufficient time and confidentiality as evidenced by signed consent and low amount of missing data. Adequate statistical power helped to ensure meaningful data of multivariate analyses to answer the research questions. Furthermore, the theoretical model of Salutogenesis (Antonovsky, 1987) was supported by the study data. Thus, sibling
96 resources that are significant to their SOC levels can be utilized to develop clinical interventions in future experimental research. Limitations A cross sectional study design cannot evaluate causation between relationships; thus, ASD severity as a pred ictor of each sibling resource is based on theory alone. Although there was a correlation and indirect effect of the TD sibling problems on the relationship of ASD severity to sibling SOC, one cannot generalize the findings because ASD severity is fairly stable and would need to be measured in a longitudinal design to establish its predictability. A second limitation of the study was inclusion/exclusion criteria (Appendix A). Although siblings with cognitive learning disabilities were excluded, siblings w ith psychiatric and chronic medical conditions were not. These conditions may indicate the presence of the benign autism phenotype (BAP), or a genetic inheritance of features that resemble one or more of the characteristics of ASD but are not severe enoug h to be diagnosed as ASD (Mazefsky et al., 2008; Pilowsky et al., 2004). Earlier studies have shown a relationship between the BAP and adjustment in siblings of individuals with ASD (Orsmond & Seltzer, 2009). In order to better understand to what extent th e environment influences siblings, siblings from families with more than one sibling on the spectrum could have been excluded (Virkud et al., 2008). Self report instruments as the only data source can limit the accuracy of the study findings and is a lim itation of this study. Multiple data sources can be obtained (direct observation, parent/teacher report) to address this but due to financial and time constraints, self report was the only method utilized in data collection. The use of required parental c onsent can introduce bias into sample selection and also limit the
97 generalizability of the research findings (Portney & Watkins, 2008). However, the nature of the study variables required parental consent for minor age siblings. In addition, a mail based survey cannot guarantee independent completion of the study instruments. It is possible that a non sibling completed the survey. This factor is not accounted for in the studies analyses and therefore is a study limitation. The composition of the sample i s a final study limitation. The use of a non random sample can limit external validity of the research findings and a purposive convenience sample was utilized to maximize the number of study participants. Although the study reached adequate power for sta tistical analyses (N=96), its findings cannot be generalized to all adolescent siblings of individuals with a brother/sister on the autism spectrum because the re was inadequate representation of minority groups or siblings from lower socioeconomic status. Future Direction Two implications for future research are offered. The first suggestion is based on study findings and the second is offered based on what was not accomplished in this study. Coping strategies was a moderating variable and siblings requ ired a greater number of them if their brother/sister had high ASD severity. The analysis of coping was limited to the total score rather than specific subscales. The majority of research in children and adolescents has focused on differences between indi viduals within a population in order to examine what types of coping contribute to successful adjustment (Blount, et al., 2008; Skinner & Zimmer Gembeck, 2007). A future direction for investigation would employ a quasi experimental research design and sys tematically test specific coping strategies under consistent conditions.
98 The study model identified that the sibling resources (coping, well being, and support networks) together contributed approximately 50% of the variance and well being was the greatest factor. Other resource variables that may have added to the variance and the science include the amount of sibling care giving, knowledge of the ASD, and an indicator for sibling self esteem and/or self concept. This cross sectional inv estigation identified sibling resources that correlated to ASD severity and TD sibling SOC levels. A next step in understanding this group requires further investigation of the instrument subscales. This information will help to develop an intervention f or enhancing TD sibling relationships with their sibling on the spectrum and/or their friends. In addition, a longitudinal, quasi experimental research design will be helpful in determining the impact of ASD severity on sibling SOC levels. Conclusion and Implications for Nursing Siblings appear to have bonds that are unique from other relationships and may play an important role for an individual with ASD. In clinical practice today, it is unusual for siblings to receive interventions specifically for them Generally speaking, the treatment plan is limited to parents. This is unfortunate since genetics, common birth ri ght and similar environmental backgrounds are contributing factors that influence their interactions and should be evaluated when providin g comprehensive care within the family environment. Siblings of individuals with ASD have surprisingly high levels of coherence and this author believes it stems from their support and protection of their brother/sister with ASD. In this study, parents we re found to be highly supportive of the TD sibling but they already have great demands on them. Thus, the TD siblings play an important role in
99 the treatment and management of the ASD and may require assistance from health care professionals or sibling sup port groups. The study findings did not identify specific criteria for intervention but they did support previous research that TD siblings who are younger, male and who have brother/sisters with high ASD severity are at the greatest risk for having diffi culty managing tension. Nurses are well positioned to help families identify and address the needs of TD siblings as well as family members with ASD. Furthermore, they can participate in future research designed to evaluate sibling interactions and support networks. This is vital for improving sibling well being and enhancing the quality of a unique sibling bond s that can last a lifetime.
100 APPENDIX A QUESTIONAIRE 1. Is there a family member w ho has a diagnosis of autism, Asperger syndrome, or pervasive developmental disorder non specified (autism spectrum disorder)? 2. Are you the legal guardian of the individual with ASD? 3. Are you the legal guardian of the adolescent sibling? 4. Do all the famil y members read and write English? 5. Is the adolescent brother or sister (sibling) between 11 18 years old? 6. Have they lived in the same home with the family member who has an autism spectrum disorder for a minimum of one year? 7. Has the adolescent sibling been diagnosed with a learning disability, chronic medical condition, or psychiatric diagnosis? Positive responses to the above questions with the exception of question 7 meet the study inclusion and exclusion criteria. Inclusion criteria for typically develo ping adolescent siblings include: A written consent from their legal guardian; The primary caregiver (parent) of the individual with ASD and the adolescent are able to read and write in English; The adolescent may be a biologic or non biologic sibling b ut must currently reside or has had previous residence of > one year with their sibling with ASD. Exclusion criteria include: The adolescent sibling has a learning disability Only one adolescent sibling enrolled per family A twin sibling
101 APPENDIX B INSTRUMENTS Demographic Information Please complete the following information about all members of your household: YES or NO GENDER AGE Father _____________________________________________________________________ Mother ____________________________________________________________________ Child ____________________________________________ __________________________ Child ______________________________________________________________________ Child ______________________________________________________________________ Child _____________________________________________________________________ Child ______________________________________________________________________ Have any children in your family been diagnosed with autism, a language disorder, learning disability, developmental problem, attention problems or a chronic illness? Yes ___________ No ___________ If YES, what specific illness (es) or type of problem (s) and which child (ren) is affected? Type of Problem ______________________________________________________________________ ____________________________________ __________________________________ ______________________________ What is your race or ethnic background? ___________________________________________________ Are any of your children adopted? YES ___________ NO__________ If YES, please speci fy which child (ren) is/are adopted. What is your family income? ________below 30,000 ________61,000 to 90,000 ________31,000 to 60,000 ________91,000 or above FOR MOTHER : What is your present marital status? (please check all that apply) ________Married ______ Separated ________Living with someone ______Never married and not living with someone ________Divorced ________Widowed
102 From the list below, please indicate the highest level of education that you have completed a) Some high school b) High school diploma c) Some post secondary, but no diploma or degree d) Post secondary diploma (ex. Technical school) e) University degree What is your occupation? ___________________________________ ______________________ Are you currently employed? Yes_____ No _____ _____ Are you involved in a professional treatment plan for your child? Yes ________NO________ __ If these include treatments include therapy, about how many hours in a week do you spend administering the therapy or providing accommodations for your child to receive them? (ex. practicing new lessons, travel, waiting) _______________________________ _______________________________________ ______________________________________________________________________ ______________________________ Aside from your time spent in the above question, about how many hours in a day are you involved with extra care you r child needs secondary to his autism? _____________________________________________ _________________________ FOR FATHER : What is your present marital status? (Please check all that apply) ________Married ______ Separated ________Living with someone ______ Never married and not living with someone ________Divorced _______Widowed From the list below, please indicate the highest level of education that you have completed a) Some high school b) High school diploma c) Some post secondary, but no diploma or degree d) Post secondary diploma (ex. Technical school) e) University degree What is your occupation? _________________________________________________________ Are you employed? Yes______ No______ Is your child participating in therapy related to their disability? Yes_______No_________ Are you involved in the treatment plan? Yes ________NO____________ About how many hours in a week do you spend related to these therapies? (Ex. Driving, practicing new les so Aside from your time spent in the above question, about how many hours in a day are you involved with extra care your child needs secondary to his autism?________________
103 The Network of Relationships Social Provision Version NRI SPV (Furman & Buhrmester, 1985) ID # ____ ____ ____ ____ Everyone has a number of people who are important in his or her life. These questions ask about your relationships with each of the following people: your parent your brother/sister with d isability, and a same sex friend. The first questions ask you to identify your parent, sibling and fri end about whom you will be answering the questions. 1. Circle the parent figure you will be describing. A. Biological/Adopted Mother B. Step C. Biological/Adopted Father D. Step E. Other _______________________ 2. How old is you sibling with an autism spectrum disorder? years old. 3. Now we would like you to choose the most important same sex friend you have now. Do not choose your sibling. How long is the friendship? _____ years ____ months ( please fill in numbers ). Now we would like you to answer the following questions about the people you have selected above. Sometimes the answers for different people may be the same but sometimes they may be different. 9. How much free time do you spend with this person? Littl e or None Some what Very Much Extre mely Much The Most Parent 1 2 3 4 5 Sibling 1 2 3 4 5 Friend 1 2 3 4 5
104 Description The NRI SPV (Furman & Buhrmester, 1985) has ten scales with three items per scale. It assesses 7 support features, 2 negative interaction features, and relative power. Scales Companionship (COM) 9 How often do you spend fun time with this person? 19 How often do you and this person go places and do things together? 29 How often do you play around and have fun with this person? Conflict (CON) 10 How often do you and this person disagree and quarrel with each other? 20 How often do you and this person get mad at or get in fights with each other? 30 How often do you and this person argue with each other? Instrumental Aid (AID) 11 know? 21 How much does this person help you figure out or fix things? 31 How much does this person help you when you need to get something done? Antagonism (ANT) 12 22 How much do you and this 33 How much do you and this person hassle or nag one another? Intimate Disclosure (DIS) 13 23 How often do you tell this person everything that you are going through? 33 How often do you share secrets and private feelings with this person? Nurturance (NUR) 14 24 How much do you protect and look out for this person? 34 How much do you take care of this person? Affection (AFF) 15 How much does this person like or love you? 25 How much does this person really care about you? 35 How much does this person have a strong feeling of affection (loving or liking) toward you? Reassurance of Worth (WOR) 16 26 36 How much does this person like or approve of the things y ou do? Relative Power (POW) 17 Who tells the other person what to do more often, you or this person? 27 Between you and this person, who tends to be the BOSS in this relationship? 37 In your relationship with this person, who tends to take charge and decide what should be done? Reliable Alliance (ALL) 18 How sure are you that this relationship will last no matter what? 28 How sure are you that your relationship will last in spite of fights? 38 How sure are you that your relationship will continue in the years to come?
105 Sense of Coherence Short Form 13 SOC 13 (Antonovsky, 1987) 1 Very seldom or never 2 3 4 5 6 7 Very often 2. Has it happened in the past that you were surprised by the behavior of people whom you thought you knew well? 1 Nev e r happened 2 3 4 5 6 7 Always happened 3. Has it happened that people whom you counted on disappointed you? 1 Never happened 2 3 4 5 6 7 Always happened 4. Until now your life has had: 1 No clear goals or purpose at all 2 3 4 5 6 7 Very clear goals and purpose 5. 1 Very often 2 3 4 5 6 7 Very seld om 6. Do what to do? 1 Very often 2 3 4 5 6 7 Not very
106 often 7. Doing the things you do every day is: 1 A source of deep pleasure and satisfaction 2 3 4 5 6 7 A source of pain and boredom 8. Do you have very mixed up feelings and ideas? 1 Very often 2 3 4 5 6 7 Very seldom or never 9. Does it happen that you have feelings inside you would rather not feel? 1 Very often 2 3 4 5 6 7 Very seldom or never 10. Many people even those with a strong character sometimes feel like sad sacks (losers) in certain situations. How often have you felt this way in the past? 1 Never 2 3 4 5 6 7 Very often 11. When something happened have you generally found that: 1 You over estimated or under 2 3 4 5 6 7 You saw things in the
107 estimated its importance right proportion do in your daily life? 1 Very often 2 3 4 5 6 7 Seldom or never control? 1 Very often 2 3 4 5 6 7 Very seldom or never
108 Adolescent Coping Orientation for Problem Experiences A COPE (McCubbin & Patterson, 1987) Scale items: When you face difficulties or feel tense, how often do you... 1. Go along with parent's requests and rules 2. Read 3. Try to be funny and make light of it all 4. Apologize to people 5. Listen to music stereo, radio 6. Talk to a teacher or counselor at school about what bothers you 7. Eat food 8. Try to stay away from home as much as possible 9. Use drugs prescribed by doctor 10. Get more involved in activities in school 11. Go shopping, buy things you like 12. Try to reason with parents and talk things out, compromise 13. Try to improve yourself (get body in shape, get better grades, etc.) 14. Cry 15. Try to think of the good things in your life 16. Be with a boyfriend or girlfriend 17. Ride around in the car 18. Say nice things ("warm fuzzies") to others 19. Get angry and yell at people 20. Joke and keep a sense of humor 21. Talk to a minister/priest/rabbi 22. Let off steam by complaining to family members 23. Go to church 24. Use drugs (not necessarily prescribed by a doctor) 25. Organize your life and what you have to do 26. Swear 27. Work hard on school work or school projects 28. Blame others for what's going on 29. Be close with someone you care about 30. Try to help other people solve their problems 31. Talk to your mother about what bothers you 32. Try, on your ow n, to figure out how to deal with your problems or tension 33. Work on a hobby you have (sewing, biking, etc.) 34. Get professional counseling (not a school teacher or school counselor) 35. Try to keep up friendships or make new friends 36. Tell yourself the problem(s) is not important 37. Go to a movie 38. Daydream about how you would like things to be 39. Talk to a brother or sister about how you feel 40. Get a job or work harder at one 41. Do things with your family
109 42. Smoke 43. Watch T. V. 44. Pray 45 Try to see the good things in a difficult situation 46. Drink beer, wine, liquor 47. Try to make your own decisions 48. Sleep 49. Say mean things to people, be sarcastic 50. Talk to your father about what bothers you 51. Let off steam by complaining to y our friends 52. Talk to a friend about how you feel 53. Play video games (Space Invaders, Pac Man), pool, pinball, etc. 54. Do a strenuous physical activity (jogging, biking, etc.) USE THE FOLLOWING RESPONSE CATEGORIES. 1. Never 2. Hardly 3. Sometimes 4. Often 5. Most of the time file:///C|/CHIPTS_local/assessment/Assessment_Instruments/Assessment_files_new/as sess_acope.htm (2 of 3) [4/7/2003 10:18:11 AM] CHIPTS Adolescent Coping Orientation for Problem Experiences
110 APPENDIX C INFORMED CONSENT FORM I NFORMED C ONSENT F ORM to Participate in Research I NTRODUCTION Name of person seeking your consent: Place of employment & position: This is a research study of adolescent siblings of individuals with an autism spectrum disorder. Could participating in this study offer any direct benefits to you? No as described on page 4. Could participating cause you any discomforts or are there any risks to you? No, as described on page 4. Please read this form which describes the study in some detail. I or one of my co workers will also describe this study to you and answer all of your questions. Your participation is entirely voluntary If you choose to participate you can change your mind at any time and withdraw from the study Y ou will not be penalized in any way or lose any benefits to which you would otherwise be entitled i f you choose not to participate in this study or to withdraw If you have questions about your rights as a research subject, please call the University of Florida Institutional Review Board (IRB) office at (352) 273 9600 If you decide to take part in this study, please sign this form on page 8 and/or 9.
111 G ENERAL I NFORMATION ABOUT THI S S TUDY 1. Name of Participant ("Study Subject") ___________________________________________________________________ 2. What is the Title of this research study? The Relationship between Resources and Sense of Coherence in Adolescent Siblings of Individuals with Autism Spectrum Disorder 3. Who do you call if you have questions about this research study? Laura O. Smith at 727 641 3890 or email firstname.lastname@example.org 4. Who is paying for this research study? The sponsor of this study is the University of Florida 5. Why is this research study being done? We do not know very much about the pre teen and teenaged brothers and sisters of children with autism spectrum disorders (ASD). The purpose of this research is to describe their psychological well being (such as frustrations, worries, behaviors, or other feelings), their support networks (which include meetings or friends where they can talk to others about their living situations and the good and bad parts of their every day living situations), their coping mechanisms (strategies used to feel better about what they may believe in a difficult situation), and coherence levels (their ability to manage tension). You are being asked to be in this research study because you have a child and/or a evelopmental Disorder Non problem as an ASD.
112 W HAT C AN YOU E XPECT IF YOU P ARTICIPATE IN THIS S TUDY ? 6. What will be done as part of your normal clinical care (even if you did not participate in this research study)? Normal clinical care is not being offered as part of this study. What will be done only because you are in this research study? Part I The legal guardian or parent of the child with ASD. If you are the parent or legal guardian, you will be asked to complete two forms. A demographic information sheet (which will tell us information about you such as your age, your education, your marital st atus and your employment), and a questionnaire that will help us to understand the severity of the ASD (this is referred to as the Child Autism Rating Scale 2 nd Edition [CARS 2]). These forms may be completed in person, over the telephone, or you may wis h to mail them back. Your response to these forms will be kept confidential. Furthermore, responses made on each of the questionnaires completed by your child with ASD will be kept confidential. This is further explained in the final paragraph of Question 17. Part II The adolescent sibling: If you are the brother or sister of a child with ASD, you will be asked to complete 4 questionnaires independent of your parent. You sho uld be able to complete all four of these forms at one interval. It is likely to take less than one hour to complete them. Your answers will not be shared with your parent. This is further explained in the final paragraph of Question 17. 8. How long will you be in this research study? Part I It should take no longer than 20 45 minutes to complete the demographic data sheet and the CARS 2. When you have completed these forms you have completed your participation in the study. Part II It should not take more than 1 hours to complete the four questionnaires. When the questionnaires have been completed, you have completed your participation in this study. 9. How many people are expected to take part in this research study? It is anticipated that 5 0 s iblings and 50 parents/legal guardians will take part in this study. Approval has been given for 160 siblings and families.
113 W HAT ARE THE R ISKS AND B ENEFITS OF THIS S TUDY AND W HAT ARE Y OUR O PTIONS ? 10. What are the possible discomforts and risks from taking part in this research study? There is a minimal risk of harm to you by participating in this study Other p ossible risks to you may include: Sensitive information about your thoughts and/or feelings may be uncomfortable for you to discuss. Researchers will take appropriate steps to protect any information they collect about you. However, there is a slight risk that information about you could be revealed inappropriately or accidentally. Depending on the nature of the information, such a re lease could upset or embarrass you. Questions 17 21 in this form discuss what information about you will be collected, used, protected, and shared. Throughout the study, the researchers will notify you of new information that may become available and m ight affect your decision to remain in the study. If you wish to discuss the information above or any discomforts you may experience, please ask questions now or call the PI or contact person listed on the front page of this form. 11a. What are the po tential benefits to you for taking part in this research study? There are no expected benefits to you for taking part in this study. 11b. How could others possibly benefit from this study? Siblings of individuals with chronic illness or ASD may identify r esources that are useful to overcome challenges associated with stressful family environments. 11c. How could the researchers benefit from this study? In general, presenting research results helps the career of a scientist. Therefore, Laura O. Smith PhD ( c), FNP C, RN may benefit if the results of this study are presented at scientific meetings or in scientific journals. 12. What other choices do you have if you do not want to be in this study? Your participation is completely voluntary. There is no penalty for not being a research participant.
114 13a. Can you withdraw from this study? You are free to withdraw your consent and to stop participating in this study at any time. If you do withdraw your consent, you will not be penalized in any way. If yo u decide to withdraw your consent to participate in this study for any reason, please contact Laura O. Smith at 727 41 3890 If you have any questions regarding your rights as a research subject, please call the Institutional Review Board (IRB) office at (352) 273 9600 13b. If you withdraw, can information about you still be used and/or collected? If you withdraw from this study, no further information will be collected from you. 13c. Can the Principal I nvestigator withdraw you from this study? You may be withdrawn from the study without your consent for the following reasons: A large number of random answers are missing on the questionnaire. W HAT ARE THE F INANCIAL I SSUES IF Y OU P ARTICIPATE ? 14. If you choose to take part in this research study, will it cost you anything? No. 15. Will you be paid for taking part in this study? Each participating sibling will receive a $20.00 gift card to WalMart. 16. What if you are injured because of the study? If you are injured as a direct result of this study, the PI, Laura O. Smith, will pay for all reasonable and necessary medical expenses required to treat your injury as long as the injury occurs during the co urse of the study and results directly from completing the questionnaires in the study. The sponsor (University of Florida) and the Principal Investigator will determine whether your injury is related to your participation in the study. No additional comp ensation is offered. The Principal Investigator and others involved in this study may be University of Florida employees. As employees of the University, they are protected under state law, which limits financial recovery for negligence. Please contact the Principal Investigator listed in question 3 of this form if you experience an injury or have questions about any discomforts that you experience while participating in this study.
115 17. How will your health information be collected, used, and shared? If you agree to participate in this study, the Principal Investigator will create, collect, and use private information about you and your health. This information is called protected health information or PHI. In order to do this the Principal Investigato r needs your authorization. The following section describes what PHI will be collected, used and shared, how it will be collected, used, and shared, who will collect, use or share it, who will have access to it, how it will be secured, and what your right s are to revoke this authorization. Your protected health information may be collected, used, and shared with others to determine if you can participate in the study, and then as part of your participation in the study. This information includes your nam e, your contact information, and your responses on the surveys you complete. Other information includes information such as age, primary language and the mental health information that your provide. This information will be stored in locked filing cabine ts in the office of Laura Smith or on computer servers with secure passwords, or encrypted electronic storage devices. Scores on the questionnaires will be collected individually. A research packet will be given a number and this number will be used on e ach form instead of your name. No one will know your answers to the questions. However, the identification number on each form can be traced back to the signed consent form. The information you provide will be kept confidential. The parent will NOT view the response of the adolescent. The adolescent will NOT be given the information the parent provides. This information will be stored in a locked filing cabinet or on computer servers with secure passwords, or encrypted electronic storage devices. Some other research purposes. If so, the limited data set will only include information that does not directly identify you. For example, the limited data set cannot include your name, address, telephone number, social security number, photographs, or other codes that link you to the information in the limited data set. If limited data sets are created and used, agreements between the parties creating and receiving the limited data set are required in order to protect your identity and confidentiality and privacy. 18. For what study related purposes will your protected health information be collected, used, and shared with others? Your PHI may be collected used and shared with othe rs to make sure y ou can p articipat e in the research, through your participation in the research, and to evaluate the results of the research study. More specifically, your PHI may be collected, used, and shared with others for the following study related purpose(s): strategies, support networks, psychological well being and coherence levels to the severity of the ASD.
116 Once this information is collected, it becomes pa rt of the research record for this study. 19. Who will be allowed to collect, use, and share your protected health information? Only certain people have the legal right to collect, use and share your research records, and they will protect the privacy a nd security of these records to the extent the law allows. These people include the: The study Principal Investigator, Laura O. Smith, PhD (c), FNP C, RN and research staff associated with this project. Professionals at the University of Florida or Shands Hospital that provide study related treatment or procedures. University of Florida Institutional Review Board (IRB; and IRB is a group of people who are responsible for lookin after the rights and welfare of people taking part in research). 20. Once coll ected or used, who may your protected health information be shared with? The study findings will be reported as a group without personal identifiers by the researchers. These may be written in professional publications, verbal presentations, and electroni c discussion for any persons interested in the topic. Your PHI may be shared with: The study sponsor, The University of Florida United States and foreign governmental agencies who are responsible for overseeing research, such as the Food and Drug Administration, the Department of Health and Human Services, and the Office of Human Research Protections. Government agencies who are responsible for overseeing public health concerns such as the Centers for Disease Control and federal, state, and local h ealth departments. Otherwise, your research records will not be released without your permission unless required by law or a court order. It is possible that once this information is shared with authorized persons, it could be shared by the persons or age ncies who receive it and it would no longer be protected by the federal medical privacy law. 21. If you agree to take part in this research study, how long will your protected health information be used and shared with others? Your PHI will be used and s hared with others until the end of the study. You are not required to sign this consent and authorization or allow researchers to collect, use, and share your PHI. Your refusal to sign will not affect your treatment, payment, enrollment, or eligibility for any benefits outside this research study. However,
117 you cannot participate in this research unless you allow the collection, use and sharing of your protected health information by signing this consent and authorization. You have the right to review and copy your protected health information. However, we can make this available only after the study is finished. You can revoke your authorization at any time before, during, or after your participation in this study. If you revoke it, no new informati on will be collected about you. However, information that was already collected may still be used and shared with others if the researchers have relied on it to complete the research. You can revoke your authorization by giving a written request with you r signature on it to the Principal Investigator. the purpose, the procedures, the possible benefits, and the risks of this research study; ed health information will be collected, used, and shared with others. privacy will be protected: ______________________________________________ _____________________ Signature of Person Obtaining Consent & Authorization Date Consenting Adults possible benefits, and risks; the alternatives to being in the study; and how your privacy will be protected. You have received a copy of this Form. You have been given the opportunity to ask questions before you sign, and you have been told that you can ask other questions at any time. Adult Consenting for Self. By signing this form, you voluntarily agree to participate in this study. By signing this form, you are not waiving any of your legal rights. ______________________________________________ _____________________ Signature of Adult Consenting for Self Date S IGNATURES
118 Parent/Adult Legally Representing the Subject. By signing this form, you voluntarily give your permission for the person named below to participate in this study. You are not waiving any legal rights for yourself or the person you are legally re presenting. After your signature, please print your name and your relationship to the subject. ______________________________________________ _____________________ Consent & Authorization Signature Date of Parent/Legal Represent ative ____ ______________________________ ___________________________________ Print: Name of Legal Representative Print: Relationship to Participant: ______________________________________________ Print: Name of Subject: Participants Who Cannot Consent But Can Read and/or Understand about the Study. Although legally you cannot "consent" to be in this study, we need to know if you want to take part. If you decide to take part in this study, and your parent or the person legally responsible for you gives permission, you both need to sign. Your signing below means that you agree to take part (assent). The signature of your parent/legal representative above means he or she gives permission (consent) for you to take part. ________ _________________________________________ __________________ Assent Signature of Participant Date
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131 BIOGRAPHICAL SKETCH Laura Smith was born in Evergreen Park, Illinois and she is one of six children. Laura began working in 1975 at St. James hospital in Chicago Heights, Illinois as a pharmacy assistant through a Marian Catholic High School education/work partnership. She worked at Foster G. McGaw Loyola Medical Center as a nursing assistant while completing her college degree. She received the Bachelor of Science degree in n ursing in 1983 from Loyola University of Chicago. Early in her career as a registered n urse, In 1997, Laura received he r Master of Science degree in n ursing from the University of South Florida. She h olds an active Florida license as an Advanced Registered Nurse Practitioner and is board certified by the American Nurses Credentialing Center. Laura has treated hundreds of patients with acute and chronic medical conditions in several primary care clinic al settings. Laura Smith received her Doctorate of Philosophy degree at the University of Florida She is a recipient of Merdinger and Charlotte Liberty scholarships. Her The Relationship of Adolescent Sibling Resources to Severity of Autism Spectrum Disorder and Sense of Coherence Levels h as opened up new interests in the health of families and young persons. She is a published scholar in the Journal of Child and Adolescent Psychiatric Nursing. Laura has been active in advocating excellenc e and professionalism for nursing to her colleagues and in her community. She is a long standing member of the Florida Nurses Association. She has is currently a member of the West Coast Council of Advanced Nurse Practitioners, Southern Nursing Research S ociety, and the
132 International Society for Autism Research. She volunteers for the Archdiocese of St. Petersburg and local events for persons with disability Laura has been married to Randall J. Smith for more than twenty five years. They have two sons and a daughter and live on the West coast of Florida. In addition to her love for science, Laura enjoys travel, gardening, and spending time with her friends and family.