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Attention and Learning in Children with Epilepsy and Co-Morbid Sleep Disturbance

Permanent Link: http://ufdc.ufl.edu/UFE0022836/00001

Material Information

Title: Attention and Learning in Children with Epilepsy and Co-Morbid Sleep Disturbance
Physical Description: 1 online resource (103 p.)
Language: english
Creator: Bongiolatti, Susan
Publisher: University of Florida
Place of Publication: Gainesville, Fla.
Publication Date: 2008

Subjects

Subjects / Keywords: attention, children, epilepsy, learning, sleep
Clinical and Health Psychology -- Dissertations, Academic -- UF
Genre: Psychology thesis, Ph.D.
bibliography   ( marcgt )
theses   ( marcgt )
government publication (state, provincial, terriorial, dependent)   ( marcgt )
born-digital   ( sobekcm )
Electronic Thesis or Dissertation

Notes

Abstract: Research suggests that children with epilepsy may be more susceptible to sleep disturbance, which has been associated with behavior problems and cognitive impairments in pediatric populations. However, few studies have examined the relationships among sleep disturbance, behavior, and cognition in children with epilepsy. Using a brief neuropsychology battery and parent questionnaires, we aimed to clarify the relationship between possible sleep disturbance and cognitive and behavioral problems in children with epilepsy. Our study hypothesized that children with epilepsy who had a higher frequency of sleep disturbance would perform more poorly on measures of attention and learning than children with epilepsy and a lower frequency of sleep disturbance. Parent report of sleep disturbance symptoms was also hypothesized to be directly related to impairment on neuropsychological measures of attention and learning. Finally, it was hypothesized that parents of children with epilepsy and a higher frequency of sleep disturbance would report greater attention problems and hyperactivity in their children. Overall, results provide limited evidence for attentional impairment in children with epilepsy and co-morbid sleep disturbance. Specifically, children whose parents reported a high frequency of sleep problems (and those who endorsed symptoms of a sleep disorder) were found to have greater difficulty sustaining attention to auditory information than children who were reported to have fewer sleep problems. Differences did not appear to be related to seizure status, seizure type, or use of antiepileptic medication, although these dissociations would need to be more directly tested in future work. Other aspects of attention and learning failed to differ between the two sleep groups, although more complex attentional control/switching approached significance. Future studies should continue to examine the relationship between sleep disturbance and cognitive performance in children with epilepsy, including research employing neuropsychological measures and objective assessments of sleep such as polysomnography and actigraphy as well as treatment-based studies in children with epilepsy and diagnosed sleep disorders.
General Note: In the series University of Florida Digital Collections.
General Note: Includes vita.
Bibliography: Includes bibliographical references.
Source of Description: Description based on online resource; title from PDF title page.
Source of Description: This bibliographic record is available under the Creative Commons CC0 public domain dedication. The University of Florida Libraries, as creator of this bibliographic record, has waived all rights to it worldwide under copyright law, including all related and neighboring rights, to the extent allowed by law.
Statement of Responsibility: by Susan Bongiolatti.
Thesis: Thesis (Ph.D.)--University of Florida, 2008.
Local: Adviser: Fennell, Eileen B.
Local: Co-adviser: Carney, Paul R.
Electronic Access: RESTRICTED TO UF STUDENTS, STAFF, FACULTY, AND ON-CAMPUS USE UNTIL 2010-12-31

Record Information

Source Institution: UFRGP
Rights Management: Applicable rights reserved.
Classification: lcc - LD1780 2008
System ID: UFE0022836:00001

Permanent Link: http://ufdc.ufl.edu/UFE0022836/00001

Material Information

Title: Attention and Learning in Children with Epilepsy and Co-Morbid Sleep Disturbance
Physical Description: 1 online resource (103 p.)
Language: english
Creator: Bongiolatti, Susan
Publisher: University of Florida
Place of Publication: Gainesville, Fla.
Publication Date: 2008

Subjects

Subjects / Keywords: attention, children, epilepsy, learning, sleep
Clinical and Health Psychology -- Dissertations, Academic -- UF
Genre: Psychology thesis, Ph.D.
bibliography   ( marcgt )
theses   ( marcgt )
government publication (state, provincial, terriorial, dependent)   ( marcgt )
born-digital   ( sobekcm )
Electronic Thesis or Dissertation

Notes

Abstract: Research suggests that children with epilepsy may be more susceptible to sleep disturbance, which has been associated with behavior problems and cognitive impairments in pediatric populations. However, few studies have examined the relationships among sleep disturbance, behavior, and cognition in children with epilepsy. Using a brief neuropsychology battery and parent questionnaires, we aimed to clarify the relationship between possible sleep disturbance and cognitive and behavioral problems in children with epilepsy. Our study hypothesized that children with epilepsy who had a higher frequency of sleep disturbance would perform more poorly on measures of attention and learning than children with epilepsy and a lower frequency of sleep disturbance. Parent report of sleep disturbance symptoms was also hypothesized to be directly related to impairment on neuropsychological measures of attention and learning. Finally, it was hypothesized that parents of children with epilepsy and a higher frequency of sleep disturbance would report greater attention problems and hyperactivity in their children. Overall, results provide limited evidence for attentional impairment in children with epilepsy and co-morbid sleep disturbance. Specifically, children whose parents reported a high frequency of sleep problems (and those who endorsed symptoms of a sleep disorder) were found to have greater difficulty sustaining attention to auditory information than children who were reported to have fewer sleep problems. Differences did not appear to be related to seizure status, seizure type, or use of antiepileptic medication, although these dissociations would need to be more directly tested in future work. Other aspects of attention and learning failed to differ between the two sleep groups, although more complex attentional control/switching approached significance. Future studies should continue to examine the relationship between sleep disturbance and cognitive performance in children with epilepsy, including research employing neuropsychological measures and objective assessments of sleep such as polysomnography and actigraphy as well as treatment-based studies in children with epilepsy and diagnosed sleep disorders.
General Note: In the series University of Florida Digital Collections.
General Note: Includes vita.
Bibliography: Includes bibliographical references.
Source of Description: Description based on online resource; title from PDF title page.
Source of Description: This bibliographic record is available under the Creative Commons CC0 public domain dedication. The University of Florida Libraries, as creator of this bibliographic record, has waived all rights to it worldwide under copyright law, including all related and neighboring rights, to the extent allowed by law.
Statement of Responsibility: by Susan Bongiolatti.
Thesis: Thesis (Ph.D.)--University of Florida, 2008.
Local: Adviser: Fennell, Eileen B.
Local: Co-adviser: Carney, Paul R.
Electronic Access: RESTRICTED TO UF STUDENTS, STAFF, FACULTY, AND ON-CAMPUS USE UNTIL 2010-12-31

Record Information

Source Institution: UFRGP
Rights Management: Applicable rights reserved.
Classification: lcc - LD1780 2008
System ID: UFE0022836:00001


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ATTENTION AND LEARNING IN CHILDR EN WITH EPILEPSY AND CO-MORBID SLEEP DISTURBANCE By SUSAN R. BONGIOLATTI A DISSERTATION PRESENTED TO THE GRADUATE SCHOOL OF THE UNIVERSITY OF FLOR IDA IN PARTIAL FULFILLMENT OF THE REQUIREMENTS FOR THE DEGREE OF DOCTOR OF PHILOSOPHY UNIVERSITY OF FLORIDA 2008 1

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2008 Susan R. Bongiolatti 2

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To children and families affected by epilepsy. 3

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ACKNOWLEDGMENTS I thank all of the people who contributed to th e completion of this project in a multitude of ways. First, I thank my mentors Dr. Eileen Fennell and Dr. Paul Carney for their guidance and steadfast support. I also thank my committee members, Dr. Fonda Eyler, Dr. Shelley Heaton, and Dr. Michael Marsiske. I am grateful to ha ve had such a collegial, supportive, and insightful committee to guide me through this endeavor. I thank the staff, nurses, and doctors of the University of Florida/Shands Hospital Departme nt of Pediatric Neurol ogy for their assistance with recruitment and administrative tasks. Partic ularly invaluable was th e support of Dr. Edgard Andrade, Dr. Zhao Liu, Christie Snively, Debbi e Ringdahl, Donna Lilly, and Debra Thomas. I am also appreciative of Wendy Gray and Jennifer W ilkinson for their assist ance with recruitment and testing. I am thankful for my parents Tren t and Ann Bongiolatti and my older brother Nick for their love and support. Finally, I cannot expr ess the depth of my gratitude to my fianc Michael Bowen for his unwavering faith, patience, encouragement, and love. 4

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TABLE OF CONTENTS page ACKNOWLEDGMENTS...............................................................................................................4LIST OF TABLES................................................................................................................. ..........7LIST OF FIGURES.........................................................................................................................8ABSTRACT.....................................................................................................................................9 CHAPTER 1 INTRODUCTION................................................................................................................. .11Overview of Pediatric Epilepsy..............................................................................................12Pediatric Epilepsy: C ognition and Behavior...........................................................................14Sleep Disturbance and Ne urobehavioral Problems................................................................21Epilepsy and Sleep Disturbance: Relationshi p to Cognitive-Behavioral Functioning?.........25Purpose of Study.....................................................................................................................302 METHODS...................................................................................................................... .......33Participants.............................................................................................................................33Measures.................................................................................................................................34Intellectual Screening......................................................................................................34Attention..........................................................................................................................35Test of Everyday Attention for Children..................................................................35Conners Continuous Performance Test, 2nd Edition...............................................36Trail Making Test.....................................................................................................37Learning: Wide Range Assessment of Memory and Learning.......................................38Processing Speed: Wechsler Intelligen ce Scale for Children, Fourth Edition Processing Speed Index...............................................................................................38Visuospatial/Visuoconstructional Ability: Beery-Buktenica Developmental Test of Visual-Motor Integration, Fifth Edition.......................................................................39Parent Questionnaires......................................................................................................39Childrens Sleep Questionnaire-Parent Report........................................................40Behavior Assessment System for Children, Second Edition...................................40Conners Parent Rating Scales-Revised, Long Form...............................................41Demographic and medical information....................................................................41Procedure................................................................................................................................42Recruitment.................................................................................................................... .42Screening and Assessment..............................................................................................43 5

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3 RESULTS...................................................................................................................... .........46Preliminary Analyses........................................................................................................... ...46Group Assignment...........................................................................................................46Demographics..................................................................................................................47Aim 1: Comparison of Attention Performan ce in Lower Frequency Sleep Problem and Higher Frequency Sleep Problem Groups..........................................................................49Selective Attention..........................................................................................................49Sustained Attention.........................................................................................................50Attentional Control/Switching.........................................................................................50Correlation between Parent Report of Sleep Disturbance and Attention Performance.................................................................................................................51Aim 2: Comparison of Learning Performance in Lower Frequency Sleep Problem and Higher Frequency Sleep Problem Groups..........................................................................52Group Comparison..........................................................................................................52Correlation Between Parent Report of Sleep Disturbance and Learning Performance.................................................................................................................52Aim 3: Comparison of Parent Report of Attention and Behavior Problems in Lower Frequency Sleep Problem and Highe r Frequency Sleep Problem Groups.........................53Additional Analyses............................................................................................................ ....54Supplementary Neuropsychological Measures: Processing Speed and Visuospatial/ Visuoconstructional Ability.........................................................................................54Comparison of Diagnostic Groups: Sleep Disorder Symptoms.....................................55Clinical Inattention Symptoms among the Two Sleep Disorder Symptom Groups........56Effect of Seizure Type.....................................................................................................57Effect of Seizure Status...................................................................................................574 DISCUSSION................................................................................................................... ......73Overview....................................................................................................................... ..........73Sleep Disturbance and Attenti on and Learning Performance.................................................73Additional Results............................................................................................................. .....80Limitations.................................................................................................................... ..........80Future Directions....................................................................................................................83 APPENDIX: CHILDRENS SLEEP QUESTIONNAIREPARENT REPORT........................86LIST OF REFERENCES...............................................................................................................96BIOGRAPHICAL SKETCH.......................................................................................................103 6

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LIST OF TABLES Table page 2-1 Study measures............................................................................................................. .....453-1 Demographic characteristics of higher and lower frequency sleep problem groups.........593-2 Epilepsy/seizure characteristics of highe r and lower frequency sleep problem groups....603-3 Mean comparison of attention performan ce between sleep problem frequency groups...613-4 Correlation of sleep disorder symptoms on the CSQ-PR and attention problems.............623-5 Mean comparison of learning performan ce between sleep problem frequency groups....633-6 Mean comparison of parent report of attention and behavior problems between sleep problem frequency groups.................................................................................................643-7 Comparison of attention and learning performance between sleep disorder diagnostic groups.................................................................................................................................653-8 Comparison of attention, learning, and sl eep disorder symptoms between partial and generalized seizure groups.................................................................................................663-9 Comparison of attention, learning, and sleep disorder symptoms between seizure status groups.......................................................................................................................67 7

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LIST OF FIGURES Figure page 3-1 Scatterplots of correlation between selec tive attention measures and Childrens Sleep Questionnaire-Parent Report (CSQ-PR)............................................................................683-2 Scatterplots of correlation between su stained attention measures and Childrens Sleep Questionnaire-Parent Report (CSQ-PR)..................................................................693-3 Scatterplots of correlation between at tentional control/swit ching measures and Childrens Sleep Questionnair e-Parent Report (CSQ-PR)................................................713-4 Scatterplots of correlation between learning measures and Childrens Sleep Questionnaire-Parent Report (CSQ-PR)............................................................................72 8

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Abstract of Dissertation Pres ented to the Graduate School of the University of Florida in Partial Fulfillment of the Requirements for the Degree of Doctor of Philosophy ATTENTION AND LEARNING IN CHILDR EN WITH EPILEPSY AND CO-MORBID SLEEP DISTURBANCE By Susan R. Bongiolatti December 2008 Chair: Eileen B. Fennell Cochair: Paul R. Carney Major: Psychology Research suggests that children with epile psy may be more susceptible to sleep disturbance, which has been associated with behavior problems and cognitive impairments in pediatric populations. However, few studies have examined the relationships among sleep disturbance, behavior, and cogni tion in children with epilepsy. Using a brief neuropsychology battery and parent questionnaires, we aimed to clarify the relationship between possible sleep disturbance and cognitive and behavioral problems in children with epilepsy. Our study hypothesized that children with epilepsy who had a higher frequency of sleep disturbance would perform more poorly on measures of attention and lear ning than children with epilepsy and a lower frequency of sleep disturba nce. Parent report of sleep disturbance symptoms was also hypothesized to be directly rela ted to impairment on neuropsychological measures of attention and learning. Finally, it was hypot hesized that parents of child ren with epilepsy and a higher frequency of sleep disturbance would report greater attention pr oblems and hyperactivity in their children. Overall, results provide limited evidence for attentional impairment in children with epilepsy and co-morbid sleep disturbance. Specifically, children whose pa rents reported a high 9

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10 frequency of sleep problems (and those who endor sed symptoms of a sleep disorder) were found to have greater difficulty sustai ning attention to auditory information than children who were reported to have fewer sleep problems. Differences did not appear to be re lated to seizure status, seizure type, or use of antiepileptic medication, although these dissociations would need to be more directly tested in future work. Other aspects of attention and learning failed to differ between the two sleep groups, although more complex attentional control/switching approached significance. Future studies should continue to examine the relationship between sleep disturbance and cognitive performance in children with epileps y, including research employing neuropsychological measures a nd objective assessments of sleep such as polysomnography and actigraphy as well as treatment-based studies in children with epilepsy and diagnosed sleep disorders.

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CHAPTER 1 INTRODUCTION Epilepsy is one of the most common neur ological disorders, currently affecting approximately 2.7 million Americans (Epilepsy Foundation of America, EFA, 2008). Each year, an estimated 45,000 children under the age of 15 deve lop epilepsy, and current statistics suggest that 326,000 school children age 14 or younger currently have epilepsy (EFA, 2008). Children with epilepsy are at increased risk for long-term educational, psychological, and social problems (Pellock, 2004), which necessitates a comprehensive approach to pediatric epilepsy management that goes beyond the typical goal of seizure control. More specifically, Besag (2004) suggests that after seizure contro l, cognition and behavior are among the most important factors in determining whether a child with epilepsy will successfully progress toward independence. Among the most commonly described neurocognitive issues for children with epilepsy are problems with attention a nd related daytime behaviors such as hyperactivity (CITE). While these difficulties have previously been hypothesized to result from the effects of seizure activity on th e brain or anti-ep ileptic medications, recent re search has suggested another potential cause. Specifically, research has suggest ed that children with epilepsy may be more susceptible to sleep disorders, which may directly contribute to beha vior problems and to attention and learning impairments. However, little to no empirical research has examined the relationships between sleep distur bance, behavior, and cognition in children with epilepsy. An established relationship would s uggest proper identification and tr eatment of sleep disorders in children with epilepsy could be one mechan ism for ameliorating daytime problems and improving cognitive performance, thereby maximizing the overall functioning of children with epilepsy. This study aims to clarify the relationship between possible sleep disturbance and cognitive and behavioral problems in children with epilepsy. 11

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Overview of Pediatric Epilepsy Epilepsy is a chronic neurological disorder characterized by recu rrent seizures. Normally, the brain continuously generates tiny electrical impulses in an orderly pattern. During an epileptic seizure, a breakdown occurs in the systems that mainta in the balance of electrical activity in the brain, resulting in abnormal elect rical discharges. This uncontrolled surge of electrical activity in the brain results in cha nges in consciousness, movement, and/or sensation, the symptoms of a seizure. In most cases (6080%), the onset of seizures in children has no apparent cause (Hauser, 2001). Both children and adults with epilepsy may e xperience one or both of two primary types of seizures: generalized and partial seizures (I nternational League Against Epilepsy, 1989). Generalized seizures emerge from cortex in both hemispheres of the brain and typically involve a disturbance of consciousness. Types of gene ralized seizures include absence (petit mal) seizures, myoclonic seizures, aton ic seizures, tonic seizures, clonic seizures, and the combined tonic-clonic (grand mal) seizur es (Mattson, 2003). In contrast to generalized seizures, partial seizures originate from an electr ical disturbance in a specific ar ea of cortex within one cerebral hemisphere. Because of this distinction, partia l seizures are also known as focal, local, or localization-related seizures. Par tial seizures are further classifi ed based upon the impairment or preservation of consciousness, su ch that simple partial seizures are those in which there is no alteration in consciousness and co mplex partial seizures are those in which consciousness is altered or lost. Depending upon the location of the electrical disturbance, virtually any movement, sensory, or emotional symptom can o ccur as part of a par tial seizure, including complex visual or auditory hallucinations. Fina lly, a partial seizure may spread to involve the entire brain, which is referred to as a secondar ily generalized seizure. Convulsive generalized 12

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seizures (e.g., tonic-clonic seizur es) appear to be the most preval ent seizure type during the first year of life, with partia l seizures becoming the most common af ter the first year (Pellock, 2004). When possible, recurrent seizures (epilepsy) are classified in epilepsy syndromes based upon the predominant seizure type and other fact ors such as the seizure triggers, seizure behaviors, genetic factors and specific brain wave patterns seen on electroencephalography. Examples of epilepsy syndromes diagnosed in children include benign childhoood epilepsy with centrotemporal spikes (BCECTS), absence ep ilepsy, juvenile myoclonic epilepsy, LennoxGastuat Syndrome, Landau-Kleffner syndrome, frontal lobe epilepsy, and temporal lobe epilepsy. The majority of childrenapproximate ly 59 percenthave a localized or partial seizure syndrome, such as frontal lobe epilepsy (Shinnar & Pellock, 2002). Approximately 29 percent of children with epilepsy have a generali zed seizure syndrome such as absence epilepsy. Epilepsy treatment typically involves use of an ti-epileptic drugs (AEDs) aimed to reduce the frequency and severity of seizures. Specific AEDs are prescribed based on many factors, including the type of seizure, the age of patient, the side effect pr ofile, and the patients ability to follow the required medication regimen. Althou gh most children with epilepsy are initially prescribed a single medication, if this medicati on is only partially effective, one or more medications may be added (typically after other types of monotherapy have been attempted). In cases of intractable epilepsythose that do not respond to medicationother treatments may be considered. These include seizure surgery invo lving the resection of the area of the seizure focus, ketogenic diet, and vagal nerve stimulator s. Prognosis for children with epilepsy has often been examined as a factor of epilepsy syndrom e. For example, benign rolandic epilepsy is known to typically completely remit, whereas Lennox-Gastaut Syndrome never remits (Camfield & Camfield, 2003). With regard to prognosis mo re generally, most patients with epilepsy 13

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become seizure-free with appropriate antiepilepti c drugs, typically within a few years of initial diagnosis (Pellock, 2004). Additionally, many are subsequently able to discontinue medication and remain free of seizures. Pediatric Epilepsy: Cognition and Behavior Despite the potential for effective seizur e control and a positive medical prognosis, children with seizures remain at risk for longterm educational and social problems (Pellock, 2004). Accordingly, to optimally manage pediatri c epilepsy it is essentia l to understand not only the neurological implications but also the neuroc ognitive effects of seizure disorders. However, the literature on cognition and be havior in children with epilepsy is relatively limited and often lacks unanimity. In general, the pediatric epilepsy literatu re has tended to focus on broad intellectual functioning (i.e., IQ) and academic achievement or on the outcomes of treatments (e.g., antiepileptic medications, epilepsy su rgery) rather than on specific aspects of neurobehavioral or cognitive functioning in children wi th seizures. Variation across th e existing studie s in terms of subjects (e.g., type of seizure or epilepsy syndrome included) and methodology also makes generalizable conclusions about neurobehavioral functioning difficu lt. Despite these challenges, some common psychiatric, behavioral, and cognitive problems have been identified in the literature on children with epilepsy, including at tention and memory problems, anxiety, and hyperactivity (e.g., Sanchez-Carpintero & Neville, 2003; Williams, 2003; Pellock, 2004) Disruption of attentional abilities is one of the most consistently described cognitive problems in children with epilepsy (Schubert, 2005; Williams, 2003). For example, Williams, Griebel, & Dykman (1998) conducted comprehens ive neuropsychological assessments including intellectual functioning, memory, attention, la nguage, academic achievement, visual motor integration and fine motor skills with a gr oup of children with epilepsy. Results of the 14

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assessments indicated that th e children performed within th e expected range given their measured intelligence on all tests except thos e measuring attention, on which they performed below expectations. Parents also reported atte ntion problems within the clinically significant range on a behavior questionnaire. Consistent with this study, Mitchell, Zhou, Chavez, and Guzman (1992) found that children with epilepsy had slower reaction times and inattention on a continuous performance task. Notably, Mitchell et al. (1992) concluded that the impaired performance of the children with epilepsy was not related to seizure severity, duration of se izure disorder, or antiepileptic drug (AED) history. Similarly, a study of Dutch ch ildren with epilepsy also found that compared to their healthy classmates, children with newly diagnosed epilepsy performed poorer on a measure of sustained attention (Oostrom, va n Teesling, Smeets-Schouten, Peters, & JennekensSchinkel, 2005). Studies such as these sugge st that attention probl ems are not simply a medication side effect or a hallmar k of particularly complicated se izure history. With regard to medication side effects, it is also important to note that Loring and M eador (2004) reviewed the cognitive side effects of AEDs in children and concluded that although some component of cognitive deficits may be attrib uted to long-term use of AEDs (particularly phenobarbital), there is insufficient evidence at this time to conclude that AEDs in general have negative cognitive side effects. This is particul arly true for the most recently developed AEDs, many of which have yet to be studied neuropsychologically. A very small number of studi es have noted reduced attention in children taking carbamazepine and, in one study, valproic acid; however, current results are far from conclusive and much of th e research has suffered fr om methodological flaws (Loring & Meador, 2004). 15

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Attention problems also do not appear to be only associated with a particular epilepsy or seizure type. Attention difficulties have been reported in children with generalized seizures as well as complex partial seizures. For example, Borgatti et al. (2003) found that both children with generalized epilepsy and children with partia l epilepsy demonstrated persistent attention difficulties as measured by a continuous performance task, despite control of seizures. In a study examining only children with generalized epile psy (absence or tonic-clonic seizures), the children with epilepsy were found to have poorer verbal and non-verbal attention than a group of ageand sex-matched healthy controls, with the absence epilepsy group demonstrating the greatest impairment (Henkin,et al, 2005). In another study, children with complex partial seizures with and without a diagnosis of attent ion deficit disorder (ADHD) were compared to a group of children with ADHD and a healthy cont rol group on a continuous performance task (Semrud-Clikeman & Wical, 1999). The children with partial seizures were found to have particular difficulty compared to the hea lthy controls, regardle ss of ADHD diagnosis. Interestingly, the children with epilepsy a nd no ADHD diagnosis performed similarly to the ADHD group, while the epilepsy with ADHD group perf ormed the worst of all four groups. Van Rijckevorsel (2006) explains this commonality of attention problems across seizure types as resulting from the frontal lobes being a prefe rential target of both generalized and focal seizures. Specifically, he states that direct neuronal dysfunction in the frontal lobes as well as indirect effects from secondary seizure spread (e.g., circuitry abnormalities) likely underlie attention and executive function proble ms in children with epilepsy. While the majority of attention research with children with epilepsy has focused on sustained attention as measured by continuous performance tests (CPTs), most current models of attention are multifocal in nature. Research with other clinical populations suggests that different 16

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aspects of attention may be variab ly affected in neurological and psychiatric disorders. For the purposes of this project, we adopted the model proposed by Mirsky, Anthony, Duncan, Ahearn, and Kellam (1991). Mirsky and colleagues used principal components an alysis to derive a model of attention with four esse ntial elements that work together in a coordinated attentional system. The four components include: focus-execute, sustain, shift, and encode. Focus-execute is the ability to efficiently and quickly scan an array to select a target and to make a verbal or manual response quickly. It requir es concentrating attentional res ources on a particular stimulus while ignoring distracters. This aspect of attention is also referred to as selective attention. Sustain is vigilance, or the capacity to mainta in focus and alertness over time. The third attentional component is attentional shiftthe abi lity to change attentive focus in a flexible or adaptive manner. Finally, Mirsky et al. also descri bed a fourth element termed encode. This component is the capacity to hold information in memory briefly in order to perform a mental operation on the information. According to Mi rsky and Duncan (2001), this is equivalent to what is often termed working memory, and may re flect more complex mechanisms than attention alone. Although CPT-based studies of sustained atte ntion have dominated the literatures, a few studies have attempted to look at the other aspects of attention with mixed results. Hernandez et al. (2003) reported that children with frontal lobe epilepsy, but not those with temporal lobe epilepsy or generalized absence epilepsy, demonstr ated impaired visual attention (i.e. selective attention) on the Coding and Symbol Search subt ests of the Wechsler Intelligence Scale for Children, Third Edition (WISC-III). A study of Is raeli children with generalized epilepsy found that children with generalized absence seizures and children with generalized tonic-clonic seizures both demonstrated poorer visual selective attention (Coding) th an healthy controls 17

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(Henkin et al., 2005). Children with benign childhood epilepsy with centrotemporal spikes (BECTS) with bilateral EEG discharges were reported to have impaired selective attention and divided attention as measured by the Stroop Color-Word task and the Trail Making Test respectively (DAlessandro et al ., 1990). Similar findings were reported in a French study of children with BECTS using a cancellation-type selective attention task from the Battery for Rapid Evaluation of Cognitive Functions (Pinton et al., 2006). In contrast, in another study of children with BECTS, no differences between child ren with seizures and healthy controls were found on the Trail Making Test, al though parents reported that the children with seizures were more distractible and had poorer concentration on parent ques tionnaires (Croona et al., 1999). Similarly, Schoenfeld et al. (1999) used the Stroop Color Word task and the Trail Making Test to assess attention in children with complex partial se izures, and reported that they demonstrated no deficits in selective or divide d attention relative to healthy si bling controls. In summary, the most consistent evidence for attention problems in children with epilepsy appears to be specifically for sustained attention difficulties, while other aspects of attention remain less understood in this population. Children with attention difficulties may be at risk for other types of cognitive and academic problems. Pollock (2004) notes that ch ildren with epilepsy may be predispose[d] to attention problems, which may in turn lead to learning difficulties. For example, Oostrom et al. (2005) reported attention problems in school child ren with newly diagnosed epilepsy. In their discussion of their findings, the researchers noted that in their sample of children with epilepsy, poorer attentional performance was also associated with having repeated a grade in school. Oostrom and colleagues concluded that the school careers of these repeater children had influenced their performance on attention ta sk. However, it seems equallyif not more 18

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plausible that the children with epilepsy prev iously did poorly in school because of their underlying attention problems and other cognitive de ficits. Given its recognized association with attention and its association with academic achie vement, learning is another important aspect of cognition to be considered in children with epilepsy. However, whereas the findings on attention sugg est the presence of impairment in children with epilepsy (with some inconsistencies in the exact nature of the defi cits), the findings on learning are much less consistent. For example, on a location learning ta sk, school-aged children with epilepsy performed similarly to a healthy control group, except for a trend toward being more susceptible to proactive interference (S chouten, Oostrom, Pestman, Peters, & JennekensSchinkel, 2002). Likewise, Williams et al. ( 2001) found intact new learning on a list learning task in children with epilepsy (although childre n with diagnosed ADHD were excluded and may represent an important sample). In contrast, Henkin et al. (2005) found that children with generalized epilepsy performed significantly poorer on a list learning task th an a healthy control group (this same group also had significantly im paired verbal and non-verbal attention). In the Dutch study described above, neuropsychol ogical testing results also indicated that in addition to the attention problems, the ch ildren with epilepsy demonstrated poorer performance on learning tasks, with proactive in terference during learning remaining an area of concern when academic history was taken into ac count (Oostrom, et al., 2005). In this same study, parent questionnaires also indicated that the children with epilepsy had a greater number of behavior and learning difficulties than the control group. Finally, in addition to direct neurops ychological testing with children, parent questionnaires frequently have been employed to assess behavior in children with chronic illnesses and neurological disorders. Such studies in children with epilepsy support the 19

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cognitive findings just described, in that they frequently indicate problems with attention as well as other behavior problems (e.g., Austin et al., 2002; Keene et al., 2005; Ott et al., 2003). In a cross-sectional parent report study of child ren with epilepsy conduc ted during regularly scheduled neurology visits, approximately 16% of the children had clinic ally elevated total behavior problem scale scores (T-score > 70), an d more than a quarter (26.9%) of the children were rated as having clinically elevated problems with atten tion (Keene et al., 2005). Dunn and colleagues (2003) had parents complete either the Childrens Sympto m Inventory or the Adolescent Symptom Inventory a nd determined that 38% of their sample of children with epilepsy met criteria for diagnosis of ADHD, wh ich exceeds the prevalence rate in the general child population. It was also noted that more children with epile psy presented with symptoms of ADHD, Inattentive type than is typically seen. In summary, while some variation exists in th e literature, the overall pattern of research findings suggests that inattention and related behavior problems pose a significant concern for children with epilepsy. Identifying the etiology of cognitive and behavior problems in children with epilepsy remains an important goal for epil epsy research, as it may improve the ability to recognize and treat these problems and accordingl y improve the overall well-being of children with epilepsy in the long-term. Recently, there has been an increased interest in possible contribution of sleep disturbance to behavior and cognitive probl ems in children with epilepsy. To better understand this possible relationship, it is helpful to fi rst briefly review what is known about the relationship between sl eep disturbance, behavior and cognition in otherwise healthy children. 20

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Sleep Disturbance and Neurobehavioral Problems The effects of chronic sleep disturbance in children has primarily been examined by studying children with sleep disord ers, particularly sleep disord ered breathing. Sleep disorders are conditions or circumstances of a physiologica l and/or psychological nature that cause sleep disturbance. Mindell & Owens (2003) broadly conceptualize sleep disorders as involving one or more basic mechanisms: inadequate duration of sl eep for age (insufficien t sleep), disruption and fragmentation of sleep (poor sleep quality), or inappropr iate timing of the sl eep period (circadian rhythm disorders). The most prevalent and most researched sleep disorder diagnosis among children is sleep disorder ed breathing (Sadeh et al., 2002), a cl ass of disruptive sleep disorders that involve varying severities of breathing difficulties including obstructive sleep apnea. Sleep disordered breathing (SDB) is characterized by repeated events of partial or complete upper airway obstruction during sleep that results in the disruption of normal ventilation, hypoxemia, and sleep fragmentation (OBrien, Mervis, Holb rook, Bruner, Smith et al., 2004). Typically, a continuum of SDB is recognized, with increasi ng severity: primary snoring (snoring with no blood gas abnormalities), upper airway resistance syndrome (snoring with increased work of breathing), and obstructive sleep ap nea (characterized by repeated episodes of pr olonged partial or complete upper airway obstruction during sl eep). Unlike in adults, in children SDB is commonly associated to adenotonsillar hypertro phy, or enlarged tons ils and adenoids. SDB has been associated with a variety of neurobehavioral consequences in children, including excessive daytime sleepiness, inatte ntiveness, impaired ex ecutive functioning and learning, and hyperactivity (Kotagal & Pianosi, 2006). Studies with children who snore have found that snorers demonstrate poorer performance on neuropsychological tests of attention when compared with non-snorers (Blunden et al., 2000; OBrien, Merv is, Holbrook, Bruner, Klaus, et al., 2004). In a sample that ex cluded snorers with diagnosed ADHD or behavior 21

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problems, attention impairment and parent-re ported hyperactivity were found, which were suggested to be related to decreased REM sl eep or sleep fragmentati oninterruption of the normal sleep cycle by brief arousals that occur throughout the night (OBrien, Mervis, Holbrook, Bruner, Klaus, et al., 2004). SDB, including snoring and obstructve sleep apnea, was also associated with lower attention, executive func tioning, and phonological processing scores in a group of non-referred first graders, although no diffe rences in parent report of behavior were found between the SDB and non-snoring group (OB rien, Mervis, Holbrook, Bruner, Smith et al., 2004). In a large survey of general pediat ric patients, endorsement of frequent snoring, increased sleepiness, and other symptoms of disr upted sleep was associated with inattention and hyperactivity (Chervin et al., 2002). In a sim ilar broad sampling of children, Kaemingk et al. (2003) conducted neuropsychologi cal testing and in-home sleep studies in a large group of school children and found that child ren with respiratory arousals c onsistent with OSA performed significantly worse (although w ithin normal limits) on learning and delayed memory tasks compared to children with fewer or no respiratory arousals. The relationship between attention problems a nd sleep disorders has also been addressed by examining sleep in children w ith known attention difficulties, specifically children diagnosed with ADHD. Corkum, Tannock, & Moldofsky (1998) suggested that the pr evalence of sleep problems in children with ADHD is between 25% and 50%, considerab ly higher than the approximately 7% prevalence of sleep problems in the general pediatric population. Based upon parent report of sleep patterns, children with ADHD have been estimated to have a 2 to 3 times greater prevalence of sleep problems such as difficulty falling asleep, restless sleep, and nighttime awakenings as compared to h ealthy control childre n (Owens, 2005). Using polysomnography (i.e., overnight sleep studies ) and other methodology, children with ADHD 22

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have been described as having l onger sleep onset latencies, mo re nocturnal and early morning awakenings, shorter average sleep duration, and increased sleepiness upon awakening (Brown & Modestino, 2000). A recent study used actigraphy to compare sleep in unmedicated children with ADHD and healthy control subjects, and fo und that children with ADHD had shorter actual sleep time as well as more interrupted sleep (Owe ns et al., 2008). The children with ADHD also reported being more sleepy during the daytime. While the nature of the relationship between ADHD and sleep problems is still being investigated, research with children with ADHD certainly suggests sleep disturbance and sleep di sorders should be cons idered as potential contributors to disrupted attentional abilities. Finally, evidence for a direct relationship betw een SDB and neurobehavi oral deficits such as inattention and hyper activity has been suggested in re search with children who undergo adenotonsillectomy (removal of tonsils and adenoids ) for the treatment of SDB. As previously noted, enlarged tonsils and adenoids are a common cause of SDB in children, and adenotonsillectomy is often a firstline treatment. Several resear chers have found that children with SDB have demonstrated neurocognitive improvement following treatment. While the children with SDB were reported to have increas ed cognitive problems and behavioral problems-including inattention and hyperactivity--prior to treatment, following ad enotonsillectomy their cognitive performance and behaviors were tested at levels similar to healthy controls (Ali, Pitson, & Stradling, 1996; Goldstein, Post, Rose nfeld, & Campbell, 2000) Although many of the early studies were limited by fairly small sample sizes, a more recent larger scale study by Chervin et al. (2006) provided further empirical support that tr eating SDB can improve behavior and cognition in children. Specifically, 76 childre n scheduled for adenotonsillectomy (primarily for OSA) and 27 children referred to other surgical clinics were evaluated for sleep, attention 23

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performance, and behavior prior to surgery a nd again one year later. The children undergoing adenotonsillectomy were reported to be significan tly more hyperactive per parent report and more inattentive based on a brief neuropsychologi cal battery (including a CPT task) relative to the control group prior to surgery. They also we re more likely to meet DSM-IV criteria for ADHD, based upon a child psychiatrists assess ment. One year later, the children who underwent adenotonsillectomy had improved signif icantly in all areas including hyperactivity and attention, eliminating the differences in performance seen prior to surgery. While relational evidence has been reporte d, the exact relations hip between SDB and cognitive and behavior problems has not been clearly defined. Most current theories suggest that sleep fragmentation or possibly nocturnal hypoxem ia likely underlie the daytime difficulties. Sadeh et al. (2002) report that sleep fragmentation negatively impacts sleep by increasing the amount of less restorative sleep and decreasing the length of d eeper, more restorative sleep stages. Additionally, Owens, Opipari, Nobile, & Spirito (1998) report that sleep fragmentation in children may not manifest as daytime sleepine ss as seen in adults, but rather as increased activity, aggression, impulsivity, acting out, inat tention, or poor con centration. Although a definitive conclusion cannot be made based on the curr ent literature, it is clear that the presence of SDB symptoms can negatively impact children s daytime functioning. As described here and in the preceding section, there are multiple simila rities in the types of cognitive and behavioral problems seen in children with epilepsy and children with sleep disorders. Given these similarities, the prevalence of sl eep disorders among children with epilepsy, and the potential for relatively easy and effective in tervention, it is not surprising th at recent resear ch has begun to look specifically at the impact of sleep di sorders in individuals with epilepsy. 24

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Epilepsy and Sleep Disturbance: Relationsh ip to Cognitive-Behavioral Functioning? There has been growing interest in the relatio nship of epilepsy and sleep disturbance, both in adults and children. A connection between slee p and seizures has long be en recognized. For example, in the 2nd century AD, the Greek physician Galen warned his patients with epilepsy to avoid sleepiness, and Sonorus hypothesized that untreated nightmares could lead to seizures, cautioning that sleep must be undisturbed in individuals with epilepsy (Temkin, 1994, p. 66). In 1885, Gowers described a relationship betwee n grand mal seizures and sleep, noting that nocturnal seizures typically o ccurred two hours after bedtime and from 4-5 AM while daytime seizures usually occurred in the first hour afte r awakening (Janz, 2000). Sleep deprivation is now well-recognized as a seizure precipitant (Jan z, 1962), and may be used clinically during the diagnostic process. Despite this longstanding recognition of a sleep -seizure relationship, there is still much that is uncertain about the interrelationship of sleep and epilepsy and the impact of comorbidity on daily functioning and epilepsy management. This is particularly true in the pediatric population, in which the limited research literature has focused pr imarily on descriptive accounts of sleep complaints among children with epilepsy and reports of sleep architecture abnormalities in relatively small samples or subp opulations with particular epilepsy syndromes characterized by sleep involvement. Much of the relatively limited amount of research about sleep disturbance and epilepsy has been conducted with adults. Drawing upon the adult literature, Bazil (2003) states there are multiple potential causes of sleep disturbance in patients with epilepsy, including general factors such as inadequate sleep hygiene, circadian rhyt hm disturbances, and co existing sleep disorders and factors specific to the epilepsy population, including seizures themselves and anti-epileptic medication side effects. Among adults, patients with epilepsy report a greater number of sleep disorder symptoms than healthy controls (Hoeppner, Garron, & Cartwright, 1984). Sleepiness in 25

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particular has been a common compla int in adults with epilepsy re lative to adults with no history of neurological disorder (Frost, Malow, & Aldrich, 1996; Hoeppner et al., 1984). Malow, Bowes, and Lin (1997) examined the predictors of excessive sleepiness in adults with epilepsy and found that participants who scored highly on the sleep apnea scale of a sleep disorders questionnaire and/or endorsed sy mptoms of restless leg syndrome also had an elevated score on the Epworth Sleepiness Scale (ESS). Interestingl y, among this sample of adults with epilepsy, symptoms supporting the presence of a treatable sleep disorder (e .g., apnea-like arousals) were stronger predictors of sleepiness than seizure frequency or numb er or type of anti-epileptic medications (Malow et al., 1997). Based on these findings, the rese archers suggested that before attributing sleepiness to anti-epil eptic medications or uncontrolled seizures, the possibility of a co-existing sleep disorder should be considere d, highlighting the importa nce of diagnosis of sleep disorders in patients with epilepsy (Devinsky, Ehrenberg, Barthlen, Abramson, & Luciano, 1994; Vaughn, DCruz, Beach, & Messenheimer, 1996). In addition, research has suggested that improving sleep by treating sleep disorders may impact seizure control. Beran, Plunkett, and Holland (1999) examined sleep disorder symptoms such as snoring in a sample of patients with epilepsy, and found that 75 percen t of the sample had a diagnosable sleep disorder, including obstructive sleep apnea, period ic leg movement syndrome, and upper airway resistance syndrome. When a small sample of these patie nts with epilepsy and obstructive sleep apnea (OSA) began treatment for their OSA, they reported at least a 50% decrease in seizures. Finally, recent research has even suggested that OSA may contribute to wo rsening seizure control or the onset of seizures in the older adult popul ation (Chihorek, Abu-Kalil, & Malow, 2007). Compared to a group of patients with resolved or improving seizures, the group with new/late onset seizures or worsening seizures had a si gnificantly higher apnea hypopnea index (consistent 26

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with OSA) and reported greater daytime sleepine ss. Similar to other studies, in a small subsample, treatment of OSA appeared to improve seizure control, but only for sleep-related (nocturnal) seizures (Chihorek et al., 2007) As previously noted, relative to the adult popul ation, less research has focused specifically on the nature of sleep disturbanc es in children with epilepsy. In general, child sleep patterns have been shown to be similar to those of adults particularly as the child approaches adolescence and adulthood (Carney, Becker, & Bongiolatti, 2005; Ferber, 1996). Likewise, there are similarities in the type of sleep disorders repo rted in the general pedi atric population (including children with epilepsy), with sleep-respirator y syndromes including obstructive sleep apnea, primary snoring, and upper airway resistance syndrome among the most commonly reported. Children with epilepsy have been reported to have higher rates of sleep problems than the general pediatric population (Zaiwalla, 1989). Pa rents are also more likely to report sleep problems in their children with epilepsy (Cor tesi, Giannoti, & Ottavi ano, 1999; Brum Batista & Lahorgue Nunes, 2007), including when they are co mpared to their nearest-aged sibing without epilepsy (Wirrell, Blackman, Barl ow, Mah, & Hamiwka, 2005). A recent study has also found that children with epilepsy self-report greater da ytime sleepiness than healthy controls (Maganti et al., 2006). In addition, polysomnography has demonstrated alterations in total sleep time, sleep latency, spontaneous awak enings, and REM latency in children with epilepsy (Becker, Fennell, & Carney, 2003; Hoeppner et al., 1984 ; Maganti et al., 2005; Sterman, Shouse, & Passousant, 1982). Interestingly, as has been re ported in adults, Koh, Ward, Meei, and Chen (2000) found that treating a sleep disorder, specifical ly sleep apnea, in children with epilepsy led to decreased seizure frequency in 5 of 9 patients aged 1-6 years. 27

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While the cognitive and behavioral effects of epilepsy and of sleep disorders have been explored separately, only a limited amount of research has examined the potential impact of sleep disorders on cognition and behavior among child ren with epilepsy. In children with sleep disturbance (primarily sleep-breathing disord ers), hyperactivity, inattention, and opposition have been reported (Zuckerman et al., 1987; Owens et al., 1998; Blunden et al 2000; Chervin et al. 2002). Similarly, children with epilepsy have been shown to exhibit more behavior problems such as inattention, poor concentration, and oppositionality than children with other chronic illnesses or healthy children (Austin, Risinger, & Beckett, 1992; Stores et al., 1998). In studies of children with concomitant epilepsy and slee p disturbance, a possibl e relationship between comorbidity and behavioral and emotional factor s has been suggested. An investigation of children ages 5 to 11 years with epilepsy f ound an association between daytime behavior problems and sleep disturbance (S tores et al., 1998). Zaiwalla (1989) also found that behavior problems are more common in children with epilep sy and are significantly associated with their sleep problems. Further, Cort esi et al. (1999) found a strong association between sleep and behavior and/or emotional proble ms in children with epilepsy, particularly in the older, overcontrolled and anxious group. However, these studies could not conclu sively state that the presence of sleep disturbance was the primary c ontributor to behavior problems. For example, the presence of a sleep disorder was often asso ciated with an increase in seizure frequency, which has also associated with behavior al problems (Hoare, 1984; Austin, 1988). Additional research has focused on behavior problems in children with epilepsy who endorse symptoms of sleep disturbance (Becker et al. 2003; Becker et al., 2004). Becker et al. (2003) employed a validated pediatric sleep ques tionnaire to examine parental report of sleep complaints in a sample of preadolescent childr en with epilepsy. Parents of children with 28

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epilepsy reported levels of daytime sleepiness, restless sleep, and snoring in their children that were comparable to parental re port of symptoms in children w ith known obstructive sleep apnea. Overnight polysomnogram results confirmed that all 14 children with epilepsy showed signs of respiratory disturbance (apnea + hyponea index > 1). Parent re port of behavior problems was also examined in the group of children with epilepsy. Based upon parent report questionnaires, sixty-four percent (64%) of the ch ildren with epilepsy were within the clinical range for attention problems and hyperactive-impulsive behavior and forty-three percent (43%) demonstrated clinical levels of exte rnalizing and oppositional behavior. The results of this study suggested that some neurobehavioral problems reported in children with epilepsy may be related to an underlying sleep disturbance. Becker et al. (2004) aimed to further explore the relations hip between sleep disturbance and daytime behavior in children with epilepsy. Parent ratings of child behavior and child selfreport of mood was examined in 30 children with epilepsy, ages 7 to 12 years. Overnight polysomngraphy was used to assess for nocturn al sleep problems, including symptoms of obstructive sleep apnea, nocturnal seizures, periodic leg movements, and sleep fragmentation. Results indicated that 80 % of the children with epilepsy exhi bited sleep disruption, due to either clinically significant obstructive sleep apnea, disturbance in sleep ar chitecture, or sleep fragmentation. Further, the majority of the sample of children with epilepsy had clinical levels of inattentive/hyperactive behavior and/or problem behaviors, as re ported on parent measures of behavior. Analyses indicated th at increased behavior problems were asso ciated with sleep disturbance, but not seizure seve rity, suggesting that sleep dist urbance may play a significant role in the development of parent-reported be havior problems in children with epilepsy. Additional research with this same sample fu rther indicated that sleep fragmentation, as 29

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measured by the number of nocturnal arousals, wa s associated with incr eased parent report of behavior problems (Bongiolatti, Fennell, & Ca rney, 2006). Increased sleep onset latency among the children whose parents reported significan t oppositional and hyperactive behaviors, also suggests the problem behaviors may be related to increased daytime sleepiness (Bongiolatti, Fennell, & Carney, 2005). Unfo rtunately, direct neuropsychologi cal testing of attention and learning was not conducted, and therefore, conclusi ons about potential cognitive difficulties were based solely on parent report. There has been very limited research a ttempting to objectively examine the possible relationship between sleep disturbance, ep ilepsy, and cognition. The only published study identified at this time compared attention pe rformance in a small sample of children with generalized epilepsy (versus hea lthy controls) using a continuous performan ce test (Maganti et al., 2005). Children with epilepsy performed signi ficantly worse on the attention measure, and were also found to have abnormal sleep archite cture, including longer rapid-eye-movement (REM) sleep onset. However, only a trend toward an association between sleep architecture and attention performance was found. The lack of neuropsychological studies examining cognitive performance and sleep disturbance in children wi th epilepsy limits our understanding of this complex relationship and suggests an opportunity for additional research. Purpose of Study Further work is needed to objectively examin e the relationship of sleep disturbance to behavioral problems and cognitive performance in children with epilepsy. The purpose of the current study was to examine the impact of sleep disturbance on attenti on, learning, and daytime behavior in children with epile psy by employing neuropsychologica l measures, parent ratings of behavior, and a comprehensive sleep questionnai re. In addition to clar ifying the nature of attention and learning problems in children with epilepsy, an established relationship would 30

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suggest treatment of sleep disorders in childre n with epilepsy could pr ovide one mechanism for ameliorating neurocognitive problems. Clarificati on of these relationships has several potential positive implications including enhancement of seizure control, reduction of daytime behavior problems, and improvement of cognitive functi oning including attention and concentration. Specific aims and hypotheses were as follows: 1. To determine whether sleep disturbance is a ssociated with increased attention problems in children with epilepsy. a. It was predicted that children with ep ilepsy and elevated levels of sleep disturbance would exhibit greater impa irment on neuropsychological tests of attention than those with lower levels of sleep disturbance. b. It was predicted that parent report of greater sleep disturbanc e would be directly correlated with impairment on measures of attention. 2. To determine whether sleep dist urbance is associated with increased learning difficulties in children with epilepsy. a. It was predicted that children with epileps y and greater levels of sleep disturbance would exhibit greater impairment on neur opsychological tests of learning than those with lower levels of sleep disturbance. b. It was predicted that parent report of greater sleep disturbanc e would be directly correlated with impairment on measures of learning. 3. To further establish the a ssociation between sleep disturbance and parent reported attention and behavior problem s in children with epilepsy. It was predicted that children with epilepsy with elevated levels of sleep problems woul d exhibit greater levels of 31

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32 attention and hyperactivity on pare nt report questionnaires than those with lower levels of sleep disturbance.

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CHAPTER 2 METHODS Participants Nineteen children diagnosed with epilepsy participated in this study. Children ranged in age from 6 to 12 years old. This age range was c hosen to reflect the greater prevalence of active seizure disorders among school-age d children, and in c onsideration of available normative data for the selected neuropsychological measures. Children were included if they carried a dia gnosis of partial or generalized epilepsy, as defined in the International L eague against Epilepsy (ILAE) Cl assification, including having previously experienced greater than one unprovoked seizure. All epilepsy diagnoses were confirmed by a University of Florida/Shands Ho spital pediatric neurologis t as documented in the childs neurology chart. Both partial and gene ralized epilepsies were included in the study as attention difficulties have been associated wi th both seizure types, and this allowed for maximized recruitment potential. Details regard ing recruitment challenge s are further discussed below. Inclusion criteria also required that a parent/caregiver was able to complete questionnaires and to provide consent for the ch ilds participation. To reduce the impact of confounding variables such as level of cognitiv e functioning and medication side effects, exclusion criteria incl uded: 1) an IQ of less than 70, as m easured by the two-subtest form of the Wechsler Abbreviated Scale of Intelligence (WASI); 2) currently taking greater than 2 antiepileptic medications or having changed medication in the past 56 days; 3) diagnosis of LennoxGastaut or other progressive brain disorder; 4) a significant history within the past two years of medical disease, (i.e., cardi ovascular, hepatic, renal, gynecologic, musculoskeletal, gastrointestinal, metabolic, endocrine, or cancer w ith a metastatic potential) which might impair participation in the study; 5) multiple physical and/or cognitive disabilities or a known diagnosis 33

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of mental retardation; and 6) a history of significant psychiat ric disease, which might impair participation in the study. Measures The full battery included brief intellectual screening, abbreviated neuropsychological testing focused on attention and learning, and pa rent-report behavior a nd sleep questionnaires (See Table 2-1). All children were administered a neuropsychological batte ry designed to assess attention and learning, the two primary areas of interest. Basic visuos patial/visuoconstructional ability and processing speed were also include d as proposed control measures. Specifically, because visuospatial/visuoconstructional impairment has not typically been reported in children with sleep disorders nor is it strongly associated with attention performance, it was anticipated that visuospatial/visuoconstructiona l skills would not be strongly a ssociated with sleep disorder symptoms in the current study (e.g., Williams, Gr iebel, & Dykman, 1998; OBrien et al., 2004; Baron, 2004). In light of controversial findings regarding the impact of AEDs on childrens processing speed (Loring & Meador, 2005), assessmen t of processing speed was also included to assess as a potential confound. Parents comple ted questionnaires rega rding their childs behavior, mood, and sleep, as well as demograp hic and medical histor y, including seizure history. Intellectual Screening To screen for cognitive ability, each child was administered the two-subtest version of the Wechsler Abbreviated Scale of Intelligence (WASI; Wechsler, 1999) This measure was chosen for its ability to reliably estimate intellectual functioning in a brief am ount of time. The WASI has been normed for children and adults ages 6 to 89 years old. The two-subtest version of the WASI provides an estimate of general intellectual ability (Full Scale IQ), and consists of the Vocabulary and Matrix Reasoning subtests. The Vocabulary subt est required defining words 34

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and the Matrix Reasoning subtest involved correc tly choosing a design to co mplete a visual array (Matrix Reasoning). The average reliability coe fficient for the two-subtest Full Scale IQ is 0.96 and test-retest reliability is 0.88 (Wechsler, 1999). For both tasks, raw scores were converted to age-adjusted T-scores. Attention Test of Everyday Attention for Children Children were administered selected subtes ts of the Test of Everyday Attention for Children (TEA-Ch; Manly, Robertson, Anders on, & Nimmo-Smith, 1999). The TEA-Ch is a standardized clinical battery designed to assess attention across multiple domains, including selective attention, sustained atte ntion, and attentional control, or switching. The TEA-Ch has been found to effectively differentiate children with Attention Deficit/Hyperactivity Disorder (ADHD) from both healthy cont rols and a non-ADHD clinical control population based upon patterns of performance across th e three attentional domains (Man ly et al., 2001; Heaton, et al., 2001). Structural equation modeling confirmed th at the three factor-model of attention assumed by the TEA-Ch formed a close fit to the patt ern of childrens performance on the measure (Manley et al., 1999). Reliability testing of the TEA-Ch indicate d moderate (.53) to high (.87) test retest correlations for all subtests (Manle y et al., 1999). The complete TEA-Ch consists of nine subtests, and is normed for use with chil dren ages 6 to 16 years based on a normative sample of 293 Australian children and adolescents. For all tasks, raw scores are converted into sex-specific, age-scaled scores, ranging from 1 through 19, with a mean of 10 (SD=3). A briefer battery was be employed for the present st udy, including Sky Search, Score!, and Creature Counting. First, Sky Search asse sses selective attention, or the ab ility to filter out or ignore distracting information in order to detect relevant information. It is a brief, timed task in which the child is instructed to find and circle pair s of target spaceships on a sheet of multiple 35

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distracter spaceships. Sky Search includes a se cond part in which ther e are no distracters; subtracting part two from part one provides a scal ed score index of the childs ability that is relatively free from the influence of motor sp eed (Sky Search Attention Score). Score! measures sustained attention, or the ab ility to maintain attention over time, even when the given task is quite boring. Children co unt the number of tones they hear played on a cassette tape, as if they were keeping track of a score in a computer game. The tones are separated by long delays, which when combined w ith the relative simplicity of the counting task, provides little to externally main tain the childs attention. Acco rdingly, it requires the child to self-sustain attention in order to successfully co mplete the task. Outcome is the number of trials correct out of ten trials. Creature Counting assesses attentional contro l/switching, which is the ability to change attentional focus flexibly and adaptively. A ttentional control/switchi ng involves efficiently stopping one task and beginning another or quic kly optimizing performa nce after changing the way that a task is performed. In Creature C ounting, the child is instructed to count aliens residing in tunnels. Occasional arro ws in the tunnels direct the ch ild to either count forward or count backward, requiring them to repeatedly switch the directi on in which they are counting. Creature Counting yields two scores, accuracy and a timing score. The timing score is essentially the average amount of time required to successfully complete a ll of the switches in a trial (total time in seconds / total number of switc hes), and is calculated on ly for correct trials. Creature Counting Timing Score was the primary variab le of interest in the current study. Conners Continuous Performance Test, 2nd Edition In addition, the children were administered the Conners Continuous Performance Test, 2nd Edition (C-CPT-II; Conners, 2000), a test of concentration and sustained attention commonly used in clinical settings. The C-CPT-II is a 14-minute computer administered task in 36

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which the respondent is required to press the space bar whenever a ny letter except the letter X appears on the computer screen. The inter-stimul us intervals (ISIs) va ry between stimulus presentations, with ISIs of either 1, 2, or 4 seconds. Several measures are provided by the CCPT-II program, including overall response times a nd errors. The primary variable of interest for this study was Hit Reaction Time (RT) Vari ability (Standard Error of Reaction Time for Hits). Increased hit RT variab ility has been strongly associat ed with the presence of ADHD symptoms (Epstein et al., 2003). In addition, number of omission and commission errors were reviewed. All variables were given as T-scor es. Performance is evaluated relative to a normative, non-clinical sample of 1920 participants. Split half reliability indices for all of the CPT performance measures range between .73 and .95, and test-retest reliabilities for a 3-month interval for a small sample of adu lts range between .55 and .84 (Conners, 2000). Trail Making Test Children were also administered the shor tened child-version of the Trail Making Test (Reitan, 1956). This brief test evaluates visual scanning ability and the ab ility to switch between two tasks (components of attention), as well as sequencing and processing speed. The Trail Making Test consists of two parts. Part A requires the child draw a line connecting 15 numbered and randomly arranged circles in order (much like connect the dots). Part B involves a page with circles containing the numbers 1 to 8 and the letters A to G, again randomly arranged. The child must draw a line that connects the circles, alternating between a number and a letter in sequential order (i.e., 1 to A to 2 to B to 3, etc.). If the child makes a mistake on either Part A or B, the mistake is quickly corrected by the examin er and the child continues. Scoring is the time required to complete each part. Timing scores were converted to age-adjusted z-scores for each child, based on normative means a nd standard deviations. 37

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Learning: Wide Range Assessment of Memory and Learning Both verbal and nonverbal learning were asse ssed using subtests from the Wide Range Assessment of Memory and Learning (WRAML; Sheslow & Adams, 1990). The WRAML is a comprehensive memory and learning battery normed for children between 5 and 17 years of age. It consists of nine subtests that assess me mory and learning of both visual and verbal information. For the present study, we employe d the two learning subtests: verbal learning, which involves word list recall, and visual learning, which involve s design location recall. On the Verbal Learning task, the child is asked to recall words from a list after each of four presentations, and following a 20minute delay. On the Visual Learning task, the child is asked to identify the locations of designs presented on a board after each of four presentations and again following a 20-minute delay. Both the Verbal and Visual Learning te sts yield a raw score that is converted to an age-ad justed scaled-score; the two scal ed scores will be used in the analyses. The WRAML was normed on a group of 2,363 children on the basis of a stratified model that reflected national demographic da ta (Sheslow & Adams, 1990). The WRAML demonstrates high reliability based on mean coe fficient alpha for Verbal Learning (.78), Visual Learning (.85) and the overall Learning Index (.91). Processing Speed: Wechsler Intelligence Scale for Children, Fourth Edition Processing Speed Index Processing speed was assessed using Coding and Symbol Search from the Wechsler Intelligence Scale for Children, Fourth Edition (WIS C-IV; Wechsler, 2003). Together these two subtests yield a Processing Speed Index, an age-ad justed Standard Score (mean of 100, standard deviation of 15), which was used as an overall measure of processing speed. The WISC-IV was normed on a sample of 2200 children from 11 one-year age bands. 38

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Coding is a motor speed task that is sensitive to deficits in focused or sustained attention. In this task, the child must tran scribe geometric symbols that are matched with the digits 1 9 as quickly as possible within a two-minute limit. The score is based upon the number completed (with possible bonus points for time), and then co nverted to an age-scaled score (mean of 10, standard deviation of 3). Test-retest reliabity conducted with a subset of 243 children was .70. Symbol Search is a match-to-sample test of speed of processing. Children are required to scan five geometric symbols to decide whether eith er one of two target symbols is present, and to then mark the appropriate yes or no box. Th ere is a 120 second time limit, and the score is the number correct minus the number incorrect (a s a correction for guessing) completed within the time limit. This score is then converted to an age-corrected scale score (mean of 10, standard deviation of 3). Symbol Search test-retest reliablity (n=243) was .76. Visuospatial/Visuoconstructional Ability: Beery-Buktenica Developmental Test of VisualMotor Integration, Fifth Edition Assessment of basic visuospatial and vis uoconstructional abilities was included as a proposed control measure, as it was predicted that this aspect of functioning should not be impacted by the presence of a sleep disorder. Visuospatial/visuoconstr uctional ability will be assessed using the Beery-Buktenica Developmenta l Test of Visual-Mot or Integration, Fifth Edition (VMI; Beery, Buktenica & Beery, 2004). The VMI is a commonly used, standardized copy forms test with an objective scoring syst em. Children are required to copy a series of increasingly complex geometric shapes within a testing booklet. Scoring is based on accuracy of each copied figure, and yields an age-adjusted standard score, which was used in analyses. Parent Questionnaires The parent/caregiver was asked to complete a pediatric sleep questionnaire as well as two behavior questionnaires, whic h are described below. 39

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Childrens Sleep Questionnaire-Parent Report An in-house questionnaire called the Childre ns Sleep QuestionnaireParent Report (CSQPR) was employed to assess the frequency of slee p disturbance symptoms (See Appendix). The CSQ-PR is a 90-item close-ended parent report measure used to assess the presence and frequency of various sleep diso rders, including sleep disord ered breathing, restless leg syndrome/period leg movement disorder as well as di srupted sleep habits. It is derived in part from existing sleep questionnaires, including the Pediatric Sleep Questionnaire (Chervin, Hedger, Dillon, & Pituch, 2000) and the Childrens Sleep Health Questionnaire (Owens et al., 2000). The questionnaire also includes a suppl emental open-ended section for assessment of general medical history and sleep history. The CSQ-PR was used to assess for the presence of sleep disorder symptoms. For the current st udy, a Total Sleep Probl ems Score was created summing the parent report of symptoms of sleep disturbance. Indivi dual subscales (e.g., Breathing Symptoms; Daytime Sleepiness Symptoms) were also summed. The measure had not yet been standardized. However, it was chos en for its more comprehensive assessment of symptoms of sleep disturbance. Behavior Assessment System for Children, Second Edition The Behavior Assessment System for Child ren, Second Edition (BASC-2) measures multiple aspects of observable behavior in children and adolescents. Parents completed the BASC-2 Parent Rating Scales (BASC-2 PRS), a co mprehensive questionnaire designed to assess a child or adolescents adaptive and problem beha viors both at home and in the community. The BASC-2 PRS is comprised of 14 subscales, which combine to yield four composite indices. Comparable forms are available for children age 6 to 11 years (PRS-C) and adolescents age 12 to 21 years (PRS-A). For the purposes of this study, we primarily examined the Attention Problems Subscale and Hyperactivity Subscale. Scores were convert ed to age-adjusted T-scores 40

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for comparison. The BASC-2 was standardized using a large, census-matched normative sample (n=3600 for PRS-C and PRS-A combined). The BASC PRS-C and PRS-A internal-consistency reliabilities are very high for the composite indices (0.900.95), and are also high across subscales (0.72-0.88; Attention: 0.87-0.88; Hyperactivity: 0.82-0.86). Conners Parent Rating Scales-Revised, Long Form The Conners Parent Rating Scales-Revised, Long Form (CPRS-R:L; Conners, 2001) uses parent ratings on 80 items to evaluate problem behavior in children and adolescents ages 3 to 17 years. For the purposes of this study, the CPRS-R:L wa s employed to assess parents perception of ADHD-related behaviors consistent with current diagnostic criter ia. The CPRS-R:L is comprised of 13 subscales. All scores are converted to ageand sex-adjust ed T-scores for comparison. The CPRS-R:L was standardized usi ng a large normative sample (n= 2200). The CPRS-R:L has very good internal consistency across scales (.75 to .94) and has been shown to effectively differentiate children with and without ADHD (s ensitivity of 92.3%, sp ecificity of 94.5%; Conners, et al, 1998). The DSM -IV Symptom subscales were used in analyses to examine clinically relevant ADHD symptoms. Demographic and medical information The parent/caregiver comp leted a brief demographic/background questionnaire that included items such as age, birth date, grade le vel, maternal and pate rnal education level, maternal and paternal occupation, significant medi cal history aside from epilepsy, and history of academic progress and learning disa bilities. Epilepsy data incl uding epilepsy type, recent AED history, age of onset and duration of illness, and a consecutive 56-day seizure frequency count (as a gross measure of seizure severity) was also obtained. Me dical and epilepsy history was supplemented and/or confirmed via medical record review as needed. 41

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Procedure Recruitment Participants were recruited from the Pediatric Neurology outpatient clinics at the University of Florida/Shands Hospital in Gain esville, Florida under th e supervision of three attending pediatric neurologists. Participants were initially identified via review of medical records of children scheduled for clinic visits (under a HIPAA Waiver of Authorization for Screening Subjects) or by the attending phys ician or nurse practitioner during routine neurological visits. Medical r ecord review was used to determ ine diagnosis (e.g., does the child have epilepsy?), age, and general functional st atus (e.g., does child have speech?) of children presenting at clinic. Participants were recruited exclusively through direct contact by the primary investigator with the assistance of th e pediatric neurologists and nurses. Based upon medical chart review or referral by the neurologist or nurse, potenti al parent(s) and children were approached during regularly scheduled visits and informed about the study opportunity. Recruitment fliers were also posted at Shands Hospital and the Shands Medical Plaza, but yielded no responses. Multiple challenges were faced during the recruitment phase of this project. The initial study design required that children be scheduled for an overnight sleep study and excluded children with a diagnosis of or symptoms consistent with ADHD (as assessed by a screening measure). These criteria proved to be overly restrictive for our sample of children, as recruitment over several months yielded only five poten tial subjects of which one consented and completed participation. Weekly sc reening of referrals to the Univ ersity of Florida Sleep Center yielded no children with epilepsy within the st udy age range over a six month period. Removal of the sleep study inclusion criterion and the ADHD exclusion criterion increased participant eligibility, although overall recruitment numbers remained lower than anticipated at the initial 42

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proposal of the project. Specifically, an esti mated 45 families were approached regarding the study, and of these, approximately 35 parents of eligible children e xpressed interest in participating. After initial expression of interest, several families were unable to participate for extraneous reasons, including the in ability to return to Gainesvill e for testing due to the expense of travel. Approximately five families failed to return telephone calls regarding scheduling a testing session or did not show for scheduled testing appointments. Twenty-four child/parent diads were consented/assented and enrolled in the study, and 19 children completed all study requirements. Following initial enrollment, th ree children were excluded based upon IQ score or the lack of basic skills required for the neuropsychological testing (e.g., counting, letter recognition). Two children who were successfully screened were unable to complete testing, one due to illness and the other due to the death of the parent. Screening and Assessment Prior to initiating testing, consent of pare nts of children meeting inclusion/exclusion criteria and assent of children age 7 and older were obtained per University of Florida and Shands Hospital IRB protocol. Participants were either tested following their regularly scheduled Pediatric Neurology clinic visit at the Shands Medical Plaza or at a later date in the UF/Shands Hospital Psychology Clinic. Two particip ants were tested in private study rooms at the public library in their hometown. Participants were first screened for general cognitive ability using th e two-subtest version of the Wechsler Abbreviated Scale of Intelligen ce (WASI; Wechsler, 1999) When possible, the screening was completed at the Pediatric Neurology Clinic visit in order to confirm eligibility prior to additional scheduling for testing. Children who met the elig ibility criteria of a Full-Scale IQ of 70 were administered the remainder of the neuropsychological test battery as described 43

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44 above while the parent/guardian completed the demographic/medical questionnaires and behavior questionnaires. The full neuropsychological test battery required approximately two hours to complete (including the screener). Breaks were provided as needed to prev ent fatigue. Test administration followed a fixed order that did no t vary between participants. Children were given a small gift (valued at less than or equal to $5) to thank them for their participation, and parents were provided with a brief written summary of the neuropsychological findings.

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Table 2-1. Study measures. Respondent Domain Measure Child Intellectual Screening Wechsler A bbreviated Scale of Intelligence (WASI) Attention Test of Everyday Attention in Children (TEA-Ch) Sky Search TEA-Ch Score! TEA-Ch Creature Counting Trail Making Test Conners Continuous Performance Test, 2nd Edition (C-CPT-II) Learning Wide Range Assessment of Learning and Memory (WRAML) Verbal Learning WRAML Visual Learning Processing Speed Wechsler Inte lligence Scale for Children, 4th edition (WISC-IV) Coding WISC-IV Symbol Search Visuospatial/Visuoconstructional Beery-Bukten ica Test of Visual-Motor Integration, 5th edition (VMI) Parent Sleep Childrens Sleep Ques tionnaireParent Report (CSQ-PR) Attention/Behavior Behavior Assessment System for Children, 2nd Edition (BASC-2) Conners Parent Rating Scale-Revised:Long Version (CPRS-R:L) 45

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CHAPTER 3 RESULTS Preliminary Analyses All statistical tests were performed using the SPSS 16.0 statistical analysis package. Raw scores were converted to Standa rd Scores (M = 100, SD = 10), T-scores (M = 50, SD = 5) or Scaled Scores (M = 10, SD = 3) for statistica l analysis, per standardized administration and scoring procedures. For all statistical te sts, the level of si gnificance was set at = 0.05. All dependent variables were evaluated for skewness as a measure of distribution symmetry. Using a cut-off of .0 as an indication of normality, al l dependent variables were determined to be within acceptable limits of normal distribution. No formal normalization corrections were necessary. Data was missing for three variable s: the C-CPT-II (one case), Trail Making Test Part B (one case), and the Cr eature Counting Timing Score (sev en cases). Missing data was attributed to functional limita tions, and therefore it was excluded casewise from statistical analyses. In addition, one case was exclude d from the C-CPT-II variable based upon its questionable validity (per standardized administra tion guidelines), which was noted to be related to the childs poor cooperation with the task. Group Assignment The primary aims of this study involve assess ing differences in at tention and learning performance in children with epilepsy with a nd without sleep disturbanc e. For the purpose of data analysis, participants were assigned to one of two groups based upon parent report of sleep problems on the Childrens Sleep Questionnaire-Parent Report (CSQ-PR). Based upon a median split of the total sleep problems score on the CSQ-PR, ten children were assigned to the Higher Frequency Sleep Problems group and nine to the Lower Frequency Sleep Problems group. The median total score on the CSQPR was 99.00 (range 31 to 202). 46

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Demographics Demographic variables between groups were compared using independent sample t-tests and Chi-square or Fishers exac t test analyses. A summary of demographic information for both groups is presented in Table 3-1. The mean age of the Higher Frequency Sleep Problems group was 9 years, 4 months (range 6 years, 3 months to 12 years, 6 months). The mean age of the Lower Frequency Sleep Problems group was 10 years, 5 months (range 6 years, 4 months to 12 years, 9 months). T-tests indicated that there were no significant differe nces between groups in age. The Higher Frequency Sleep Problems group c onsisted of five females, and five males, while the Lower Frequency Sleep Problems group c onsisted of six females, and three males. Among the Higher Frequency Sleep Problems group, ni ne of the children were reported by their parent to be Caucasian and one was reported to be African-American /Hispanic (mixed ethnicity). The Lower Frequency Sleep Problems group was composed of seven Caucasian children, one Hispanic child, and one African-American/Caucasian (mixed ethnicity) child. Initial Chi-square analyses indicated that Fishers Exact test should be employed, which revealed no significant differences in the male:female ratio or the ratio of various racial/ethnic classifications between the two groups. Maternal education was used as a marker for socioeconomic status. Mothers of children in the Higher Frequency Sleep Problems group had an average of 14 years of education, and the mothers of the children in the Lower Fr equency Sleep Problems group had an average of 12 years of education. T-tests indicated that th ere were no differences in maternal education. Children were screened for eligibility using the Wechsler Abbreviated Scale of Intelligence (WASI) 2-Subtest Form. In one case the child was administered the Wechsler Intelligence Scale for Children, Fourth Edition (WISC -IV). Results of an independent samples ttest comparing the two groups on intellectual func tioning revealed that th ere was no significant difference in mean Full Scale IQ betw een the two groups (See Table 3-1). 47

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Medical and epilepsy information was also collected from the parents and the medical record. In total, twelve child ren with generalized epilepsy and seven children with partial epilepsy participated in the study. Among the children with generalized epilepsy, six were diagnosed with absence epilepsy, five had pr imary generalized epilepsy, and one child had myoclonic epilepsy. In the Higher Frequenc y Sleep Problems group, eight children had generalized epilepsy and two children had part ial epilepsy. In the Lower Frequency Sleep Problems group, four children had generalized epile psy and five children had partial epilepsy. Fishers Exact test indicated that the ratio of generalized epilepsy to partial epilepsy did not differ significantly between the two groups (see Ta ble 3-2). Seizure status was also recorded, and categorized based upon whether the child has had a seizure in the past two months. The result of Fishers Exact analysis revealed that th e ratio of children who ha d seizures in the past two months and those who had no seizures in the past two months did not differ between the two sleep groups. Finally, anti-epileptic medication information wa s also collected for all children. Children were predominantly on monotherapy for the trea tment of epilepsy (57.9% of total sample). Three children were treated with two anti-epileptic medications (15.8% of total sample), while five children were not currently taking any anti -epileptic medication (26.3 % of total sample). Anti-epileptic medications were also compared in the two groups (See Table 3-2). In the Lower Frequency Sleep Problems group, fo ur children were taking no AEDs four children were taking one AED, and one child was taking two AEDs. In the Higher Freque ncy Sleep Problem group, one child was taking no AEDs, seven children we re taking one AED, and two children were taking two AEDs. Because of the small sample size, AED status was reduced to a two-factor 48

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variable (on or off AEDs). Fish er Exact test analysis revealed that there were no differences between groups. Aim 1: Comparison of Attention Performa nce in Lower Frequency Sleep Problem and Higher Frequency Sleep Problem Groups Means, standard deviations, and statistical results for the attentional tasks are presented in Table 3-3. Independent samples t-tests were employed to test whether children with epilepsy with a higher frequency of sleep problems pe rformed more poorly on a ttentional tests than children with epilepsy and a lower frequency of sleep problems. Initial dependent variables for selective attention included th e TEA-Ch Sky Search Attention sc ore (time per target) and Trail Making Test Part A. For sustained attenti on, the dependent variables included the TEA-Ch Score! number correct and the C-CPT-II Hit Rate Variability score. Dependent variables for attentional control/switching were the TEA-Ch Creature Counting Accuracy score and Trail Making Test Part B. Selective Attention The Higher Frequency Sleep Problems group and the Lower Frequency Sleep Problems group did not differ on either measure of selectiv e attention. On the TEA-Ch Sky Search test, the mean performance of both groups fell within the low average range relative to age-based norms, but did not differ significantly from one a nother, (See Table 3-3) On the secondary measure of selective attention, Tr ail Making Test Part A, the two groups also did not differ from each other. The mean of both groups fell with in the average range relative to age-based normative data. Cohens d effect sizes (Cohen, 1977) were ca lculated using pooled standard deviations (Rosnow & Rosenthal, 1996). Effect sizes for both Sky Search and Trails A were very small. 49

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Sustained Attention Two measures were used to assess sustaine d attention, TEA-Ch Score! auditory sustained attention test and the C-CPT-II visual sustained a ttention test. Results are presented in Table 33. The Higher Frequency Sleep Problems Group performed significantly worse than the Lower Frequency Sleep Problems group on the auditory sustained attention test (TEA-Ch Score!). Notably, the Higher Frequency Sleep Problems gr oup also performed within the clinically significant range (scaled score < 7) on this ta sk, whereas the Lower Frequency Sleep Problems group was within normal limits. Cohens d effect size was in the very large range. It should be acknowledged that given the tota l number of t-tests employed in this study, there is a high experiment-wise error rate. If the p -value for the t-tests examini ng all attention variables were Bonferroni-corrected (.05/9), the critical value of alpha would be adjusted to p < .00556, and this finding would no longer be significant. In contrast to the auditory sustained atte ntion findings, the two groups did not differ on the visual sustained attention task, the C-CPT -II. Hit Reaction Time (RT) Variability was similar in both the Higher Frequency Sleep Problems group and the Lower Frequency Problems group. In addition, the number of Omissionswhich is strongly and specifically associated with poor vigilancedid not differ betw een the groups. The performance of both groups on both CCPT-II measures were within normal limits. Eff ect sizes were in the small to medium range. Attentional Control/Switching Finally, the groups were compared on two meas ures of attentional control and switching (See Table 3-3). The first proposed measure of attentional control/switching was the Timing score from the TEA-Ch Creature Counting subtes t. The Timing score takes into account both speed, accuracy, and the number of switches made during the task, and can be calculated when greater than 2 of the 7 test items are correct. However, only 12 of the 19 children in the study 50

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were able to correctly complete 3 or more items. Because these missing cases were due to limitations in task performance, it was determin ed that standardized methods for replacing missing data would not be appropriate. Among this smaller sample (6 children per group), the Creature Counting Timing Score did not differ between the Higher Frequency Sleep Problems group and the Lower Frequency Sleep Problems group. A medium effect si ze was calculated. Both groups performed within broad normal lim its, although the mean score of the Higher Frequency Sleep Problems group was lower than the Lower Frequency Sleep Problems group. In light of the apparent difficulty that severa l children had on this task, the Creature Counting Accuracy score was also examined. Again, there was no significant differe nce in the number of items correct between the two Sleep Problem groups and the effect size was small. Both groups performed generally in the low average range (mean scaled scores of 7 to 8). Trail Making Test Part B was included as a second measure of attentional control/switching. There was no significant diff erence in performance be tween the two groups. Both groups performed within normal limits relative to age-based normative data, although it should be noted that the Higher Frequency Sl eep Problems group performed somewhat better than the Lower Frequency Sleep Problems group. The effect size was small. Correlation between Parent Report of Sleep Disturbance and Attention Performance Pearsons correlations using the Total Sleep Problems Score from the CSQ-PR and the attention variables from the TEA-Ch, C-CPT-II, and the Trail Making Test were conducted to examine the relationship between parent report of child sleep disturbance and attention performance. TEA-Ch Score! a nd the CSQ-PR were moderately negatively correlated, although the correlation did not quite reach statistical significance (See Table 3-4 and Figure 3-1). Otherwise, results indicated that there were no significant relationships between the Total Score on the CSQ-PR and the attention variables in this sample of children with epilepsy. The 51

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correlations between the CSQ-PR an d each of the attention variables are graphically presented in Figures 3-1, 3-2, and 3-3. Employing scatterplots enabled closer review of the data correlations, and suggested that some correlations, such as that between the Creatur e Counting scaled score and the CSQ-PR, may have been adversel y skewed by outliers (See Figure 3-3). Aim 2: Comparison of Learning Performa nce in Lower Frequency Sleep Problem and Higher Frequency Sleep Problem Groups Group Comparison Independent sample t-tests were conducte d to compare verbal and visual learning performance in children with epilepsy and a hi gher frequency of sleep problems and children with epilepsy and a lower frequency of sleep problems. Means, standard deviations, and statistical results for the learni ng variables are presented in Tabl e 3-5. The dependent variables for learning included the Verbal Learning score and the Visual Learning score from the Wide Range Assessment of Memory and Learning (WRAML). The Lower Frequency Sleep Problems group di d not perform significantly better than the Higher Frequency Sleep Problem group on the WRAML Verbal Learning test. The mean scores for both groups also fell solidly within the aver age range relative to ag e-adjusted norms. A similar result was found for Visual Learni ng. The Lower Frequency Sleep Problems group performed only slightly better (not significantly) than the Hi gher Frequency Sleep Problems group on the Visual Learning test, and scores for both remained within normal limits. Effect sizes for both Verbal Learning and Visual Learni ng were in the small to medium range. Correlation Between Parent Report of Sleep Disturbance and Learning Performance As were done for the attention scores, Pearsons correlations using the Total Sleep Problems Score from the CSQ-PR and the WRAML Verbal and Visual learning scores were conducted to examine the relationship between pa rent report of child sleep disturbance and 52

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learning performance (See Figure 3-4). There was no significant correlation between sleep problems and verbal learning (r = -0.30, N = 19, p = 0.10, one-tailed) or vi sual learning (r = 0.00, N = 19, p = 0.49, one-tailed). Although the relati onship was not significant, it was noted that there was a trend towards verbal learning and sl eep problems being negatively correlated, which could bear out with a larger sample size (See Figure 3-4). Aim 3: Comparison of Parent Report of A ttention and Behavior Problems in Lower Frequency Sleep Problem and Higher Frequency Sleep Problem Groups The final study aim was to replicate prior findi ngs of an association between parent report of sleep problems and parent report of inatte ntion and behavior problems in children with epilepsy. For the purpose of this study, hyperact ivity was chosen as the primary behavior of interest because of its frequent association with inattention. Independent sample t-tests were again employed to test whether the children with epilepsy reported to have Higher Frequency Sleep Problems and the children with epilepsy reported to have Lower Frequency Sleep Problems differed on parent report measures of a ttention and hyperactivity. Dependent variables included the Attention subscale score and Hype ractivity subscale score from the Behavior Assessment System for Children, Second Edition, Parent Rating Scales (BASC-2:PRS). In addition, to more specifically l ook at diagnostic symptoms associ ated with Attention Deficit Hyperactivity Disorder (ADHD), the DSM-IV: In attentive subscale, the DSM-IV: HyperactiveImpulsive subscale, and the DSM-IV: Total subs cale from the Conners Parent Rating ScalesRevised, Long Form (CPRS-R:L) were in cluded as dependent variables. Means, standard deviations, a nd statistical results for the pa rent report questionnaires are presented in Table 3-6. Parents reported a greater degree of a ttention problems in the Higher Frequency Sleep Problems group than the Lo wer Frequency Sleep Problems group on the BASC-2 Attention subscale; however, this difference was not significant. Cohens d effect size 53

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was medium to large in size. Although th e Higher Frequency Sleep Problems group was relatively elevated on this scale, neither group fell with in the clinically elevated range on the measure (T-score > 65). The Higher Frequency Sleep Problems group also did not differ significantly from the Lower Frequency Sleep Problems group on the BASC-2 Hyperactivity scale. Cohens d was of medium size. The means for both groups were solidly within the average range for this scale and not indicative of clinical concern. Endorsement of symptoms of attention and hyperactivity/impulsivity consistent with a DSM-IV diagnosis of ADHD were compared in the two groups using the CPRS-R:L. The Higher Frequency Sleep Problems group scored higher than the Lower Frequency Sleep Problems group across all three DSM-IV subscale s, but the difference between the two sleep problems groups was not sta tistically signifi cant (See Table 3-6). Cohens d estimates of effect were small to medium in size. Although neith er group exceeded clinical cutoff scores on the measure (T-score > 65), the mean of the Higher Frequency Sleep Problems group was at least one standard deviation above the normative mean (T-score = 50) on all three subscales. Additional Analyses Supplementary Neuropsychological Measures: Processing Speed and Visuospatial/ Visuoconstructional Ability In addition to the primary study battery, supplementary neuropsychological measures were included as potential control measures. Specifically, the Processing Speed Index composed of Coding and Symbol Search from the W echsler Intelligence Scale for Children, 4th Edition (WISC-IV) was included to assess for potential deficits in pro cessing speed that might underlie performance on attention tasks. The Beery-Bukt enica Test of Visual Motor Integration (VMI) was included as a proposed dissociation control, as it was anticipated that the children with 54

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epilepsy would perform similarly on this visuospa tial/visuoconstructional measure regardless of the presence or absence of sleep problems. In order to test whether the Higher Fr equency Sleep Problems group and the Lower Frequency Sleep Problems group demonstrated si milar processing speed skills, an Independent Samples t-test was conducted using the Processing Speed Index score as the dependent variable. Although the difference was not st atistically significant, the Higher Frequency Sleep Problems group ( M = 80.80, SD = 21.81) performed worse on the proce ssing speed measures than the Lower Frequency Problem Sleep group ( M = 92.67, SD = 13.26), t(17) = 1.45, n.s. An independent samples t-test was also conducted to assess whether the two groups differed in visuospatial/visuoconstr uctional skills. The ability to reproduce line drawings did not significantly differ between the children in the Lower Frequency Sleep Problems ( M = 87.33, SD = 10.55) group and the Higher Freq uency Sleep Problems group ( M = 79.80, SD = 9.88), t(17) = 1.61, n.s. Clinically, the Lower Frequency Sleep Problem group performed within broad normal limits; however, the Higher Frequency Sleep Problems group performed below average. Comparison of Diagnostic Groups: Sleep Disorder Symptoms As described above, the two group of interest in this study were created using a median split employing the parent responses to a comp rehensive sleep questionnaire (Total Sleep Problem Score), the CSQ-PR. This method was a dopted in part because the small sample size precluded the use of other anti cipated statistical approaches, including regression analyses. Subsequently, a more qualitative, diagnostic me thod was employed to better ensure that the two groups represented clinicallyand not just statisticallydifferent groups of children with regard to sleep disorder symptoms. Specifically, re view of the CSQ-PR was conducted to select specific items from the Breathing Subscale, Movement Subscale, and Daytime Sleepiness Subscale that were representative of key dia gnostic signs or symptoms of the more common 55

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pediatric sleep disorders. A list of these items is available in Appendix A. Children who were described as often or very often experienci ng the majority of symptoms from either the Breathing Subscale or the Movement Subscale and the Daytime Sleepiness Subscale were assigned to the Sleep Disorder Symptoms gr oup (n = 8), while the children described as predominantly sometimes, rarely, or never experiencing the symptoms were assigned to the Non-symptomatic group (n = 11). The performance of these two groups on the neuropsychological measures of attention and learning were then compared as described above for the Higher Frequency and Lower Freque ncy Sleep Problems groups. Specifically, independent samples t-tests comparing th e Sleep Disorder Symptoms group and Nonsymptomatic group were conducted using the fo llowing dependent variables: TEA-Ch Sky Search Attention score, TEA-Ch Score!, TEA-C h Creature Counting Accuracy, C-CPT-II Hit RT Variability, C-CPT-II Omissions, C-CPT-Commissions, WRAML Verbal Learning, and WRAML Visual Learning. Results are presented in Table 3-7. Consistent with the prior analyses, the Sleep Disorder Symptoms group dem onstrated poorer auditory sustained attention on the TEA-Ch Score! test, relative to the N on-symptomatic group. Th e two diagnostic groups did not differ significantly on any other m easures of attention and learning. Clinical Inattention Symptoms among the Two Sleep Disorder Symptom Groups To reduce the multiple attention variables to a single variable that could be examined across the two diagnostic sleep groups (Sleep Disorder Symptoms group and Non-symptomatic group), a similar approach was used to assign ch ildren to either a clinical or non-clinical attention group. TEA-Ch scaled scores and C-CPT-II HIT RT Variability were examined to identify children who demonstrated impairment on at least two of the measures (TEA-Ch scaled score < 7, C-CPT-II T-score > 65). Based upon these criteria, five children were assigned to the Clinical Inattention group and the remaining 14 were assigned to the Non-Clinical group. 56

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The frequency of clinical inattention in the two sleep disorder symp toms groups was then examined. Only one of the 11 children (9.1%) in the Non-symptomatic Sleep group had signs of clinically significant attention problems. In contra st four of the eight chil dren (50%) in the Sleep Disorder Symptoms group had clin ically significant atte ntion problems. A Fishers Exact test was employed to examine the association betwee n symptoms of sleep di sorder and attention impairment. There was no statistically significan t relationship between sl eep disorder symptoms and tendency toward attention impairment (p = .07). Effect of Seizure Type In order to determine whether there we re unique effects of seizure type on neuropsychological performance or sleep dist urbance, independent samples t-tests were employed comparing children with generalized seizures (n = 12) and children with partial seizures (n = 7). The dependent variables incl uded the attention and learning variables used in the primary analyses as well as the CSQ-PR Total Sleep score, Breathing subscale score, Movement Related subscale score, and Daytime Sleepiness subscale score. Means, standard deviations, and statistical results are presented in Table 3-9. The children with partial seizures performed significantly worse than the children with generalized seizures on the TEA-Ch Sky Search measure of selective attention. The two groups did not differ on any other measure of attention or learning. Effect of Seizure Status The final sample for the study included both chil dren who have recently been seizure-free and children who are currently experiencing seizures on a more frequent basis. To determine whether there were differences in neuropsychol ogical performance or sleep problems based upon current/recent seizure activity, independent samples t-tests employing two levels of the independent variablechildren with recent seizures (n = 8) and without recent seizures (n = 57

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11)were employed. Neuropsychological dependent variables were the attention and learning variables included in the primary analyses. Sleep complaint variables included the CSQ-PR Total Sleep score, Breathing subscale, Moveme nt Related subscale, and Daytime Sleepiness Subscale. Means, standard deviations, and statis tical results are presented in Table 3-10. The children who have had seizures in the past two months did no t differ significantly from the children who have not had seizures on any of the measures. 58

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Table 3-1. Demographic characteristics of higher and lower frequency sleep problem groups Variable Higher Frequency (n = 10) Lower Frequency (n = 9) Test Statistic p-value Age (months) 112.20 (30.69) 125.00 (22.34) 1.031 NS Sex (# males) 5 3 NA2 NS Race/ethnicity (# Caucasian) 9 7 NA2 NS Maternal Education (years) 14.2 (3.0) 12.2 (2.4) -1.38s1 NS Full Scale IQ 92.10 (10.27) 93.67 (16.87) 0.261 NS Note: Values presented as Mean ( SD ) unless otherwise noted; 1 t -value; 2Fishers Exact test was used, which yields no test statistic. 59

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60 Table 3-2. Epilepsy/seizure char acteristics of higher and lower frequency sleep problem groups Variable Higher Frequency (n = 10) Lower Frequency (n = 9) p-value Seizure type (# generalized) 8 4 NS Seizure status (# seizure-free) 5 3 NS AED Status (# medication-free) 1 4 NS Note: Analyses conducted using Fishers Exact test which yields no test statistic; AED = Antiepileptic drug.

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Table 3-3. Mean comparison of attention perfor mance between sleep problem frequency groups Domain Variable Higher Frequency (N = 10) Lower Frequency (N = 9) One-tailed p-value Effect size (Cohens d) Selective Attention Sky Search Time per Target 7.80 (2.15) 7.56 (3.40) 0.42 0.08 Trails A 0.04 (0.92) 0.09 (1.07) 0.47 0.05 Sustained Attention Score! 6.80 (2.30) 9.56 (2.51) 0.01* 1.15 C-CPT-II Hit RT Variability 53.56 (9.98)1 50.78 (8.79) 0.28 0.30 C-CPT-II Omissions 53.00 (8.40)1 49.54 (5.25) 0.16 0.49 C-CPT-II Commissions 54.78 (2.34)1 53.14 (9.12) 0.31 0.25 Creature Counting Accuracy 7.50 (2.99) 8.44 (3.54) 0.27 0.29 Attentional Control/Switching Creature Counting Timing 8.83 (1.72) 2 10.00 (1.90)2 0.15 0.65 Trails B 0.10 (0.86)3 -0.27 (1.18) 0.23 0.05 61 Note: Mean ( SD ); *p<.05; 1n = 8, 2n = 6, 3n = 9

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Table 3-4. Correlation of sleep disorder symptoms on the CSQ-PR and attention problems Domain Variable Pearsons r p -value Selective Attention Sky Search Time per Target 0.10 0.35 Trails A 0.03 0.46 Sustained Attention Score! -0.38 0.05 C-CPT-II Hit RT Variability 0.08 0.38 C-CPT-II Omissions 0.04 0.45 C-CPT-II Commissions 0.23 0.18 Creature Counting Accuracy -0.08 0.38 Attentional Control/Switching Creature Counting Timing -0.24 0.23 Trails B 0.20 0.22 62

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63 Table 3-5. Mean comparison of learning perfor mance between sleep problem frequency groups Variable Higher Frequency (N = 10) Lower Frequency (N = 9) One-tailed p-value Effect size (Cohens d) WRAML Verbal Learning 9.60 (1.96) 10.44 (3.40) 0.22 0.36 WRAML Visual Learning 8.30 (3.02) 9.89 (3.79) 0.16 0.46 Note: Mean ( SD )

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Table 3-6. Mean comparison of parent re port of attention and behavior problems between sleep problem frequency groups Measure Variable Higher Frequency (N = 10) Lower Frequency (N = 9) One-tailed p-value Effect size (Cohens d) BASC-2 Attention 58.60 (6.59) 50.89 (12.96) 0.07 0.75 Hyperactivity 55.00 (5.16) 48.67 (12.04) 0.06 0.68 CPRS-R:L DSM-IV Inattentive 60.90 (11.82) 55.56 (15.22) 0.20 0.39 DSM-IV Hyperactive/Impulsive 60.60 (8.11) 55.67 (10.65) 0.13 0.52 DSM-IV Total 62.40 (11.26) 56.56 (13.69) 0.07 0.55 Note: Mean ( SD ); BASC-2 = Behavior Assessment System for Children, 2nd Edition; CPRS-R:L = Conn ers Parent Rating ScaleRevised, Long Form; DSM-IV = Dia gnostic and Statistical Manual, 4th Edition 64

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Table 3-7. Comparison of attenti on and learning performance between sleep disorder diagnostic groups Domain Variable Sleep Disorder Symptoms (N = 8) Non-symptomatic (N = 11) One-tailed p-value Effect size (Cohens d) Selective Attention Sky Search Time per Target 8.00 (2.33) 7.45 (3.08) 0.34 0.20 Trails A -0.04 (0.77) 0.14 (1.12) 0.35 0.19 Sustained Attention Score! 6.25 (1.98) 9.45 (2.42) 0.01* 1.45 C-CPT-II Hit RT Variability 50.48 (9.26)1 52.97 (9.49) 0.30 0.27 C-CPT-II Omissions 50.64 (6.36)1 51.45 (7.48) 0.41 0.12 C-CPT-II Commissionsa 54.27 (2.45)1 53.71 (8.27) 0.42 0.09 Creature Counting Accuracy 7.88 (3.14) 8.09 (3.33) 0.44 0.06 Attentional Control/Switching Creature Counting Timing 8.40 (1.52)2 10.14 (1.77)3 0.05* 1.05 Trails B 0.10 (0.76)3 -0.21 (1.17) 0.27 0.31 Learning WRAML Verbal Learning 9.12 (1.89) 10.64 (2.46) 0.08 0.69 WRAML Visual Learning 9.00 (2.98) 9.09 (3.83) 0.48 0.03 65 Note: Mean ( SD ); aUnequal variances for this measure; *p<.05; 1n = 6, 2n = 5, 3n = 7

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66Table 3-8. Comparison of attention, lear ning, and sleep disorder symptoms between partial and genera lized seizure groups Domain Variable Partial (N = 7) Generalized (N = 12) p-value Effect size (Cohens d) Selective Attention Sky Search Time per Target 6.00 (2.77) 8.67 (2.27) 0.04* 1.05 Trails A 0.39 (0.62) -0.12 (1.10) 0.28 0.57 Sustained Attention Score! 9.00 (3.00) 7.58 (2.54) 0.29 0.51 C-CPT-II Hit RT Variability 51.53 (10.62)1 52.39 (8.84)2 0.86 0.09 C-CPT-II Omissions 48.42 (4.51)1 52.66 (7.70) 2 0.24 0.67 C-CPT-II Commissions 56.95 (7.95)1 52.25 (5.60)2 0.17 0.68 Creature Counting Accuracy 9.43 (3.10) 7.17 (3.01) 0.17 0.74 Attentional Control/Switching Creature Counting Timing 10.20 (1.79)3 8.86 (1.77)4 0.23 0.75 Trails B 0.20 (1.15)1 -0.23 (0.97) 0.42 0.40 Learning WRAML Verbal Learning 11.00 (2.31) 9.42 (2.19) 0.16 0.70 WRAML Visual Learning 10.00 (4.08) 8.50 (3.00) 0.37 0.42 CSQ-PR Total Score 78.43 (27.23) 99.00 (50.36) 0.34 0.51 Sleep Disorder Symptoms CSQ-PR Breathing 7.86 (4.45) 8.83 (6.65) 0.73 0.17 CSQ-PR Movement Related 9.86 (7.43) 13.33 (8.48) 0.38 0.44 CSQ-PR Daytime Sleepinessa 10.86 (7.65) 21.42 (15.38) 0.06 0.87 Note: Mean ( SD ); aUnequal variances for this measure; *p<.05; 1n = 6, 2n = 11, 3n = 5; 4n = 7

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Table 3-9. Comparison of attention, learning, and sleep disorder sympto ms between seizure status groups Domain Variable Seizure Free (N = 8) Recent Seizures (N = 11) p-value Effect size (Cohens d) Selective Attention Sky Search Time per Targeta 8.25 (1.28) 7.27 (3.44) 0.40 0.38 Trails A 0.00 (1.18) 0.11 (0.83) 0.81 0.11 Sustained Attention Score! 8.25 (3.58) 8.00 (2.10) 0.85 0.09 C-CPT-II Hit RT Variability 52.04 (11.12) 52.13 (7.76)1 0.98 0.01 C-CPT-II Omissions 53.22 (8.14) 49.34 (5.45) 1 0.26 0.56 C-CPT-II Commissions 53.41 (8.67) 54.36 (4.82)1 0.78 0.14 Creature Counting Accuracy 9.62 (3.20) 6.82 (2.68) 0.05 0.95 Attentional Control/Switching Creature Counting Timing 9.71 (1.98)2 9.00 (1.73)3 0.53 0.38 Trails B -0.47 (1.06) 0.22 (0.92)4 0.16 0.70 Learning WRAML Verbal Learning 9.88 (2.03) 10.09 (2.59) 0.85 0.09 WRAML Visual Learning 9.12 (4.29) 9.00 (2.83) 0.95 0.03 CSQ-PR Total Score 77.62 (34.59) 101.45 (48.18) 0.25 0.57 Sleep Disorder Symptoms CSQ-PR Breathing 8.38 (5.40) 8.55 (6.27) 0.95 0.03 CSQ-PR Movement Related 9.38 (5.78) 14.00 (9.18) 0.23 0.60 CSQ-PR Daytime Sleepiness 12.12 (10.44) 21.45 (15.10) 0.15 0.72 67 Note: Mean ( SD ); aUnequal variances for this measure; 1n = 9, 2n = 7, 3n = 5; 4n = 10

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A B 68Figure 3-1. Scatterplots of corre lation between selective attenti on measures and Childrens Slee p Questionnaire-Parent Report ( CSQPR). A) Sky Search Attention score from Test of Everyday Attention for Children and CSQ-PR. B) Trail Making Test Part A and CSQ-PR.

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A B 69Figure 3-2. Scatterplots of corre lation between sustained attention measures a nd Childrens Sleep Questi onnaire-Parent Report ( CSQPR). A) Score! from Test of Everyday Attention for Child ren and CSQ-PR. B) Conners C ontinuous Performance Test, 2nd Edition (C-CPT-II) Hit Reaction Time Variability CSQ-PR. C) C-CPT-II Omissons and CSQ-PR. D) C-CPT-II Comissions and CSQ-PR.

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C D 70Figure 3-2 Continued.

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A B 71Figure 3-3. Scatterplots of corre lation between attentional contro l/switching measures and Child rens Sleep Questionnaire-Paren t Report (CSQ-PR). A) Creature Counting Ti ming score from Test of Everyday A ttention for Children and CSQ-PR. B) Trail Making Test Part B and CSQ-PR.

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72A B Figure 3-4. Scatterplots of corre lation between learning measures and Childrens Sleep Questionnaire-Parent Report (CSQ-PR). A) WRAML Verbal Learning and CSQ-PR. B) WRAML Visual Learning and CSQ-PR.

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CHAPTER 4 DISCUSSION Overview The current study examined the relationship between sleep disturba nce and attention and learning in children with epilepsy. Prior research has suggested that children with epilepsy may be at greater risk for sleep disturbance (B ecker, Fennell, & Carney, 2003), and that sleep disturbance can negatively impact attention and learning performance (e.g., Stores, Wiggs, & Campling, 1998; Kotagal & Pianosi, 2006). This study aimed to determine whether children with epilepsy and co-morbid sleep disturbance demonstrate attention a nd learning impairment relative to children with epilepsy with few or no sleep complaints. Children with epilepsy and co-morbid sleep diso rders have been reported to have increased attention problems and other behavior problems in questionnaire-based studies. However, to date only one other study has b een published looking specifica lly at sleep disturbance and attention performance on a neuropsyc hological measure in children wi th seizures. In addition to expanding the neuropsychological understanding of attention in this population, this study was unique in that it examined multiple domains of attention and assessed performance in the related domain of learning. The study also attempted to replicate earlier research associating increased sleep disturbance with parent report of hyperactivity and inattention. Sleep Disturbance and Attentio n and Learning Performance Consistent with most current models, a multimodal approach to attention assessment was taken in this study. Drawing from the model by Mirsky et al. (1991) selective attention, sustained attention, and attentiona l control/switching were all exam ined. It was predicted that children with epilepsy who had a greater report of sleep problems would demonstrate greater impairment than children with fewer sleep proble ms across the measures of attention. However, 73

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the Higher Frequency Sleep Problem group only performed significantly worse on one measure of auditory sustained attention. Although this finding no longer met significance with Bonferroni correction, it offers at least trend support for the study hypothesis. On the other measures of attention, the ch ildren with epilepsy who had a greater frequency of sleep disturbance performed statistically similar to the children with epilepsy who had few or no complaints of sleep disturbance. Sustained attention deficits have been desc ribed in children with epilepsy and children with sleep disorders in prior studies, including in children with generalized epilepsy and identified abnormal sleep architecture (Maganti et al., 2005). While it was predicted that the epilepsy group with higher frequency sleep pr oblems would demonstr ate greater sustained attention impairment, it was not anticipated that this differe nce would be found only for the auditory sustained attention task (TEA-Ch Score!) and not the visu al sustained attention task (CCPT-II). Given the magnitude of the differen ce between the two groups on C-CPT-II Omissions (approximately 0.5 standard deviation), it is possibl e that the failure to find significant results was due to a lack of power resulting from the sma ll sample size. In a larger sample both auditory and visual sustained attention differences would likely be found. It should also be acknowledged that in the present study the scores for both groups on the visual sustained attention task were largely within normal limits for their age, partic ularly on Omissions, the more specific measure of inattention. In contrast, othe r studies of sustained attention in children with epilepsy have reported clinically impaired performance (e .g., Hernandez et al., 2003; Williams, Griebel, & Dykman, 1998; Mitchell et al., 1992). This suggests that the sample of children with epilepsy in the current study might be atypical in their overall performance (i.e., have relatively better attention skills). Alternatively, it could reflect differences in the continuous performance task 74

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parameters and outcome variables, which have be en cited as a source of inconsistent findings between studies of attention (Fletcher, 1998). Given the dearth of research in this particular area, further study is necessary to better characterize sustained a ttention performance in children with epilepsy and co-morbid sleep disturbance. As previously noted, neither selective attention performan ce nor attentional control/switching performance differed significantly between the two sleep frequency groups. Although previous research has sugg ested that children with sleep disorders, particularly sleep disordered breathing, tend to demonstrate grea ter impairment on selec tive attention tasks, (Owens et al., 2000; Beebe et al., 2004), this did not bear out in this sa mple of children with seizures. In fact, both sleep problem groups pe rformed below average relative to the normative sample on the TEA-Ch Sky Search task (but with in normal limits on Trailmaking Part A). While selective attention deficits have been described in some studies of children with epilepsy, the literature as a whole has been inconsistent, ma king conclusive interpretations difficult. One possible interpretation is that ep ilepsy alone is a risk factor for poorer performance on the more complex selective attention task and sleep di sruption in the higher frequency group did not additionally contribute to the chil drens performance. Additional research, particularly involving control groups such as children only diagnosed with sleep disorders is needed to further clarify this relationship. While not statistically signifi cant, the Higher Frequency Sl eep Problems group did tend to perform worse on the TEA-Ch measure of atten tional control/switching, Creature Counting. As indicated by the effect size analysis, the childr en with more frequent sleep problems performed over a half a standard deviation below the children with few or no sleep complaints as assessed by the Creature Counting timing score, a comprehe nsive score that takes into account accuracy, 75

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time, and the number of times the child had to sw itch between sets. A la rger sample size would likely provide sufficient power to detect the di fferences between these groups. Notably, across both groups, several cases had to be excluded from the analysis because low accuracy precluded the calculation of the timing score (four from the Higher Frequency group, and three from the Lower Frequency group). Although the excluded cases could not be statisti cally compared given the small samples, review of poten tial contributing features such as seizure type, seizure status, and medication did not suggest any clear patterns Qualitatively, these children were the most impaired on the task but could not be included in statistical analyses due to test validity limitations. The exclusion of these lower perf ormers limited the comprehensive assessment and comparison of attentional cont rol between the two groups. In contrast, children from both groups performed within normal limits on the le ss cognitively demanding Trailmaking Part B switching task. The contrast in the performance patterns of the two sleep problem groups on Creature Counting and Trailmaking Part B might reflect the involveme nt of abilities better described as executive function and working memory than attention. Trailmaking Part B is a fairly straightforward task and easily instructed task, involving switching back and forth between two sequential sets (numbers and alphabet) in a repetitive manner (1-A, 2-B, etc.). Creature Counting, however, involves what are arguab ly more complex cognitive skills including inhibiting the pre-potent res ponse in order to switch coun ting direction mid-sequence and maintaining the last number spoke n while identifying the new dir ection for counting. Manly et al. (2001) recognized this complexity in descri bing the Creature Countin g task as reflecting a broad executive or attentiona l control factor in their in itial description of the TEA-Ch normative sample (p. 1074). Although beyond the scope of the present study, executive 76

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functioning is an aspect of cogn ition that may merit further resear ch in children with epilepsy and co-morbid sleep disorders. Both the Behavior Assessment System for Children, 2nd Edition (BASC-2) and the Conners Parent Rating Scales-Revised: Long Edit ion (CPRS) were employed to look at parent report of varying aspects of attentional perf ormance. For example, items on the BASC-2 Attention scale are more narrowly targeted to atte ntion problems, such as is easily distracted, has a short attention span, and listens careful ly. In contrast, the DSM-IV Inattention scale looks more broadly at the signs and symptoms of ADHD, Inattentive type, including poor sustained attention as well as including items such as avoiding mental effort, making careless errors, being poorly organized, and losing things. Contrary to th e study hypothesis, parent report of attention problems did not di ffer significantly between the two groups on either measure. As was found for the neuropsychological measures of visual sustained attention and attentional control, review of analyses s uggested that low power may have contributed to the lack of findings. In a larger sample, we predict that a significant difference would be found supporting our hypothesis that children with epilepsy with increased sl eep problems would demonstrate higher levels of parent reported attention problems. C linically significant le vels of hyperactivity were also not endorsed on either measure, and no differences in hyperac tive behavior were found between the higher frequency and lowe r frequency sleep problem groups. Notably, not only were scores in the present study below clinical levels, scores on the CPRS-R:L fell well below those reported in prio r research examining children with epilepsy and sleep disruption. Specifically, B ecker, Fennell, and Carney (2004) reported that their sample of children with epilepsy and sleep complaints had a mean score on the Total DSM-IV scale of the CPRS-R:L nearly one full standard deviation a bove what was reported in the present study. 77

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Additionally, over 70 percent of th e children in the Becker sample had individual scores within the clinical range (T-score > 65). In contrast, only 5 of 19 (26%) ch ildren in the total sample of this study had DSM-IV Total scores exceeding this clinical cutoff score. This finding raises interesting questions about the na ture of the current study sample. Specifically the children in the Becker, Fennell and Carney (200 4) study differed from those in the present study in that they were recruited on the basis of being referred for sleep studies, and 80 percent ultimately demonstrated signs of disorder ed sleep during polysomnography. In contrast, the current study recruited children via their regu larly scheduled neurology appointments, without regard to sleep complaints or concerns. The results of the Beck er, Fennell, and Carney ( 2004) study suggest that clinical attention proble ms may be most strongly associated with more significant sleep disorder symptoms. Particularly given the relatively small sample size it is possible the current study under-sampled children with epilepsy with more severe sleep distur bance and more profound attention problems. Attempts were made in post-hoc analyses to more stringently define the two sleep problems groups to ensure that the higher fr equency sleep problem group captured children with significant sleep concerns (more similar to those that would have presented for sleep studies in the Becker, Fennell, and Carney study). Using questionnaire items cons istent with clinical screening for sleep disordered breathing and move ment-related sleep disorders, children were reassigned to the Sleep Disorder Symptoms group and the Non-symptomatic group. Group assignments remained largely the same, with only two children being reassigned to the Nonsymptomatic group. Similarly, the pattern of findings remained the same in the new groups, with the Sleep Disorder Symptoms group demonstrat ing statistically significant impairment for sustained attention and a trend to ward greater impairment for a ttentional control. Finally, a 78

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greater percentage of the children with in the Sleep Disorder Symptoms group also presented with symptoms of clinical at tention problems than in the No n-symptomatic group (50% versus 9%). Although this difference did not meet si gnificance, the finding is promising in that it reflects the pattern seen in earlier research. In addition to attention, the present study aimed to examine learning performance in children with epilepsy and co-morbid sleep dist urbance. Results did not support the hypothesis that children with epilepsy and sleep disturbance would perform worse on measures of verbal and visual learning. Further, the mean scores of both groups fell within the average range for both verbal learning and visual learning, suggesting that there were no clinically relevant performance problems. In light of the differenc es found for auditory sustained attention, it was somewhat surprising that similar findings were not found for verbal learning. Learning was chosen as a variable of interest because of its frequently described c onnection to attention, and sustained attention in particular Specifically, in order to succe ssfully encode information on a supraspan learning task like those used in this study, the child must be able to sustain his or her attention to the task, both within a trial and over the successive presentation trials. While certainly other cognitive processes are involved in learning, it was suspected that if children with sleep problems exhibited poor sustained atten tion, they would also exhibit poor learning. The small sample size precluded any statistical ex amination of the direct relationship between attention performance and learning performance; however, it is possible to speculate why the hypothesis was not supported. One hypothesis is that the learning tasksespecially the visual learning testwere experienced as more engaging or fun by the children, and thus they allocated more attention to the task at hand. In fact, during testing, many of the children commented that the visual memory task reminded them of a ga me, and several children demonstrated eagerness 79

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to improve their performance over trials of both tasks (e.g., asking did I get more?, or stating Im getting better). In addition, the verbal learning task involves calculating a total score across four trials. While a child might miss encoding informing during a transient moment of inattention on a single trial, he or she has the opportunity to recuperate the information on a following trial. Subtle difficulties in encoding acro ss the trials might be missed in a measure that uses only a summation score like that of th e WRAM Verbal and Visual Learning tests. Additional Results Subsequent to addressing the primary analys es, additional analyses were undertaken to examine potential study confounds. Learning performance and sleep problems were found not to differ based upon seizure type (generalized versus part ial) or seizure status (seizure-free or not in past 56 days). Among the attention variables, on ly selective attention differed between the two seizure types. While more direct regressional analyses were not feasible gi ven the sample size, these results suggest that the modest findings in the study are not likely at tributable to epilepsy characteristics. Additionally, the children with mo re frequent sleep problems tended to perform more slowly on measures of processing speed than the children with less frequent sleep problems, although the difference was not statistica lly significant. It should be acknowledged that while Symbol Search and Coding were chos en as measures of processing speed, both tasks also involve attentional abilities, particularly visual scanni ng and selective attention. However, the processing speed differences did not translat e to differences on the primary measures of selective attention, and would not be expected to influence the differences on the motor-free, auditory sustained attention task. Limitations This study has several limitations that are important to consider with regards to the present outcomes and planning for future endeavors. First and foremost, th e current study is best 80

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conceptualized as an exploratory pilot study due to the small sample of participants. Multiple barriers to recruitment and study completion negatively impacted th e final sample size, which in turn limited the options for st atistical analysis of the original study aims and hindered interpretation of findings. Ideally, a larger sample would have enabled planned regressional analyses to more directly examine the relatio nship between sleep prob lems and attention and learning performance. Instead, a suboptimal approach employing a median-split (with subsequent symptom-based classi fication in follow-up analyses), was necessitated in order to create groups for comparison. Although there wa s sufficient power to find group differences on auditory sustained attention, the small sample size limited the ability to make conclusions regarding other aspects of perf ormance including visual sustai ned attention and attentional control/switching. Importantly, effect size calcu lations indicated that with a larger sample, significant difference would have been found for one measure of a ttentional control as well as for parent report of at tention problems. The results of the study may have been affect ed by outliers, which can be particularly influential when using central tendency-based sta tistics with small samples. As noted in the review of correlations, auditory sustained attention (Score!) and the sleep measure demonstrated a fairly clear, albeit not significant, negative linear relationship (See Figure 3-1). However, other correlations (e.g., Creature Counting) appeared to be skewed by su spected outliers that distorted the results by pulling the mean in their direction. Reviewing scat terplots also suggested that there were atypical, non-linear relationships between the sleep questionnaire and some variables such as Sky Search that would not have been detected by t-tests. It is possible that such atypical relationships might have become more normalized in a larger sample. 81

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The present study was also hindered by the la ck of a well-defined control group. Although the implementation of a two-group design was a post-hoc decision, it contributed to the diminished statistical power and the challenge of interpretation of findings Unfortunately, it is difficult to establish a control group consisti ng of children who have epilepsy but no sleep disorder without using objective measures of slee p quality. Similarly, the lack of objective sleep measures and the reliance on parent report of sleep problems on a newly developed measure is another study limitation. As is true of other such measures, the parents report on the CSQ-PR may be confounded by the subjectivity of the informant. Without established normative data or diagnostic guidelines, it was necessary to a dopt a more qualitative approach to using the measure. The gold standard for sleep disorder diagnosis is the polysomnogram (PSG), or overnight sleep study, which allows a more precise examination of sleep disorder symptoms. Inclusion of PSGs in the present study was not possible due to the significant cost involved and the low rates of children eligible for the study w ho were being evaluated for sleep problems as part of their standard care. In fact, while access to the data from existing PSGs was allowed under the study IRB, none of the children enroll ed in the study underwent an overnight sleep study during the project timeframe. In addition to their diagnostic utility, PSGs would also have been valuable because sleep studies can capture s ources of disruption that would not be detected by parents, including microarousal s that can occur in children th roughout the night. Frequent arousals during sleep can cause sleep fragmentation, or disruption of the sleep cycle, which has been hypothesized to underlie neurobehaviora l problems in children with sleep disorders (OBrien and Gozal, 2004). While not studied extensively, sleep fragmentation has been associated with increased daytime behavior problems in children with epilepsy (Bongiolatti, Fennell, & Carney, 2006). It is pos sible that some children assigned to the Lower Frequency 82

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sleep problems group currently expe rience these more subtly pres enting, yet impactful, types of sleep disturbance or have a sleep disorder that has not been recognized by the parent. Unfortunately, without the aid of PSG this could not be determined. Finally, epilepsy is a diverse diagnosis, a nd children with epilepsy differ in terms of seizure presentation, severity, course of illn ess, and medication re gimen. For the primary analyses, the current study did not separate children with epileps y on the basis of any disorderrelated factors such as partial versus genera lized seizures. While we tried to account for potential differences between the two groups, incl uding the number of AEDs and seizure status, it would be advantageous to limit the potential contribution of these additional variables by focusing on more narrow subgroups and thereby increasing interpreta bility of findings. Unfortunately, recruiting this type of selectiv e sample requires a grea ter timeframe than was available for this study. Future Directions The relationship between sleep disorders a nd neurobehavioral func tioning in children with epilepsy has only recently become a topic of research interest. Accordingly, there are multiple avenues for future research. Although the findings in the current study were limited, they provide support for conti nued investigation. Combined with the existi ng literature associating parent report of inat tention and sleep problems in children with epilepsy, as well as the growing research associating sleep disorders such as sleep apnea and periodic leg movement disorder to daytime attention problems, it seems likely that future studies will find further support for a connection between sleep problems and attention impairment in children with seizures. A large sample study examining attent ion performance in child ren with epilepsy and co-morbid sleep disorders is needed in order to further elucidate this relationship. A large sample should allow for comparisons across seizure types, as well as more appropriate controls 83

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for seizure severity and medications. Additi onally, because so little is known about the relationship between sleep and cognition in ch ildren with epilepsy and other neurological disorders, it would be adva ntageous to employ more comp rehensive neuropsychological assessments to create a profile of performance across domains. It will be vital to employ polysomnography (PSG) in future studies to objectively characterize sleep disturbance in children with epilepsy. Use of PSGs would allow for more diagnosis and classification of children into sleep disorder groups for comparison, and examination of sleep architecture. Ideally, fu ture studies should us e PSG and parent-report measures (as well as self-report measures when age appropriate) in c onjunction to thoroughly examine sleep in children with epilepsy. Use of standardized sleep measures may facilitate interpretation of findings in future studies, and when used in collaboration with PSG will begin to inform the development of scr eening measures that can be used in clinical settings. Research to develop an effective screener for use in pedi atric medical and psycholog ical settings would aid health care providers in identifying children who ar e most at risk for a sleep disorder and comorbid neurobehavioral problems. Finally, future studies should examine the eff ects of treating sleep disorders in children with epilepsy. Unlike adults, children with obs tructive sleep apnea (O SA) most frequently present with enlarged tonsils and adenoids, and first line treatment tends to be adenotonsillectomy. This treatment protocol provid es an opportune structure for testing children with epilepsy before and after intervention that would help to clarify the specific relationship between sleep and seizures. Recent studies in adults with epilepsy and OSA have focused primarily on the seizure control benefits associated with tr eating OSA (e.g., Malow et al., 2008). However, based upon the pediatric sleep apnea lite rature, it appears likely that treating sleep 84

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apnea in children with epileps y would not only improve seizure control (Koh et al., 2000), but also improve daytime attention and behavior. 85

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APPENDIX CHILDRENS SLEEP QUESTIONNAIREPARENT REPORT Childs Name: _______________________________________ Todays Date: ___________________ Childs Date of Birth: ______________________ Childs Age: ___________ Childs Grade: ________ Childs Ethnicity: Caucasian African American Hispanic Asian American/Pacific Islander Native American Other (please specify _______________________________) Name of Person Completing Form: _____________________ Relationship to Child: ________________ Directions for Caregivers: The following statements are about your childs sleep habits and possible difficulties with sleep. Think about a typical week during the past month in your childs life when answering these questions. If there were no typical weeks in the past month for your child or the family (e.g., child was sick, visitors in the home, family vacation, etc.), please base your responses on the most recent typical week Answer Very Often if something occurs 5-7 days or nights during a week Answer Often if something occurs 2-4 days or nights during a week Answer Sometimes if something occurs 1 day or night during a week Answer Rarely if something occurs less than 1 day or night a week but at least once in a month Answer Never if something never occurs during a typical week or month (i.e., does not happen) Also, at the end of each section, we will ask you about how sure you we re about your answers in that section. Please answer every question. If you are not sure of an answer, provide your best guess. General Sleep Section Your child.. Very Often (5-7) Often (2-4) Sometimes (1) Rarely (< 1) Never (0) 1. Sleeps too little 2. Sleeps the right amount 3. Sleeps too much 4. Sleeps about the same amount each day (combining nighttime and naps) 5. Has a regular bedtime routine 6. Goes to bed at the same time at night 7. Falls asleep within 20 minutes after going to bed? 8. Has difficulty falling asleep 9. On this page, how sure are you about your answers? very sure somewhat sure very unsure (guessing) 86

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Answer Very Often if something occurs 5-7 days or nights during a week Answer Often if something occurs 2-4 days or nights during a week Answer Sometimes if something occurs 1 day or night during a week Answer Rarely if something occurs less than 1 day or night a week but at least once in a month Answer Never if something never occurs during a typical week or month (i.e., does not happen) Breathing Section While sleeping your child: Very Often (5-7) Often (2-4) Sometimes (1) Rarely (< 1) Never (0) 10. Snores 11. Snores loudly 12. Sleeps with the mouth open 13. Has heavy or loud breathing 14. Snorts and/or gasps during sleep 15. Has trouble breathing, or struggles to breathe 16. Seems to stop breathing during sleep 17. Needs shaking to get him/her to breathe or wake up and breathe In the morning, your child: 18. Wakes up with a snorting sound 19. Has a dry mouth upon waking up in the morning 20. On this page, how sure are you about your answers? very sure somewhat sure very unsure (guessing) 87

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Answer Very Often if something occurs 5-7 days or nights during a week Answer Often if something occurs 2-4 days or nights during a week Answer Sometimes if something occurs 1 day or night during a week Answer Rarely if something occurs less than 1 day or night a week but at least once in a month Answer Never if something never occurs during a typical week or month (i.e., does not happen) Movement Related Section At night time your child.. Very Often (5-7) Often (2-4) Sometimes (1) Rarely (< 1) Never (0) 21. Is restless and moves a lot during sleep 22. Has twitches or jerks while sleeping 23. Has brief kicks of one leg or both legs while sleeping 24. Becomes sweaty at night or pajamas become wet with perspiration 25. Has trouble sleeping because he/she feels too hot 26. Has trouble sleeping because he/she feels too cold 27. Has leg pains that are worst in bed 28. Describes funny feelings in his/her legs when lying down or sitting (e.g., creeping, crawling, tingling, or tickling sensations) 29. If your child sleeps with you, he/she kicks you hard enough that it disrupts your sleep or wakes you (If child does not sleep with you, check here:_______) During the day, your child. 30. Complains of leg pains that are relieved by moving 31. Becomes weak in the legs, or anywhere else, after laughing or being surprised by something 32. On this page, how sure are you about your answers? very sure somewhat sure very unsure (guessing) 88

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Answer Very Often if something occurs 5-7 days or nights during a week Answer Often if something occurs 2-4 days or nights during a week Answer Sometimes if something occurs 1 day or night during a week Answer Rarely if something occurs less than 1 day or night a week but at least once in a month Answer Never if something never occurs during a typical week or month (i.e., does not happen) Night Time Waking Section At night time your child.. Very Often (5-7) Often (2-4) Sometimes (1) Rarely (< 1) Never (0) 33. Awakes once during the night 34. Awakes more than once during the night 35. Awakens during the first 2 hours after falling asleep 36. Wakes up saying he/she is scared 37. Wakes up complaining of an upset stomach 38. Awakens during night screaming, sweating and inconsolable 39. Awakens alarmed by a frightening dream 40. Awakens, but appears confused or disorientated (e.g., does not respond to you, talks about things that dont make sense) 41. Has trouble falling back asleep if wakes up during the night 42. Moves to someone elses bed during the night (parent, brother, sister, etc.) 43. Reports feeling unable to move for a short period of time, although he/she is awake and can look around 44. Describes dreaming while still awake at night (i.e., sees images or hears sounds) 45. Talks during sleep 46. Grinds teeth during sleep (your dentist may have told you this) 47. Sleepwalks during the night 48. Gets out of bed night (for any reason) 49. Gets out of bed to use the bathroom 50. Wets the bed at night 51. On this page, how sure are you about your answers? very sure somewhat sure very unsure (guessing) 89

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Answer Very Often if something occurs 5-7 days or nights during a week Answer Often if something occurs 2-4 days or nights during a week Answer Sometimes if something occurs 1 day or night during a week Answer Rarely if something occurs less than 1 day or night a week but at least once in a month Answer Never if something never occurs during a typical week or month (i.e., does not happen) Morning Time Waking Section In the morning, your child. Very Often (5-7) Often (2-4) Sometimes (1) Rarely (< 1) Never (0) 52. Wakes up very early in the morning and has difficulty going back to sleep 53. Has difficulty getting out of bed in the morning 54. Falls back to sleep after being awakened in the morning 55. Wakes up by him/herself in the morning 56. Adults or siblings wake up child 57. Wakes up in the morning tangled up in the sheets 58. Wakes up with headaches in the morning 59. Wakes up in a negative mood (e.g., cranky, irritable) 60. Complains of still being tired 61. Takes a long time to become alert in the morning 62. Takes a long time to get ready in the morning 63. On this page, how sure are you about your answers? very sure somewhat sure very unsure (guessing) 90

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Answer Very Often if something occurs 5-7 days or nights during a week Answer Often if something occurs 2-4 days or nights during a week Answer Sometimes if something occurs 1 day or night during a week Answer Rarely if something occurs less than 1 day or night a week but at least once in a month Answer Never if something never occurs during a typical week or month (i.e., does not happen) Daytime Sleepiness Section During the daytime, your child.. Very Often (5-7) Often (2-4) Sometimes (1) Rarely (< 1) Never (0) 64. Has a problem with sleepiness during the day 65. Seems tired 66. Teachers or other supervisor comment that he/she appears sleepy during the day 67. Feels an irresistible urge to take a nap during the day 68. When awake, disrupts family activities because of sleepiness 69. Yawns a lot during the day 70. Takes a nap during the day 71. Falls asleep if sent to room for misbehaving 72. Is very sleepy while watching TV 73. Falls asleep while watching TV 74. Is very sleepy while riding in a car 75. Falls asleep while riding in a car 76. On this page, how sure are you about your answers? very sure somewhat sure very unsure (guessing) 91

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Answer Very Often if something occurs 5-7 days or nights during a week Answer Often if something occurs 2-4 days or nights during a week Answer Sometimes if something occurs 1 day or night during a week Answer Rarely if something occurs less than 1 day or night a week but at least once in a month Answer Never if something never occurs during a typical week or month (i.e., does not happen) Sleep Habits Section Your child.. Very Often (5-7) Often (2-4) Sometimes (1) Rarely (< 1) Never (0) 77. Falls asleep alone in own bed 78. Falls asleep in parents or siblings bed 79. Needs parent in the room to fall sleep 80. Is afraid of sleeping alone 81. Is afraid of sleeping in the dark 82. Sleeps with a light or night-light on 83. Has trouble sleeping away from home (visiting relatives or friends, on vacation) 84. Falls asleep with rocking, rhythmic or head banging movements 85. Struggles at bedtime (e.g., cries, refuses to stay in bed, etc.) 86. Has temper tantrums at bedtime 87. Eats, drinks, or exercises within 2 hours of going to bed 88. Watches TV or plays video or computer games within 1 hour of going to bed 89. Is punished by going to bed or being placed in his/her bed 90. On this page, how sure are you about your answers? very sure somewhat sure very unsure (guessing) 92

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CHILDS GENERAL MEDICAL HISTORY Childs Height: _____feet _____inches Childs Weight: _______pounds Does your child still have his/her tonsils and/or adenoids? Yes No Has your child ever sustained a head injury where they were knocked out? Yes No If yes, describe what happened & how long unconscious: Does your child have any problems with their hearing or eyesight? Yes No If yes, describe: Does your child have any medical or neurological conditions, such as seizures, sickle cell anemia, diabetes, cancer, or autism? Yes No If yes, describe: Has your child been diagnosed with or treated for prob lems with attention or hyperactivity, such as ADHD or ADD? Yes No If yes please answer the following questions: At what age was he/she diagnosed? _____________ Are they diagnosed as Primarily Inattentive Subtype, Primarily Hyperactive/Impulsive Subtype, Combined Subtype, or do not know subtype? Does your child currently take medication(s) for ADHD? Yes No Do you think your childs medication helps, hurts, or not have an effect on his/her sleep? Has your child ever been enrolled in classes where they get special assistance? Yes No If yes, describe: Has your child been diagnosed with or treated for a learning disability? Yes No If yes, describe: Have you ever been told that your child had a low IQ or was mentally handicapped? Yes No If yes, describe: Is your child currently taking any medications? Yes No If yes, please list names and what t hey are treating (e.g., asth ma, sleep problems, ADHD): ___________________________________ _______________________________________ ___________________________________ _______________________________________ ___________________________________ ______________________________________ 93

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CHILDS SLEEP RELATED MEDICAL HISTORY Has your child ever been diagnosed or treated for a sleep disorder? Yes No If yes, describe: Does your child have respiratory or breathing-related allergies? Yes No If yes, do these allergies a ffect your childs ability to breathe through the nose? Yes No For the most recent typical week how many nights did your child have a congested or stuffed nose? 0 1 2 3 4 5 6 7 For the most recent typical week did your child tend to breathe through his/her mouth? Yes No CHILDS SLEEP HISTORY Weekday Sleep Schedule Your childs usual bedtime on weekday nights is between _____ : _____ PM and _____ : _____ PM Your child usually falls asleep on weekday nights between _____ : _____ PM and _____ : _____ PM/AM Your childs usual waketime on weekday mornings is between _____ : _____ AM and _____ : _____ AM Weekend/Vacation Sleep Schedule Your childs usual bedtime on weekend/vacation nights is between _____ : ____ PM and ____ : ____ PM Your child usually falls asleep on weekend/vacation nights between ___ : ___ PM and ___ :_____ PM/AM Your childs usual waketime on weekend/vacation mornings is between ____ : ___ AM and ___ : ____ AM Naps How many naps does your child take in a typical day ? 0 1 2 3 4 other:______ If your child naps, how long is a typical nap? _______hour(s) ________minutes General Sleep Child is usually put to bed by: Mother Father Both Parents Self Others Write in the amount of time the child spends in his/her bedroom before going to sleep: ___ hours ____ minutes What time does your childs school typi cally start in the morning? _____ : _____ AM How many days each week is your child late to school because of difficulty getting going in the morning? 0 1 2 3 4 5 6 7 Write the number of times that you think your child wakes up each night: ________ If your child wakes up at night, on average, how long does it take him/her to fall back to sleep? _____ hours ______ minutes On a typical day, how long does it usually take for your child to become fully alert and oriented after waking up in the morning: _______hour(s) _________minutes 94

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95 On a typical day, how many cups or cans (12 ounces ) of caffeinated beverages (e.g., soda, tea, coffee) does your child drink? __________ If it is easier to estimate based on ounces (because some beverages are sold in 16, 20, and 32 ounce sizes), please write in the number of ounces here: _____________ FAMILYS SLEEP HISTORY Does anyone in the family have a sleep disorder? Yes No If yes, mark the disorder(s): Insomnia Mother Father Brother/sister Grandparent Snoring Mother Father Brother/sister Grandparent Sleep apnea Mother Father Brother/sister Grandparent Restless legs syndrome Mother Father Brother/sister Grandparent Periodic limb movement disorder Mother Father Brother/sister Grandparent Sleepwalking/sleep terrors Mother Father Brother/sister Grandparent Sleep talking Mother Father Brother/sister Grandparent Narcolepsy Mother Father Brother/sister Grandparent Other: Mother Father Brother/sister Grandparent OTHER FAMILY INFORMATION Do any of the family members smoke in the house? Yes No Do any of the family members smoke in the car? Yes No Highest level of education completed by Female caregiver : (check one) Grade School: 1 2 3 4 5 Junior High: 6 7 8 High School: 9 10 11 12 College: 13 14 15 16 Graduate School: 17 18 19 20 21 22 Other: ____________ Highest level of education completed by Male caregiver : (check one) Grade School: 1 2 3 4 5 Junior High: 6 7 8 High School: 9 10 11 12 College: 13 14 15 16 Graduate School: 17 18 19 20 21 22 Other: ____________ Some questions have been adapted from the PSQ (Chervin et al., 2000), CSHQ (Owens et al., 2000), ESS (Johns MW, 1991), OSAS (Franco et al., 2000), PDSS (Drake et al., 2003), PRSP (Marcott e et al., 1998), OSA-QOL (Cohen et al., 1998), PSQI (Buysse et al., 1989), & PSS (Owens, 2001).

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LIST OF REFERENCES Ali, N. J., Pitson, D., & Stradling, J. R. ( 1996). Sleep disordered breathing: Effects of adenotonsillectomy on behaviour an d psychological functioning. European Journal of Pediatrics, 155(1), 56-62. Austin, J. K. (1988). Childhood epilepsy: Child adaptation and family resources. Journal of Child and Adolescent Psychiatric and Mental Health Nursing, 1, 18-24. Austin, J. K., Dunn, D. W., Caffrey, H. M., Perkin s, S. M., Harezlak, J., & Rose, D. F. (2002). Recurrent seizures and behavior problems in children with first recognized seizures: A prospective study. Epil epsia, 43(12), 1564-1573. Austin, J. K., Risinger, M. W., & Beckett, L. A. (1992). Correlates of behavior problems in children with epilepsy. Epilepsia, 33 1115-1122. Beebe, D. W., Wells, C. T., Jeffries, J., Chini, B., Kalra, M., & Amin, R. (2004). Neuropsychological effects of pedi atric obstructive sleep apnea. Journal of the International Neuropsyc hological Society, 10, 962-975. Besag, F. M. C. (2004). Behavioral asp ects of pediatric epilepsy syndromes. Epilepsy & Behavior, 5(Suppl. 1) S3-S13. Brum Batista, B.H. & Lahorgue Nunes, M. L. (2007). Evaluation of sleep habits in children with epilepsy. Epilepsy & Behavior, 11, 60-64. Bazil C.W. (2003). Epilepsy and sleep disturbance. Epilepsy & Behavior, 4 S39-S45. Becker, D. A., Fennell, E. B., & Carney, P. R. (2003). Sleep disturba nce in children with epilepsy. Epilepsy & Behavior, 4, 651-658. Becker, D. A., Fennell, E. B., & Carney, P. R. (2004). Daytime behavior and sleep disturbance in childhood epilepsy. Epilepsy & Behavior, 5, 708-715. Beery, K. (1997). The Developmental Test of Visual-Motor Integration: Administration Scoring and Teaching Manual (4th ed. Revised). Parsippany, NJ: Modern Curriculum Press. Beran, R. G., Plunkett, M. J., & Holland, G. J. (1999). Interface of epilepsy and sleep disorders. Seizure, 8, 97-102. Besag, F. M. (2004). Behavioral aspects of pediatric epilepsy syndromes. Epilepsy & Behavior, 5, S3-13. Bongiolatti, S.R., Fennell, E.B., & Carney, P.R. ( 2005). Sleepiness as a fact or in varying degrees of behavior problems in children with epile psy. Poster presented at the joint annual meeting of the American Epilepsy Societ y and American Clinical Neurophysiology Society, Washington, D.C. 96

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Bongiolatti, S.R., Fennell, E.B., & Carney, P.R. (2006). The role of sleep fragmentation in disruptive behaviors in children with epileps y and comorbid sleep breathing disorders. Poster presented at the annual meeting of the International Neuropsychology Society, Boston, MA. Borgatti, R., Piccinelli, P., Montirosso, R., Donati, G., Rampani, A., Molteni, L., et al. (2003). Study of attentional processes in children with idiopathic epilepsy by Conners Continuous Performance Test. Journal of Child Neurology, 19(7), 509-515. Blunden, S., Lushington, K., Kennedy, D., Marti n, J., & Dawson, D. (2000). Behavior and neurocognitive performance in children aged 5-10 yrs who snore compared to controls. Journal of Clinical & Ex perimental Neuropsychology, 22(5), 554-568. Brown, T.E. & Modestino, E.J. (2000). Atten tion-deficit disorders with sleep/arousal disturbances. In T.E. Brown (Ed.), Attention-deficit disorders and comorbidities in children, adolescents, and adults (pp. 341-362). Washington, DC : American Psychiatric Publishing. Camfield, P. & Camfield, C. (2003). Childhood epileps y: What is the evidence for what we think and what we do? Journal of Child Neurology, 18(4), 272-287. Carney, P. R., Becker, D. & Bongiolatti, S. (2005) Ontogeny of sleep. In P. R. Carney, R. B. Berry, & J. D. Geyer (Eds.), Clinical Sleep Disorders (pp. 95-98). Philadelphia: Lippincott, Willams, & Wilkins. Chervin, R. D., Archbold, K. H., Dillon, J.E., Pana hi, P., Pituch, K.J., Da hl, R.E., et al. (2002). Inattention, hyperactivity, and sympto ms of sleep-disordered breathing. Pediatrics, 109(3), 449-456. Chervin, R. D., Hedger, K. M., Dillon, J. E., & Pituch, K. J. (2000). Pediatric Sleep Questionnaire (PSQ): Validity and reliability of scales for sleep-disordered breathing, snoring, sleepiness, and behavioral problems. Sleep Medicine, 1, 21. Chervin, R. D., Ruzicka, D. L., Giordani, B. J., Weatherly, R. A., Dillon, J. E., Hodges, E. K. et al. (2006). Sleep-disordered breathing, behavior, and cognition in children before and after adenotonsillectomy. Pediatrics, 117 769-778. Chihorek, A.M., Abou-Khalil, B., & Malow, B.A. (2007). Obstructive sleep apnea is associated with seizure occurrence in older adults with epilepsy. Neurology, 69, 1823. Conners, C. K. (2000). Conners' Continuous Performance Test II North Tonawanda, NY: Mental Health Systems. Corkum, P., Tannock, H., & Moldofsky, R. (1998). Sl eep disturbances in children with Attention Deficit/Hyperactivity Disorder. Journal of the American Academy of Child and Adolescent Psychiatry, 37, 637-646. 97

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Cortesi, F., Giannoti, F., & Ottaviano, S. ( 1999). Sleep problems and daytime behavior in childhood idiopathic epilepsy. Epilepsia, 40(11), 1557-1565. Croona, C., Kihlgren, M., Lundberg, S. Eeg-Olofsson, O., & Eeg-Olofson, K.E. (1999). Neuropsychological findings in childre n with benign childhood epilepsy with centrotemporal spikes. Developmental Medicine and Child Neurology, 41 813-818. D'Alessandro, P., Piccirilli, M., Tiacci C., Ibba A., Maiotti M., Sciarma T., et al. (1990). Neuropsychological features of be nign partial epilepsy in children. Italian Journal of Neurological Sciences, 11 265. Devinsky, O., Ehrenberg, B., Barthlen, G. M., Abramson, H. S., & Luciano, D. (1994). Epilepsy and sleep apnea syndrome. Neurology, 44 2060-2064. Epilepsy Foundation of America (20 08). Epilepsy and seizure sta tistics. Retrieved September 1, 2008, from http://www.epilepsyfoundation.o rg/answerplace/s tatistics.cfm. Epstein, J. N., Erkanli, A., Conners, C. K., Kl aric, J., Costello, J. E., & Angold, A. (2003). Relations between continuous performance test performance measures and ADHD behaviors. Journal of Abnormal Child Psychology, 31 543-554. Ferber, R. (1996). Childhood sleep disorders. Neurologic Clinics, 14 493-511. Frost, M., Malow, B., & Aldrich, M (1996). A su rvey of sleep disorders in epilepsy patients [Abstract]. Neurology 46, A120. Goldstein, N.A., Post, J.C., Rosenfeld, R.M., & Ca mpbell, T.F. (2000). Impact of tonsillectomy and adenoidectomy on child behavior. Archives of OtolaryngologyHead & Neck Surgery, 126(4), 494-498. Hauser, W.A. (2001). Epidemiology of epilepsy in children. In J. M. Pellock, W. E. Dodson & B. F. D. Bourgeois (Eds.), Pediatric epilepsy: Diagnosis and therapy (pp. 81-96). New York: Demos. Heaton, S. C., Reader, S. K., Preston, A. S., Fenne ll, E. B., Puyana, O. E., Gill, N., et al. (2001). The Test of Everyday Attention for Childre n (TEA-Ch): Patterns of performance in children with ADHD and clinical controls. Child Neuropsychology, 7(4), 251-264. Henkin, Y., Sadeh, M., Kivity, S., Shabtai, E., Kishon-Rabin, L., & Gadoth, N. (2005). Cognitive function in idiopathic generalized epilepsy of childhood. Developmental Medicine & Child Neurology, 47, 126. Hernandez, M., Sauerwein, H. C., Jambaque, I., de Guise, E., Lussier, F., Lortie, A., et al. (2003). Attention, memory, and behavioral adju stment in children with frontal lobe epilepsy. Epilepsy and Behavior, 4, 522. Hoare, P (1984). The development of psychiat ric disorder among school children with epilepsy. Developmental Medicine & Child Neurology, 26, 3-13. 98

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Hoeppner, J. B., Garron, D. C., & Cartwright, R. D. (1984). Self-reported sleep disorder symptoms in epilepsy. Epilepsia 25 434-437. International League Against Epilepsy (1989). Proposal for revised classification of epilepsies and epileptic syndromes. Epilepsia, 30, 389-399. Janz, D. (1962). Grand mal epileps ies and the sleep-waking cycle. Epilepsia, 3 69-109. Janz, D. (2000). Epilepsy with grand ma l on awakening and sleep-waking cycle. Clinical Neurophysiology, 111, S103-S110. Kaemingk, K.L., Pasvogel, A.E., Goodwin, J.L., Mulvaney, S.A., Martinez, F., Enright, P.L., et al. (2003). Learning in children and sleep di sordered breathing: Findings of the Tucson Children's Assessment of Sleep Apnea (TuCASA) Prospective Cohort Study. Journal of the International Neuropsychological Society, 9(7) 1016-1026. Keene, D.L., Manion, I., Whiting, S., Belanger, E., Brennan, R., Jacob, P., et al. (2005). A survey of behavior problems in children with epilepsy. Epilepsy & Behavior, 6(4), 581586. Koh, S., Ward, S. L., Meei, L., & Chen, L. S. (2002). Sleep apnea treatment improves seizure control in children with neurodevelopmental disorders. Pediatric Neurology, 22 36-39. Kotagal, S. & Pianosi, P. (2006). Sleep disorders in children and adolescents. British Medical Journal, 332, 828-832. Loring, D.W., & Meador, K.J. (2004). Cognitive side effects of antiepileptic drugs in children. Neurology 62, 872-877. Maganti, R., Hausman, N., Kochn, M., Sandok, E., Glurich, I., & Mukesh, B. N. (2006). Excessive daytime sleepiness and sleep complaints among children with epilepsy. Epilepsy & Behavior, 8, 272-277. Maganti, R., Sheth, R. D., Hermann, B. P., Webe r, S., Gidal, B. E., & Fine, J. (2005). Sleep architecture in children with idiopathic generalized epilepsy. Epilepsy, 46 104-109. Malow, B. A., Bowes, R. J., & Lin, X. (1997). Predictors of sleepiness in epilepsy patients. Sleep, 20, 1105-1110. Malow, B. A., Foldvary-Schaefer, N., Vaughn, B. V., Selwa, L. M., Chervin RD, Weatherwax, K. J., et al. (2008). Treating obstruc tive sleep apnea in adults with epilepsy: a randomized pilot trial. Neurology, 71 572-527. Manly, T., Anderson, V., Nimmo-Smith, I., Turn er, A., Watson, P. & Robertson, I.H. (2001). The differential assessment of children's atte ntion: The Test of Everyday Attention for Children (TEA-Ch), normative sample and ADHD performance. Journal of Child Psychology & Psychiatry & Allied Disciplines, 42(8) 1065-1081. 99

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Manly, T., Robertson, I. H. Anderson, V., & Nimmo-Smith, I. (1999). TEA-Ch: The Test of Everyday Attention for Children. Bury St. Edmunds, England: Thomas Valley Test Company Limited. Mattson, R. H. (2003). Overview: idi opathic generalized epilepsies. Epilepsia, 44(Suppl. 2), 2-6. Mindell, J. & Owens, J. (2003). A Clinical Guide to Pediatri c Sleep: Diagnosis and Management of Sleep Problems in Children and Adolescents. Philadelphia: Lippincott, Williams, and Wilkins. Mitchell, W.G., Zhou, Y., Chavez, J. M., & Guzman, B. L. (1992). Reaction time, attention, and impulsivity in epilepsy. Pediatric Neurology, 8(1), 19-24. Mirsky A. F., Anthony, B. J., Duncan, C. C., Ahear n, M. B., & Kellam, S.G. (1991). Analysis of the elements of attention: a neuropsychological approach. Neuropsychological Review 2, 109-45. Mirsky, A.F. & Duncan, C.C. (2001). A nosology of disorders of attention. Annals of the New York Academy of Sciences, 931, 17. O'Brien. L. M. & Gozal, D. (2004). Neurocognitive dysfunction and sleep in children: from rodents to man. Pediatric Clinics of North America, 51, 187. O'Brien, L.M., Mervis, C.B., Holbrook, C.R., Bruner, J.L., Klaus, C.J., Rutherford, J., et al. (2004). Neurobehavioral implications of habitual snoring in children. Pediatrics, 114(1), 44-9. O'Brien, L.M., Mervis, C.B., Holbrook, C.R., Br uner, J.L., Smith, N.H., McNally, N. et al. (2004). Neurobehavioral correlates of sleep-disordered breathing in children. Journal of Sleep Research, 13(2), 165-72. Oostrom, K. J., van Teeseling, H., Smeets-Schoute n, A., Peters, A. C., & Jennekens-Schinkel, A. (2005). Three to four years after diagnos is: Cognition and behaviour in children with epilepsy only. A prospective, controlled study. Brain, 128(7), 1546-1555. Ott, D., Caplan, R., Guthrie, D., Siddarth, P., Kom o, S., Shields, W.D. et al. (2001). Measures of psychopathology in children with complex pa rtial seizures and primary generalized epilepsy with absence. Journal of the American Acade my of Child and Adolescent Psychiatry, 40(8), 907-914. Owens, J. (2005). The ADHD and sleep conundrum: A review. Journal of Developmental & Behavioral Pediatrics, 26, 312-322. Owens, J., Opipari, L., Nobile, C., & Spirito, A. (1998). Sleep and daytime behavior in children with obstructive sleep apnea and behavioral sleep disorders. Pediatrics, 102(5), 1178-84. 100

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Owens, J., Sangal, R. B., Sutton, V. K., Bakken, R., Allen, A. J., & Kelsey, D. (2008). Subjective and objective measures of sleep in children w ith attention-deficit/ hyperactivity disorder. Sleep Medicine, in press. Epub ahead of prin t retrieved September 2008, from www.elsevier.com/locate/sleep. Owens, J., Spirito, A., Marcotte, A. C., McGuinn, M., & Berkelhammer, L. (2000). Neuropsychological and behavi oral correlates of obstructive sleep apnea syndrome in children: A preliminary study. Sleep and Breathing, 4, 67-68. Pellock, J. M. (2004). Defining th e problem: psychiatric and behavi oral comorbidity in children and adolescents with epilepsy. Epilepsy & Behavior, 5(Suppl. 3), 3-9. Pinton, F., Ducot, B., Motte, J., Arbus, A.S., Barondiot, C., Barthez, M.A., et al. (2006). Cognitive functions in children with benign ch ildhood epilepsy with centrotemporal spikes (BECTS). Epileptic Disorders, 8, 11-23. Reynolds, C. R., & Kamphaus, R. W. (2004). Behavior Assessment System for Children Second. Edition (BASC-2). Circle Pines, MN: American Guidance Service. Sadeh, A., Gruber, R., & Raviv, A. (2002). Slee p, neurobehavioral functioning, and behavior problems in school-age children. Child Development, 73(2), 405-417. Sanchez-Carpintero, R. & Neville, B. G. R. (2003), Attentional ability in children with epilepsy. Epilepsia, 44(10), 1340-1349. Schoenfeld, J., Seidenberg, M., Woodard, A., Hec ox, K., Inglese, C., Mack, K., et al. (1999). Neuropsychological status of childre n with complex partial seizures. Developmental Medicine and Child Neurology, 41, 724. Schouten, A., Oostrom, K. J., Pestman, W. R., Peters, A. C. B., & Jennekens-Schinkel, A. (2002). Learning and memory of school children with epilepsy: a prospective controlled longitudinal study. Developmental Medicine & Child Neurology 44, 803. Schubert, R. (2005). Attention de ficit disorder and epilepsy. Pediatric Neurology, 32(1) 1-10. Semrud-Clikeman, M. & Wical, B. (1999). Compone nts of attention in children with complex partial seizures with and without ADHD. Epilepsia, 40(2) 211-215. Sheslow, D., & Adams, W. (1990). Wide Range Assessment of Memory and Learning. Wilmington, DE: Jastak Associates. Shinnar, S. & Pellock, J. M. (2002). Update on the epidemiology and prognosis of pediatric epilepsy. Journal of Child Neurology, 17 (Suppl. 1), S4-S17. Sterman, M. B., Shouse, M. N., & Passousant, P. (1982). Sleep and Epilepsy. New York: Academic. 101

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103 BIOGRAPHICAL SKETCH Susan R. Bongiolatti was born in Springfield, Missouri. She spent her childhood years in Fairfield, Iowa, and later moved to Paducah, Ke ntucky, where she graduated valedictorian from Lone Oak High School in 1995. Susan attended Wash ington University in St. Louis, Missouri. She completed an honors thesis examining worki ng memory in children with Attention Deficit/ Hyperactivity Disorder (ADHD) with Sandra Ha le, Ph.D., and graduated summa cum laude with a bachelors degree in psychology. Susan remained at Washington University for two years as a cognitive neuroscience research assistant, work ing with Todd Braver, Ph.D. She entered the University of Floridas doctoral program in Clinical and Health Psychology in 2001, and earned her M.S. in clinical psychology in May 2003. Her primary area of study is neuropsychology, with a focus on pediatric populations includi ng children with epilepsy. In June 2008, Susan completed a pre-doctoral clinical psychology internship at the Childrens Hospital of Philadelphia. She will earn her Ph.D. in clinical psychology in December 2008 with a concentration in neuropsychology. Upon comple tion of her dissertation, Susan will begin a postdoctoral fellowship in clinical neuropsychology at the Clevel and Clinic, with a continued focus on pediatric populations.