1 S OF REPAIRED CLEFT LIP By DAFNE ELLIS A THESIS PRESENTED TO THE GRADUATE SCHOOL OF THE UNIVERSITY OF FLORIDA IN PARTIAL FULFILLMENT OF THE REQUIREMENTS FOR THE DEGREE OF MASTER OF SCIENCE UNIVERSITY OF FLORIDA 2012
2 2012 Dafne Ellis
3 To my mom, Nancy Jorquera for insp iring me to reach for the stars and to m y husband, Paul Ellis f or helping me achieve my dreams
4 ACKNOWLEDGMENTS I thank my committee members, Calogero Dolce, D.D.S., Ph .D.; Timothy T. Wheeler, D.M.D., Ph.D. ; and Sue McGorray, Ph.D. for their guidance and support. I would also like to thank Aylin Ockular for her assistance and willingness to help.
5 TABLE OF CONTENTS P age ACKNOWLEDGME NTS ................................ ................................ ................................ .. 4 LIST OF TABLES ................................ ................................ ................................ ............ 6 LIST OF FIGURES ................................ ................................ ................................ .......... 7 ABSTRACT ................................ ................................ ................................ ..................... 8 CHAPTER 1 INTRODUCTION ................................ ................................ ................................ ...... 9 2 MATERIALS AND METHODS ................................ ................................ ................ 13 3 RESULTS ................................ ................................ ................................ ............... 17 4 DISCUSSION ................................ ................................ ................................ ......... 24 5 CONCLUSION ................................ ................................ ................................ ........ 28 LIST OF REFERENCES ................................ ................................ ............................... 29 BIOGRAPHICAL SKETCH ................................ ................................ ............................ 32
6 LIST OF TABLES Table P age 3 1 Parents demographic comparison ................................ ................................ .... 20 3 2 Reliability ................................ ................................ ................................ ........... 20 3 3 Overall agreement (score value) between first and second evaluation ............. 21 3 4 Pearson correlation coefficients ................................ ................................ ......... 21
7 LIST OF FIGURES Figure P age 2 1 Example of cleft lip patient set of images from picture b ooklet ........................... 16 2 2 Example of non cleft lip patient set of images from picture booklet .................... 16 3 1 Mean ratings o f unilateral cleft lip pa tients ................................ .......................... 22 3 2 Mean ra tings of non cleft lip patients ................................ ................................ .. 22 3 3 Average ratings versus gold standard for unilateral cleft lip patie nts and non cleft lip patients ................................ ................................ ................................ ... 23
8 Abstract of Thesis Presented to the Graduate School of the University of Florida in Partial Fulfillment of the Requirements for the Degree of Master of Science ERSPECTIVE S OF REPAIRED CLEFT LIP By Dafn e Ellis May 2012 Chair: Calogero Dolce Major: Dental Sciences Orthodontics Introduction : One of the goals in the treatment of children with cleft lip and palate (CLP) is to improve the esthetic appearance of s tructures affected by the cleft. However, patients invariably demonstrate some degree of deformation, asymmetry, scarring, or an uneven junction. Comparison of the perception of the esthetic outcome of treatment between families who have a child that has C LP and families who do not would be beneficial to determine if any additional procedures, such as lip or nose revision surgery, are advisable. Methods : 30 participants with a child with CLP and 30 who did not were asked to rate the esthetic appeal of facia l and profile pictures of children with CLP. Different parts of the face (upper lip, lower lip, nose and chin) were rated separately on a five point scale indicating very good (1), good (2), fair (3), poor (4) or very poor (5) appearance. Results : When ass essing patients with CLP, the parents without children with CLP were more critical than parents with children with CLP. These findings were statistically significant for the upper lip and no significance was found on other facial features scored. Conclusio ns : In this study, parents with a child with a cleft lip report greater satisfaction with the esthetic outcome mainly of lip, and evaluate the cleft esthetics more favorably, than parents who do not have children with a cleft lip.
9 CHAPTER 1 INTRODUCTION C left lip with or without cleft palate are congenital malformations characterized by an incomplete formation of those structures which separate nasal from oral cavities (i.e. lip, alveolus, hard and soft palate), and can affect the right, the left, or both sides. This malformation is one of the most common cranio facial defects in humans; cleft lip and palate occurs in approximately 1 out of 750 live births in the United States 1,2,3,4,5,6 Although cleft lip and cleft palate often occur together, they can oc cur separately. Approximately 70 percent of infants who have a unilateral cleft lip and 85 percent of infants with a bilateral cleft lip also have a cleft palate. 7 Clefts are further classified as either non syndromic or syndromic 7 Patients affected by or ofacial clefts require multidisciplinary treatment. Traditionally cleft lip repair is performed at approximately 3 months of life. Subsequent palatoplasty is performed at 1 year and alveolar bone grafting at 8 to 9 years of age. Finally, orthodontic treatm ent occasionally involving orthognathic surgery with or without rhinoplasty is completed when patients reach adolescence. 8,9 The goal s of the surgical corrections of a unilateral cleft lip are function, symmetry and esthetics. This requires the surgeon to approximate the lip segments, forming a effect on the tip of the nose and attempting to camouflage the surgical scar. 10 However, repairing a unilateral cleft lip and pal ate rarely produces exact facial esthetics. Despite advanced surgical technique, post procedural distortion of the lip, nose, and dentition still occur. Surgical repair of the lip and palate also leads to a series of well recognized secondary growth distur bances that lead to anomalies in nasal form, nasal asymmetry,
10 and distortion of the upper lip. This can also produce recognized scarring of the philtral area, with a diminished or absent philtral groove. 11,12 Nasolabial appearance is one of the most impor tant measures of success involving treatment outcome for cleft lip patients. Methods described in studies assessing nasolabial appearance can be divided into qualitative and quantitative categories. Quantitative methods objectively analyze the extent of a bnormal morphology. Qualitative methods are more subjective and analyze facial esthetics and appearance using scales, indices, scoring systems, and rankings. Nevertheless, if any scoring index is to be a useful tool analyzing cleft lip and palate outcome, it must satisfy the requirements of scientific reproducibility. 13 Several indices in literature specifically evaluate the esthetic outcome of nasolabial appearance in the repaired cleft lip and palate patient. Asher McDade et al. in 1991 developed a me thod for rating nasolabial appearance of unilateral cleft repairs from photographs. Four nasolabial components (nasal form, nose symmetry, vermilion border, and nasal profile) were rated separately on five point scales by a panel of orthodontists all famil iar with cleft lip deformity. In this method the nasolabial areas were masked, thus reducing the subjective influence of surrounding facial features 11,14 Previous studies suggest that experimental judges are biased towards overall facial attractiveness wh ich varies among individual judges. The standardized rating system outlined by Asher McDade may be used to differentiate treatment outcome in patients from different treatment centers 11 The Asher McDade index also improved reproducibility of another esthe tic index described by Johnson and Sandy in 2003. It enhanced rater objectivity by including text criteria for each index category 13 The reproducibility of the 2003 study also compared favorably with other established esthetic
11 indices and was also not aff ected by subject age. This improved the versatility for both direct and indirect esthetic evaluations. 13 There are limitations to utilizing photographic methods to rate nasolabial appearance which hav e been addressed in literature. 15,16 These limitations i nclude variability in the quality of the photography, and the lack of a standard scale defining the categories used among judges in different studies. It is important to consider, however, that current photographic rating methods of cleft lip repairs can b e frustrating to employ because the data they generate may be difficult to analyze. Therefore, it cannot be concluded that patients in these studies who were awarded favorable result scores were actually treated by superior surgical methods compared to tho se patients who were rated less favorably. 17 Finally, after surgical closure patients may demonstrate some degree of nose and upper lip deformation, nasal asymmetry, scarring of the philtral area, and an uneven muco cutaneous junction. These craniofacial impairments can result in negative psychological consequences, including low self esteem and the stigma of social rejection based on cosmetic appearance. Investigating parental perception of esthetic treatment outcome comparing families who have a child wi th CLP and families who do not may be beneficial to determine if additional procedures are advisable. The purpose of this study was to investigate the perception of the esthetics of repaired cleft lip of parents of children with CLP and compare it to non c understanding of the differences in perceptions of facial esthetics between professional members of a cleft repair team, parents of these patients, and individuals with a cleft lip would be an invaluable tool in treat ment planning. It would also facilitate discussion of treatment outcomes with the patient and the individuals that play a role in the
12 management of this pathology. This information would be an asset in cleft treatment planning, discussion of treatment outc omes, as well as management of patient expectations so that optimal treatment results and patient satisfaction are achieved.
13 CHAPTER 2 MATERIALS AND METHODS The materials consisted of a questionnaire and a picture booklet evaluating esthetic appearance o f cleft lip of patients with CLP. Photographic records were collected from the University of Florida Orthodontic Clinic database, and cropped so that only the nose, lip and chin were visible (Figure 2 1 and 2 2). Photographs were then altered to remove pat ient identity then organized in files with a case number specific for the project. A picture booklet was prepared which contained 33 sets of frontal and profile views of each patient. The booklet contained images of cleft lip patients, non cleft lip patien ts, and included both males and females of different race in order to minimize bias. The final picture booklet comprised a total of 33 sets of photos that were organized randomly. Twenty five of the photos featured patients with CLP, five photos depicted p atients without CLP, and three photos were duplicates in order to assess intra rater reliability (Figure 2 1 and 2 2). Participants completed the survey questionnaire using the survey picture booklet. Subjects were asked to examine each set of patient phot ographs then rate the esthetic appeal of each of them using an esthetic index. This index incorporated different parts of the face (upper lip, lower lip, nose and chin) which were rated separately on a five point scale. The scale used a numerical score whi Visual Analogue Scale, or VAS, even though both methods have comparable responsiveness and validity in a randomized trial settin g; a five point scale offers ease of administration and interpretation. 18 The questionnaire also included demographic questions such as age, sex, highest level of education completed, ethnicity or race,
14 number of family members, number of family members li ving in the household, family history of cleft lip or palate, and relationship of this CLP family member to the participant. The evaluators were randomly selected from the parents of patients of the Graduate Orthodontic Clinic at UF and ultimately consist ed of 60 study participants. The first group (n=30), CLP parent group, were comprised of parents with at least one child with a history of a repaired cleft lip. These individuals were preselected from a patient database of the Graduate Orthodontic Clinic, and were contacted in person at the comprised of parents of children with no history of CLP. This group was selected from volunteers that were matched to experimental particip age and gender. The control subjects did not have any close family members with a history of cleft lip. Parents were approached to solicit permission for voluntarily participation in the study, and were asked to sign an i nformed consent letter. This project was approved by the Institutional Review Board. Kappa statistics were used to examine the intra rater reliability, comparing the 3 duplicate image scores. The data was summarized by evaluating the discrepancies for eac h duplicate image for each feature evaluated, across images. A total discrepancy score was calculated for each rater. Overall kappa statistics, unweighted and weighted, were calculated based on all duplicate calls across raters. The weighted kappa stati stic yields higher agreement for small discrepancies, with larger departure from agreement for more extreme discrepancies. Kappa statistics were also calculated for each facial component. A kappa statistic was calculated to assess intra examiner reliabili ty while
15 taking into account the agreement occurring by chance. The strength of agreement is marginal when the kappa statistic is 0 0.2, fair between 0.21 0.4, moderate between 0.41 0.6, substantial between 0.61 0.8, and almost perfect when the kappa stat istic approaches 0.81 1.00. 19 Summary statistics were calculated for each rater and facial feature for the CLP images (n=25) and then non CLP images (n=5). Average scores were averaged across facial features. Two sample t tests were used to assess diffe rences between CLP parents and control parents. Two sample t tests were used to examine whether factors (sex and race of parent completing the ratings) affected average scores. Correlation was examined to assess patterns in scoring related to age. Two sam ple t tests were used to examine Non Cleft versus CLP ratings within parent type. One sample t tests were used to compare ratings to the gold standard
16 Figure 2 1. Example of cleft l ip patient set of images from picture booklet Figure 2 2 Example of n on cleft l ip patient set of images from picture booklet
17 CHAPTER 3 RESULTS Sample characteristics are summarized in Table 3 1. The experimental and control groups were distributed similarly regarding gender, age, race and education level. Specifically, the cleft lip group had more males (40%) than the control group (23%), but this was not statistically significant. Race and/or ethnicity also did not differ significantly: both groups comprised near even numbers of Caucasians, African Americans, and Hispan ics. The age distribution was similar for the two groups: cleft lip parents had a mean age of 42.5 (s.d. 9.0, range 28 63), while the control parents mean age was 41.7 (s.d. 7.7, range 30 65). Education was of borderline significance, and control participa nts had more education on average. The intra rater agreement was evaluated using three sets of duplicate photographs placed randomly throughout the picture booklet. Discrepancy scores were generated by assigning an absolute value of the difference between the first and second calls for each of the three sets of duplicates for each component (upper lip, lower lip, nose and chin). Therefore four calls were considered for each subject involving three calls for each facial component, totaling twelve calls per parent. Note that each discrepancy ranged from 0 to 4. (Table 3 2) 3 3) Overall, the raters wer e consistent with themselves 56% of the time (represented by one asterisk (*) in the table), and their answers were within +/ 1 point 90.9 % of the time (represented by one asterisk (*) plus two asterisks (**) in the table). When the Kappa score was calculated it agreement.
18 For th e initial comparison of facial ratings of cleft lip parents and control parents, average ratings were calculated for each facial component. For children with unilateral cleft lip, ratings of twenty five sets of photographs were averaged based on participan t groups. For non cleft lip children, ratings of five sets of photographs were also averaged based on participant groups in a similar fashion. A gold standard rating was assigned to each component of each set of photographs with the purpose of comparing th e facial assessment of the two groups to a standard defined by two calibrated orthodontists. The principal investigators were calibrated to one another and generated standard scores that represented standard base ratings for all the photograph sets in the picture booklet. When rating photographs of children with CLP, parents without children with CLP gave higher ratings (were more critical) than parents with children with CLP. For ratings of the upper lip involving pictures of children with unilateral clef t lip, the experimental group rating was lower than the control group. This was also true for photographs of only the upper lip but not fo r the nose. (Figures 3 1 and 3 2 ) A Pearson correlation was calculated to determine the correlation between the rating assessments of the different facial components. As hypothesized, there was a high degree of correlation between the component scores and overall averages. This indicated that a high or low score for the upper lip appearance corresponded, respectively, wit h a high or low score for other components of the face. (Table 3 4) Generally, control parents were less satisfied compared to parents who have had children with a history of cleft lip repair. However, in most of the comparisons the
19 differences were not si gnificant. Likewise, these results indicate that the both groups of parents were more critical than the gold standard. (Figure 3 3 ) Possible covariates were examined to determine if facial ratings were influenced by differences in age, sex or race of the parents. Pearson correlation coefficients and two sample t test values indicated that there are not significant relationships to these variables.
20 Table 3 1. Parents demographic c omparison Cleft Lip Parent Control Parent Significance Sex female: 60% female: 77% Chi square test p= 0.17 male: 40% male: 23% Race Caucasian: 80% Caucasian 80% Fisher exact test p=1.00 African American: 10% African American 7% Hispanic: 10% Hispanic 10% Native American 3% Age mean: 42.5 mean 41.7 t test p=0.6 9 S.D.: 9.0 S.D. 7.7 WRS test p=0.65 range: 28 63 range 30 65 Education HS grad or less: 55% HS grad or less: 27% Chi square test p= 0.06 Some college: 28% Some college: 33% College grad or more: 17% College grad or more: 40% Table 3 2. Reliability Variable N Median Mean Std. Dev. Minimum Maximum Discrepancy for duplicate subject 1 58 2.0 2.1 1.1 0 5 Discrepancy for duplicate subject 2 60 2.5 2.7 2.4 0 11 Discrepancy for duplicate subject 3 60 2.0 1.7 2.1 0 12 Discrepancy Upper Lip 60 1.0 1.5 1.3 0 2 Discrepancy Lower Lip 60 1.5 1.6 1.4 0 6 Discrepancy Nose 60 2.0 1.8 1.3 0 6 Discrepancy Chin 60 1.0 1.5 1.7 0 7 Discrepancy Total 58 6.0 6.6 4.4 0 21
21 Table 3 3. Overall agreement (score value) between first an d second evaluation Score 2 Frequency 1 2 3 4 5 Total Percent Score 1 1 1 34 66 1 7 6 3 226 1 8.8 9.3 ** 2. 4 0.8 0.4 31.7 2 33 14 8 53 10 3 247 4.6 ** 20.8 7.4 ** 1. 4 0. 4 34.7 3 5 41 66 16 5 133 0.7 5.7 ** 9.3 2.3 ** 0. 7 18.7 4 0 7 25 24 9 65 0. 0 1.0 3.5 ** 3.4 1.3 ** 9.1 5 0 0 2 13 26 41 0. 0 1. 0 0. 3 1.8 ** 3.7 5.8 Total 1 72 2 62 1 6 3 69 46 712 24.2 3 6 .8 2 2.9 9.7 6.5 ** 100.0 (*). Percentage of correct agreement (**). Percentage of agreement within one value to eith er side Table 3 4. Pearson c orrelation coefficients cleft upper lip cleft lower lip cleft nose cleft chin cleft average score cleft upper lip 1.00 0.77 0.87 0.06 0.91 < .0001 < .0001 < .0001 < .0001 N = 60 Prob. > |r| under H0: Rho=0
22 Figure 3 1. Mean r atings of unilateral cleft lip p atients. Assessments were made using a five point sca le. The higher the number is on the scale, the less favorable the assessment is. (P< 0.05) Figure 3 2. Mean ratings of non cleft lip p atients Assessments wer e made using a five point scale. The higher the number is on the scale, the less favorable the assessment is. (P< 0.05)
23 Figure 3 3. Average ratings versus g old standard for unilateral cleft lip p atients and n on cleft lip patients ( P< 0.05)
24 CHAPTER 4 D IS CUSSION Understanding the perceptual differences of facial esthetics between providers, patients with a cleft lip, and laypersons improves management of cleft disorders. Similarly, discussion of these treatment outcomes with the patient, health care tea m, and parents optimizes patient expectations and achieves superior treatment outcomes and improves patient satisfaction. This study evaluated the differences in parental perception of repaired unilateral cleft lip by subjectively rating photographs of chi ldren. In general, patients with a cleft lip undergo extensive treatments involving sequential surgeries that may result in scarring and disfigurement. Several studies have evaluated subjective assessment of post surgical treatment results. 20 21,22,23,24 T his study attempted to compare the parental perception of post procedural esthetic success of children with cleft lip repair to those with no history of cleft lip. Different life dings in a variety of ways. It was hypothesized that parents who had a child with cleft lip would perceive cleft lip repair differently than those parents who did not have a child with a history of a cleft lip or palate. The parents of babies with clefts a re usually shocked and confused just after the birth. 25 One might theorize that parents in the cleft lip group might want their children to look more like non cleft lip children and might be conditioned to perceive a cleft lip with negative connotations. I n contrast to this hypothesis, it is possible that these parents might be content with any surgical closure after experiencing the birth of a child with an open, unrepaired lip, and actually ignore the subtle esthetic appearance of the lip and nose. It has been suggested that patients and parents hold different views about the perception of the patient with a cleft lip.
25 However, the literature illustrates contradictory conclusions with respect to this matter. Previous studies had also evaluated difference i n perception between professionals and laypeople and cleft patients versus non cleft patients. In a study in 1988, Strauss et al reported that there was no signif icant difference between parent and patient rated satisfaction regarding lip appearance. 26 Pa tient ratings of facial appearance showed that most patients were very pleased or moderately pleased. Nearly all the patients felt that their operations had accomplished what they expected, though some of the cleft lip patients and their parents were less appraisals of treatment outcome and appearance. In addition, a questionnaire survey in 1991, Noar found that the parents were correct in believing that they we re happier than their children and were also more satisfied with the appearance of the lip. 27 Noar reported that patients were happy with their overall facial appearance and speech, although noted that they were less satisfied with features which are direc tly affected by the cleft: the nose, lips, profile, smile, and teeth. In general patients and parents are satisfied with the treatment outcome and the facial appearance. Van Lierde et al. in 2011 reported that there is no significant difference between par ents and the children regarding the appearance of facial esthetics. However, the parents of the children with a cleft have a decreased satisfaction with the facial esthetics in comparison with the controls. 28 Furthermore, differences exist in the ratings of facial esthetics among professionals and laypeople for patients with and without a cleft lip. Therefore, a
26 perceived need for further surgery should also be considered when managing cleft treatment expectations. Sinko et al in 2005 found no difference between the medical and non medical professionals in their perception of the need for further surgery, and the professionals tended to deem further treatment unnecessary. 29 A possible reason ay be due to their better understanding of the limitations of the surgery in correcting facial deformities. Even when the professionals rated the facial esthetics less favorably, they may have considered that realistic esthetic results have been achieved f or the cleft patients, therefore the higher disagreement on the need for further surgery. This finding was corroborated in a recent study in 2011 by Sinko et al. which evaluated perceived need for further treatment between professionals and laypersons. The professional raters perceived further surgery was not required to correct all components of the face, while the lay raters perceived the opposite. 29 It should also be considered that the difference in rating panel composition may influence the general p ultimately affected by their own experience with cleft lip and/or palate. A study by Foo et al. compared the esthetic perceptions in individuals who had a cleft lip, with those who did not, and disc overed that the esthetic ratings in the former population were significantly more attractive than those individuals who did not have cleft pathology. 30 The principal goal of that study was to compare professionals versus laypeople; however, the number of l ay raters with a cleft was small, and therefore the ratings in that study may not truly represent a larger population of individuals with a cleft lip.
27 Further investigations with a greater number of raters with a cleft are warranted, in order to test the v alidity of these findings The results of the study illustrated that control parents were actually more critical assessing the esthetics of the repaired cleft lip. Although this was true in general for all of the facial components, it was only statistically significant for the upper lip. These data also suggest that the parents of children who have a cleft lip reported greater satisfaction with the esthetic outcome of the repair. However, these data failed to illustrate statistical significance to suggest th at parents in the experimental group are merely content with a lip closure and disregard facial esthetics. gold standard rating it was found that the parents were more critical in their ratings. This was true w ith regards to both parents groups which could be expected considering that professionals report greater satisfaction from the treatment outcome and evaluate cleft consequences with less severity than laypeople. 31 This could be attributed to the fact that specialists are more familiar with the esthetic consequences of the cleft and the difficulties of treating them, and thus evaluate clefts with less severity than laypeople. Statistical analysis revealed moderate agreement between the parents. This could be attributed to the fact that raters were not calibrated and were not given specific instructions. Adding objective criteria to the five point scale would have been too complex for non professional raters. Moreover, this would have required calibrating each parent which would have been challenging due to time limitations.
28 CHAPTER 5 C ONCLUSION In this study, the cleft lip parent group reported greater satisfaction with the esthetic outcome of cleft lip and evaluated the cleft esthetics more favorably than parents who do not have children or close relatives with a cleft lip. These findings were statistically significant for the upper lip, but might not be clinically significant for any of the facial components (including the upper lip) since both the control and cleft lip parent Raters were consistent with themselves and both groups rated facial esthetics significantly higher that the trained orthodontists. These findings are in agreement with contemporary literature. Further research is needed to objectively assess the primary esthetic needs of individuals with cleft lip, which along with the subjective needs defined by the patient, should determine further treatment interven tions.
29 LIST OF REFERENCES 1. Yu W, Serrano M, San Miguel S, Ruest LB, Svoboda KKH. Cleft lip and palate genetics and application in early embryological development. Indian J Plast Surg. 2009; (1)42:S35 S50. 2. Merritt L. Understanding the embryology and genetics of cleft lip and palate. Adv Neonatal Care. 2005;(5)2:64 71. 3. Tolarova M, Cervenka J. Classification and birth prevalence of orofacial clefts Am J Med Genet. 1998;75:126 137. 4. Canfield MA, Honein MA, Yuskiv N, Xing J, Mai CT, Collins JS, Devine O, Petrini J Ramadhani TA, Hobbs CA, Kirby RS. National Birth Defects Prevention Network. National Estimates and Race/Ethnic Specific Variation of Selected Birth Def ects in the United States, 1999 2001. Birth Defects Res A Clin Mol Teratol. 2006;76: 747 756. 5. Croen LA, Shaw GM, Wasserman CR, Tolarova MM. Racial and Ethnic Variations in the Prevalence of Orofa cial Clefts in California, 1983 1992. Am J Med Genet. 1998;79: 42 47. 6. Rullo R, Carinci F, Mazzarella N, Maria Festa V, Farina A, Morano D, Carls F, Gombos F. A Delai scored according to the EUROCLEFT guidelines. Int J Pe diatr Otorhinolaryngol. 2006;70: 463 468. 7. Murray JC. Gene/environment causes of cleft lip and /or palate. Clin Genet. 2002;61: 248 256. 8. Esper LA, Sbrana MC, Ribeiro IWJ, De Siqueira EN, De Almeida A. Esthetic Analysis of Gingival Components of Smile and Degree of Satisfaction in Individuals With Cleft Lip and Palate Cleft Pal. Craniofac. J. 2009;(46)4:381 387. 9. Tollefson TT and Gere RR. Presurgical Cleft Lip Management: Nasal Alveolar Molding Facial Plast Surg. 2007;23:(2)113 122. 10. Clark JM, Skoner JM, Wang TD. Repair of the Unilateral Cleft Lip/Nose Deformity. Facial Plast Surg. 2003;(19)1:29 39. 11. Asher McDade C, Roberts C, Shaw WC, Gallager C: Devel opment of a method for rating nasolabial appearance in patients with clefts of the lip and palate Cleft Pal Craniofac J. 1991;28: 385 391 14. 12. Jacobsen A. Psychological aspects of dentofacial esthetics and orthognathic surgery. Angle Orthod. 1984;54:18 35.
30 13. Johnson N, Sandy J. An aesthetic index for evaluation of cleft repair. Euro J Orthod. 2003; 25(3):243 249. 14. Tobiasen J M 1991 Commentary. Cleft Palate Craniofacial Journal 28:193 194. 15. Asher McDade C, Brattstrm V, Dahl E, McWilliam J, Mlsted K, Plint DA, P rahl Andersen B, Semb G, Shaw WC. The RPS. A six center international study of treatment outcome in patients with clefts of the lip and palate: Part 4. Assessment of nasolabial appearance. Cleft Palate Craniofac J 1992; 29:409 412. 16. Markus AF, Delaire J. F unctional primary closure of cleft lip Br J Oral Maxillofac Surg. 1993;31:281 291. 17. William S. Garrett Jr., M.D. Director of Medical Service University of Pittsburgh Cleft Palate Center Pittsburgh, Pennsylvania. Commentary on of a method for rating nasolab ial appearance in patients with clefts of the lip and palate. Asher McDade C, Roberts C, Shaw WC, Gallager C: Development of a method for rating nasolabial appearance in patients with clefts of the lip and palate Cleft Pal Craniofac J. 1991;28:385 391. 18. Ja eschke R, Singer J, Guyatt GH. A comparison of seven point and visual analogue scales. Con trolled Clinical Trials 1990;11: 43 51. 19. Landis JR, Koch GG. The measurement of observer agreement for categorical data. Biometrics 1977;33:159 174. 20. Broder HL, Smith FB Strauss RP. Habilitation of patients with cleft: parent and child ratings of satisfaction with appearance and speech. Cleft Palate Craniofac J 1992;29:262 267. 21. Hunt O, Burden D, Hepper P, Stevenson M, Johnston C. Parent reports of the psychosocial functi oning of children with cleft lip and/or palate. Cleft Palate Craniofac J 2007;44:304 311. 22. Thomas PT, Turner SR, Rumsey N, Dowell Y, Sandy JR. Satisfaction with facial appearance among subjects affected by a cleft. Cleft Palate Craniofac J 1997;34:226 231. 23. Christofides E, Potgieter A, Chait L. Along term subjective and objective assessment of the scar in unilateral cleft lip repairs using the Millard technique without revisional surgery. J Plast Reconstr Aesthet Surg. 2006;59(4):380 6. 24. Richman LC, Holmes CS Eliason MJ. Adolescents with cleft lip and palate: self perceptions of appearance and behavior related topersonality adjustment. Cleft Palate J. 1985;Apr;22(2):93 6.
31 25. Clifford E. Psychosocial aspects of orofacial anomalies: speculations in search of data. ASHA report No. 8. American Speech and Hearing Association, 1973. 26. Strauss RP, Broder H, Helms RW. Perceptions of appearance and speech by adolescent patients with cleft lip and palate and by their parents. Cleft Palate J. 1988 Oct; 25(4):335 42. 27. Noar JH. Questionnaire survey of attitudes and concerns of patients with cleft lip and palate and their parents. Cleft Palate Craniofac J. 1991 Jul;28(3):279 84. 28. Van Lierde KM, Dhaeseleer E, Luyten A, Van De Woestijne K, Vermeersch H, Roche N. Parent and child rati ngs of satisfaction with speech and facial appearance in Flemish pre pubescent boys and girls with unilateral cleft lip and palate. Int J Oral Maxillofac Surg. 2011 Nov 27. 29. Sinko K, Jagsch r, Prechtl V, Watzinger F, Hollmann K, Baumann A. Evaluation of est hetic functional and quality of life outcome in adult cleft lip and palate patients. Celft Palate Craniofac J. 2005 Jul;42(4):355 61. 30. Foo P, Sampson W, Roberts R, Jamieson L, David D. Facial aesthetics and perceived need for further treatment among adult s with repaired cleft as assessed by cleft team professionals and laypersons. Eur J Orthod. 2011 Nov 2. 31. Papamanou DA, Gkantidis N, Topouzelis N, Christou P. Appreciation of cleft lip and palate treatment outcome by professionals and laypeople. Eur J Orthod 2011 Jul 19.
32 BIOGRAPHICAL SKETCH Dafne Ellis is a native of Argentina and grew up in the capital of the San Juan province. She initiated her dental studies at the University of Cordoba but immigrated to the United States in 2002 with her husband, Dr. M ichael Ellis. She subsequently matriculated to the University of Arkansas in Little Rock and earned Magna Cum Laude upon completion of a baccalaureate degree in h ealth p rofessions She was accepted into the dental program at the University of Florida in Ga inesville in 2005 and earned a d octorate of d ental m edicine s degree in 2009. Mo st recently, she was awarded a m aster of s cience s degree by the University of Florida Orthodontics program in the May of 2012.