1
PARENTS’ PERSPECTIVES 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 inspiring me to reach for the stars, and to my
husband, Paul Ellis, for 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 Page
ACKNOWLEDGMENTS .................................................................................................. 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 Page 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 Page 2-1 Example of cleft lip patient set of images from picture booklet ........................... 16
2-2 Example of non-cleft lip patient set of images from picture booklet .................... 16
3-1 Mean ratings of unilateral cleft lip patients .......................................................... 22
3-2 Mean ratings of non-cleft lip patients .................................................................. 22
3-3 Average ratings versus gold standard for unilateral cleft lip patients 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
PARENTS’ PERSPECTIVES OF REPAIRED CLEFT LIP
By
Dafne 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 structures 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 CLP 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 facial 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 assessing 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. Conclusions: 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
Cleft 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 occur 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 orofacial 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 treatment occasionally involving
orthognathic surgery with or without rhinoplasty is completed when patients reach
adolescence.8,9
The goals 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
continuous and functional orbicularis oris muscle, while being wary of the procedure’s
effect on the tip of the nose and attempting to camouflage the surgical scar.10 However,
repairing a unilateral cleft lip and palate 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 disturbances 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 important 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 abnormal 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 method 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 familiar 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 which 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 esthetic 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 affected 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 have been addressed in literature.15,16
These limitations include 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 be 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 those 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 with 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-cleft lip parents’ perception. A better
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 treatment 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 outcomes, 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 of 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 patient 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 patients, 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
patients 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 photographs 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 which was designated as follows: 1: ‘very
good’, 2: ‘good’, 3: ‘fair’, 4: ‘poor’; and 5: ‘very poor’. This index was chosen over a
Visual Analogue Scale, or VAS, even though both methods have comparable
responsiveness and validity in a randomized trial setting; 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 living 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 consisted 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
child’s orthodontic appointment. The second group (n=30), the control group, was
comprised of parents of children with no history of CLP. This group was selected from
volunteers that were matched to experimental participants according to their children’s
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 informed 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
each 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 statistic
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 reliability 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 statistic
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 differences 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-sample 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 lip patient set of images from picture booklet.
Figure 2-2. Example of non-cleft lip 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
Hispanics. 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
participants 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)
Cohen’s Kappa coefficient was calculated. (Table 3-3) Overall, the raters were
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
yielded a score of 0.40, and a weighted Kappa of 0.56, which indicated a “moderate”
agreement.
18
For the 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 participant
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 the 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 cleft lip, the experimental
group rating was lower than the control group. This was also true for photographs of
only the upper lip but not for 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, with 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 significant. 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 comparison.
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=0.69
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 and second evaluation.
Score 2
Frequency 1 2 3 4 5 Total
Percent
Sc
ore
1
1 134 66 17 6 3 226
18.8*
9.3** 2.4 0.8 0.4 31.7
2 33 148 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 172 262 163 69 46 712
24.2 36.8 22.9 9.7 6.5** 100.0
(*). Percentage of correct agreement (**). Percentage of agreement within one value to either side
Table 3-4. Pearson correlation 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 ratings of unilateral cleft lip patients. Assessments were made using a five point scale. 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 patients. Assessments were 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 gold standard for unilateral cleft lip patients and non-cleft lip patients. (P< 0.05)
24
CHAPTER 4 DISCUSSION
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 team,
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 children.
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 This 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
circumstances influence one’s personal perception of others and one’s surroundings 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 are 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. In 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 in 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 significant difference
between parent and patient-rated satisfaction regarding lip appearance.26 Patient
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 than ‘very
pleased’ with the appearance of the lip suggesting that those ratings were realistic
appraisals of treatment outcome and appearance.
In addition, a questionnaire survey in 1991, Noar found that the parents were
correct in believing that they were 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 directly 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 parents 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
for the professional’s lower perceived need for further surgery may 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 for 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 public’s perception. Namely, the perception of a person is
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 discovered 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 lay 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 validity 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 that parents in the experimental group are
merely content with a lip closure and disregard facial esthetics.
When comparing the parents’ ratings with the gold standard rating it was found
that the parents were more critical in their ratings. This was true with 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 CONCLUSION
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
groups had similar mean ratings. The upper lip was rated by both groups as ‘fair.’
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 interventions.
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 Defects 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 Orofacial 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 Delaire’s cheilorhinoplasty: Unilateral cleft aesthetic outcome scored according to the EUROCLEFT guidelines. Int J Pediatr 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: 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 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, Brattström V, Dahl E, McWilliam J, Mølsted K, Plint DA, Prahl-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. Functional 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 nasolabial 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. Jaeschke R, Singer J, Guyatt GH. A comparison of seven-point and visual analogue scales. Controlled 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 functioning 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.
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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 ratings 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 esthetic, 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 adults 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.
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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. Michael Ellis. She subsequently
matriculated to the University of Arkansas in Little Rock and earned Magna Cum Laude
upon completion of a baccalaureate degree in health professions. She was accepted
into the dental program at the University of Florida in Gainesville in 2005 and earned a
doctorate of dental medicine’s degree in 2009. Most recently, she was awarded a
master of science’s degree by the University of Florida Orthodontics program in the May
of 2012.