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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
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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

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© 2012 Dafne Ellis

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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

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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.

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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

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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

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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

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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.

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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,

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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

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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

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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.

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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,

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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

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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.

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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.

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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.

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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

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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.

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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

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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

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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)

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Figure 3-3. Average ratings versus gold standard for unilateral cleft lip patients and non-cleft lip patients. (P< 0.05)

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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.

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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

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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.

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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.

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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.

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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.

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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.

Page 31: PARENTS’ PERSPECTIVES OF REPAIRED CLEFT LIPufdcimages.uflib.ufl.edu/UF/E0/04/41/42/00001/ELLIS_D.pdf · Although cleft lip and cleft palate often occur together, they can occur

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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.


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