Sex Differences in Perceived Stigmatization, Body Image Disturbance, and Satisfaction with Facial Appearance and Speech Among Adolescents with Craniofacial Conditions

Abstract

Youth with craniofacial conditions often have appearance and speech differences and are vulnerable to social stigmatization and body image disturbances. Given sociocultural pressures for female attractiveness, adolescent girls with craniofacial conditions may be especially vulnerable to body dissatisfaction and appearance-related social stigmatization, though such sex differences have been infrequently studied in this population. This study aimed to: (1) examine sex differences in body image disturbance, satisfaction with speech and facial appearance, and perceived stigmatization among adolescents with craniofacial conditions; and (2) evaluate whether stigmatization perceptions are predictive of body image disturbance and satisfaction with facial appearance and speech. Using a cross-sectional design, 110 adolescents from two craniofacial centers completed measures of body image disturbance, satisfaction with speech and facial appearance, and perceived stigmatization. Females reported significantly greater levels of body image disturbance and lower satisfaction with facial appearance compared to males. There were no significant sex differences for satisfaction with speech or perceived stigmatization. Perceived stigmatization was a significant predictor of body image disturbance, and satisfaction with facial appearance and speech while controlling for sex, body mass index, and age. Interventions to prevent and/or address body image and stigmatization concerns are clinically indicated for both sexes.

1. Introduction

Craniofacial conditions including cleft lip and palate are relatively common congenital anomalies characterized by facial bone and soft tissue deficiencies that can impact a number of facial functions including speech, hearing, feeding, and in some instances, breathing (Buchanan et al., 2014). Dentition can also be affected due to missing or misaligned teeth and dental malocclusion. Staged reconstructive procedures, typically beginning in infancy and ending in adolescence or young adulthood, are often performed with the goal of restoring facial function and improving facial appearance. Facial appearance differences are common across craniofacial conditions and are due to the nature of the congenital soft tissue and bone deficiencies as well as scarring from corrective surgeries (Losee & Kirschner, 2015).

The functional and aesthetic problems associated with craniofacial conditions can significantly affect the psychosocial functioning of individuals. For example, risks for anxiety, depression, reduced quality of life, and low-self-esteem have been documented in craniofacial populations (Feragen & Stock, 2017). These psychosocial problems are thought to be related, in part, to the experience of living with facial appearance and/or speech differences (Broder, Smith, & Strauss, 2001; Rumsey & Harcourt, 2007; Watterson, Mancini, Brancamp, & Lewis, 2013). Having a facial or speech difference can negatively affect self-perceptions including body image (Crerand, Sarwer, Kazak, Clarke, & Rumsey, 2017; Langlois, Kalakanis, Larson, Hallam, & Smoot, 2000; Shapiro, Waljee, Ranganathan, Buchman, & Warschausky, 2015). Among youth with craniofacial conditions, some studies have found high rates of dissatisfaction with facial appearance (Hunt, Burden, Hepper, & Johnston, 2005; Noar, 1991; Thomas, Turner, Rumsey, Dowell, & Sandy, 1997); others have found evidence of fewer appearance concerns (Slifer et al., 2003).

These disparate findings likely reflect methodological limitations, including a reliance on general measures of appearance satisfaction or unidimensional assessments of body image, which may not capture the type and impact of body image concerns experienced by youth with craniofacial conditions. Further, as Feragen and Stock (2018) observed, exceedingly limited research in the craniofacial field has been grounded in theory or used appropriate models to understand body image in this population, which likely contributes to inconsistent findings. In an effort to address such limitations, they advocate for using Cash’s cognitive behavioral model of body image development (Cash, 2011) as a framework for understanding body image and potential factors that may impact appearance evaluations in craniofacial populations (Feragen & Stock, 2018). A recent study of adolescents with craniofacial conditions and a comparison group of non-affected youth, which was informed by Cash’s model and assessed multiple dimensions of body image, found that youth with craniofacial conditions reported significantly more concerns about facial features; however, there were no significant differences between groups with respect to body image or overall satisfaction with appearance and quality of life (Crerand et al., 2017).

In Cash’s model, cultural socialization is an important and relevant predisposing factor that can impact body image, including cultural norms and ideals for appearance (Cash, 2011). Among the general population, females are particularly vulnerable to body image dissatisfaction because they are typically socialized at a young age to value and prioritize their physical appearance over other characteristics (Jones, Vigfusdottir, & Lee, 2004; Murnen, 2011; Striegel-Moore & Franko, 2002). Sociocultural messages about the importance of attractiveness are often internalized and can affect self-perceptions about appearance, particularly during adolescence (Levine & Chapman, 2011; Levine & Smolak, 2002) when physical changes and increased interest in dating and fitting in with peers may contribute to self-consciousness.

Given these known sociocultural factors, girls with craniofacial conditions are often thought to be more vulnerable to appearance concerns. Recent studies (e.g., Crerand et al., 2017; Feragen & Stock, 2016; Shapiro et al., 2015) report greater dissatisfaction with facial appearance among girls; dissatisfaction with facial appearance was associated with greater symptoms of depression (Feragen & Stock, 2016; Shapiro et al., 2015) as well as anxiety and lower quality of peer relationships only for girls (Shapiro et al., 2015). Crerand et al. (2017) observed that females with craniofacial conditions reported higher levels of appearance investment (or the degree to which appearance is important to their self-concept), which may place them at increased risk for body image disturbances, facial appearance dissatisfaction, and more general psychosocial problems (Cash, 2011; Cash, Phillips, Santos, & Hrabosky, 2004; Pruzinsky, 2002). An understanding of factors that increase risk for poor psychosocial functioning in youth with craniofacial conditions is needed in order to better target screening, prevention, and intervention for this patient population.

Commensurate with Cash’s model (Cash, 2011), negative interpersonal experiences (e.g., teasing) constitute other important predisposing factors that contribute to the development of body image problems (e.g., Cash, 1995; Cash et al., 2004; Feragen & Stock, 2016; Weingarden, Curley, Renshaw, & Wilhelm, 2017). Facial appearance and/or speech differences such as articulation disorders and velopharyngeal dysfunction are known to negatively impact speech intelligibility and consequently, social interactions (Rumsey & Harcourt, 2007; Watterson et al., 2013). High rates of teasing, bullying, and other forms of social stigmatization such as staring and avoidance have been reported in craniofacial populations (Feragen & Stock, 2016; Rumsey & Harcourt, 2007; Strauss et al., 2007), with females reporting greater stigmatization than males in at least two studies (Feragen & Stock, 2016; Strauss et al., 2007).

However, as with studies of body image in craniofacial populations, few studies have utilized instruments that are sensitive to the range of stigmatizing behaviors commonly experienced by persons with visible appearance differences beyond overt bullying or teasing. Similarly, few investigations have examined relationships between perceived stigmatization and body image disturbance in youth with craniofacial conditions, or potential sex differences in these relationships. Finally, studies have largely neglected to assess perceptions of functional aspects of craniofacial conditions that may impact body image and social stigmatization, such as satisfaction with speech. Pruzinsky (2004) argued that studies of body image in persons with medical conditions should incorporate not only perceptions of physical appearance but also bodily function, and speech is arguably one of the most significant aspects of physical functioning affected by craniofacial conditions.

In an effort to address some of the limitations of the extant literature as described above, the present study was informed by Cash’s cognitive behavioral model of body image development with the goal of examining sex differences in body image disturbance, satisfaction with facial appearance and speech, and perceptions of stigmatization among adolescents with craniofacial conditions, and utilized measures that are sensitive to the concerns of youth with facial differences. Owing to the sociocultural emphasis placed on physical appearance and attractiveness for females, female adolescents with craniofacial conditions were anticipated to report more body parts of concern, greater levels of body image disturbance, lower satisfaction with facial appearance and speech, and greater perceived stigmatization compared to males. Perceived stigmatization was hypothesized to be predictive of greater body image disturbance and lower satisfaction with speech and appearance.

2. Method

2.1. Participants

Participants were recruited from two pediatric hospital-based craniofacial centers in the U.S. At Site 1, 97 adolescents were approached, 76 of whom provided usable data (78%). The remaining 21 adolescents either did not return packets (n = 17) or had incomplete data (n = 4). At Site 2, 59 youth were identified as being potentially eligible for the study. Two families actively declined participation, and another 15 were contacted but never returned calls about the project (passive decline). Forty-two families consented (71%), and 34 had usable data (81%).

In order to be included in the study, youth at both sites had to be between 14 and 18 years of age, have a diagnosis of cleft lip and palate or another congenital craniofacial condition characterized by visible appearance differences (e.g., craniofacial microsomia), and have a history of craniofacial-related reconstructive surgery. Youth were excluded if they had a comorbid cognitive impairment (e.g., intellectual disability, developmental delay or disorder such as autism), if they were unable to read or understand English, or if they had undergone surgery within the last three months prior to study enrollment (to account for post-operative appearance changes such as swelling which could inflate appearance concerns).

2.2. Procedure

At Site 1, recruitment procedures entailed sending adolescents and their parents a letter describing the study (signed by the adolescent’s attending surgeon and the principal investigator) in advance of in-person office visits, and/or approaching families directly during the adolescent’s scheduled visit. After eligibility was confirmed and informed consent and assent were obtained by research assistants, participants received questionnaires to complete during their office visit and were provided with stamped return envelopes if they did not finish before the end of the visit.

At Site 2, eligible adolescents were mailed a letter from the principal investigator and their attending surgeon describing the study and giving the family the option to opt out of further contact by leaving a voicemail for the study team. A member of the research team then called potential participants who did not opt out, screened them for eligibility, and reviewed study procedures. Eligible, consenting participants then completed questionnaires.

Reminder telephone calls and letters were used to prompt enrolled adolescents who had not returned questionnaires within two weeks. Participants received compensation ($25 U.S. dollars) after returning their completed questionnaires. Information about participants’ medical and surgical histories were abstracted from medical charts by trained research assistants.

Institutional review board approval was obtained at both pediatric hospitals. Consent was obtained from parents or legal guardians of youth 14-17 years, and all adolescents provided assent (or consent if 18 years old).

2.3. Measures

2.3.1. Appearance-related distress.

The Derriford Appearance Scale (DAS) is a 59-item instrument that measures appearance-related distress and problems (Carr, Harris, & James, 2000; Harris, Carr, & Moss, 2004). Respondents are asked to indicate how often they feel or behave in response to their appearance concern (e.g., “Iam self-conscious about my feature.”) using a response scale (e.g., 1= almost never; 5 = almost always). The DAS has been used to assess appearance concerns in adolescents and adults in plastic surgery and community settings. Higher scores indicate more appearance-related distress and difficulties. Scores on the DAS have demonstrated good psychometric properties including convergent, divergent, and construct validity as well as good test-retest reliability and internal consistency (Harris, Carr, & Moss, 2004). Internal consistency for this measure was high (α = .94).

2.3.2. Body image disturbance.

The Body Image Disturbance Questionnaire (BIDQ) assesses symptoms of body image distress and related impairment in functioning (Cash et al., 2004). Seven quantitative items evaluate: (1) concern about body part(s) felt to be unattractive; (2) preoccupation with the concern(s); (3) experiences of emotional distress about appearance; (4) impairment in social, occupational, or other areas of functioning; (5) interference with social life, school, job, or role functioning; (6) avoidance of activities because of appearance; and (7) behavioral avoidance (Cash et al., 2004). Quantitative items are rated on a scale from 1-5 (e.g., 1 = not at all concerned; 5 = extremely concerned) and averaged to provide a total score. Higher scores are indicative of greater symptoms of body image disturbance; clinical levels of body image disturbance are defined as mean scores ≥ 3 on this instrument. Items 1, 2, 5, 6, and 7 have corresponding qualitative questions which permit respondents to describe their specific appearance concerns and their impact on distress levels and/or functioning. For this study, qualitative responses to Item 1 regarding specific concerns about body parts thought to be unattractive were tabulated.

Scores on the BIDQ have demonstrated good psychometric properties including convergent validity with other body image measures (Cash et al., 2004) and reliability (Cash & Grasso, 2005). Internal consistency for this measure in the present sample was acceptable (α = .85).

2.3.3. Perceived stigmatization.

The Perceived Stigmatization Questionnaire (PSQ) is a 21-item questionnaire which is used to assess stigmatizing behavior (e.g., staring, teasing, avoidance) commonly experienced by youth with disfigurements due to burns or other conditions (Lawrence, Rosenberg, Rimmer, Thombs, & Fauerbach, 2010). Participants are asked to rate how often they experienced a behavior on a 5-point scale (never, almost never, sometimes, often, always). The measure yields a total score as well as three subscales: (1) Absence of Friendly Behavior (e.g., “People I don’t know say hi to me”); (2) Confused/Staring Behavior (“People don’t know what to say to me”); (3) Hostile Behavior (e.g., “People call me names”). Internal consistency for the total score in the current sample was .90. Scores on the PSQ have demonstrated good construct validity in youth (Lawrence, Rosenberg, Rimmer, Thombs, & Fauerbach, 2010) and convergent and divergent validity in adults (Lawrence, Fauerbach, Heinberg, Doctor, & Thombs, 2006).

Perceived stigmatization was also assessed by a subscale on the Youth Quality of Life Instrument-Facial Difference Module (YQOL-FD). This measure assesses quality of life in adolescents with facial differences (Patrick et al., 2007). It consists of 48 items that evaluate domains of quality of life. Its scores have demonstrated good internal consistency and convergent validity with other measures of quality of life (Patrick et al., 2007). For this study, the 10-item Stigma subscale (e.g., “Because of how my face looks, people think I am stupid”) was used. Scores for this subscale range from 0 to 100 with higher scores reflecting lower quality of life. Internal consistency for the Stigma subscale in the present sample was high (α = .93).

2.3.4. Appearance and speech satisfaction.

Developed by the UK Cleft Psychology Special Interest Group, the Satisfaction with Appearance Scale (SWA) is comprised of 20 items that evaluate satisfaction with aspects of facial appearance (e.g., nose, eyes, overall facial appearance) and other physical factors that may be impacted by craniofacial conditions (e.g., speech) using a rating scale ranging from 0 (very unhappy dissatisfied) to 10 (very happy satisfied) (Emerson, Spencer-Bowdage, & Bates, 2004). Twelve items specifically focus on facial features, and the mean of these items is used to calculate the total score. One item assesses satisfaction with speech (“How happy are you with your speech?”). Evidence of psychometric properties for this measure including internal consistency and construct validity have been reported (Emerson et al., 2004). Internal consistency for this measure in the current sample was high (α = .92).

2.4. Statistical Analyses

REDCap electronic data capture tools (Harris et al., 2009) were used to collect and maintain all study data. Descriptive statistics (means, frequencies) were utilized for demographic and clinical characteristics, outcome measures, and for the BIDQ Item 1 qualitative responses regarding specific appearance concerns. Percentages of adolescents who reported clinical levels of body image disturbance as measured by the BIDQ were calculated by dividing the sample into low and high BIDQ scores (low: scores ≤ 3; high: scores > 3) in line with recommendations by Cash et al. (2004). Chi-square tests were then used to test for differences by sex for dichotomized BIDQ scores and for body parts of concern derived from BIDQ Item 1.

To evaluate whether there were sex differences on BIDQ (continuous total score) and the other outcome measures, regression analyses were conducted controlling for age and body mass index (BMI). For the second hypothesis, regression analyses were conducted such that stigma variables (PSQ total score and YQOL-FD Stigma subscale) were entered as predictors of BIDQ, SWA, and DAS total scores while controlling for sex, age, and BMI. Initial examination of the outcome variables of interest showed that some of these variables were substantially nonnormally distributed before and after variable transformations were performed. Consequently, two-tailed tests of significance (p-values) and 95% confidence intervals were calculated for the parameters of interest based on two approaches: (a) the standard approach to statistical inference in linear regression and (b) a non-parametric bootstrap approach based on 100,000 bootstrap samples. Results were similar across both approaches (e.g., all the reported p-values were within .01), therefore only the results from the standard approach to statistical inference for linear regression are reported here. A p-value < .05 was considered statistically significant. Statistical analyses were performed using SAS 9.4.

3. Results

As shown in Table 1, there were no significant differences between males and females on any demographic or clinical characteristics. The average age for youth in this sample was 15.56 ± 1.42 years, with 54% identifying as male. Most of the sample identified as Non-Hispanic and Caucasian. With respect to craniofacial diagnosis, 82% of the sample had a history of combined cleft lip and palate.

Table 1.

Demographic and Clinical Characteristics for the Total Sample and by Sex

Variable	Total (n = 110) M ± SD	Males (n = 59) M ± SD	Females (n = 51) M ± SD	p
Age (years)	15.66 ± 1.42	15.61 ± 1.47	15.73 ± 1.36	.67
BMI (kg/m2)	21.91 ± 4.72	21.48 ± 3.93	22.41 ± 5.49	.31
	n (%)	n (%)	n (%)	p
Ethnicity
Hispanic	8 (7.3)	3 (5.1)	5 (9.8)	.34
Not Hispanic	63 (92.7)	56 (94.9)	46 (90.2)
Race
Asian	9 (8.2)	8 (13.6)	1 (2)	.26
African American/Black	9(8.2)	2 (3.4)	7 (13.7)
Caucasian/White	84 (76.4)	47 (79.7)	37 (72.5)
American Indian/Alaskan Native	1 (0.9)	0 (0)	1 (0.9)
More than one race	3 (2.7)	1 (1.7)	2 (3.9)
Unknown/not reported	4 (3.6)	1 (1.7)	3 (5.9)
Diagnosis				.92
Cleft lip and palate	91 (82.7)	49 (83.1)	42 (82.4)
Other craniofacial condition	19 (17.3)	10 (16.9)	9 (17.6)

3.1. Body image disturbance and satisfaction with facial appearance and speech.

Table 2 presents descriptive data for males and females with respect to identified appearance concerns as assessed by the BIDQ (Item 1 qualitative and quantitative questions). For the entire sample, the most commonly reported areas of concern were the nose (23.6%), lips (15.5%), and face (10%). For females, the most common features of concern were the nose (35.3%), lips (17.6%), and stomach (15.7%); for males, concerns about the nose (13.6%) and lips (13.6%) were most common, followed by overall concerns with facial appearance (8.5%) (see Table 2). On the quantitative part of Item 1 on the BIDQ, which asks whether the respondent is concerned about the appearance of some part(s) of his/her body which is considered especially unattractive, 59.3% of males endorsed being “not at all concerned” in comparison to only 13.7% of females (p < .01). Females were significantly more likely than males to report any facial appearance concern (56.9% vs. 27.1%, p < .01), or any type of body appearance concern (43.1% vs. 11.9%, p < .01). Nine percent of the total sample (n = 10) met criteria for clinically significant body image disturbance on the BIDQ (score of 3 or higher). While more females than males fell into the clinical range on the BIDQ (7 females vs. 3 males), this difference was not statistically significant (p = .18).

Table 2.

Appearance Concerns in Male and Female Adolescents

Measure - BIDQ	Male Adolescents (n = 59)		Female Adolescents (n = 51)		Overall (N = 110)
	n	%	n	%	n	%
Concern about Unattractive Body Parts
Not at All Concerned	35	59.3	7	13.7	42	38.2
Somewhat Concerned	7	11.9	19	37.3	26	23.6
Moderately Concerned	9	15.3	15	29.4	24	21.8
Very Concerned	3	5.1	8	15.7	11	10.0
Extremely Concerned	3	5.1	2	3.9	5	4.5
Concerns Endorsed
Face	5	8.5	6	11.8	11	10.0
Profile	1	1.7	2	3.9	3	2.7
Weight	1	1.7	6	11.8	7	6.4
Hair	0	0.0	0	0.0	0	0.0
Forehead	1	1.7	0	0.0	1	0.9
Eyes	2	3.4	2	3.9	4	3.6
Ears	1	1.7	0	0.0	1	0.9
Nose	8	13.6	18	35.3	26	23.6
Cheeks	0	0.0	1	2.0	1	0.9
Skin	0	0.0	0	0.0	0	0.0
Lips	8	13.6	9	17.6	17	15.5
Teeth	1	1.7	4	7.8	5	4.5
Jaw	1	1.7	1	2.0	2	1.8
Scars	3	5.1	4	7.8	7	6.4
Chin	1	1.7	1	2.0	2	1.8
Chest	2	3.4	1	2.0	3	2.7
Arms	1	1.7	1	2.0	2	1.8
Stomach	2	3.4	8	15.7	10	9.1
Hips	0	0.0	4	7.8	4	3.6
Lower Back	0	0.0	1	2.0	1	0.9
Butt	0	0.0	1	2.0	1	0.9
Thighs/Legs	0	0.0	7	13.7	7	6.4
Feet	1	1.7	0	0.0	1	0.9
Nonspecific Concern	3	5.1	3	5.9	6	5.5

Table 3 presents means and standard deviations for the BIDQ, SWA, and DAS total scores for the total sample and stratified by sex. Regression analyses (see Table 3) indicated that sex was a significant predictor of BIDQ (Δ = 0.48, 95% CI: [0.22, 0.74], p < .01), DAS (Δ = 15.47, 95% CI: [3.62, 27.32], p < .01), and SWA (Δ = −0.78, 95% CI: [−1.47, −0.09], p = .03) total scores while controlling for age and BMI. Females reported significantly higher levels of body image disturbance on both the BIDQ and DAS, as well as significantly lower satisfaction with facial appearance on the SWA compared to their male counterparts. Satisfaction with speech was not significantly different for males and females.

Table 3.

Body Image and Stigma Measure Raw Means and Standard Deviations for the Total Sample and by Sex and Results for Adjusted Sex Mean Differences Controlling for BMI

	M(SD)			Adjusted Sex Mean Difference
Scale	Total Sample	Female (n = 51)	Male (n = 59)	ΔA,Sex	95% CI	p	ΔR2
BIDQ
Total Score	1.73 (0.71)	1.98 (0.69)	1.52 (0.67)	0.48	.22, .74	<.01	.11
SWA
Total Score	7.14 (1.85)	6.74 (1.84)	7.47 (1.80)	−0.79	−1.49, −.10	.03	.05
Satisfaction With Speech	7.15 (2.67)	6.88 (2.80)	7.39 (2.55)	−0.59	−1.60, .42	.25	.01
DAS
Total Score	91.86 (31.94)	99.78 (33.16)	85.02 (29.43)	15.60	3.77, 27.44	.01	.06
YQOL-FD
Stigma	26.26 (25.01)	28.96 (23.66)	23.85 (26.13)	6.02	−3.61, 15.65	.22	.01
PSQ
Total Score	1.92 (0.57)	1.92 (0.59)	1.91 (0.55)	0.01	−.21, .23	.91	.00
Absence Friendly Behavior	2.25 (0.65)	2.28 (0.65)	2.22 (0.66)	0.06	−.20, .31	.66	.00
Confused Starting Behavior	1.70 (0.69)	1.63 (0.67)	1.75 (0.71)	−0.12	−.38, .15	.38	.01
Hostile Behavior	1.74 (0.85)	1.82 (0.95)	1.66 (0.76)	0.15	−.18, .48	.36	.01

Note: BIDQ = Body Image Disturbance Questionnaire; SWA = Satisfaction with Appearance Scale; DAS = Derriford Appearance Scale; YQOL-FD = Youth Quality of Life Instrument-Facial Difference Module; PSQ = Perceived Stigmatization Questionnaire

3.2. Perceived stigmatization.

Table 3 presents means and standard deviations for the stigma measures for the whole sample and stratified by sex. There were no significant differences for sex on any of the stigma measures (YQOL Stigma or PSQ total score or subscales) before or after controlling for age and BMI (see Table 3).

3.3. Stigmatization as a predictor of body image disturbance and satisfaction with facial appearance and speech.

Finally, regression analyses were used to examine whether perceived stigmatization (as measured by the PSQ total score and YQOL-FD Stigma subscale) was predictive of body image disturbance, satisfaction with facial appearance, and satisfaction with speech while controlling for BMI, age, and sex. Specifically, we examined the relationships between the BIDQ, DAS, SWA total and satisfaction with speech scores with each of the PSQ total score and YQOL-FD Stigma measures of stigma separately while controlling for sex, age, and BMI. All eight of these analyses yielded statistically significant positive relationships (all ps < .01) between body image disturbance, satisfaction with facial appearance and speech, and stigma (see Table 4 for corresponding effect sizes and confidence intervals), indicating that perceived stigmatization was a significant predictor of body image disturbance, satisfaction with facial appearance, and speech.

Table 4.

Regression Analyses for Stigmatization as a Predictor of Satisfaction with Speech and Appearance, Body Image Disturbance^†

	PSQ Total Score as Predictor
Scale	b	95% CI	p	ΔR2
BIDQ
Total Score	0.70	0.51, 0.88	<.01	.31
DAS
Total Score	27.83	18.98, 36.67	<.01	.25
SWA
Total Score	−1.81	−2.30, −1.31	<.01	.31
Satisfaction with Speech	−1.47	−2.31, −0.64	<.01	.10
	YQOL-FD Stigma as Predictor
Scale	b	95% CI	p	ΔR2
BIDQ
Total Score	0.017	0.013, 0.021	<.01	.35
DAS
Total Score	0.75	0.56, 0.95	<.01	.33
SWA
Total Score	−0.047	−0.058, −0.037	<.01	.40
Satisfaction with	−0.045	−0.064, −0.027	<.01	.17

Note: BIDQ = Body Image Disturbance Questionnaire; SWA = Satisfaction with Appearance Scale; DAS = Derriford Appearance Scale; YQOL-FD = Youth Quality of Life Instrument-Facial Difference Module; PSQ = Perceived Stigmatization Questionnaire

^†Statistical Inference and Regression Coefficients for Two Measures of Stigma (PSQ Total Score or YQOL-FD Stigma). Stigma measures are entered into separate regression models. Regression coefficients are adjusted for sex and BMI.

4. Discussion

This study evaluated sex differences in body image disturbance, satisfaction with facial appearance and speech, and perceived stigmatization in adolescents with craniofacial conditions. In line with the hypotheses, adolescent girls reported significantly greater levels of dissatisfaction with facial appearance and body image disturbance compared to males. Adolescent girls were significantly more likely to have any type of appearance concern and specifically, concerns about facial appearance, but also about other body parts as well (e.g., stomach, thighs). Males also reported concerns about facial appearance but were significantly less likely to report any appearance concerns (facial or other body parts). These sex differences could reflect differences in appearance investment which tends to be higher in females (Crerand et al., 2017; Moss, Lawson, White, & the Appearance Research Collaboration, 2014), or the influence of sociocultural pressures for female attractiveness which are difficult to avoid particularly with the popularity of social media (Fardouly & Vartanian, 2016). Girls with craniofacial conditions are not just vulnerable to facial appearance concerns, but also more “normative” concerns about weight and body shape, which may make them more vulnerable overall to body image disturbance (Crerand et al., 2017).

Similar sex differences with respect to appearance concerns have also been found in studies of youth with craniofacial conditions and other differences requiring reconstructive surgery (Feragen & Borge, 2010; Simis, Verhulst, & Koot, 2001). For example, Shapiro et al. (2015) reported that girls in their sample reported lower overall satisfaction with facial appearance compared to boys. These findings highlight the need to monitor body image concerns and potential impacts on psychosocial functioning among youth with craniofacial conditions, particularly among girls.

Both males and females reported concerns about nasal and lip appearance, consistent with other reports of specific appearance concerns in the craniofacial literature (e.g., Hunt et al., 2005). These findings could reflect the fact that the majority of youth in this sample had cleft lip and palate, a diagnosis that commonly impacts lip and nasal appearance, and these features frequently require additional surgical revision particularly during adolescence and/or young adulthood.

Nine percent of youth had BIDQ scores in the clinical range, suggesting that they are experiencing clinically significant levels of distress and impairment in functioning related to their appearance concerns. Although there were no significant differences between sexes, more females than males had scores within this range. These findings are consistent with other studies that have evaluated rates of body image disturbance in adults seeking reconstructive surgery due to craniofacial or other congenital or acquired appearance differences (Crerand et al., 2004; Sarwer et al., 1999). Clinically, it is important to ask patients about how their appearance concerns are impacting both their emotional and daily functioning (e.g., how upset or anxious are they about their appearance concerns? Are they avoiding social activities or school because of appearance concerns?). The BIDQ is a brief, easily scored instrument which could be integrated into team assessments in order to better understand the nature and impact of appearance concerns in youth with craniofacial conditions and may help identify youth who would benefit from further psychological evaluation and treatment. Cognitive-behavioral therapy may be indicated to improve body image concerns for some youth regardless of whether they are candidates for additional craniofacial-related surgery (Clarke, Thompson, Jenkinson, Rumsey, & Newell, 2014).

In this study, no significant sex differences were noted in satisfaction with speech. However, perceived stigmatization was a significant predictor of speech dissatisfaction. These findings add to the growing body of research evaluating perceptions of speech in relationship to psychosocial outcomes in craniofacial populations. Child self-assessments of speech satisfaction have been found to correlate with psychosocial adjustment (Feragen, Stock, & Kvalem, 2015) and quality of life (Bickham et al. 2017). Our findings highlight speech-related psychosocial risks in craniofacial populations and underscore the need for screening and monitoring in clinical settings. Future studies that incorporate objective as well as subjective speech assessments are needed.

Contrary to hypotheses, there were no significant differences between males and females with respect to perceptions of stigmatization. Masnari et al. (2012) found no evidence of sex differences for perceived stigmatization in a sample of youth with congenital or acquired disfigurements; although in their sample, parents completed the PSQ, which may have biased their assessment. However, in the craniofacial literature, sex differences have been observed. For example, Feragen and Borge (2010) observed greater peer harassment in Norwegian adolescent females with cleft lip and/or palate compared to males. Strauss et al. (2007) also reported that females with congenital or acquired facial differences were more likely than males to report stigma experiences using the YQOL-FD, and they observed concordance between maternal and self-reports of stigma. Sex differences could be due to females being more willing to acknowledge stigma experiences and their resulting impact on emotional well-being and perceptions of appearance (Strauss et al., 2007). Reasons for the lack of difference observed in this sample are unknown but suggest that stigmatization was a concern for both males and females and highlight the need for ongoing assessment of stigmatization experiences for both sexes in clinical settings.

Importantly, perceived stigmatization was predictive of body image disturbance and dissatisfaction with speech and facial appearance for both males and females, underscoring that stigma experiences can be detrimental for body image among both sexes. Stigmatization has also been found to be predictive of internalizing problems and health-related quality of life in youth with acquired and congenital facial disfigurements (Masnari et al., 2013). In the craniofacial literature, studies have documented associations between teasing and lower appearance satisfaction (e.g., Feragen & Stock, 2018; Hunt, Burden, Hepper, Stevenson, & Johnston, 2006), as well as social withdrawal and anxiety (Endriga & Kapp-Simon, 1999; Hunt et al., 2006).

These findings highlight the need for prevention and intervention approaches to address risks for social stigmatization and body image disturbances in youth with craniofacial conditions. Given that questions from peers about a speech or appearance difference can be easily interpreted as teasing, these interventions should include education about normative curiosity about differences and assist youth with differentiating curiosity from cruelty (Feragen & Borge, 2010; Feragen, Borge, & Rumsey, 2009). Teaching youth to be prepared with brief explanations about their differences that can be delivered in a matter-of-fact way may help ease social interactions and increase confidence about handling these types of situations. For example, “Explain-Reassure-Distract/Deflect” is a helpful tactic developed by Changing Faces (https://www.changingfaces.org.uk/), a United Kingdom-based charity dedicated to supporting individuals with appearance differences due to any cause. Strategies for managing staring should also be provided, including smiling and making eye contact with the person who is staring, as well as learning assertiveness skills to confront individuals more directly. Youth and their caregivers should also be counseled from an early age about teasing and bullying and effective strategies for addressing these concerns.

It is also important to consider societal-level interventions to support acceptance of facial differences and reduce stigmatization. A recent scoping review found that stigmatizing responses appear to be more common among individuals with limited exposure to people with visible differences (Jewett, Gumuchian, Pepin, Rice, et al., 2018). Thus, public awareness campaigns and advocacy efforts, such as those championed by Changing Faces in the United Kingdom, may help increase positive portrayals of persons with visible differences in the media and reduce negative attitudes and discriminatory behaviors towards those with craniofacial and other types of differences.

Similarly, prevention and intervention strategies are also indicated for body image disturbances. Care should be taken about how differences are discussed with youth from an early age including use of neutral, non-stigmatizing language (e.g., using name of the craniofacial condition rather than “birth defect” or “anomaly”) to describe differences both at home and in clinical settings. Furthermore, families should be counseled about strategies for supporting healthy body image development from an early age. For example, caregivers should be mindful of how they talk about their own physical appearance as well as comments about the physical attributes of others (e.g., limit “fat talk” and criticisms about appearance). Strategies to enhance appreciation of one’s body including both aesthetic and functional attributes can also be helpful. Finally, for youth with body image disturbance, cognitive-behavioral strategies can also be effective in addressing appearance-related distress and impairment in daily functioning (Clarke et al., 2014; Harcourt et al., 2018). In the U.K., an online program called YP Face IT has shown efficacy in improving appearance and related social concerns in youth with a range of disfiguring conditions (Williamson, Hamlet, & White, 2016). However, this program is not yet available in the U.S., nor has it been evaluated specifically for craniofacial populations. Given the known risks for social stigmatization and body image concerns in youth with craniofacial conditions, it is imperative that these interventions be incorporated into routine clinical practice. Addressing and/or preventing these risks could ultimately reduce risks for depression and anxiety and improve quality of life. Support for health professionals who provide appearance-related psychosocial care to children and adolescents with visible differences (e.g., prioritizing the development of appearance-related support and intervention, increasing appearance-related knowledge) is imperative (Gee, Maskell, Newcombe, Kimble, & Williamson, 2019).

While the cross-sectional design of this study precludes making statements about causal relationships between stigmatization and body image disturbance, these findings nonetheless suggest that stigmatization experiences may contribute to risk for body image concerns in youth with craniofacial conditions. In a large, longitudinal study of the impact of teasing on appearance evaluations and psychosocial functioning in youth with cleft lip and/or palate (N = 340), experiences of teasing after age 10 significantly impacted appearance evaluations and depressive symptoms in adolescent females (Feragen & Stock, 2016). Similarly, longitudinal studies in youth without visible differences have found that peer victimization in childhood is related to body shame in adolescence, with greater shame evident in girls compared to boys (Lunde & Frisén, 2011).

This study has several limitations including the cross-sectional design, the lack of a control group, and reliance on self-reported data. Longitudinal studies are needed to understand how perceptions of appearance, speech, and stigmatization change over time and in relationship to surgical and other craniofacial-related treatments (e.g., speech therapy). Multi-informant, mixed methods approaches are also needed to further assess body image and stigmatization experiences, as well as other factors from Cash’s model (e.g., personality attributes) which may impact body image in craniofacial populations.

Other limitations relate to socio-economic status which was not assessed, and potentially selection bias and social desirability. It is possible that participants who volunteered for the study did so because they were more concerned about their appearance or were more comfortable with reporting their concerns. It could also be that adolescents with more significant appearance concerns and related distress were reluctant to participate because of fear or embarrassment about sharing their experiences. However, results suggest that participants with a variety of appearance and stigmatization-related concerns took part in the study. Finally, the participants in this study were recruited from pediatric craniofacial centers with interdisciplinary teams. Adolescents who are still engaged in treatment may differ from those who have discontinued interdisciplinary care whether due to family or individual choice, or access to care barriers (e.g., transportation, insurance).

Despite these limitations, this study has several notable strengths, including its relatively large sample size (compared to Shapiro et al., 2015) recruited from two sites, and its use of instruments that were developed specifically for youth with craniofacial and/or other visible differences, as well as the DAS and BIDQ, which were developed for use in both clinical and non-clinical populations. Use of both condition-specific and general measures of body image has been recommended (Crerand et al., 2017) in order to capture specific concerns while also promoting future comparisons with normative and/or other medical population samples. Additional strengths include the study’s application of Cash’s cognitive-behavioral body image model to an understudied population at risk for body image concerns, and inclusion of perceptions of both appearance and physical functioning relevant to craniofacial populations. This study’s findings underscore that social stigma and body image concerns are common in youth with craniofacial concerns, and that females may be especially vulnerable to body image disturbances.

4.1. Conclusions

This study adds to the body of research that suggests that social stigma and body image concerns are common in youth with craniofacial concerns and require intervention. Females may be especially vulnerable to body image disturbances, although stigmatization is associated with body image disturbance and dissatisfaction with speech and facial appearance for both sexes. These results underscore Feragen and Stock (2016)’s recommendation for the early identification of perceived teasing in all children with cleft or craniofacial conditions to prevent development of emotional problems and dissatisfaction with appearance. They also highlight the need for prevention and intervention strategies to be further integrated into routine craniofacial care.

Highlights.

• Body image disturbance varies by sex in youth with craniofacial conditions.

• Girls report more body image disturbance and appearance dissatisfaction than boys.

• Speech satisfaction and stigmatization do not differ by sex.

• Stigmatization predicts body image disturbance, speech and appearance satisfaction.