Abstract
Purpose:
To examine differences in community participation and environmental support for youth with and without craniofacial microsomia.
Methods:
This study involved secondary analyses of a subset of data (n = 396) from a longitudinal cohort study. Multiple linear and Poisson regression analyses and Wilcoxon Mann-Whitney tests were used to estimate differences in community participation and environmental support between youth with craniofacial microsomia and youth without craniofacial microsomia, stratified based on their history of education and health-related service use. Chi-square analyses were used to explore item-level group differences in change desired across community activities.
Results:
Statistically significant differences were found in community participation frequency (ES = −0.52; p < 0.001), level of involvement (r = −0.16; p = 0.010), and desire for change in participation when comparing youth with craniofacial microsomia and non-affected peers not receiving services (p < 0.001). There were no statistically significant differences between youth with craniofacial microsomia and non-affected peers receiving services.
Conclusions:
Results suggest lower community participation in youth with craniofacial microsomia as compared to non-affected peers not receiving services. This may suggest opportunities for designing and testing interventions to promote community participation among youth with craniofacial microsomia, so as to support their transition to adulthood.
Keywords: leisure activities, caregivers, patient reported outcome measures, Goldenhar syndrome
Introduction
Children with craniofacial microsomia (CFM) present with asymmetric underdevelopment of craniofacial structures such as the mandible, maxilla, ears, soft tissues, and facial nerves [1–4]. CFM is a highly variable condition that can lead to impairments in vision, hearing, speech [5], eating [5,6], and/or sleeping [7].
Prior studies have shown that children with CFM experience challenges in neurodevelopmental and psychosocial outcomes in childhood [8–10]. For example, children with CFM were reported to have significantly lower scores than their age-matched peers in receptive vocabulary and perceptual motor abilities, and academic achievement per caregiver and teacher report [10]. Furthermore, teachers report that children with CFM show significantly more internalizing and externalizing behavior problems and lower social competence, as compared to their non-affected peers [11]. Similarly, caregivers of children with CFM reported on their children as having significantly more social problems when compared to caregivers of children without CFM [11]. Finally, Khetani and colleagues [12] showed that caregiver-rated quality of life was significantly lower for children with CFM than for non-affected peers across physical, social, and school functioning domains. Effect sizes for these differences were small but consistently show that children with CFM are at higher risk for experiencing neurodevelopmental and psychosocial difficulties and lower quality of life when compared to their peers without CFM.
Speltz and colleagues [13] showed that as children with CFM transition into adolescence and young adulthood they exhibit lower neurodevelopmental outcomes than their age-matched peers, specifically verbal processing including vocabulary, reading and writing. However, these differences were smaller in magnitude relative to earlier phases of study with this same cohort [10,13]. Similarly, behavior problems detected in early school-aged children were largely attenuated in adolescents [11,14]. There was some evidence of persistent social impact for youth with CFM, with continued differences between youth with CFM and their non-affected peers in quality of life specific to youth’s social functioning and caregiver-report of their child’s social competence [13,14]. Other studies have also found differences in quality of life for youth with and without craniofacial conditions, whether congenital (e.g., CFM) or acquired due to burn and trauma [15,16]. Qualitative research involving youth with CFM support these findings by illustrating ongoing concern around the stigma and negative psychosocial impact of managing daily life with CFM [9,17].
Community participation need among youth with CFM has not been studied, but may be clinically relevant for informing health-related service need. Participation in home, school, and community activities is a patient-centric and intervenable outcome for children and youth receiving pediatric health-related services (e.g., rehabilitation and habilitation services) [18,19]. For youth, participation in activities has been linked to positive development of their skills and capacities, social networks, and self-efficacy, which are key to a successful transition into adulthood [20]. There is a growing body of evidence on disparities between children and youth with and without disabilities in community participation that increase with age [21] and the significant role of the environment on child and youth participation [22–25]. Therefore, knowledge about the community participation need and the community environment as a potential target for intervention can support tailored health-related services to meet the functional need of youth with CFM during their transition to adulthood [12,26].
The aims of this study were to examine differences between youth with CFM and their peers without CFM in (1) community participation, and (2) environmental support for community participation. We hypothesized that youth with CFM will experience greater restrictions in community participation (i.e., lower frequency, lower level of involvement, higher desire for change) as compared to their non-affected peers. We also expected caregivers of youth with CFM to report less environmental support for their youth’s community participation as compared to youth without CFM.
Methods
Study Design
This study employed secondary analyses of a subset of data collected for a longitudinal cohort study of children and youth with CFM (2010–2015). These data were collected as part of the second phase of follow-up with children and families [13]. The study was approved by the institutional review boards of Boston University and Seattle Children’s Hospital prior to data collection. Ethics approval was obtained at the University of Illinois at Chicago prior to undertaking analyses for this study.
Participants
Youth with CFM.
Participants were initially recruited between 1996–2002, as part of the first phase of an epidemiological study [27]. Children with CFM and their families were recruited from craniofacial specialty clinics using the following inclusion criteria: Children were less than 36 months of age and were diagnosed with CFM by a craniofacial physician, according to the criteria for hemifacial microsomia, facial asymmetry, unilateral microtia, oculo-auriculo-vertebral syndrome, or Goldenhar syndrome. Families were excluded if their child was diagnosed with chromosomal anomalies, Mendelian-inherited disorders, or in utero isotretinoin exposure, if their child was adopted or if their child was older than 36 months. This resulted in 279 children with CFM and their families for the first phase [13,27]. Families were then approached to enroll in a follow-up study (2004–2009) [10–12]. One hundred and ninety-eight children with CFM and their families enrolled and were then approached again to participate in the current phase (2011–2015). Of the 198 participants, 18 could not be contacted, 30 declined to participate and eight were not approached or found to be ineligible. This resulted in a sample size of 142 [13,14].
Youth without CFM.
Initial recruitment during the first phase of study involved enrolling three children without CFM per each child with CFM, through pediatricians of children with CFM or other pediatric practices. Children were eligible as youth without CFM if no known birth defect was present, if they were not adopted, and they were within two months of the age of children with CFM at the time of recruitment [27]. In total, 884 children without CFM participated during the first phase and 568 children and families decided to continue during the second phase. For the current third phase, two youth without CFM per youth with CFM were contacted [13,14]. A total of 169 families could not be approached or contacted, 79 declined, four were not approached due to non-matching reasons (e.g., ineligibility or child death) and one was diagnosed with grade 2 unilateral microtia and therefore excluded, resulting in 315 youth without CFM [13].
Measures
Child and Family Characteristics.
Caregivers were asked to report on their annual income, formal education, and age at the time of child’s birth. Caregivers were also asked whether their child has received or is receiving health-related services (e.g., occupational therapy, physical therapy, speech therapy, vision therapy, hearing services, mental health services) and/or special education services. Caregivers were also asked to update information previously collected, including child race/ethnicity and medical history.
Community Participation and Environmental Support.
The Participation and Environment Measure for Children and Youth (PEM-CY) [28] is a proxy measure of a child’s participation as well as environmental support for participation of children or youth (5–17 years). For this study, data collected from the PEM-CY community section were used.
The PEM-CY community section consists of 10 items focusing on participation in types of community activities (e.g., neighborhood outings, going to a movie). For each type of community activity, caregivers were asked to report on the following: 1) the frequency of their child’s participation on an 8-point scale (from never = 0 to daily = 7); 2) their child’s level of involvement on a 5-point scale (from minimally involved = 1 to very involved = 5); and 3) whether they desire change in their child’s participation (yes, no). After evaluating their child’s community participation, caregivers were administered 16 items to evaluate environmental factors and resources (e.g., physical layout, sensory qualities, attitudes and actions of others) according to whether they help or make it harder for the child to participate in community activities, on a 3-point scale (3 = usually helps/usually yes/no impact/not an issue/not needed; 2 = sometimes helps, sometimes makes harder/sometimes yes, sometimes no; 1 = usually makes harder/usually no). The PEM-CY community scales have acceptable internal consistency reliability (α = 0.70 – 0.83) [21,29], similar to the estimated value for this study (α = 0.67 for participation frequency, α = 0.84 for level of involvement, α = 0.83 for desire of change, α = 0.81 for environmental support). Furthermore, the PEM-CY has moderate to excellent test-retest reliability (ICC = 0.66 – 0.96) [21,29]. For individual items, test-retest reliability ranges from .73 to .93. PEM-CY community scales can also distinguish between children with and without disabilities (ES = 0.76 – 1.86) [21,29].
For this study, four scores were calculated. For community frequency and level of involvement, a mean score across the 10 community participation items was computed. The score for desire for change is based on count data and was calculated by taking the sum of all “yes, desire change” responses across the 10 community participation items. A community environmental support score was derived by calculating the sum of ratings across the environmental items, dividing by the maximum possible score, and then multiplying by 100.
Procedure
Between 2011 and 2015, trained psychometrists traveled to participants for the purpose of administering a 4–5 hour battery of standardized developmental assessments [13,14]. While the youth were being tested, caregivers were administered a series of questionnaires including the demographic questionnaire and the electronic version of the PEM-CY [28]. Participating caregivers were reimbursed with a $35 gift card.
Data Analysis
Data were analyzed using SAS 9.4 (SAS Institute Inc., Cary NC). Initially, data were examined (via histogram, descriptive analyses) to ensure that data met the underlying assumptions for the chosen statistical analyses. Participants with missing values on all PEM-CY community items were excluded from analyses (N=50).
Due to known associations between service use and participation [30], for the current study, youth without CFM were further grouped according to whether or not they currently receive and/or have received health-related and/or special education services (i.e., physical therapy, occupational therapy, speech language therapy, visual therapy, hearing services, mental health services, special education). Eleven youth without CFM were excluded due to missing data on their service use that precluded assignment into one of the groups for youth without CFM. Because of insuffient sample size, analyses were not pursued specific to youth with CFM receiving and not receiving services. Sensitivity analyses of youth with CFM who receive services (N=94) were conducted to test whether results were significantly different when only including youth with CFM who receive services versus all youth with CFM. Analyses revealed no significant differences but in environmental support when compared to youth without CFM and not receiving services, whereby caregivers of youth with CFM perceived their child’s community environment to be less supportive than their non-affected peers who were not receiving services.
Descriptive statistics were used to summarize the characteristics of the youth and families in each of the three groups (youth with CFM; youth without CFM and receiving services; youth without CFM and not receiving services).
Based on model selection and previous studies with the same cohort [10–14] as well as studies on child and youth participation [31,32], the following covariates were selected for main analyses: youth gender, age, and race/ethnicity; caregiver age at time of child’s birth, education, and marital status; and annual income.
Four sets of analyses were performed to examine between-group differences in community participation frequency, level of involvement, desire for change (Aim 1), and environmental support (Aim 2). For continuous response variables sensitivity analyses were performed using mean substitution, which revealed no significant differences. Therefore, missing values were ignored for analyses. After performing regression diagnostics analyses to examine selected model fitting (i.e., residual analyses, test for collinearity), multiple linear regression was applied to test for differences in community participation frequency and perceived environmental support for participation. For categorical covariates, the reference groups were those having the largest sample size in the sub-group of youth with CFM (i.e., married, white non-Hispanic, ≥$65,000, at least high school/general education diploma). To estimate the magnitude of group differences, effect sizes were calculated using Cohen’s d and interpreted based on Cohen’s reference values (0.2=small, 0.5=medium, 0.8=large) [33]. To examine group differences in the child’s level of involvement during community activities, Wilcoxon-Mann Whitney tests (two-sided) were used because of skewed data distribution and an insufficient regression model fit. Additionally, effect sizes were calculated using r, based on z-score (0.3=medium, 0.5=large) [34]. Differences in the number of activities in which change is desired were tested using Poisson regression model. Exploratory analyses revealed no significant differences (p > 0.05) in community participation frequency, level of involvement, or desire for change by CFM phenotype (i.e., microtia only, microtia and mandibular hypoplasia, other).
Descriptive statistics were applied to item-level data to report on mean and median results for youth with CFM and youth without CFM not receiving services. Participation need is captured through caregivers’ desired change in community participation (i.e., desire for change indicates dissatisfaction) and is of high clinical relevance. Thus, significance testing using chi-square analyses were used to test for item-level group differences in caregiver desire for change only.
Since little is known about community participation in youth with and without CFM, we did not adjust for multiple comparisons to avoid missing potentially important associations that warrant further study.
Results
Participants were 120 caregivers of youth with CFM, 140 caregivers of youth without CFM receiving services, and 136 caregivers of youth without CFM not receiving services. Analyses comparing participants who did and did not enroll for this study revealed that non-enrolled participants were more likely to be non-white or Hispanic, and Spanish speaking [13]. Sampled youth were between 11–17 years of age. As shown in table 1, more than half of the youth were male, and the majority were White, non-Hispanic (82.35% - 82.86%). Most of the youth’s caregivers were married (75.94% - 85.59%), and earned more than the annual median U.S. income (62.61% - 72.73%) [35].
Table 1.
Youth and family characteristics and service use
| Youth with CFM | Youth without CFM receiving services | Youth without CFM not receiving services | |
|---|---|---|---|
| n = 120 | n = 140 | n = 136 | |
| Youth gender, n (%) | |||
| Male | 69 (57.50) | 74 (52.86) | 61 (44.85) |
| Female | 51 (42.50) | 66 (47.14) | 75 (55.15) |
| Youth age in years, M (SD) | 13.39 (1.25) | 13.52 (1.52) | 13.15 (1.33) |
| Receiving at least one type of service, n (%)† | 94 (78.99) | 140 (100.00) | 0 (0.00) |
| Type of service received, n (%)† | |||
| Rehabilitation services (OT, PT, ST) | 81 (68.07) | 69 (50.74) | 0 (0.00) |
| Vision therapy | 9 (7.56) | 24 (17.14) | 0 (0.00) |
| Hearing services | 39 (32.77) | 3 (2.16) | 0 (0.00) |
| Mental health services | 17 (14.29) | 37 (26.43) | 0 (0.00) |
| Special education services | 37 (31.09) | 44 (32.12) | 0 (0.00) |
| Other services | 28 (23.73) | 65 (46.76) | 0 (0.00) |
| Youth race/ethnicity, n (%) | |||
| White, non-Hispanic | 99 (82.50) | 116 (82.86) | 112 (82.35) |
| White, Hispanic | 15 (12.50) | 9 (6.43) | 12 (8.82) |
| African-American | 0 (0.00) | 8 (5.71) | 8 (5.88) |
| Other | 6 (5.00) | 7 (5.00) | 4 (2.94) |
| Marital status, n (%)† | |||
| Married | 101 (85.59) | 101 (75.94) | 114 (84.44) |
| Single | 5 (4.24) | 10 (7.52) | 4 (2.96) |
| Divorced, separated, widowed | 12 (10.17) | 22 (16.54) | 17 (12.59) |
| Annual income, n (%)† | |||
| <$25,000 | 7 (6.09) | 14 (10.77) | 7 (5.30) |
| $25,000-$34,999 | 17 (14.78) | 8 (6.15) | 9 (6.82) |
| $35,000-$64,999 | 19 (16.52) | 16 (12.31) | 20 (15.15) |
| ≥$65,000 | 72 (62.61) | 92 (70.77) | 96 (72.73) |
| Caregiver education† | |||
| At least high school/GED | 45 (38.46) | 28 (20.90) | 42 (31.11) |
| associates degree | 17 (14.53) | 25 (18.66) | 19 (14.07) |
| bachelor’s degree | 41 (35.04) | 44 (32.84) | 50 (37.04) |
| graduate degree | 14 (11.97) | 37 (27.61) | 24 (17.78) |
| Caregiver age at time of child’s birth, M (SD) | 29.93 (6.00) | 30.49 (5.47) | 29.89 (4.89) |
Note. CFM = Craniofacial microsomia; GED=General education diploma; OT=Occupational therapy; PT=Physical therapy; ST=Speech/language therapy; M=Mean; SD=Standard deviation; n=number of participants
missing data
CFM and Community Participation Frequency for Youth
Youth with CFM participated less frequently in community activities as compared to their non-affected peers who were not receiving services (ES = −0.52; 95% CI = −0.78, −0.26; p < 0.001), even after adjusting for covariates (see table 2). Item-level results showed that the largest differences in mean participation frequency pertained to “organized physical activities” and “getting together with other children in the community” (Mean differences ≥ 0.79; see table 3). There was no statistically significant difference in participation frequency between youth with and without CFM who received services (ES = −0.20; 95% CI = −0.46, 0.07; p = 0.147).
Table 2.
Community participation of youth with and without craniofacial microsomia (CFM)
| Youth with CFM versus youth without CFM receiving services | Youth with CFM versus youth without CFM not receiving services | ||||||||
|---|---|---|---|---|---|---|---|---|---|
| Variables | b /exp (b) (95%CI) | ES (95% CI) | b /exp (b) (95%CI) | ES (95% CI) | |||||
| Participation frequency †• | −0.18 (−0.43, 0.07) | −0.20 (−0.46, 0.07) | −0.49** (−0.73, −0.25) | −0.52 (−0.78, −0.26) | |||||
| Desire for change in community participation †§ | 1.03 (0.90, 1.17) | 1.35 **(1.17, 1.54) | |||||||
| Environmental support in community participation †• | −1.60 (−4.15, 0.95) | −0.17 (−0.43, 0.10) | −2.32 (−4.81, 0.18) | −0.24 (−0.50, 0.02) | |||||
| Youth with CFM | Youth without CFM receiving services | Youth without CFM not receiving services | |||||||
| Mdn (Q1, Q3) | Mdn (Q1, Q3) | Mdn (Q1, Q3) | |||||||
| Involvement in community-based activities ‡ | 4.32 (3.76, 4.78) | 4.38 (3.71, 4.80) | 4.57 (4.10, 4.90)* | ||||||
Note. CFM = Craniofacial microsomia; b=Beta; ES = Effect size; CI = Confidence interval; Mdn = Median; Q1 = First quartile; Q3 = Third quartile
Adjusted for youth gender, youth age, youth race/ethnicity, caregiver age, caregiver education, annual income, and marital status.
Calculated using multiple linear regression
Significance testing using Wilcoxon Mann-Whitney test was done between youth with CFM and each of the groups of youth without CFM.
Calculated using Poisson regression; exponential beta reported (exp (B))
p < 0.05
p < 0.001
Table 3.
Participation across activities and environmental support of youth with and without craniofacial microsomia (CFM)
| Youth with CFM | Youth without CFM receving services | Youth without CFM not receiving services | ||
|---|---|---|---|---|
| n = 120 | n = 140 | n = 136 | p | |
| Participation frequency, M (SD) | ||||
| Neighborhood outings | 5.19 (1.20) | 5.18 (1.18) | 5.31 (1.27) | - |
| Community events | 3.10 (1.75) | 3.57 (1.74) | 3.51 (1.60) | - |
| Organized physical activities | 3.83 (2.74) | 4.44 (2.53) | 4.69 (2.37) | - |
| Unstructured physical activities | 4.70 (2.00) | 4.76 (1.83) | 5.07 (1.89) | - |
| Classes and lessons (not schoolsponsored) | 2.01 (2.55) | 2.49 (2.50) | 2.55 (2.54) | - |
| Organizations, groups, clubs, and volunteer or leadership activities | 1.84 (2.26) | 2.08 (2.26) | 2.38 (2.31) | - |
| Religious or spiritual gatherings and activities | 3.02 (2.46) | 3.06 (2.35) | 3.53 (2.33) | - |
| Getting together with other children in the community | 3.99 (2.04) | 4.43 (1.86) | 4.79 (2.03) | - |
| Working for pay | 2.38 (2.40) | 2.50 (2.38) | 2.71 (2.27) | - |
| Overnight visits or trips | 2.37 (1.57) | 2.79 (1.35) | 2.93 (1.48) | - |
| Level of involvement, Mdn (IQR) | ||||
| Neighborhood outings | 5.00 (1.00) | 4.00 (1.00) | 5.00 (1.00) | - |
| Community events | 5.00 (2.00) | 5.00 (1.00) | 5.00 (1.00) | - |
| Organized physical activities | 5.00 (1.00) | 5.00 (1.00) | 5.00 (0.50) | - |
| Unstructured physical activities | 5.00 (1.00) | 5.00 (2.00) | 5.00 (1.00) | - |
| Classes and lessons (not schoolsponsored) | 5.00 (1.00) | 5.00 (2.00) | 5.00 (1.00) | - |
| Organizations, groups, clubs, and volunteer or leadership activities | 5.00 (2.00) | 4.50 (2.00) | 5.00 (1.00) | - |
| Religious or spiritual gatherings and activities | 4.00 (2.00) | 4.00 (2.00) | 4.00 (2.00) | - |
| Getting together with other children in the community | 5.00 (1.00) | 5.00 (1.00) | 5.00 (1.00) | - |
| Working for pay | 4.00 (2.00) | 5.00 (2.00) | 5.00 (1.00) | - |
| Overnight visits or trips | 5.00 (1.00) | 5.00 (1.00) | 5.00 (0.00) | - |
| Desire for change in participation, n (%) | ||||
| Neighborhood outings | 19 (15.83) | 26 (18.57) | 20 (14.71) | 0.802 |
| Community events | 39 (32.50) | 46 (32.86) | 36 (26.47) | 0.290 |
| Organized physical activities | 52 (43.33) | 55 (39.29) | 32 (23.53) | 0.001 |
| Unstructured physical activities | 61 (50.83) | 75 (53.57) | 53 (38.97) | 0.057 |
| Classes and lessons (not schoolsponsored) | 58 (48.33) | 66 (47.14) | 53 (38.97) | 0.131 |
| Organizations, groups, clubs, and volunteer or leadership activities | 67 (55.83) | 72 (51.43) | 51 (37.50) | 0.003 |
| Religious or spiritual gatherings and activities | 54 (45.00) | 72 (51.43) | 57 (41.91) | 0.619 |
| Getting together with other children in the community | 50 (41.67) | 52 (37.14) | 33 (24.26) | 0.003 |
| Working for pay | 57 (47.50) | 70 (50.00) | 54 (39.71) | 0.209 |
| Overnight visits or trips | 35 (29.17) | 36 (25.71) | 21 (15.44) | 0.008 |
| Environmental supports, M (SD) | ||||
| Physical layout | 98.02 (10.03) | 93.05 (16.29) | 95.56 (15.13) | - |
| Sensory quality | 89.64 (20.24) | 93.29 (16.63) | 97.79 (8.32) | - |
| Physical demands of activity | 93.79 (16.28) | 95.44 (14.02) | 98.28 (8.43) | - |
| Cognitive demands of activity | 94.68 (15.02) | 93.96 (16.23) | 98.77 (6.30) | - |
| Social demands of activity | 89.55 (19.83) | 94.24 (16.02) | 96.57 (11.67) | - |
| Relations with peers | 90.40 (18.54) | 95.68 (11.93) | 97.06 (11.09) | - |
| Attitudes | 93.79 (14.42) | 95.68 (12.58) | 98.53 (6.87) | - |
| Weather conditions | 92.37 (15.35) | 88.25 (18.33) | 90.44 (18.55) | - |
| Safety | 95.24 (13.92) | 92.33 (18.96) | 93.38 (18.06) | - |
| Personal transportation | 97.76 (9.44) | 97.12 (11.69) | 98.52 (8.98) | - |
| Public transportation | 92.72 (19.96) | 91.37 (21.36) | 91.36 (22.29) | - |
| Programs and services | 96.61 (13.35) | 94.69 (15.20) | 95.06 (14.97) | - |
| Information | 94.64 (13.09) | 93.87 (14.17) | 93.99 (15.46) | - |
| Equipment and supplies | 95.76 (12.04) | 93.33 (16.19) | 94.40 (15.11) | - |
| Time | 90.77 (16.87) | 91.24 (16.30) | 92.97 (16.01) | - |
| Money | 90.86 (17.39) | 89.78 (19.62) | 91.41 (18.82) | - |
Note. CFM = Craniofacial microsomia; Mdn=Median; IQR=Interquartile range; n=number of participants; M=Mean; SD= Standard deviation
P-values are based on comparison between youth with CFM and youth without CFM who do not receive services
CFM and Youth Level of Involvement in Community Activities
Youth with CFM (Mdn = 4.32) were less involved as compared to their non-affected peers who were not receiving services (Mdn = 4.57; W = 13846.00; z = −2.59; r = −0.16; p = 0.010). Item-level results showed a difference in median level of involvement for “working for pay” (Median differences = 1; see table 3). There was no statistically significant difference in levels of involvement when comparing youth with CFM and those without CFM who received services (Mdn = 4.38; W = 15242.50; z = −0.30; r = −0.02; p = 0.764).
CFM and Desire for Change in Youth Community Participation
When compared to youth without CFM not receiving services, having CFM was significantly associated with 34% increase in the mean number of community activites for which change is desired (exp(b) = 1.35; 95% CI = 1.17, 1.54; p < 0.001). On average, caregivers of youth with CFM and youth without CFM who received services were similar in wanting their child’s participation to change in 4 out of 10 community activities. Item-level comparisons of youth with CFM and their non-affected peers who did not receive services reveal significant differences for four community activities and one trending towards significance: overnight visits or trips (p = 0.008); getting together with other children in the community (p = 0.003); organizations, groups, clubs, and volunteer/leadership activities (p = 0.003); and organized physical activities (p = 0.001); unstructured physical activities (p = 0.057; see table 3).
CFM and Perceived Environmental Support for Youth Community Participation
The difference in environmental support for community participation between youth with CFM and youth without CFM who do not receive services trended towards statistical significance (ES = −0.24; 95%CI = −0.50, 0.02; p = 0.069), when adjusting for select child and family characteristics. Item-level results showed that differences in mean environmental support were highest for three items (Mean differences > 6.50), with youth with CFM showing lower scores than youth without CFM who do not receive services. These items include ‘social demands of typical activities’, ‘relationships with peers’, and ‘sensory qualities (e.g., noise, crowds, lighting)’ (see table 3).
Discussion
One pathway through which CFM might affect a child’s overall quality of life is its impact on their participation in valued activities of everyday life. For transition-aged youth, understanding community participation difficulties is particularly valuable for informing the need to intervene for successful transition to adulthood which is marked by full community integration. Our study examined participation difficulty among youth with and without CFM with varying health-realted and/or education service use. Results suggest that there are differences between youth with CFM and youth without CFM who did not receive services according to how often and how much they participate, as well as caregiver satisfaction with youths’ participation across diverse community activities. However, environmental support for youth participation in community activities were perceived similar.
CFM and Youth Community Participation
Study results suggest that youth with CFM participate less frequently and are less involved in community activities only when compared to youth without CFM who do not receive services. Effect size for the difference in participation frequency was medium, meaning that half of the youth with CFM have lower participation frequency than 69% of youth without CFM who do not receive services. In contrast, effect size was small for level of involvement, meaning that half of youth with CFM have lower scores than around 58% of youth without CFM who do not receive services [33]. Effect size and the magnitude of mean difference in participation frequency are similar to prior studies of community participation among children and youth with mixed disabilities when compared to similar aged peers without disabilities [21,28]. For level of involvement, effect sizes and magnitude of median differences were smaller than in previous studies [21,28]. This finding might be related to youth with CFM experiencing both the support of their parents, extended family [8,17], and close friends [17]. In addition, due to previously described challenges such as bullying because of differences in appearance, withdrawn behavior, and challenges socializing with peers due to hearing loss [9], youth with CFM might also be selective in choosing community activities where they feel involved, possibly reflecting the found moderate difference in participation frequency between youth with CFM and their non-affected peers not receiving services. Youth in this prior study also described low self-esteem and insecurities [9], which are participation-related constructs [23] that may limit how often and how much youth take part in activities. This relates to the current findings indicating that youth with CFM participate less frequently and that caregiver satisfaction in participation is lower with respect to getting together with other children as well as organized physical activities, when compared to their non-affected peers who do not receive services (see table 3). From a clinical perspective, this may indicate the need to pay attention to both participation frequency and involvement to appraise current participation among youth, as they may influence each other [23].
As compared to frequency and level of involvement, caregiver desire for change in their child’s participation is a direct indicator of service need and therefore of high clinical relevance. Results of this study suggest that caregivers of youth with CFM express greater need for their child’s participation to change when compared to their non-affected peers who do not receive services. This finding is similar to prior studies of quality of life among children and youth with CFM, another subjective marker of a child’s health and well-being. For example, Khetani and colleagues [12] and Wallace and colleagues [14] found significantly lower caregiver-rated quality of life among children with CFM when compared to their non-affected peers. The most common activities in which change was desired were activities that place a higher demand on involvement with others (i.e., overnight visits or trips; getting together with other children in the community; organizations, groups, clubs, and volunteer or leadership activities; organized as well as unstructured physical activities). This might be also reflected by the slightly lower perceived environmental support with respect to the social demands of these activities for youth with CFM when compared to their peers without CFM who do not receive services. Differences in unstructured as well as structured community activities were also present in a prior study involving children with and without mixed disabilities [21], and together suggest that youth with CFM may be most likely to have unmet need specific to both types of social activities. These findings are useful for targeting health-related services to meet the need of youth with CFM during their transition to adulthood.
Across all dimesions of participation under study, no significant differences were found between youth with CFM and youth without CFM who receive services. This finding indicates that youth with CFM may have similar community participation need as their peers who receive services. These group similarities may generalize beyond the community setting, as similar findings were found with respect to school participation of students with CFM and those without CFM who receive services [36].
CFM and Perceived Environmental Support for Youth Community Participation
Contrary to our hypothesis, group differences in perceived environmental support for participation were found to, at most, trend towards statistical significance. This was found despite the growing body of evidence on disparities between children and youth with and without disabilities in environmental support for participation across home [37], school [38], and community [21] settings, and the significant mediating role of environment on participation beginning in early childhood [22–25]. For example, Bedell and colleagues [21] found that caregivers of children with diverse disabilities perceived their child’s community environment to be less supportive than caregivers of children without disabilities.
This discrepancy may reflect age-related differences across samples, as the mean age for youth included in this study is higher than prior studies. As youth age, there is increasing emphasis of school-based services on self-determination [39], whereby youth decide what activities they will participate in and may lean towards activities that take place in supportive environments. Contemporary participation-focused interventions emphasize youth appraising and intervening on their environment to improve participation outcomes [40,41]. Therefore, the older children included in this study may have and/or be receiving environmentally-focused interventions (e.g., Equipment and supplies, see table 3), resulting in overall similar perceptions of environmental support for community participation between groups.
Alternatively, it is possible that for youth with CFM, some aspects of the community environment (e.g., negative attitudes, stigmatization, sensory qualities) [15,9] are more salient in their impact on participation, similar to how physical barriers are more salient in hindering participation for individuals with mobility restrictions [42]. This explanation is supported by lower mean environmental support scores for youth with CFM in items pertaining to social demands of activities, peer relations, and sensory qualities when compared to youth without CFM who do not receive services. Relatively high means of perceived environmental support for youth with CFM on items such as attitudes may also reflect successful strategies that youth with CFM use to cope with potential environmental stressors [17]. Further studies on strategy use of youth with CFM to support participation could reveal the extent to which these strategies are environmentally focused. Futhermore, given the disparities in community participation detected in this study, future studies should model community participation as a function of child and family characteristics, service use, and perceptions of environmental support. For these studies, it may be helpful to parse out the relative effect of the child’s physical and social environment on community participation to best inform the scope of participation-focused care with this population.
Study results should be interpreted in light of several limitations. First, by using non-parametric testing for community involvement we were not able to control for covariates, which could have led to bias in those results. Second, there were missing data on service use (e.g., reason or duration of services) among youth without CFM, which limits us to examining service receipt (yes, no) relative to outcome. Finally, this study is limited to caregiver perspectives of community participation, whereas caregiver and youth perspectives about current and desired community participation may differ as shown in prior studies of quality of life [12]. Youth self-report assessment options are needed [43].
Conclusion
This study contributes new knowledge about caregiver perceptions of participation need for youth with CFM. Results indicate that youth with CFM have more restricted community participation, as denoted by lower frequency and level of involvement in community activities. These differences in the youth’s current community participation are of concern to caregivers of youth with CFM who display higher rates of dissatisfaction with their child’s community participation, when compared to their non-affected peers who do not receive education and health-related services. Similar analyses are underway to examine group differences in school participation to further strengthen knowledge about the full scope of participation difficulty across relevant settings for youth with CFM.
IMPLICATIONS FOR REHABILIATION.
Youth with craniofacial microsomia may have unmet rehabilitation needs related to their community participation.
Rehabilitation professionals should pay attention to participation of youth with craniofacial microsomia in activities that place a higher demand on involvement with others.
Rehabilitation professionals should appraise participation frequency and involvement of youths with craniofacial microsomia to gain accurate insight into their current community participation.
Acknowledgements:
The authors thank the families who participated in this research. We also thank Dr. Jinsong Chen for statistical support.
Declaration of interest statement: We have no interest that could be perceived as posing a conflict or bias. Data for this study was collected with funding from NIH grant R01 DE 11939 from the National Institute of Dental and Craniofacial Research to Dr. Werler.
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