We found that parents of infants with clefts provided caregiver-centered language useful in describing their daily observations. No additional domains of infant health were noted by parents. We will use these observations and language to develop and validate a parent-reported observation diary that can augment assessments of the impacts of interventions on infants who are receiving care for CL±P.

Access to specialized medical care is critical to decrease complications and minimize long-term morbidity, yet racial disparities in cleft surgery persist as time to initial reconstruction remains delayed among minority patients. Research has demonstrated an average 3-week delay in surgery for minority patients nationally. A retrospective chart review of patient demographics, visit timing, and surgical history was performed for patients who underwent primary cleft lip with or without palate (CL + P) reconstruction between 2002 and 2016 at an urban craniofacial center. Of the 89 children who underwent surgery, 87% were ethnic minorities (58% Hispanic, 25% African-American, 4% Asian/Other). Caucasian children were the earliest to receive CL (3.5 months) and CP (13-months) repair. Minority children trended toward a delay in CL repair, with surgery for African-Americans at 5-months (P = 0.06) and Hispanics at 4.8-months (P = 0.07). Time from first visit to CL surgery showed significant delays for minority, non-English speaking, and public insurance patients; however, for CP repair, male children were delayed from first visit to surgery compared to females (P = 0.03). While there was no statistical difference in age at CL or CP surgical repair among our racial/ethnic cohorts, there were significant racial/ethnic differences in timing spent in the preoperative period for CL. However, racial/ethnic differences decreased as the patients spent more time within the healthcare system. Thus, established, interdisciplinary cleft/craniofacial centers well versed in minority patients can minimize the complex social and cultural factors that contribute to delays in cleft care.

Objective To compare relative levels of stress reported by mothers and fathers in families containing infants with and without single-suture craniosynostosis. Design Case-control study. Participants Mothers and fathers of 246 infants with recently diagnosed single-suture craniosynostosis and 253 frequency-matched control infants completed the Parenting Stress Index just prior to their infant’s cranioplastic surgery. Family demographic information and mothers’ ratings of the severity of their child’s single-suture craniosynostosis were obtained. Results Average Parent Domain scores for parents of infants with single-suture craniosynostosis differed little from those reported by parents of control infants; however, Child Domain scores among parents of infants with single-suture craniosynostosis were higher on some subscales, primarily related to unexpected infant health and appearance issues. In both groups, fathers reported higher Child Domain stress than mothers, and mothers reported higher Parent Domain stress than fathers. Case mothers reported greater stress if they perceived their child’s condition as more noticeable to others. Conclusions Prior to cases’ cranioplastic surgery, parents of children with and without single-suture craniosynostosis reported similar levels of stress in relation to their parenting roles and the behavioral characteristics of their infants. Visibility of condition should be considered a risk for increased stress for mothers of infants with single-suture craniosynostosis. Stress differences between mothers and fathers were far more discernible than those associated with the presence or absence of single-suture craniosynostosis.

Importance: Knowledge of unmet school participation needs for students with craniofacial microsomia (CFM) can inform decisions regarding intervention support. Objective: To compare students with and without CFM on school participation (i.e., frequency, involvement, desire for participation to change) and caregivers’ perceptions of environmental support for participation in occupations. Design: Cross-sectional design using secondary analyses of a subset of data. Setting: Multisite cohort study. Participants: Caregivers of students with CFM ( n = 120) and of students without CFM ( n = 315), stratified by history of education- and health-related service use. Outcomes and Measures: School participation and environmental support, obtained with the Participation and Environment Measure–Children and Youth. Results: Significant group differences were found in frequency of school participation (effect size [ES] = −0.38, 95% confidence interval [−0.64, −0.12], p = .005), level of involvement (ES = −0.14, p = .029), and desired change ( p = .001), with students with CFM exhibiting greater participation restriction than students without CFM and no history of service use. No statistically significant group differences were found in environmental support for participation in the school setting. Item-level findings showed statistically significant higher desire for participation to change in three of five school occupations (odds ratio = 1.77–2.39, p = .003–.045) for students with CFM compared with students without CFM and no history of service use. Conclusions and Relevance: The results suggest that students with CFM experience restriction in participation at school. What This Article Adds: Students with CFM may benefit from targeted school-based interventions to optimize their inclusion.

Objective To examine family functioning related to sociodemographic and clinical characteristics in youth with cleft lip and/or palate (CL/P). Design Cross-sectional, multisite investigation. Setting Six U.S. cleft centers. Patients/Participants A diverse sample of 1200 children with CL/P and their parents. Main Outcome Measure Parents completed the Family Environment Scale (FES), which assesses three domains of family functioning: cohesion (or closeness), expressiveness (open expression of feelings), and conflict. Demographic and clinical characteristics were also assessed including race, ethnicity, type of insurance, and surgical recommendations. Results The FES scores for families seeking team evaluations for their youth with CL/P (mean age = 11.6 years) fall within the average range compared with normative samples. Families receiving surgical recommendations for their youth also had FES scores in the average range, yet families of children recommended for functional surgery reported greater cohesion, expressiveness, and less conflict compared with those recommended for aesthetic surgery (P < .05). For cohesion and expressiveness, significant main effects for race (P = .012, P < .0001, respectively) and ethnicity (P =.004, P < .0001, respectively) were found but not for their interaction. No significant differences were found on the conflict domain. Families with private insurance reported significantly greater cohesion (P < .001) and expressiveness (P < .001) than did families with public insurance. Conclusions Family functioning across domains was in the average range. However, observed differences by race, ethnicity, type of insurance, and surgical recommendation may warrant consideration in clinical management for patients and families.