Jennifer A Hansen-Moore1,2, Hillary M Kapa3, Jennifer L Litteral3, Leena Nahata1,2,3, Justin A Indyk1,2, Venkata R Jayanthi1,4, Yee-Ming Chan5,6, Amy C Tishelman5,7, Canice E Crerand1,2,3,8. 1. Nationwide Children's Hospital. 2. Department of Pediatrics, The Ohio State University College of Medicine. 3. Center for Biobehavioral Health, The Abigail Wexner Research Institute at Nationwide Children's Hospital. 4. Department of Urology, The Ohio State University College of Medicine. 5. Division of Endocrinology, Department of Pediatrics, Boston Children's Hospital. 6. Department of Pediatrics, Harvard Medical School. 7. Department of Psychiatry, Harvard Medical School. 8. Department of Plastic Surgery, The Ohio State University College of Medicine.
Abstract
OBJECTIVE: To assess health-related quality of life (HRQoL), psychosocial adjustment, and family functioning of children with differences of sex development (DSD) or cleft lip and/or palate (CL/P). METHODS: In this cross-sectional study, parents of children with DSD (n = 67), CL/P (n = 121), and a comparison group of unaffected youth (n = 126) completed standardized measures assessing family functioning and their children's HRQoL and psychosocial adjustment. Medical charts were abstracted for youth with either congenital condition. RESULTS: Children with DSD were rated as having significantly lower HRQoL and greater internalizing problems compared to youth with CL/P and unaffected youth. Children in the DSD group were also significantly more likely to fall into the clinical risk categories for total and internalizing problems relative to the CL/P and unaffected groups. Caregivers of children with DSD were significantly more likely to endorse items about child suicidality compared with caregivers in the CL/P and unaffected groups. No significant differences were found between groups for externalizing problems or the expressiveness domain of family functioning; parents of children with DSD reported significantly less family conflict relative to the other groups and greater cohesion relative to the unaffected group. Conclusions Youth with DSD appear to be at greater risk for psychosocial problems relative to children with CL/P and unaffected peers. Results underscore the need for integrated interdisciplinary care and ongoing psychosocial risk monitoring in youth with DSD.
OBJECTIVE: To assess health-related quality of life (HRQoL), psychosocial adjustment, and family functioning of children with differences of sex development (DSD) or cleft lip and/or palate (CL/P). METHODS: In this cross-sectional study, parents of children with DSD (n = 67), CL/P (n = 121), and a comparison group of unaffected youth (n = 126) completed standardized measures assessing family functioning and their children's HRQoL and psychosocial adjustment. Medical charts were abstracted for youth with either congenital condition. RESULTS: Children with DSD were rated as having significantly lower HRQoL and greater internalizing problems compared to youth with CL/P and unaffected youth. Children in the DSD group were also significantly more likely to fall into the clinical risk categories for total and internalizing problems relative to the CL/P and unaffected groups. Caregivers of children with DSD were significantly more likely to endorse items about child suicidality compared with caregivers in the CL/P and unaffected groups. No significant differences were found between groups for externalizing problems or the expressiveness domain of family functioning; parents of children with DSD reported significantly less family conflict relative to the other groups and greater cohesion relative to the unaffected group. Conclusions Youth with DSD appear to be at greater risk for psychosocial problems relative to children with CL/P and unaffected peers. Results underscore the need for integrated interdisciplinary care and ongoing psychosocial risk monitoring in youth with DSD.
Authors: Michael Goodman; Rami Yacoub; Darios Getahun; Courtney E McCracken; Suma Vupputuri; Timothy L Lash; Douglas Roblin; Richard Contreras; Lee Cromwell; Melissa D Gardner; Trenton Hoffman; Haihong Hu; Theresa M Im; Radhika Prakash Asrani; Brandi Robinson; Fagen Xie; Rebecca Nash; Qi Zhang; Sadaf A Bhai; Kripa Venkatakrishnan; Bethany Stoller; Yijun Liu; Cricket Gullickson; Maaz Ahmed; David Rink; Ava Voss; Hye-Lee Jung; Jin Kim; Peter A Lee; David E Sandberg Journal: BMJ Open Date: 2022-09-21 Impact factor: 3.006