Frontiers in dental medicine
Caregiver-reported oral health-related quality of life in children with Mowat-Wilson syndrome: a comparative study
By Cirio S, Salerno C, Allam A and 3 others.
Abstract
Background
Mowat-Wilson syndrome (MWS) is a rare genetic disorder characterized by intellectual disability, distinctive craniofacial features, behavioral impairments, and congenital anomalies. Although oral and craniofacial abnormalities are frequently reported, their impact on oral health-related quality of life (OHRQoL) has not been investigated. This study aimed to evaluate OHRQoL in children with MWS and compare it with peers with autism spectrum disorder (ASD) and neurotypical controls. Secondary analyses explored caregiver functional limitations and dental care characteristics.
Methods
A cross-sectional questionnaire-based study was conducted involving caregivers of children aged 6-18 years with MWS (n = 19), ASD (n = 19), and neurotypical controls (n = 38). OHRQoL was assessed using the short-form Parental-Caregiver Perception Questionnaire (P-CPQ) and Family Impact Scale (FIS). Additional questions explored socioeconomic characteristics, dental care setting and attendance, and parental perceptions of oral health. Group comparisons were performed using non-parametric and parametric tests, with significance set at p < 0.05.