Child and Parent-Reported Outcomes in Children Historically Managed as Nonsyndromic Pierre Robin Sequence: Feeding, Speech, and Sleep-Disordered Breathing
Bruno Matos Santos, Alexis Poirier, Hassan Youssef, Anthony S. de Buys RoessinghObjective
To assess child- and parent-reported quality of life and functional outcomes in children managed as nonsyndromic Pierre Robin sequence (nsPRS), using child-reported and parent-reported outcome measures.
Design
Retrospective observational cross-sectional study.
Setting
Single tertiary craniofacial center.
Participants
Children aged 11-20 years followed for nsPRS between 2010 and 2021. Twelve child-parent dyads participated. A pre-specified sensitivity analysis was restricted to children meeting strict nsPRS criteria.
Outcome Measure(s)
Child-reported CLEFT-Q scores for feeding, speech, psychological well-being, and social function; parent-reported Pediatric Sleep Questionnaire (PSQ) scores and Behavioral Pediatrics Feeding Assessment Scale (BPFAS) child-behavior frequency scores calculated from the 24 available child-behavior frequency items.
Results
Mean age was 13.5 ± 3.9 years. CLEFT-Q scores showed feeding as the best-preserved child-reported domain (86.3 ± 12.3), followed by social function (77.0 ± 15.0), psychological well-being (69.9 ± 18.3), and speech (66.6 ± 15.9). Suspected obstructive sleep apnea syndrome according to PSQ screening was identified in 1/12 patients (8.3%), although habitual snoring was reported in 5/12 (41.7%). Parent-reported feeding behavior was classified as problematic in 6/12 families (50.0%). Descriptive cross-instrument comparison showed that 4/12 children reported high CLEFT-Q feeding scores despite parent-reported BPFAS concerns. Findings were directionally similar in the strict nsPRS sensitivity subgroup.
Conclusion
In our small cohort, child-reported quality of life at follow-up was globally satisfactory, with preserved feeding scores but lower speech scores. Parent-reported feeding concerns and residual sleep-related symptoms support follow-up that includes both child-reported and parent-reported outcomes, with particular attention to speech rehabilitation, feeding behavior, and sleep-related symptoms.