Nasal CPAP is associated with increased midfacial retrusion in children with FGFR-related craniosynostoses: risk factors and biomechanical determinants
- 1Hopital Necker-Enfants Malades: Hopital universitaire Necker-Enfants malades
Abstract
BackgroundChildren with FGFR-related craniosynostoses frequently require prolonged nasal continuous positive airway pressure (CPAP) for severe obstructive sleep apnea while definitive craniofacial procedures are delayed. We investigated whether sustained CPAP use alters midfacial growth and identified the morphological and biomechanical determinants of this effect.
MethodsIn this retrospective observational study, we combined longitudinal 3D morphometric analyses of craniofacial shape in CPAP-treated and untreated children with FGFR-related craniosynostoses with experimental measurements of mask-induced forces and pressure distribution using 3D-printed patient-specific facial models instrumented with sensors. Multivariate analyses characterized growth trajectories, and structural equation modeling (SEM) disentangled the effects of three major risk factors: age, initial morphology, and CPAP duration.
ResultsCPAP-treated patients exhibited a distinct growth trajectory characterized by greater midfacial retrusion than untreated patients with similar baseline morphology. Mask use generated mechanical loads of 6-10 N. Intentional overtightening markedly increased the total force applied to the face, but no notable difference was observed between expert and non-expert operators. Craniofacial morphology was the main determinant of force and pressure distributions and of growth response, identifying morphological risk factors. CPAP duration exaggerated these effects, particularly at younger ages. SEM supported links between morphology, mechanical loading, and deviation from expected growth.
ConclusionsNasal CPAP is associated with additional midfacial growth deviation in children with FGFR-related craniosynostoses, with susceptibility strongly dependent on baseline morphology, treatment duration, and age at initiation. These findings support systematic craniofacial growth monitoring during prolonged CPAP and consideration of respiratory management and interface fit within longitudinal craniofacial treatment planning.
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BibTeX
@misc{taverne2026nasal,
title = {{Nasal CPAP is associated with increased midfacial retrusion in children with FGFR-related craniosynostoses: risk factors and biomechanical determinants}},
author = {M. Taverne and M. Ntirubuza and Y. Zhong and A. Roussel and S. Beukes and O. Delassus and L. Griffon and M. Moazen and S. Laporte and C. Muth-Seng and P. Rouch and G. Paternoster and E. Arnaud and C. Collet and B. Fauroux and S. Khirani and R. H. Khonsari},
howpublished = {medRxiv},
year = {2026},
month = oct,
doi = {10.64898/2026.10.21.26366177},
url = {https://www.medrxiv.org/content/10.64898/2026.10.21.26366177v1},
note = {CC BY},
}
RIS
TY - UNPB
TI - Nasal CPAP is associated with increased midfacial retrusion in children with FGFR-related craniosynostoses: risk factors and biomechanical determinants
AU - M. Taverne
AU - M. Ntirubuza
AU - Y. Zhong
AU - A. Roussel
AU - S. Beukes
AU - O. Delassus
AU - L. Griffon
AU - M. Moazen
AU - S. Laporte
AU - C. Muth-Seng
AU - P. Rouch
AU - G. Paternoster
AU - E. Arnaud
AU - C. Collet
AU - B. Fauroux
AU - S. Khirani
AU - R. H. Khonsari
PY - 2026
DA - 2026/10/21
PB - medRxiv
DO - 10.64898/2026.10.21.26366177
UR - https://www.medrxiv.org/content/10.64898/2026.10.21.26366177v1
AB - BackgroundChildren with FGFR-related craniosynostoses frequently require prolonged nasal continuous positive airway pressure (CPAP) for severe obstructive sleep apnea while definitive craniofacial procedures are delayed. We investigated whether sustained CPAP use alters midfacial growth and identified the morphological and biomechanical determinants of this effect. MethodsIn this retrospective observational study, we combined longitudinal 3D morphometric analyses of craniofacial shape in CPAP-treated and untreated children with FGFR-related craniosynostoses with experimental measurements of mask-induced forces and pressure distribution using 3D-printed patient-specific facial models instrumented with sensors. Multivariate analyses characterized growth trajectories, and structural equation modeling (SEM) disentangled the effects of three major risk factors: age, initial morphology, and CPAP duration. ResultsCPAP-treated patients exhibited a distinct growth trajectory characterized by greater midfacial retrusion than untreated patients with similar baseline morphology. Mask use generated mechanical loads of 6-10 N. Intentional overtightening markedly increased the total force applied to the face, but no notable difference was observed between expert and non-expert operators. Craniofacial morphology was the main determinant of force and pressure distributions and of growth response, identifying morphological risk factors. CPAP duration exaggerated these effects, particularly at younger ages. SEM supported links between morphology, mechanical loading, and deviation from expected growth. ConclusionsNasal CPAP is associated with additional midfacial growth deviation in children with FGFR-related craniosynostoses, with susceptibility strongly dependent on baseline morphology, treatment duration, and age at initiation. These findings support systematic craniofacial growth monitoring during prolonged CPAP and consideration of respiratory management and interface fit within longitudinal craniofacial treatment planning.
ER -