Skip to content
DraconiticResearch
Search Sources API
203,234 studies31 sourcesUpdated 26 min ago
Peer-reviewedCC BY 4.0BMC Pediatrics

Exploring extracraniofacial anomalies in relation to facial morphology in craniofacial microsomia and microtia

  • Elsa M. Ronde1,2,3,4,5,6,7,8
  • Guido A. de Jong5,6
  • Jitske W. Nolte2,3,4
  • Marloes E. L. Nienhuijs5
  • Neil W. Bulstrode7,8
  • Thomas J. J. Maal5,6
  • Alfred G. Becking2,3,4
  • Corstiaan C. Breugem1,3,4
  • J. Bolks
  • J. Bolks
  • D. Dunaway
  • FA Ebbens
14 more authors
  • FH Kruisinga
  • TJJ Maal
  • EM Ongkosuwito
  • BJ van Royen
  • S. Schievano
  • A. Stadhouder
  • M. Tjaberinga
  • EM Ronde
  • GA de Jong
  • JW Nolte
  • MEL Nienhuijs
  • NW Bulstrode
  • AG Becking
  • CC Breugem
8 affiliations
  1. 1Department of Plastic, Reconstructive and Hand Surgery, Amsterdam UMC, University of Amsterdam, Amsterdam, the Netherlands
  2. 2Department of Oral and Maxillofacial Surgery, Amsterdam UMC, University of Amsterdam and ACTA, Academic Centre Dentistry Amsterdam, Amsterdam, the Netherlands
  3. 3Amsterdam Reproduction and Development Research Institute, Amsterdam, the Netherlands
  4. 4Amsterdam UMC Expert Center for Cleft, Craniofacial and Airway Disorders, Amsterdam, the Netherlands
  5. 5Department of Oral and Maxillofacial Surgery, Radboudumc, Nijmegen, the Netherlands
  6. 6Radboudumc 3D Lab, Radboudumc, Nijmegen, the Netherlands
  7. 7Department of Plastic Surgery, Great Ormond Street Hospital, London, UK
  8. 8UCL Great Ormond Street Institute of Child Health, London, UK
Read full textDOICite

Abstract

Background: Craniofacial microsomia (CFM) is associated with extracraniofacial anomalies, obstructive breathing, and feeding difficulties. This study aimed to explore whether patients with extracraniofacial anomalies, obstructive breathing, or feeding difficulties can be distinguished from patients without these anomalies or difficulties, based on their facial morphology.

Methods: This cross-sectional study used three-dimensional photographs and asymmetry index (ASI), facial signature (FS) scores, and principal components (PC) derived from FS scores to measure facial morphology. Univariate analyses were performed to assess significant differences in ASI, FS, and PC scores, as well as patient characteristics between patients with extracraniofacial anomalies, or feeding and/or breathing difficulties, and patients without these anomalies or difficulties. Multivariate logistic regression analyses were performed for both outcomes.

Results: Extracraniofacial anomalies were found in 84 (47%) out of 179 patients. In the multivariate analysis, the number of associated groups of craniofacial anomalies (OR 2.62 [95% CI 1.72–4.15]) and higher PC30 scores, depicting increasingly negative FS scores around the chin, maxilla, and inferior orbital rims (OR 1.06 [95% CI 1.01–1.11]) were significant independent predictors of extracraniofacial anomalies. For feeding and/or breathing difficulties, independent predictors were clefting (OR 6.45 [95% CI 2.72–16.05]), anomalies of the respiratory or gastrointestinal tracts (OR 7.63 [95% CI 2.33–28.06]), and lower PC34 scores (OR 0.91 [95% CI 0.84–0.97]), corresponding to increasingly positive FS scores of the inferior orbital rims and increasingly negative FS scores of the forehead.

Conclusions: Patients with extracraniofacial anomalies and patients with breathing or feeding difficulties had more severe facial phenotypes. Extracraniofacial anomalies were significantly and independently associated with morphological patterns possibly corresponding to bilateral orbital involvement and chin retrusion, as well as other associated craniofacial anomalies.

Supplementary Information: The online version contains supplementary material available at https://doi.org/10.1186/s12887-026-07445-7.

Related

  1. Experiences of family caregivers regarding the health of children with congenital craniofacial anomalies in Colombia
    María Mercedes Lafaurie, Lina María Vargas-Escobar, María Clara González +1
    PLOS Global Public HealthPeer-reviewedCC BY 4.0Full textDevelopmental biologyEpidemiology & public healthPLOS
  2. Deep Learning-Based Anatomical Level Classification of Craniofacial CT Slices: Comparison of CNN Architectures and Ensemble Methods
    S. Nishimoto, K. Nakajima, H. Yoshiike +3
    medRxivPreprintCC BY-NCFull textMedical imaging & radiologymedRxiv
  3. Craniofacial Arteriovenous Malformation Diagnosed in the Neonatal Period: A Case Report
    Mohammed A Obeid, Omran A Saeed, Naeem Alkheamy +2
    CureusPeer-reviewedCC BY 4.0Full textObstetrics, gynaecology & paediatricsPMC
  4. Site-Specific Survival and Complications of Extra-oral Craniofacial Implants in Irradiated and Non-irradiated Patients: A Systematic Review and Meta-Analysis
    V Srishti, Sanjeev Singh, Saumya Sharma +2
    CureusPeer-reviewedCC BYFull textOncologyEurope PMCPMC
  5. Bringing high-fidelity 3D craniofacial assessment to the chairside: A low-cost smartphone pipeline via 2D Gaussian splatting
    Fengcong Wang, Yang Lyu, Ping Liu +1
    Digital Dentistry JournalPeer-reviewedCC BYFull textGeneral medicineDOAJ

Cite

BibTeXRIS
BibTeX
@article{ronde2026exploring,
  title = {{Exploring extracraniofacial anomalies in relation to facial morphology in craniofacial microsomia and microtia}},
  author = {Elsa M. Ronde and Guido A. de Jong and Jitske W. Nolte and Marloes E. L. Nienhuijs and Neil W. Bulstrode and Thomas J. J. Maal and Alfred G. Becking and Corstiaan C. Breugem and J. Bolks and J. Bolks and D. Dunaway and FA Ebbens and FH Kruisinga and TJJ Maal and EM Ongkosuwito and BJ van Royen and S. Schievano and A. Stadhouder and M. Tjaberinga and EM Ronde and GA de Jong and JW Nolte and MEL Nienhuijs and NW Bulstrode and AG Becking and CC Breugem},
  journal = {BMC Pediatrics},
  year = {2026},
  month = aug,
  doi = {10.1186/s12887-026-07445-7},
  url = {https://doi.org/10.1186/s12887-026-07445-7},
  note = {CC BY 4.0},
}
RIS
TY  - JOUR
TI  - Exploring extracraniofacial anomalies in relation to facial morphology in craniofacial microsomia and microtia
AU  - Elsa M. Ronde
AU  - Guido A. de Jong
AU  - Jitske W. Nolte
AU  - Marloes E. L. Nienhuijs
AU  - Neil W. Bulstrode
AU  - Thomas J. J. Maal
AU  - Alfred G. Becking
AU  - Corstiaan C. Breugem
AU  - J. Bolks
AU  - J. Bolks
AU  - D. Dunaway
AU  - FA Ebbens
AU  - FH Kruisinga
AU  - TJJ Maal
AU  - EM Ongkosuwito
AU  - BJ van Royen
AU  - S. Schievano
AU  - A. Stadhouder
AU  - M. Tjaberinga
AU  - EM Ronde
AU  - GA de Jong
AU  - JW Nolte
AU  - MEL Nienhuijs
AU  - NW Bulstrode
AU  - AG Becking
AU  - CC Breugem
PY  - 2026
DA  - 2026/08/07
JO  - BMC Pediatrics
DO  - 10.1186/s12887-026-07445-7
UR  - https://doi.org/10.1186/s12887-026-07445-7
AB  - Background: Craniofacial microsomia (CFM) is associated with extracraniofacial anomalies, obstructive breathing, and feeding difficulties. This study aimed to explore whether patients with extracraniofacial anomalies, obstructive breathing, or feeding difficulties can be distinguished from patients without these anomalies or difficulties, based on their facial morphology.  Methods: This cross-sectional study used three-dimensional photographs and asymmetry index (ASI), facial signature (FS) scores, and principal components (PC) derived from FS scores to measure facial morphology. Univariate analyses were performed to assess significant differences in ASI, FS, and PC scores, as well as patient characteristics between patients with extracraniofacial anomalies, or feeding and/or breathing difficulties, and patients without these anomalies or difficulties. Multivariate logistic regression analyses were performed for both outcomes.  Results: Extracraniofacial anomalies were found in 84 (47%) out of 179 patients. In the multivariate analysis, the number of associated groups of craniofacial anomalies (OR 2.62 [95% CI 1.72–4.15]) and higher PC30 scores, depicting increasingly negative FS scores around the chin, maxilla, and inferior orbital rims (OR 1.06 [95% CI 1.01–1.11]) were significant independent predictors of extracraniofacial anomalies. For feeding and/or breathing difficulties, independent predictors were clefting (OR 6.45 [95% CI 2.72–16.05]), anomalies of the respiratory or gastrointestinal tracts (OR 7.63 [95% CI 2.33–28.06]), and lower PC34 scores (OR 0.91 [95% CI 0.84–0.97]), corresponding to increasingly positive FS scores of the inferior orbital rims and increasingly negative FS scores of the forehead.  Conclusions: Patients with extracraniofacial anomalies and patients with breathing or feeding difficulties had more severe facial phenotypes. Extracraniofacial anomalies were significantly and independently associated with morphological patterns possibly corresponding to bilateral orbital involvement and chin retrusion, as well as other associated craniofacial anomalies.  Supplementary Information: The online version contains supplementary material available at https://doi.org/10.1186/s12887-026-07445-7.
SN  - 1471-2431
ER  -