Hyoid bone morphology in patients with isolated robin sequence – A case-control study utilizing 3D morphable models
C.P.O.M. Van Den Berg,
K. El Ghoul,
E. O'Sullivan,
P.K. Guntaka,
C.M. Resnick,
B. Pullens,
R.H. Khonsari,
D.J. Dunaway,
E.B. Wolvius,
L.S. Van de Lande,
M.J. Koudstaal
Affiliations
C.P.O.M. Van Den Berg
Department of Oral and Maxillofacial Surgery, Erasmus University Medical Center, Rotterdam, the Netherlands
K. El Ghoul
Department of Oral and Maxillofacial Surgery, Erasmus University Medical Center, Rotterdam, the Netherlands; Corresponding author.
E. O'Sullivan
Department of Computing, Imperial College London, London, United Kingdom
P.K. Guntaka
Department of Plastic and Oral Surgery, Boston Children's Hospital, Boston, United States of America
C.M. Resnick
Department of Plastic and Oral Surgery, Boston Children's Hospital, Boston, United States of America
B. Pullens
Department of Otorhinolaryngology, Erasmus University Medical Center, Rotterdam, the Netherlands
R.H. Khonsari
Service de Chirurgie Maxillofaciale et Chirurgie Plastique, Hôpital Necker - Enfants Malades, Assistance Publique - Hôpitaux de Paris, Faculté de Médecine, Université Paris Cité, Paris, France
D.J. Dunaway
Craniofacial Unit, University College London, Great Ormond Street Hospital, London, United Kingdom
E.B. Wolvius
Department of Oral and Maxillofacial Surgery, Erasmus University Medical Center, Rotterdam, the Netherlands
L.S. Van de Lande
Department of Oral and Maxillofacial Surgery, Erasmus University Medical Center, Rotterdam, the Netherlands; Service de Chirurgie Maxillofaciale et Chirurgie Plastique, Hôpital Necker - Enfants Malades, Assistance Publique - Hôpitaux de Paris, Faculté de Médecine, Université Paris Cité, Paris, France; Craniofacial Unit, University College London, Great Ormond Street Hospital, London, United Kingdom
M.J. Koudstaal
Department of Oral and Maxillofacial Surgery, Erasmus University Medical Center, Rotterdam, the Netherlands; Department of Plastic and Oral Surgery, Boston Children's Hospital, Boston, United States of America; Craniofacial Unit, University College London, Great Ormond Street Hospital, London, United Kingdom
Background: Abnormalities of the hyoid bone are associated with impairment of oropharyngeal functions including feeding, swallowing, and breathing. Few studies have characterized anatomic abnormalities of the hyoid in patients with Robin sequence (RS), e.g. a less mineralized and voluminous hyoid. The purpose of this study was to compare normal hyoid bone morphology and hyoid bone morphology in children with isolated RS. Methods: Three-dimensional (3D) reconstructions of the hyoid bone were obtained from CT-imaging of children with RS and unaffected controls. A 3D morphable model was constructed using Principal Component Analysis (PCA). Partial least squares – Discriminant Analysis (PLS-DA) and multivariate analysis of variance (MANOVA) were used to characterize and compare hyoid shape differences between patients with RS and an age-matched control group. Results: The study included 23 subjects with RS (mean age 9.8 ± 10.3 months) and 46 age-matched control samples. A less voluminous hyoid was observed for the RS group with a larger lateral divergence of the greater horns compared to controls (MANOVA, p-value<0.001). The first shape variable from the PLS-DA model showed a significant correlation for the observed variance between the two groups (Spearman R = −0.56, p-value<0.001). The control samples and 151 CT-scans of subjects up to age 4 years were used to create a 3D morphable model of normal hyoid shape variation (n = 197, mean age 22.1 ± 13.1 months). For the normal 3D morphable model, a high degree of allometric shape variation was observed along the first principal component. Conclusions: The 3D morphable models provide a comprehensive and quantitative description of variation in normal hyoid bone morphology, and allow detection of distinct differences between patients with isolated RS and controls.
