Van Den Berg C P O M, El Ghoul K, O'Sullivan E, Guntaka P K, Resnick C M, Pullens B, Khonsari R H, Dunaway D J, Wolvius E B, Van de Lande L S, Koudstaal M J
Department of Oral and Maxillofacial Surgery, Erasmus University Medical Center, Rotterdam, the Netherlands.
Department of Computing, Imperial College London, London, United Kingdom.
Bone Rep. 2024 Jan 13;20:101738. doi: 10.1016/j.bonr.2024.101738. eCollection 2024 Mar.
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.
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.
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, -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 = -0.56, -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 ( = 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.
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.
舌骨异常与口咽功能受损有关，包括进食、吞咽和呼吸。很少有研究描述罗宾序列征（RS）患者舌骨的解剖学异常，例如矿化程度较低且体积较小的舌骨。本研究的目的是比较孤立性RS患儿的正常舌骨形态与舌骨形态。
通过对RS患儿和未受影响的对照儿童进行CT成像，获得舌骨的三维（3D）重建图像。使用主成分分析（PCA）构建3D可变形模型。采用偏最小二乘判别分析（PLS-DA）和多变量方差分析（MANOVA）来表征和比较RS患者与年龄匹配的对照组之间舌骨形状的差异。
该研究纳入了23名RS患者（平均年龄9.8±10.3个月）和46个年龄匹配的对照样本。与对照组相比，RS组观察到舌骨体积较小，大角的侧向发散更大（MANOVA，P值<0.001）。PLS-DA模型的第一个形状变量显示两组之间观察到的方差具有显著相关性（Spearman ρ = -0.56，P值<0.001）。使用对照样本和151例4岁以下受试者的CT扫描数据创建了正常舌骨形状变异的3D可变形模型（n = 197，平均年龄22.1±13.1个月）。对于正常的3D可变形模型，沿着第一主成分观察到高度的异速生长形状变异。
3D可变形模型提供了正常舌骨形态变异的全面定量描述，并允许检测孤立性RS患者与对照组之间的明显差异。
