Department of Oral and Maxillofacial Surgery, Magna Graecia University of Catanzaro, Viale Europa, Catanzaro, 88100, Italy; Department of Maxillofacial and Plastic Surgery, Necker-Enfants Malades Hospital, 75015 Paris, France.
Department of Pediatric Radiology, Necker-Enfants Malades Hospital, 75015 Paris, France.
J Stomatol Oral Maxillofac Surg. 2020 Sep;121(4):357-362. doi: 10.1016/j.jormas.2019.08.014. Epub 2019 Sep 6.
The aim of the study was to investigate hyoid bone anomalies in patients with Pierre Robin sequence (PRS) compared to the control group, using computed tomography (CT) examination and three-dimensional reconstruction of the hyoid bone and mandible.
A retrospective study was performed of patients between birth and 12 months old with isolated PRS (i-PRS) and syndromic PRS (ni-PRS), who had undergone CT examination, and whose results were compared to the control group of the same age. DICOM data was processed to highlight bone tissue. The mandible and hyoid bones were the main targets of the three-dimensional reconstruction. The study outcomes were the analysis of hyoid bone ossification, volume, and position (distance between hyoid and mandibular symphysis). Univariate and multivariate statistical analyses were performed with α=0.05 as level of significance.
The study sample included 29 i-PRS and 21 ni-PRS patients, while 43 infants in the control group. Hyoid ossification was present in 26/50 (52%) PRS patients (14 i-PRS; 12 ni-PRS) but in 31/43 controls (72%). Statistical analysis showed that absence of hyoid ossification was significantly associated with the diagnosis of PRS (P<0.05). Only ni-PRS patients showed a significant reduction of the distance between hyoid and mandible compared to the control group (P<0.001). Hyoid volume was significantly lower only in the ni-PRS group than in controls (P<0.001).
I-PRS and ni-PRS patients differ both etiologically and clinically. Ni-PRS patients confirmed their worst clinical condition than i-PRS with severe anomalies of hyoid development, helping for their ontogeny classification.
本研究旨在通过 CT 检查和三维重建对患有 Pierre Robin 序列（PRS）的患者的舌骨异常进行研究，并与对照组进行比较。
对 50 例单纯 PRS（i-PRS）和综合征型 PRS（ni-PRS）患者进行回顾性研究，年龄在出生至 12 个月之间，所有患者均行 CT 检查，并与同年龄的对照组进行比较。处理 DICOM 数据以突出骨组织。下颌骨和舌骨是三维重建的主要目标。研究结果为舌骨骨化、体积和位置（舌骨与下颌联合的距离）分析。采用单变量和多变量统计分析，显著性水平为α=0.05。
研究样本包括 29 例 i-PRS 和 21 例 ni-PRS 患者，对照组 43 例。50 例 PRS 患者中有 26 例（52%）存在舌骨骨化（14 例 i-PRS；12 例 ni-PRS），而 43 例对照组中有 31 例（72%）存在舌骨骨化。统计分析表明，舌骨骨化缺失与 PRS 诊断显著相关（P<0.05）。只有 ni-PRS 患者的舌骨与下颌骨之间的距离明显低于对照组（P<0.001）。仅 ni-PRS 组的舌骨体积明显低于对照组（P<0.001）。
i-PRS 和 ni-PRS 患者在病因学和临床方面存在差异。ni-PRS 患者比 i-PRS 患者的临床状况更差，其舌骨发育异常严重，有助于对其进行胚胎发生分类。
