Article record

Pierre Robin序列征婴儿下颌骨牵张成骨术后残余阻塞性睡眠呼吸暂停的预测因素：对综合征分层的启示

Predictors of residual obstructive sleep apnea after mandibular distraction osteogenesis in infants with Pierre Robin sequence: Insight on syndromic stratification.

临床研究咽喉科IF 1.7Q2

文献信息

PMID	42906227
原文	在 PubMed 查看原文 ↗
发表日期	2026
作者	Michael Oca 查看其他 6 位作者 Suraj M Rajan；Alexis MacDonald；Melissa Kanack；Rakesh Bhattacharjee；Daniel Lesser；Tzyynong L Friesen
作者单位	Department of Otolaryngology Head and Neck Surgery, University of California San Diego, San Diego, CA, USA; Division of Otolaryngology, Department of Surgery, Rady Children's Hospital, San Diego, CA, USA. 查看其他 4 条单位信息 Department of Plastic and Reconstructive Surgery, University of Utah, Salt Lake City, UT, USA.；Division of Plastic Surgery, Department of Surgery, University of California San Diego, Rady Children's Hospital, San Diego, CA, USA.；Division of Respiratory Medicine, Department of Pediatrics, University of California San Diego, Rady Children's Hospital, San Diego, CA, USA.；Department of Otolaryngology Head and Neck Surgery, University of California San Diego, San Diego, CA, USA; Division of Otolaryngology, Department of Surgery, Rady Children's Hospital, San Diego, CA, USA. Electronic address: tfriesen@health.ucsd.edu.
期刊	International journal of pediatric otorhinolaryngology
SCI 分区	Q2
IF	1.7
研究类型	临床研究 · 临床
所属专科	咽喉科

中文摘要

背景： 下颌骨牵张成骨（MDO）可改善Pierre Robin序列征（PRS）婴儿的阻塞性睡眠呼吸暂停（OSA），但残余疾病常见。我们试图确定MDO术后残余重度OSA的预测因素。
方法： 我们开展了一项回顾性队列研究，纳入2019年至2025年间在一家三级儿科中心接受MDO及术后多导睡眠监测（PSG）的PRS婴儿。残余重度OSA预先定义为术后阻塞性呼吸暂停-低通气指数（oAHI）>10次/小时或术后需要气管切开。采用单因素logistic回归评估候选预测因素。Stickler综合征因其舌根气道生理类似于孤立性PRS，分别在综合征类别内及排除后进行分析。
结果： 33名婴儿符合纳入标准，其中11名（33.3%）存在残余重度OSA；1名需要气管切开。排除Stickler综合征的综合征诊断（比值比[OR] 5.95，95%置信区间[CI] 1.22-28.95，p=0.03）、神经性肌张力低下（OR=5.40，95%CI 1.08-26.93，p=0.04）和较高的术前oAHI（每增加1次/小时，OR=1.04，95%CI 1.01-1.07，p=0.02）与残余重度OSA相关。探索性受试者工作特征分析显示区分能力中等（AUC=0.70，95%CI 0.43-0.97），最佳术前oAHI截断值为96次/小时（敏感性50%，特异性95%）。在25名有配对PSG的婴儿中，非综合征/Stickler组与综合征组的中位oAHI下降幅度相似（90.5% vs 87.6%，p=0.3）。
结论： 三分之一的婴儿在MDO术后存在残余重度OSA。较高的术前oAHI、神经性肌张力低下以及排除Stickler综合征的综合征诊断与残余疾病相关，可能有助于识别需要更密切术后PSG监测的婴儿。

英文摘要

BACKGROUND: Mandibular distraction osteogenesis (MDO) improves obstructive sleep apnea (OSA) in infants with Pierre Robin sequence (PRS), but residual disease is common. We sought to identify predictors of residual severe OSA after MDO.
METHODS: We performed a retrospective cohort study of infants with PRS who underwent MDO and postoperative polysomnography (PSG) at a tertiary pediatric center between 2019 and 2025. Residual severe OSA was defined a priori as postoperative obstructive apnea-hypopnea index (oAHI) > 10 events/hour or postoperative need for a tracheostomy. Candidate predictors were evaluated using univariate logistic regression. Stickler syndrome was analyzed both within and excluded from the syndromic category based on its tongue-base airway physiology resembling isolated PRS.
RESULTS: Thirty-three infants met inclusion criteria, of whom 11 (33.3%) had residual severe OSA; one required tracheostomy. Syndromic diagnosis excluding Stickler syndrome (odds ratio [OR] 5.95, 95% confidence interval [CI] 1.22-28.95, p = 0.03), neurologic hypotonia (OR = 5.40, 95%CI 1.08-26.93, p = 0.04) and higher preoperative oAHI (OR = 1.04 per event/hour, 95%CI 1.01-1.07, p = 0.02) were associated with residual severe OSA. Exploratory receiver operating characteristic analysis demonstrated modest discrimination (AUC = 0.70, 95%CI 0.43-0.97) with an optimal preoperative oAHI cutoff of 96 events/hour (50% sensitivity, 95% specificity). Among 25 infants with paired PSGs, median oAHI reduction was similar between the non-syndromic/Stickler and syndromic groups (90.5% vs 87.6%, p = 0.3).
CONCLUSIONS: One-third of infants had residual severe OSA after MDO. Higher preoperative oAHI, neurologic hypotonia, and syndromic diagnosis excluding Stickler syndrome were associated with residual disease and may help identify infants who warrant closer postoperative PSG surveillance.