Pediatric Otolaryngology and Head and Neck Surgery Department, Necker Enfants-Malades Assistance Publique - Hôpitaux de Paris, Université de Paris, Paris, France.
Pediatric Otolaryngology and Head and Neck Surgery Department, Necker Enfants-Malades Assistance Publique - Hôpitaux de Paris, Université de Paris, Paris, France.
Int J Pediatr Otorhinolaryngol. 2021 Jul;146:110764. doi: 10.1016/j.ijporl.2021.110764. Epub 2021 May 11.
Microtia is a congenital auricular malformation, often part of a syndromic form (35%-55% of cases). The accurate prevalence of associated malformations remains to be determined with regard to the heterogeneous results of the previous studies. This study aims to describe in a large population cohort the abnormalities associated with microtia and to determine the most suitable assessment for these children.
This is a retrospective and observational cohort study collecting data from the medical records of children affected by microtia, diagnosed or followed-up between 2007 and 2017. Data were collected via a computer database. Clinical data, as well as imaging or genetic results, were noted.
Six hundred ninety four children were included, 587 (84.6%) with unilateral and 107 (15.4%) with bilateral microtia. Inner ear malformations were observed in 14.1% of the ears. The main associated anomalies were hemifacial microsomia (29%), velopharyngeal insufficiency (9%), ophthalmologic (6.2%), vertebral (5.9%), cardiac (5.5%) and kidney (3%) abnormalities. Main identified entities were Goldenhar, Treacher-Collins and Guion-Almeida syndromes.
A comprehensive clinical assessment must be completed when microtia is diagnosed. Besides screening well-known oculo-auriculo-vertebral spectrum malformations, velopharyngeal insufficiency should be systematically sought. Specialized care must be provided to the very frequently associated hemifacial macrosomia. Mild forms of this last malformation may correspond to Guion-Almeida syndrome, especially in cases of learning disability.
小耳畸形是一种先天性耳廓畸形,常为综合征形式的一部分(35%-55%的病例)。先前研究的结果存在异质性,因此仍需确定相关畸形的准确患病率。本研究旨在描述大样本队列中小耳畸形相关的异常,并确定这些儿童最适合的评估方法。
这是一项回顾性和观察性队列研究,收集了 2007 年至 2017 年间诊断或随访的患有小耳畸形的儿童的病历数据。数据通过计算机数据库收集。记录了临床数据以及影像学或遗传学结果。
共纳入 694 名儿童,587 名(84.6%)为单侧小耳畸形,107 名(15.4%)为双侧小耳畸形。14.1%的耳朵存在内耳畸形。主要相关异常为颜面半侧短小症(29%)、腭咽闭合不全(9%)、眼科(6.2%)、脊柱(5.9%)、心脏(5.5%)和肾脏(3%)异常。主要识别出的病种为 Goldenhar、Treacher-Collins 和 Guion-Almeida 综合征。
诊断小耳畸形时必须进行全面的临床评估。除了筛查已知的眼-耳-脊椎发育不良综合征外,还应系统寻找腭咽闭合不全。必须为非常常见的颜面半侧短小症提供专门护理。这种最后一种畸形的轻度形式可能对应于 Guion-Almeida 综合征,尤其是在存在学习障碍的情况下。