Department of Pediatrics and Pediatric Neurology, Georg August University, Robert Koch Str. 40, 37075 Göttingen, Germany.
Am J Med Genet A. 2009 Dec;149A(12):2832-7. doi: 10.1002/ajmg.a.33111.
The oromandibular limb hypogenesis syndromes (OLHS) represent a group of rare conditions characterized by congenital malformations involving the tongue, mandible, and limbs. There is considerable overlap between the syndromes gathered under the term OLHS, and a marked variability of face and limb anomalies as well as additional malformations. In this report we describe a girl with gastroschisis and pulmonary hypoplasia in addition to features of Moebius syndrome comprising hypoplasia of the tongue and mandible, brachydactyly of halluces, cranial nerve palsies with bilateral facial paralysis and an inability to execute horizontal eye movements. Karyotyping and array-based comparative genomic hybridization were normal. This observation confirms an overlap between Moebius syndrome and OLHS and widens the spectrum of associated malformations. Intrauterine environmental factors including vascular insufficiency, high maternal fever, and drug abuse are likely to play a crucial role in the pathogenesis of this condition.
口颌肢体发育不良综合征(OLHS)是一组罕见的疾病,其特征为先天性畸形,涉及舌头、下颌骨和四肢。在 OLHS 这一术语下收集的综合征之间存在相当大的重叠,并且面部和肢体畸形以及其他畸形的表现具有明显的可变性。在本报告中,我们描述了一名女孩,其具有先天性腹裂和肺发育不良,此外还具有包括舌和下颌骨发育不良、大脚趾短缩、颅神经麻痹伴双侧面肌瘫痪和无法进行水平眼球运动的 Moebius 综合征特征。核型分析和基于阵列的比较基因组杂交正常。这一观察结果证实了 Moebius 综合征和 OLHS 之间存在重叠,并扩大了相关畸形的范围。宫内环境因素,包括血管功能不全、母体高热和药物滥用,可能在这种疾病的发病机制中起关键作用。