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超越戈麦斯-洛佩斯-埃尔南德斯综合征:菱脑结合发育不良中反复出现的表型主题。

Beyond Gómez-López-Hernández syndrome: recurring phenotypic themes in rhombencephalosynapsis.

机构信息

Division of Pediatric Neurology, Department of Neurology, University of Washington, Seattle, USA.

出版信息

Am J Med Genet A. 2012 Oct;158A(10):2393-406. doi: 10.1002/ajmg.a.35561. Epub 2012 Sep 10.

Abstract

Rhombencephalosynapsis (RES) is an uncommon cerebellar malformation characterized by fusion of the hemispheres without an intervening vermis. Frequently described in association with Gómez-López-Hernández syndrome, RES also occurs in conjunction with VACTERL features and with holoprosencephaly (HPE). We sought to determine the full phenotypic spectrum of RES in a large cohort of patients. Information was obtained through database review, patient questionnaire, radiographic, and morphologic assessment, and statistical analysis. We assessed 53 patients. Thirty-three had alopecia, 3 had trigeminal anesthesia, 14 had VACTERL features, and 2 had HPE with aventriculy. Specific craniofacial features were seen throughout the cohort, but were more common in patients with alopecia. We noted substantial overlap between groups. We conclude that although some distinct subgroups can be delineated, the overlapping features seen in our cohort suggest an underlying spectrum of RES-associated malformations rather than a collection of discrete syndromes.

摘要

脑桥小脑发育不良(RES)是一种罕见的小脑畸形,表现为半球融合而无蚓部间隔。常与戈麦斯-洛佩斯-埃尔南德斯综合征相关联,RES 也与 VACTERL 特征和全前脑畸形(HPE)相关联。我们试图在一个大的患者队列中确定 RES 的全表型谱。通过数据库回顾、患者问卷调查、影像学和形态评估以及统计分析来获取信息。我们评估了 53 名患者。33 名患者有脱发,3 名患者有三叉神经麻醉,14 名患者有 VACTERL 特征,2 名患者有伴有脑室的 HPE。整个队列中都能看到特定的颅面特征,但在有脱发的患者中更为常见。我们注意到各群组之间存在明显的重叠。我们得出结论,尽管可以划定一些不同的亚组,但我们队列中观察到的重叠特征表明存在 RES 相关畸形的潜在谱,而不是一系列离散的综合征。

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