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22q11.2缺失综合征的神经影像学表现。

Neuroradiographic findings in 22q11.2 deletion syndrome.

作者信息

Bohm Lauren A, Zhou Tom C, Mingo Tyler J, Dugan Sarah L, Patterson Richard J, Sidman James D, Roby Brianne B

机构信息

University of Minnesota, Minneapolis, Minnesota.

ENT and Facial Plastic Surgery, Children's Minnesota, Children's Specialty Center, Minneapolis, Minnesota.

出版信息

Am J Med Genet A. 2017 Aug;173(8):2158-2165. doi: 10.1002/ajmg.a.38304. Epub 2017 Jun 3.

Abstract

22q11.2 deletion syndrome (22q11.2DS) is a common genetic disorder with enormous phenotypic heterogeneity. Despite the established prevalence of developmental and neuropsychiatric issues in this syndrome, its neuroanatomical correlates are not as well understood. A retrospective chart review was performed on 111 patients diagnosed with 22q11.2DS. Of the 111 patients, 24 with genetically confirmed 22q11.2 deletion and brain MRI or MRA were included in this study. The most common indications for imaging were unexplained developmental delay (6/24), seizures of unknown etiology (5/24), and unilateral weakness (3/24). More than half (13/24) of the patients had significant radiographic findings, including persistent cavum septi pellucidi and/or cavum vergae (8/24), aberrant cortical veins (6/24), polymicrogyria or cortical dysplasia (4/24), inner ear deformities (3/24), hypoplastic internal carotid artery (2/24), and hypoplastic cerebellum (1/24). These findings reveal the types and frequencies of brain malformations in this case series, and suggest that the prevalence of neuroanatomical abnormalities in 22q11.2DS may be underestimated. Understanding indications for imaging and frequently encountered brain malformations will result in early diagnosis and intervention in an effort to optimize patient outcomes.

摘要

22q11.2缺失综合征(22q11.2DS)是一种常见的遗传性疾病,具有极大的表型异质性。尽管该综合征中发育和神经精神问题的既定患病率较高,但其神经解剖学相关性尚未得到很好的理解。对111例诊断为22q11.2DS的患者进行了回顾性病历审查。在这111例患者中,本研究纳入了24例经基因证实有22q11.2缺失且进行了脑部MRI或MRA检查的患者。最常见的影像学检查指征是不明原因的发育迟缓(6/24)、病因不明的癫痫发作(5/24)和单侧无力(3/24)。超过一半(13/24)的患者有显著的影像学表现,包括持续的透明隔腔和/或穹窿腔(8/24)、异常的皮质静脉(6/24)、多小脑回或皮质发育异常(4/24)、内耳畸形(3/24)、颈内动脉发育不全(2/24)和小脑发育不全(1/24)。这些发现揭示了该病例系列中脑畸形的类型和频率,并表明22q11.2DS中神经解剖学异常的患病率可能被低估。了解影像学检查指征和常见的脑畸形将有助于早期诊断和干预,以优化患者的治疗效果。

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