单卵双胎GRIN2B神经发育障碍的不同表现:具有独特影像表型的病例报告。

IF 1.1 4区 医学 Q4 CLINICAL NEUROLOGY
Jernej Avsenik, Mirjana P Benedik, Mihael Rogač, Asthik Biswas, Sniya Sudhakar, Felice D'Arco, Ulrike Löbel, Kshitij Mankad
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引用次数: 0

摘要

我们描述了一组患有GRIN2B相关神经发育障碍(GRIN2B- nd)的同卵双胞胎,由于GRIN2B基因的新杂合致病变异(C . 2453t >C, p.Met818Thr),他们表现出不同的临床和影像学特征。双胞胎A表现出广泛对称的皮质发育畸形(MCD),类似于多小回症,伴有浅脑沟、侧脑室扩张以及基底节、胼胝体和海马的发育异常。在双胞胎B中,畸形特征,如脑容量减少,MCD,浅沟和侧脑室扩张,局限于左半球。结合先前发表的数据,我们的报告强调了与p.(Met818Thr)致病变异相关的可变表型,特别是具有不对称甚至单侧表现的可能性。我们讨论了遗传和环境因素之间潜在的相互作用,在同卵双胞胎的背景下这种现象。此外,我们还强调在评估明显单侧脑畸形时认识潜在遗传基础的重要性。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Divergent Presentation of GRIN2B Neurodevelopmental Disorder in Monozygotic Twins: Case Report with Unique Imaging Phenotypes.

We describe a set of monozygotic twins with Glutamate Ionotropic Receptor N-methyl-D-aspartate Type Subunit 2B-related neurodevelopmental disorder (GRIN2B-ND) who exhibited distinct clinical and imaging characteristics due to a de novo heterozygous pathogenic variant in the GRIN2B gene (c.2453T > C, p.Met818Thr). Twin A displayed extensive symmetric malformation of cortical development (MCD) resembling polymicrogyria, accompanied by shallow sulci, dilated lateral ventricles, and dysplastic appearances of the basal ganglia, corpus callosum, and hippocampi. In twin B, malformative features, such as reduced brain volume, MCD, shallow sulci, and dilated lateral ventricle, were confined to the left hemisphere. In combination with previously published data, our report highlights variable phenotypes associated with the p.(Met818Thr) pathogenic variant, specifically with a potential for asymmetric or even unilateral presentation. We discuss the potential interplay between genetic and environmental factors underlying this phenomenon within the context of monozygotic twins. In addition, we also highlight the importance of recognizing potential genetic underpinnings in the assessment of apparently unilateral brain malformations.

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来源期刊
Neuropediatrics
Neuropediatrics 医学-临床神经学
CiteScore
2.80
自引率
0.00%
发文量
94
审稿时长
>12 weeks
期刊介绍: For key insights into today''s practice of pediatric neurology, Neuropediatrics is the worldwide journal of choice. Original articles, case reports and panel discussions are the distinctive features of a journal that always keeps abreast of current developments and trends - the reason it has developed into an internationally recognized forum for specialists throughout the world. Pediatricians, neurologists, neurosurgeons, and neurobiologists will find it essential reading.
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