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与胼胝体发育不全和导水管狭窄相关的KIDINS220变异体

KIDINS220 Variant Associated With Hypoplasia of the Corpus Callosum and Aqueduct Stenosis.

作者信息

Ghannad-Zadeh Kimia, Shannon Patrick, Jobling Rebekah, Miller Elka, Chong Karen, Mathews Erin, Chitayat David, Shinar Shiri

机构信息

Temerty Faculty of Medicine, University of Toronto, Toronto, Canada.

Department of Pathology and Laboratory Medicine, Mount Sinai Hospital, University of Toronto, Toronto, Canada.

出版信息

Prenat Diagn. 2025 Jun;45(6):825-828. doi: 10.1002/pd.6804. Epub 2025 May 2.

DOI:10.1002/pd.6804
PMID:40317787
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12137044/
Abstract

KIDINS220 plays a key role in neuronal survival, differentiation, and synaptic function. Abnormalities in its expression have been linked postnatally to neurodevelopmental disorders and SINO syndrome though prenatal presentations are rarely described. We report a novel de novo heterozygous KIDINS220 variant identified prenatally associated with bilateral ventriculomegaly, abnormal anterior horns, aqueductal stenosis, and a hypoplastic corpus callosum. This is the first prenatal case of such findings in KIDINS220, emphasizing the value of trio WES/WGS for diagnosis and counseling.

摘要

KIDINS220在神经元存活、分化及突触功能中起关键作用。其表达异常在出生后与神经发育障碍和SINO综合征相关,不过产前表现鲜有描述。我们报告了一种新的产前鉴定出的KIDINS220杂合变异,与双侧脑室扩大、前角异常、导水管狭窄及胼胝体发育不全有关。这是KIDINS220相关此类发现的首例产前病例,强调了三联体全外显子组测序/全基因组测序在诊断和咨询中的价值。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/955c/12137044/a182794dbaf6/PD-45-825-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/955c/12137044/a182794dbaf6/PD-45-825-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/955c/12137044/a182794dbaf6/PD-45-825-g001.jpg

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1
KIDINS220 Variant Associated With Hypoplasia of the Corpus Callosum and Aqueduct Stenosis.与胼胝体发育不全和导水管狭窄相关的KIDINS220变异体
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本文引用的文献

1
Prenatal Diagnosis of a KIDINS220 De Novo Heterozygous Variant in a Fetus With a Complex CNS Anomaly.胎儿复杂中枢神经系统异常的 KIDINS220 从头杂合变异的产前诊断。
Prenat Diagn. 2024 Nov;44(12):1518-1521. doi: 10.1002/pd.6682. Epub 2024 Oct 4.
2
TrkA mediates effect of novel KIDINS220 mutation in human brain ventriculomegaly.TrkA介导新型KIDINS220突变在人脑脑室扩大中的作用。
Hum Mol Genet. 2021 Feb 4;29(23):3757-3764. doi: 10.1093/hmg/ddaa245.
3
Prenatal delineation of a distinct lethal fetal syndrome caused by a homozygous truncating KIDINS220 variant.
产前诊断一种由 KIDINS220 纯合截短变异引起的致死性胎儿综合征。
Am J Med Genet A. 2020 Dec;182(12):2867-2876. doi: 10.1002/ajmg.a.61858. Epub 2020 Sep 10.
4
Homozygous KIDINS220 loss-of-function variants in fetuses with cerebral ventriculomegaly and limb contractures.患有脑室扩大和肢体挛缩的胎儿中纯合子KIDINS220功能丧失变异体。
Hum Mol Genet. 2017 Oct 1;26(19):3792-3796. doi: 10.1093/hmg/ddx263.
5
Heterozygous KIDINS220/ARMS nonsense variants cause spastic paraplegia, intellectual disability, nystagmus, and obesity.杂合的KIDINS220/ARMS无义变异导致痉挛性截瘫、智力残疾、眼球震颤和肥胖。
Hum Mol Genet. 2016 Jun 1;25(11):2158-2167. doi: 10.1093/hmg/ddw082. Epub 2016 Mar 22.
6
Kidins220/ARMS mediates the integration of the neurotrophin and VEGF pathways in the vascular and nervous systems.Kidins220/ARMS 介导神经营养因子和 VEGF 通路在血管和神经系统中的整合。
Cell Death Differ. 2012 Feb;19(2):194-208. doi: 10.1038/cdd.2011.141. Epub 2011 Nov 3.