• 文献检索
  • 文档翻译
  • 深度研究
  • 学术资讯
  • Suppr Zotero 插件Zotero 插件
  • 邀请有礼
  • 套餐&价格
  • 历史记录
应用&插件
Suppr Zotero 插件Zotero 插件浏览器插件Mac 客户端Windows 客户端微信小程序
定价
高级版会员购买积分包购买API积分包
服务
文献检索文档翻译深度研究API 文档MCP 服务
关于我们
关于 Suppr公司介绍联系我们用户协议隐私条款
关注我们

Suppr 超能文献

核心技术专利:CN118964589B侵权必究
粤ICP备2023148730 号-1Suppr @ 2026

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验

胎儿肾脏异常的一种新型 NONO 无义变异。

A novel NONO nonsense variant in a fetus with renal abnormalities.

机构信息

Biochemistry and Molecular Genetics Department, Hospital Clinic of Barcelona and Fundacio de Recerca Clínic Barcelona-Institut d'Investigacions Biomediques August Pi i Sunyer (FRCB-IDIBAPS), Barcelona, Spain.

CIBER of Rare Diseases (CIBERER), Instituto de Salud Carlos III, Madrid, Spain.

出版信息

Prenat Diagn. 2024 Jan;44(1):77-80. doi: 10.1002/pd.6500. Epub 2023 Dec 18.

DOI:10.1002/pd.6500
PMID:38110236
Abstract

At 16 + 6-weeks a fetal scan performed in the second pregnancy of a 42 y.o. woman identified a right multicystic dysplastic kidney, left renal agenesis, absent urinary bladder, myocardial hypertrophy, increased nuchal fold, a single umbilical artery, and oligohydramnios. Trio exome sequencing analysis detected a novel pathogenic NONO variant. Postmortem examination after the termination of pregnancy confirmed the ultrasound findings and also revealed pulmonary hypoplasia, retrognathia and low-set ears. The variant was a novel de novo hemizygous pathogenic loss-of-function variant in NONO [NM_007363.5], associated with a rare X-linked recessive neurodevelopmental disorder, named intellectual developmental disorder, X-linked syndromic 34 (OMIM#300967). The postnatal characteristic features of this disorder include intellectual disability, developmental delay, macrocephaly, structural abnormalities involving the corpus callosum and/or cerebellum, left ventricular noncompaction and other congenital heart defects. In the prenatal setting, the phenotype has been poorly described, with all described cases presenting with heart defects. This case highlights the need of further clinical delineation to include renal abnormalities in the prenatal phenotype spectrum.

摘要

在第二次妊娠 16+6 周时,对一位 42 岁女性进行了胎儿扫描,发现右侧多囊性发育不良肾、左侧肾发育不全、无膀胱、心肌肥厚、颈后皮肤皱褶增厚、单脐动脉和羊水过少。三核苷酸外显子组测序分析发现了一个新的致病性 NONO 变异。妊娠终止后的尸检检查证实了超声检查的结果,还发现了肺发育不良、小下颌和低位耳。该变异是 NONO[NM_007363.5]中的新型新生纯合致病性功能丧失变异,与一种罕见的 X 连锁隐性神经发育障碍有关,命名为智力发育障碍、X 连锁综合征 34(OMIM#300967)。该疾病的产后特征包括智力残疾、发育迟缓、大头畸形、胼胝体和/或小脑结构异常、左心室致密化不全和其他先天性心脏缺陷。在产前环境中,该表型描述较差,所有描述的病例均存在心脏缺陷。本病例强调需要进一步的临床描述,将肾脏异常纳入产前表型谱中。

相似文献

1
A novel NONO nonsense variant in a fetus with renal abnormalities.胎儿肾脏异常的一种新型 NONO 无义变异。
Prenat Diagn. 2024 Jan;44(1):77-80. doi: 10.1002/pd.6500. Epub 2023 Dec 18.
2
Further delineation of the phenotypic spectrum associated with hemizygous loss-of-function variants in NONO.进一步描绘与 NONO 中杂合功能丧失变异相关的表型谱。
Am J Med Genet A. 2020 Apr;182(4):652-658. doi: 10.1002/ajmg.a.61466. Epub 2019 Dec 28.
3
Expanding the genetic and clinical spectrum of the NONO-associated X-linked intellectual disability syndrome.扩展 NONO 相关的 X 连锁智力障碍综合征的遗传和临床谱。
Am J Med Genet A. 2019 May;179(5):792-796. doi: 10.1002/ajmg.a.61091. Epub 2019 Feb 17.
4
Congenital heart defects and left ventricular non-compaction in males with loss-of-function variants in NONO.患有 NONO 功能丧失变异的男性的先天性心脏缺陷和左心室心肌致密化不全
J Med Genet. 2017 Jan;54(1):47-53. doi: 10.1136/jmedgenet-2016-104039. Epub 2016 Aug 22.
5
Case Report: Non-ossifying fibromas with pathologic fractures in a patient with -associated X-linked syndromic intellectual developmental disorder.病例报告:一名患有与X连锁综合征相关的智力发育障碍患者的非骨化性纤维瘤伴病理性骨折。
Front Genet. 2023 Jul 18;14:1167054. doi: 10.3389/fgene.2023.1167054. eCollection 2023.
6
Novel hemizygous loss-of-function variant in NONO identified in a South African boy.在一名南非男孩中发现的 NOVO 基因新型杂合性功能丧失变异。
Am J Med Genet A. 2022 Jan;188(1):373-376. doi: 10.1002/ajmg.a.62509. Epub 2021 Sep 22.
7
Genetic and phenotypic spectrum in the NONO-associated syndromic disorder.NONO 相关综合征性疾病的遗传与表型谱。
Am J Med Genet A. 2023 Feb;191(2):469-478. doi: 10.1002/ajmg.a.63044. Epub 2022 Nov 25.
8
Genomic study of severe fetal anomalies and discovery of GREB1L mutations in renal agenesis.严重胎儿畸形的基因组研究及 GREB1L 基因突变在肾发育不全中的发现。
Genet Med. 2018 Jul;20(7):745-753. doi: 10.1038/gim.2017.173. Epub 2017 Oct 26.
9
Prenatal diagnosis of a 15q24.1 microdeletion in a fetus with cerebral and urogenital abnormalities.一名患有脑和泌尿生殖系统异常胎儿的15q24.1微缺失的产前诊断
Clin Genet. 2024 Nov;106(5):537-544. doi: 10.1111/cge.14592. Epub 2024 Jul 16.
10
Prenatal diagnosis of Myhre syndrome with a heterozygous pathogenic variant in SMAD4 gene presented with thick nuchal translucency and cardiac abnormalities.SMAD4 基因杂合致病性变异导致的 Myhre 综合征的产前诊断表现为颈项透明层增厚和心脏异常。
Prenat Diagn. 2023 Sep;43(10):1366-1369. doi: 10.1002/pd.6414. Epub 2023 Aug 2.