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病例报告:TTC7A基因复合杂合突变导致的胃肠道缺陷和免疫缺陷综合征的产前诊断

Case Report: Prenatal diagnosis of gastrointestinal defects and immunodeficiency syndrome caused by compound heterozygous mutations in TTC7A gene.

作者信息

Han Shuning, Wang Miaomiao, Jin Pengzhen, Hong Jiawei, Xu Chunfei, Dong Minyue

机构信息

Women's Hospital, School of Medicine, Zhejiang University, Hangzhou, China.

Key Laboratory of Reproductive Genetics (Zhejiang University), Ministry of Education, Hangzhou, China.

出版信息

Front Immunol. 2025 Aug 18;16:1611155. doi: 10.3389/fimmu.2025.1611155. eCollection 2025.

DOI:10.3389/fimmu.2025.1611155
PMID:40901475
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12399662/
Abstract

Gastrointestinal defects and immunodeficiency syndrome (GIDID) is a rare and complex disorder characterized by concurrent dysfunction of the digestive and immune systems. Typically manifesting in infancy or early childhood, GIDID carries a severe prognosis with high early mortality rates. The syndrome has been specifically linked to mutations in the TTC7A gene located on chromosome 2p21. Although GIDID can present during the fetal period, reports of prenatal diagnosis remain exceptionally rare. In this study, we investigated a case involving a fetus with gastrointestinal abnormalities detected during prenatal screening, conceived by a consanguineous couple. Following termination of the pregnancy, whole-exome sequencing of the affected fetus revealed compound heterozygous variants (c.2378del and c.2357G>T) in the TTC7A gene (OMIM:609332). These findings provide critical insights for the prenatal diagnosis of GIDID and enhance fetal detection rate. Furthermore, this study expands the spectrum of known pathogenic mutations in the TTC7A gene and underscores the significant utility of fetal whole-exome sequencing for diagnosing this condition.

摘要

胃肠道缺陷与免疫缺陷综合征(GIDID)是一种罕见且复杂的疾病,其特征为消化系统和免疫系统同时出现功能障碍。GIDID通常在婴儿期或幼儿期表现出来,预后严重,早期死亡率高。该综合征与位于2号染色体2p21上的TTC7A基因突变有特定关联。虽然GIDID可在胎儿期出现,但产前诊断的报告极为罕见。在本研究中,我们调查了一例病例,该病例涉及一对近亲结婚夫妇所怀胎儿,在产前筛查中检测到胃肠道异常。终止妊娠后,对受影响胎儿进行全外显子测序,发现TTC7A基因(OMIM:609332)存在复合杂合变异(c.2378del和c.2357G>T)。这些发现为GIDID的产前诊断提供了关键见解,并提高了胎儿检测率。此外,本研究扩展了TTC7A基因已知致病突变的范围,并强调了胎儿全外显子测序在诊断这种疾病方面的重要作用。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5c34/12399662/a44db6ede0f0/fimmu-16-1611155-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5c34/12399662/1bae1f209104/fimmu-16-1611155-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5c34/12399662/738447c8fed3/fimmu-16-1611155-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5c34/12399662/a44db6ede0f0/fimmu-16-1611155-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5c34/12399662/1bae1f209104/fimmu-16-1611155-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5c34/12399662/738447c8fed3/fimmu-16-1611155-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5c34/12399662/a44db6ede0f0/fimmu-16-1611155-g003.jpg

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本文引用的文献

1
TTC7A Variants Results in Gastrointestinal Defects and Immunodeficiency Syndrome: Case Series and Literature Review.TTC7A基因变异导致胃肠道缺陷和免疫缺陷综合征:病例系列及文献综述
Clin Rev Allergy Immunol. 2025 Jan 28;68(1):7. doi: 10.1007/s12016-024-09017-y.
2
Systematic review of phenotypes and genotypes of patients with gastrointestinal defects and immunodeficiency syndrome-1 (GIDID1) (related to TTC7A).胃肠道缺陷与免疫缺陷综合征-1(GIDID1)(与TTC7A相关)患者的表型和基因型的系统评价
Intractable Rare Dis Res. 2024 May 31;13(2):89-98. doi: 10.5582/irdr.2023.01109.
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Drug Screen Identifies Leflunomide for Treatment of Inflammatory Bowel Disease Caused by TTC7A Deficiency.
药物筛选发现来氟米特可治疗 TTC7A 缺陷引起的炎症性肠病。
Gastroenterology. 2020 Mar;158(4):1000-1015. doi: 10.1053/j.gastro.2019.11.019. Epub 2019 Nov 16.
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TTC7A: Steward of Intestinal Health.TTC7A:肠道健康的守护者。
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Novel Mutations of the Gene and Phenotype/Genotype Comparison.该基因的新型突变及表型/基因型比较
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Hypomorphic mutation in TTC7A causes combined immunodeficiency with mild structural intestinal defects.TTC7A基因的次等位基因突变导致伴有轻度肠道结构缺陷的联合免疫缺陷。
Blood. 2015 Mar 5;125(10):1674-6. doi: 10.1182/blood-2014-08-595397.
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Immune deficiency-related enteropathy-lymphocytopenia-alopecia syndrome results from tetratricopeptide repeat domain 7A deficiency.免疫缺陷相关肠病-淋巴细胞减少-脱发综合征由四肽重复结构域 7A 缺陷引起。
J Allergy Clin Immunol. 2014 Dec;134(6):1354-1364.e6. doi: 10.1016/j.jaci.2014.07.019. Epub 2014 Aug 28.
10
Mutations in tetratricopeptide repeat domain 7A result in a severe form of very early onset inflammatory bowel disease.四肽重复结构域 7A 突变导致一种严重形式的极早发性炎症性肠病。
Gastroenterology. 2014 Apr;146(4):1028-39. doi: 10.1053/j.gastro.2014.01.015. Epub 2014 Jan 11.