一个新的突变在转谷氨酰胺酶-1基因鉴定在一个collodion婴儿:一个病例报告。

IF 1.5 4区 医学 Q4 MEDICINE, RESEARCH & EXPERIMENTAL
Journal of International Medical Research Pub Date : 2025-10-01 Epub Date: 2025-10-08 DOI:10.1177/03000605251382379
Wang Lixiang, Li Xianghong, Yang Lexia, Xi Hongmin, Li Liangliang
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引用次数: 0

摘要

常染色体隐性遗传性鱼鳞病是一组以异常角化为特征的皮肤疾病。胶体婴儿表型是一种罕见的常染色体隐性先天性鱼鳞病的表型,其特征是新生儿被一层紧密的半透明膜包裹,这导致了重大的医学挑战。本病例报告描述了一名在妊娠35周零6天出生的男婴,他出现了collodion婴儿综合征。本病例表现为非大疱性先天性鱼鳞样红皮病样表型,以弥漫性红斑和细鳞为特征。遗传分析显示,TGM1中存在两个复合杂合突变:第3外显子C . 425g >T (p.a g142leu),这是先前报道的非大疱性先天性鱼鳞状红皮病的致病热点;第8外显子C . 1198a >C (p.a n400his),这一突变不仅与板层状鱼鳞病有关,而且在非大疱性先天性鱼鳞状红皮病中也检测到。以减轻症状和支持性护理为重点的综合治疗策略显著改善,患儿住院6天后出院。本病例强调了早期诊断、多学科护理和基因检测在管理常染色体隐性先天性鱼鳞病中的重要性。新TGM1突变的鉴定有助于扩大突变谱,并可能为未来的诊断和治疗方法提供信息。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
A novel mutation in the transglutaminase-1 gene identified in a collodion baby: A case report.

Autosomal recessive congenital ichthyosis is a group of skin disorders characterized by abnormal keratinization. The collodion baby phenotype is a rare phenotype of autosomal recessive congenital ichthyosis characterized by a tight, translucent membrane that encases the newborn, which leads to significant medical challenges. This case report describes a male infant born at 35 weeks and 6 days of gestation who presented with the collodion baby syndrome. The present case exhibited a nonbullous congenital ichthyosiform erythroderma-like phenotype, characterized by diffuse erythema and fine scaling. Genetic analysis revealed two compound heterozygous mutations in TGM1: c.425G>T (p.Arg142Leu) in exon 3, previously reported as a pathogenic hotspot in nonbullous congenital ichthyosiform erythroderma, and a novel mutation, c.1198A>C (p.Asn400His) in exon 8, which has not only been associated with lamellar ichthyosis but has also been detected in nonbullous congenital ichthyosiform erythroderma. A comprehensive treatment strategy focused on symptom alleviation and supportive care led to significant improvement, and the infant was discharged after 6 days of hospitalization. This case highlights the importance of early diagnosis, multidisciplinary care, and genetic testing in managing autosomal recessive congenital ichthyosis. The identification of novel TGM1 mutations contributes to the expanding mutation spectrum and may inform future diagnostic and therapeutic approaches.

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来源期刊
CiteScore
3.20
自引率
0.00%
发文量
555
审稿时长
1 months
期刊介绍: _Journal of International Medical Research_ is a leading international journal for rapid publication of original medical, pre-clinical and clinical research, reviews, preliminary and pilot studies on a page charge basis. As a service to authors, every article accepted by peer review will be given a full technical edit to make papers as accessible and readable to the international medical community as rapidly as possible. Once the technical edit queries have been answered to the satisfaction of the journal, the paper will be published and made available freely to everyone under a creative commons licence. Symposium proceedings, summaries of presentations or collections of medical, pre-clinical or clinical data on a specific topic are welcome for publication as supplements. Print ISSN: 0300-0605
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