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角蛋白16(KRT16)1A结构域中的p.Arg127Pro突变在中国患者中导致先天性厚甲症:1例与肢端黑色素瘤相关的先天性厚甲症病例报告

Mutation p.Arg127Pro in the 1A Domain of KRT16 Causes Pachyonychia Congenita in Chinese Patient: A Case Report of PC Associated with Acral Melanoma.

作者信息

Ge Wei-Wei, Chen Zai-Ming, Chou Meng-Wei, Ismail Ferina, Chen Guang, Wu Li-Ming, Yang Jian-Qiang

机构信息

Department of Dermatology, Taizhou Second People's Hospital (Mental Health Center Affiliated to Taizhou University School of Medicine), Taizhou University, Taizhou, Zhejiang, 318000, People's Republic of China.

Department of Dermatology, The First Affiliated Hospital of Huzhou University, Huzhou, Zhejiang, People's Republic of China.

出版信息

Clin Cosmet Investig Dermatol. 2024 May 16;17:1111-1116. doi: 10.2147/CCID.S462273. eCollection 2024.

DOI:10.2147/CCID.S462273
PMID:38770089
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11104379/
Abstract

Pachyonychia congenita (PC) is a group of rare hereditary disorders, characterised by hypertrophic nails and palmoplantar keratoderma (PPK), particularly localised to the pressure areas of the feet. At a molecular level, it is caused by mutations in genes encoding KRT6A, KRT6B, KRT6C, KRT16, or KRT17. To identify the underlying gene mutation in a Chinese family with PC presenting with disabling palmoplantar keratoderma and subsequent associated acral melanoma. Genomic DNA was extracted from peripheral blood samples of three available individuals in the Chinese family, which included the patient and his two unaffected sisters. The index patient presented with severe palmoplantar keratoderma as well as a newly diagnosed acral malignant melanoma (MM). Whole-exome sequencing (WES) was carried out with amplification of exon 1 of KRT16 by polymerase chain reaction (PCR). PCR products were then sequenced to identify potential mutations. We identified the proline substitution mutation p.Arg127Pro (c.380G>C) in our patient's 1A domain of KRT16. The same mutation was not found in his sisters or unrelated healthy controls. The mutation (p.Arg127Pro (c.380G>C)) in KRT16 has been reported in Dutch patients with PC. However, it is the first such report of a patient with a PC of Chinese origin. In addition, the acral MM occurred under the background of genetic PPK caused by KRT16 mutation in this patient.

摘要

先天性厚甲症(PC)是一组罕见的遗传性疾病,其特征为指甲肥厚和掌跖角化病(PPK),尤其是局限于足部的受压部位。在分子水平上,它是由编码KRT6A、KRT6B、KRT6C、KRT16或KRT17的基因突变引起的。为了鉴定一个患有致残性掌跖角化病并继发相关肢端黑色素瘤的中国PC家系中的潜在基因突变。从该中国家系中三名可用个体(包括患者及其两名未受影响的姐妹)的外周血样本中提取基因组DNA。索引患者表现为严重的掌跖角化病以及新诊断的肢端恶性黑色素瘤(MM)。通过聚合酶链反应(PCR)扩增KRT16的外显子1进行全外显子测序(WES)。然后对PCR产物进行测序以鉴定潜在突变。我们在患者的KRT16的1A结构域中鉴定出脯氨酸替代突变p.Arg127Pro(c.380G>C)。在他的姐妹或无关健康对照中未发现相同突变。KRT16中的突变(p.Arg127Pro(c.380G>C))已在荷兰PC患者中报道。然而,这是首例关于中国血统PC患者的此类报告。此外,该患者的肢端MM发生在由KRT16突变引起的遗传性PPK背景下。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d58a/11104379/fe3706823dd7/CCID-17-1111-g0003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d58a/11104379/d3296848bd7c/CCID-17-1111-g0001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d58a/11104379/71f8f47324db/CCID-17-1111-g0002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d58a/11104379/fe3706823dd7/CCID-17-1111-g0003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d58a/11104379/d3296848bd7c/CCID-17-1111-g0001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d58a/11104379/71f8f47324db/CCID-17-1111-g0002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d58a/11104379/fe3706823dd7/CCID-17-1111-g0003.jpg

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本文引用的文献

1
A recurrent missense mutation in the KRT16 gene causing pachyonychia congenita in a patient.一名患者中KRT16基因的复发性错义突变导致先天性厚甲症。
Int J Dermatol. 2024 Feb;63(2):e47-e49. doi: 10.1111/ijd.16975. Epub 2023 Dec 13.
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A KRT6A and a Novel KRT16 Gene Mutations in Chinese Patients with Pachyonychia Congenita.中国先天性厚甲症患者中的KRT6A和一种新型KRT16基因突变
Int J Gen Med. 2021 Mar 17;14:903-907. doi: 10.2147/IJGM.S280160. eCollection 2021.
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Biomed Res Int. 2020 Aug 31;2020:1840415. doi: 10.1155/2020/1840415. eCollection 2020.
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Generalized bullae in a young girl with KRT6A-related pachyonychia congenita.一名年轻女性患 KRT6A 相关先天性厚甲症,出现广泛性大疱。
Pediatr Dermatol. 2020 Sep;37(5):974-976. doi: 10.1111/pde.14285. Epub 2020 Jul 14.
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A role for keratins in supporting mitochondrial organization and function in skin keratinocytes.角蛋白在支持皮肤角质形成细胞中线粒体的组织和功能中的作用。
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