Ge Wei-Wei, Chen Zai-Ming, Chou Meng-Wei, Ismail Ferina, Chen Guang, Wu Li-Ming, Yang Jian-Qiang
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.
Clin Cosmet Investig Dermatol. 2024 May 16;17:1111-1116. doi: 10.2147/CCID.S462273. eCollection 2024.
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.
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。