Peter A Johansson, Jane M Palmer, Lindsay McGrath, Sunil Warrier, Hayley R Hamilton, Timothy Beckman, Matthew G D'Mellow, Kelly M Brooks, William Glasson, Nicholas K Hayward, Antonia L Pritchard
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引用次数: 0
摘要
葡萄膜黑色素瘤(UM)和非骶尾部皮肤黑色素瘤(CM)尽管都是由黑色素细胞引起的,但它们的遗传特征各不相同。不过,它们也有相似之处,即它们最常影响欧洲血统的人,而且 BAP1、POT1 和 CDKN2A 的高穿透性种系变异已被证明易导致 UM 和 CM。本研究旨在进一步探索 UM 和 CM 患者的种系变异,揭示导致这些疾病的潜在遗传机制。通过外显子组测序,我们分析了澳大利亚 83 名被诊断为 UM 和 CM 患者的种系 DNA 样本。结果发现,8 名患者(10%)携带已知黑色素瘤易感基因 POT1、MITF、OCA2、SLC45A2 和 TYR 的致病突变。3例(4%)患者携带以前与其他癌症综合征相关的基因(ATR、BRIP1和MSH6)的致病变异,另有3例携带隐性癌症基因(色素沉着病和范可尼贫血症)的单偶致病变异,这表明这些人的表型穿透性降低可能会导致UM和CM的发生。这些发现凸显了进一步研究这些基因在黑色素瘤易感性中的作用的必要性。
Germline Variants in Patients Affected by Both Uveal and Cutaneous Melanoma.
Uveal melanoma (UM) and nonacral cutaneous melanoma (CM) are distinct entities with varied genetic landscapes despite both arising from melanocytes. There are, however, similarities in that they most frequently affect people of European ancestry, and high penetrance germline variants in BAP1, POT1 and CDKN2A have been shown to predispose to both UM and CM. This study aims to further explore germline variants in patients affected by both UM and CM, shedding light on the underlying genetic mechanism causing these diseases. Using exome sequencing we analysed germline DNA samples from a cohort of 83 Australian patients diagnosed with both UM and CM. Eight (10%) patients were identified that carried pathogenic mutations in known melanoma predisposition genes POT1, MITF, OCA2, SLC45A2 and TYR. Three (4%) patients carried pathogenic variants in genes previously linked with other cancer syndromes (ATR, BRIP1 and MSH6) and another three cases carried monoallelic pathogenic variants in recessive cancer genes (xeroderma pigmentosum and Fanconi anaemia), indicating that reduced penetrance of phenotype in these individuals may contribute to the development of both UM and CM. These findings highlight the need for further studies characterising the role of these genes in melanoma susceptibility.
期刊介绍:
Pigment Cell & Melanoma Researchpublishes manuscripts on all aspects of pigment cells including development, cell and molecular biology, genetics, diseases of pigment cells including melanoma. Papers that provide insights into the causes and progression of melanoma including the process of metastasis and invasion, proliferation, senescence, apoptosis or gene regulation are especially welcome, as are papers that use the melanocyte system to answer questions of general biological relevance. Papers that are purely descriptive or make only minor advances to our knowledge of pigment cells or melanoma in particular are not suitable for this journal. Keywords
Pigment Cell & Melanoma Research, cell biology, melatonin, biochemistry, chemistry, comparative biology, dermatology, developmental biology, genetics, hormones, intracellular signalling, melanoma, molecular biology, ocular and extracutaneous melanin, pharmacology, photobiology, physics, pigmentary disorders