Nagashima Akari, Morimura Sohshi, Hamada Toshihisa, Shiomi Takayuki, Mori Ichiro, Sato Naoko, Nomoto Junko, Tanaka Masaki, Tsuji Shoji, Sugaya Makoto
Department of Dermatology, International University of Health and Welfare, Narita 286-8520, Japan.
Department of Pathology, International University of Health and Welfare, Narita 286-8520, Japan.
Diagnostics (Basel). 2024 Jun 17;14(12):1279. doi: 10.3390/diagnostics14121279.
The diagnosis of hereditary skin tumors is difficult for "old" diagnostic tools such as immunohistochemistry. Whole-exome sequencing analysis as a "new" diagnostic tool enables us to make a final diagnosis in spite of unknown hereditary diseases in the past. Hereditary leiomyomatosis and renal cell cancer are autosomal dominant hereditary cancer syndromes characterized by uterine myomas, cutaneous leiomyomas, and aggressive renal cell cancer. The syndrome is associated with pathogenic germline variants in the fumarate hydratase gene. Herein, we demonstrate a pathogenic germline variant of the fumarate hydratase gene in a 60-year-old woman with multiple cutaneous leiomyomas, leading to the diagnosis of hereditary leiomyomatosis and renal cell cancer. Whole-exome sequencing analysis using genomic DNA extracted from peripheral blood leukocytes revealed one germline variant in the FH gene on chromosome 1 (c.290G>A, p.Gly97Asp). She received total hysterectomy due to uterine myoma, which strongly supported the diagnosis. No tumor was detected in her kidney by computed tomography and ultrasound examination. Genetic examination for the mutation of the fumarate hydratase gene is important in order to reach the correct diagnosis and to detect renal cancer at its early stage.
对于免疫组化等“传统”诊断工具而言,遗传性皮肤肿瘤的诊断颇具难度。全外显子测序分析作为一种“新型”诊断工具,即便面对过去未知的遗传性疾病,也能让我们做出最终诊断。遗传性平滑肌瘤病和肾细胞癌是常染色体显性遗传癌症综合征,其特征为子宫肌瘤、皮肤平滑肌瘤以及侵袭性肾细胞癌。该综合征与延胡索酸水合酶基因的致病性种系变异相关。在此,我们在一名患有多发性皮肤平滑肌瘤的60岁女性中证实了延胡索酸水合酶基因的致病性种系变异,从而确诊为遗传性平滑肌瘤病和肾细胞癌。使用从外周血白细胞中提取的基因组DNA进行全外显子测序分析,结果显示1号染色体上的FH基因存在一个种系变异(c.290G>A,p.Gly97Asp)。她因子宫肌瘤接受了全子宫切除术,这有力地支持了诊断。通过计算机断层扫描和超声检查,在她的肾脏中未检测到肿瘤。对延胡索酸水合酶基因的突变进行基因检测,对于做出正确诊断以及早期发现肾癌至关重要。