Kapila Vaishali, Kalra Arjun G, Stockman David L
Internal Medicine, Central Michigan University College of Medicine, Mt. Pleasant, USA.
Internal Medicine, Brooke Army Medical Center, San Antonio, USA.
Cureus. 2021 Feb 15;13(2):e13344. doi: 10.7759/cureus.13344.
Hereditary leiomyomatosis and renal cell carcinoma (HLRCC) syndrome is believed to result from an autosomal dominant mutation in the fumarate hydratase (FH) gene on chromosome 1. It is characterized by leiomyomas, mainly uterine or cutaneous, and renal cell carcinoma (RCC). The most common type of RCC associated with HLRCC is type II papillary RCC although other types are seen. Of note, chromophobe RCC has not been described in previously documented cases of HLRCC. HLRCC is typically associated with germline mutations with occasional somatic mutations reported, however, to the best of our knowledge, none have yielded the full phenotype until now. Herein, we report a case of a 45-year-old woman who underwent a hysterectomy following a year of heavy vaginal bleeding, yielding a diagnosis of uterine leiomyomas. Eight months later, the patient presented with hematuria and was subsequently found to have a left renal mass. Following a left radical nephrectomy, histologic exam revealed a chromophobe RCC with FH deficiency.
遗传性平滑肌瘤病和肾细胞癌(HLRCC)综合征被认为是由1号染色体上的富马酸水合酶(FH)基因的常染色体显性突变引起的。其特征是存在平滑肌瘤,主要为子宫平滑肌瘤或皮肤平滑肌瘤,以及肾细胞癌(RCC)。与HLRCC相关的最常见的肾细胞癌类型是II型乳头状肾细胞癌,不过也可见其他类型。值得注意的是,在先前记录的HLRCC病例中尚未描述过嫌色性肾细胞癌。HLRCC通常与种系突变相关,不过偶尔也有体细胞突变的报告,但据我们所知,到目前为止尚无产生完整表型的情况。在此,我们报告一例45岁女性病例,该患者在经历一年严重阴道出血后接受了子宫切除术,诊断为子宫平滑肌瘤。八个月后,患者出现血尿,随后被发现左肾有肿物。在进行左肾根治性切除术后,组织学检查显示为伴有FH缺乏的嫌色性肾细胞癌。