Fukuoka K, Nishikawa K, Mizumoto Y, Shimoyama T, Mikasa K, Kounoike Y, Sawaki M, Narita N, Oka R, Tsuruta Y
Rinsho Ketsueki. 1989 Nov;30(11):1992-6.
A 39-year-old woman was admitted to our hospital complaining of hemosputum and right neck swelling. Pharyngography, neck CT scan and laryngoscopy revealed moderately differentiated squamous cell carcinoma of the right pyriform sinus. After series of examinations, it was found that she had pancytopenia, hypoplastic bone marrow, hyperpigmentation of the skin, cardiac anomaly, small stature, hypogonadism, chromosomal aberrations and consanguinity in her parents. These findings suggested that she was the congenital aplastic anemia, that is Fanconi's anemia, variant form. Although pepleomycin and corticosteroids were administrated for the treatment of squamous cell carcinoma and aplastic anemia, she died of cardiovascular shock due to massive hemorrhage. Flow cytometric analysis of squamous cell carcinoma showed an unusual aneuploidy DNA histogram. This is the first report on Fanconi's anemia with squamous cell carcinoma in Japan. It is said that chromosomal aberrations and impairment of DNA cross-links repair may play an important role developing of malignancy in Fanconi's anemia.
一名39岁女性因咯血和右颈部肿胀入住我院。咽造影、颈部CT扫描和喉镜检查显示右侧梨状窝中度分化鳞状细胞癌。经过一系列检查,发现她全血细胞减少、骨髓发育不全、皮肤色素沉着、心脏异常、身材矮小、性腺功能减退、染色体畸变以及父母近亲结婚。这些发现提示她患有先天性再生障碍性贫血,即范可尼贫血的变异型。尽管使用了培普利欧霉素和皮质类固醇治疗鳞状细胞癌和再生障碍性贫血,但她因大量出血死于心血管休克。鳞状细胞癌的流式细胞术分析显示出异常的非整倍体DNA直方图。这是日本首例关于范可尼贫血合并鳞状细胞癌的报告。据说染色体畸变和DNA交联修复受损可能在范可尼贫血的恶性肿瘤发生中起重要作用。