Department of Surgery, Jyväskylä Central Hospital, Jyväskylä, Finland.
Fam Cancer. 2012 Sep;11(3):395-401. doi: 10.1007/s10689-012-9526-6.
Increased risk for urological tumors has been observed in mutation carriers with Lynch syndrome (LS). In this study, we evaluated the clinical features of uroepithelial (bladder and ureter) and kidney cancers in 974 Finnish mutation carriers. Altogether 30 patients had a total of 34 urological tumors: 12 ureter, 12 bladder, and 10 kidney cancers. Urological tumor was the only tumor in 9 (30 %) patients, and metachronous other tumor occurred in 21 (70 %). The occurrence of uroepithelial cancers was significantly higher in MSH2 mutation carriers (6 %; 95 % CI, 2.7-11.0) than in MLH1 carriers (2 %; 95 % CI, 1.1-3.2) and MSH6 mutation carriers (0 %) (p = 0.014). The mean ages of patients at the time of diagnosis were: bladder cancer, 57 years; ureter cancer, 58 years; and kidney cancer, 64 years. Overall 5-year survival rates were 70 % (95 % CI, 0.32-0.89) in bladder cancer, 81 % (95 % CI, 0.45-0.95) in ureter cancer, and 75 % (95 % CI, 0.31-0.93) in kidney cancer. Cancer-specific 5-year survival rates were 70 % (95 % CI, 0.32-0.89) in bladder cancer, 91 % (95 % CI, 0.51-0.98) in ureter cancer, and 100 % in kidney cancer. In conclusion, early age of onset was observed in patients with uroepithelial tumors, but not in patients with kidney cancer. The frequency of uroepithelial tumors was significantly higher in MSH2 mutation carriers than in MLH1 carriers. Further studies with larger numbers of patients, however, are needed to evaluate the potential benefit of surveillance of urological tumors in LS.
在林奇综合征(LS)的突变携带者中已经观察到泌尿系统肿瘤的风险增加。在这项研究中,我们评估了 974 名芬兰突变携带者的尿路上皮(膀胱和输尿管)和肾脏癌症的临床特征。共有 30 名患者总共患有 34 种泌尿系统肿瘤:12 例输尿管癌,12 例膀胱癌和 10 例肾癌。9 名(30%)患者仅患有泌尿系统肿瘤,21 名(70%)患者发生了其他器官的肿瘤。MSH2 突变携带者中泌尿系统肿瘤的发生率明显高于 MLH1 携带者(6%;95%CI,2.7-11.0)和 MSH6 突变携带者(0%)(p=0.014)。患者诊断时的平均年龄分别为:膀胱癌 57 岁;输尿管癌 58 岁;肾癌 64 岁。膀胱癌总体 5 年生存率为 70%(95%CI,0.32-0.89),输尿管癌为 81%(95%CI,0.45-0.95),肾癌为 75%(95%CI,0.31-0.93)。膀胱癌的癌症特异性 5 年生存率为 70%(95%CI,0.32-0.89),输尿管癌为 91%(95%CI,0.51-0.98),肾癌为 100%。总之,患有尿路上皮肿瘤的患者发病年龄较早,但患有肾癌的患者并非如此。MSH2 突变携带者的尿路上皮肿瘤发生率明显高于 MLH1 携带者。然而,需要进行更多的患者研究来评估 LS 中泌尿系统肿瘤监测的潜在获益。