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芬兰林奇综合征家系突变携带者的死因。

Causes of death of mutation carriers in Finnish Lynch syndrome families.

机构信息

Department of Surgery, Jyväskylä Central Hospital, Jyväskylä, Finland.

出版信息

Fam Cancer. 2012 Sep;11(3):467-71. doi: 10.1007/s10689-012-9537-3.

Abstract

Lynch syndrome (LS) is an autosomal dominant cancer syndrome including increased life-long risk for colorectal (CRC) and endometrial (EC) cancer, but also for cancers of other types. The risk for CRC is up to 70-80 % and for EC up to 50-60 %. Due to screening and early diagnosing the mortality related to CRC and EC seems to be low. In spite of many studies on surveillance of mutation carriers, there is no comprehensive evaluation on causes of death in LS families. The disease history and cause of death of all the deceased, tested mutation carriers and their mutation negative relatives in the Finnish LS families (N = 179) was examined utilizing hospital records and relevant national registries. Out of 1069 mutation carriers 151 had succumbed; 97 (64 %) from cancer. Out of 1146 mutation-negative family 44 members had died; 11 (25 %) of them from cancer. In 12 (7.7 %) of the deceased mutation carriers no cancer had been diagnosed. The mean age of death from cancer was 63.2 years vs. 68.8 years from non-cancer causes. Only 7.9 % of the patients with CRC had died from CRC and 5 % of those with EC, respectively. 61 % of the cancer deaths were related to extra-colonic, extra-endometrial cancers. The cumulative overall and cancer specific death rates were significantly increased in Mut+ compared to Mut- family members. Even surveillance yields decrease in the life-long risk and mortality of the most common cancers CRC and EC in LS, almost all mutation carriers will contract with cancer, and two thirds of the deceased have died from cancer. This should be taken in account in genetic counseling. Mutation carriers should be encouraged to seek help for abnormal symptoms.

摘要

林奇综合征(LS)是一种常染色体显性遗传的癌症综合征,包括终生增加结直肠癌(CRC)和子宫内膜癌(EC)的风险,但也增加其他类型癌症的风险。CRC 的风险高达 70-80%,EC 高达 50-60%。由于筛查和早期诊断,CRC 和 EC 的死亡率似乎较低。尽管对突变携带者的监测进行了许多研究,但对 LS 家族中死亡原因尚未进行全面评估。利用医院记录和相关的国家登记处,检查了所有已故、经测试的突变携带者及其突变阴性亲属(芬兰 LS 家族,N=179)的病史和死因。在 1069 名突变携带者中,有 151 人死亡;其中 97 人(64%)死于癌症。在 1146 名突变阴性家族成员中,有 44 人死亡;其中 11 人(25%)死于癌症。在 12 名(7.7%)已故的突变携带者中,没有诊断出癌症。癌症相关死亡的平均年龄为 63.2 岁,而非癌症相关死亡的平均年龄为 68.8 岁。CRC 和 EC 患者的死亡人数分别仅占 CRC 和 EC 患者的 7.9%和 5%。61%的癌症死亡与结直肠外、子宫内膜外癌症有关。与 Mut-家族成员相比,Mut+家族成员的总体和癌症特异性死亡率均显著增加。即使监测使 LS 中最常见的 CRC 和 EC 癌症的终生风险和死亡率降低,几乎所有突变携带者都会患上癌症,三分之二的死者死于癌症。这在遗传咨询中应予以考虑。应鼓励突变携带者寻求异常症状的帮助。

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