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Tumor development mechanism and its risk evaluation in Werner syndrome

Werner综合征肿瘤发生机制及其风险评估

基本信息

批准号：
15590326
负责人：
ISHIKAWA Yuichi
金额：
$ 2.3万
依托单位：
Japanese Foundation for Cancer Research
依托单位国家：
日本
项目类别：
Grant-in-Aid for Scientific Research (C)
财政年份：
2003
资助国家：
日本
起止时间：
2003 至 2004
项目状态：
已结题

来源：
https://kaken.nii.ac.jp/en/grant/KAKENHI-PROJECT-15590326/
关键词：
Werner syndrome aging malignant tumors hereditary diseases tumor spectrum tumorigenesis tumor risk non-epithelial tumors 廊下

项目摘要

Werner syndrome is an interesting model for researches both of aging and cancer. We have so far revealed that six kinds of rare tumors, i.e. thyroid carcinoma, bone sarcoma, soft part sarcoma, myeloid disorders/MDS, meningioma and melanoma, were increased and that tumors common in general populations were not increased in Werner syndrome.In the current study, we have investigated (1) relationships between WRN helicase deficit and tumorigenesis and (2) evaluation of tumor risk in Werner syndrome.Regarding the first issue, for further analysis in the future, we collected seven cases of lung malignant tumors because relations between tumor causes and histology/genetic changes were relatively well established in lung tumors.For the second issue, we gathered data and/or materials of 133 non-epithelial (including 110 malignant) and 75 epithelial tumors from our related institutions and research collaborators or through examining public data bases such as the PubMed and the Igaku-Chuo-Zasshi. As a result, we updated our tumor files, providing the new tumor spectrum : 37 soft part sarcoma (including subcutaneous ones), 34 melanomas, 23 leukemia/MDS, 21 meningiomas, 14 bone sarcomas, 4 brain tumors as well as 28 thyroid carcinomas, 10 skin carcinomas, 7 liver carcinomas, 6 breast carcinomas, 6 gastric carcinomas, 5 lung carcinomas, 1 colonic carcinoma and no prostatic carcinoma. As compared with the previous statistics published in 1996, increased soft part sarcoma and skin cancers other than melanoma were noted. Particularly, it was interesting that we had now as many as 39 malignant tumors related to skin, including subcutaneous ones. Details were 23 melanomas, 5 basal cell carcinomas, 3 squamous cell carcinomas, 1 sebaceous gland carcinoma, 1 Bowen's disease, and 1 carcinoma NOS. Benign skin tumors were 1 solar keratosis, 1 nevocellular nevus, and subcutaneous sarcomas included 2 malignant fbrous histioctomas and 1 acute T cell leukemia.

Werner综合征是研究衰老和癌症的有趣模型。到目前为止，我们已经发现六种罕见肿瘤，即甲状腺癌，骨肉瘤，软组织肉瘤，髓系疾病/MDS，脑膜瘤和黑色素瘤，在Werner综合征中增加，而在一般人群中常见的肿瘤没有增加。我们已经研究了（1）WRN解旋酶缺陷和肿瘤发生之间的关系和（2）Werner综合征中肿瘤风险的评估关于第一个问题，为了将来进一步分析，我们收集了7例肺恶性肿瘤病例，因为在肺肿瘤中肿瘤原因与组织学/遗传学变化之间的关系相对较好地建立。对于第二个问题，我们从我们的相关机构和研究合作者或通过检查公共数据库如PubMed和Igaku-Chuo-Zasshi收集了133例非上皮性（包括110例恶性）和75例上皮性肿瘤的数据和/或材料。因此，我们更新了肿瘤文件，提供了新的肿瘤谱：37例软组织肉瘤（包括皮下肿瘤），34例黑色素瘤，23例白血病/MDS，21例脑膜瘤，14例骨肉瘤，4例脑肿瘤，以及28例甲状腺癌，10例皮肤癌，7例肝癌，6例乳腺癌，6例胃癌，5例肺癌，结肠癌1例，无前列腺癌。与1996年发表的统计数字比较，软组织肉瘤和黑色素瘤以外的皮肤癌的发病率有所上升。特别有趣的是，我们现在有多达39个与皮肤有关的恶性肿瘤，包括皮下肿瘤。详细信息包括23例黑色素瘤、5例基底细胞癌、3例鳞状细胞癌、1例皮脂腺癌、1例Bowen病和1例癌NOS。皮肤良性肿瘤包括日光性角化病1例，痣细胞痣1例，皮下肉瘤包括恶性纤维组织细胞瘤2例，急性T细胞白血病1例。