van der Post Rachel S, Vogelaar Ingrid P, Carneiro Fátima, Guilford Parry, Huntsman David, Hoogerbrugge Nicoline, Caldas Carlos, Schreiber Karen E Chelcun, Hardwick Richard H, Ausems Margreet G E M, Bardram Linda, Benusiglio Patrick R, Bisseling Tanya M, Blair Vanessa, Bleiker Eveline, Boussioutas Alex, Cats Annemieke, Coit Daniel, DeGregorio Lynn, Figueiredo Joana, Ford James M, Heijkoop Esther, Hermens Rosella, Humar Bostjan, Kaurah Pardeep, Keller Gisella, Lai Jennifer, Ligtenberg Marjolijn J L, O'Donovan Maria, Oliveira Carla, Pinheiro Hugo, Ragunath Krish, Rasenberg Esther, Richardson Susan, Roviello Franco, Schackert Hans, Seruca Raquel, Taylor Amy, Ter Huurne Anouk, Tischkowitz Marc, Joe Sheena Tjon A, van Dijck Benjamin, van Grieken Nicole C T, van Hillegersberg Richard, van Sandick Johanna W, Vehof Rianne, van Krieken J Han, Fitzgerald Rebecca C
Department of Pathology, Radboud University Medical Center, Nijmegen, The Netherlands.
Department of Human Genetics, Radboud University Medical Center, Nijmegen, The Netherlands.
J Med Genet. 2015 Jun;52(6):361-74. doi: 10.1136/jmedgenet-2015-103094. Epub 2015 May 15.
Germline CDH1 mutations confer a high lifetime risk of developing diffuse gastric (DGC) and lobular breast cancer (LBC). A multidisciplinary workshop was organised to discuss genetic testing, surgery, surveillance strategies, pathology reporting and the patient's perspective on multiple aspects, including diet post gastrectomy. The updated guidelines include revised CDH1 testing criteria (taking into account first-degree and second-degree relatives): (1) families with two or more patients with gastric cancer at any age, one confirmed DGC; (2) individuals with DGC before the age of 40 and (3) families with diagnoses of both DGC and LBC (one diagnosis before the age of 50). Additionally, CDH1 testing could be considered in patients with bilateral or familial LBC before the age of 50, patients with DGC and cleft lip/palate, and those with precursor lesions for signet ring cell carcinoma. Given the high mortality associated with invasive disease, prophylactic total gastrectomy at a centre of expertise is advised for individuals with pathogenic CDH1 mutations. Breast cancer surveillance with annual breast MRI starting at age 30 for women with a CDH1 mutation is recommended. Standardised endoscopic surveillance in experienced centres is recommended for those opting not to have gastrectomy at the current time, those with CDH1 variants of uncertain significance and those that fulfil hereditary DGC criteria without germline CDH1 mutations. Expert histopathological confirmation of (early) signet ring cell carcinoma is recommended. The impact of gastrectomy and mastectomy should not be underestimated; these can have severe consequences on a psychological, physiological and metabolic level. Nutritional problems should be carefully monitored.
种系CDH1突变会使患弥漫性胃癌(DGC)和小叶性乳腺癌(LBC)的终生风险升高。组织了一次多学科研讨会,以讨论基因检测、手术、监测策略、病理报告以及患者在多个方面的观点,包括胃切除术后的饮食。更新后的指南包括修订后的CDH1检测标准(考虑一级和二级亲属):(1)任何年龄有两名或更多胃癌患者的家庭,其中一名确诊为DGC;(2)40岁之前患DGC的个体;(3)同时诊断为DGC和LBC的家庭(其中一项诊断在50岁之前)。此外,对于50岁之前患双侧或家族性LBC的患者、患有DGC和唇腭裂的患者以及具有印戒细胞癌前体病变的患者,可考虑进行CDH1检测。鉴于侵袭性疾病相关的高死亡率,建议在专业中心对携带致病性CDH1突变的个体进行预防性全胃切除术。建议对携带CDH1突变的女性从30岁开始每年进行乳腺MRI检查以监测乳腺癌。对于目前选择不进行胃切除术的患者、携带意义未明的CDH1变异的患者以及符合遗传性DGC标准但无种系CDH1突变的患者,建议在经验丰富的中心进行标准化内镜监测。建议进行专家组织病理学确认(早期)印戒细胞癌。胃切除术和乳房切除术的影响不应被低估;这些手术在心理、生理和代谢层面可能会产生严重后果。应仔细监测营养问题。