Hughes L J, Michels V V
University of Health Sciences, College of Osteopathic Medicine, Kansas City, Missouri.
Am J Med Genet. 1992 Aug 1;43(6):1023-5. doi: 10.1002/ajmg.1320430621.
Infantile and childhood hepatoblastoma occurs more frequently in persons heterozygous for the familial adenomatous polyposis (FAP) gene than in the general population. This observation is based on numerous case reports plus the results of an international survey of FAP registries. However, the frequency of this rare tumor in FAP patients is unknown. In a retrospective review of our family history data, 2/470 (0.42%) children born to 241 patients with FAP had hepatoblastoma. This figure is significantly higher than the 1/100,000 incidence of hepatoblastoma in the general population. However, for genetic counseling purposes, an empiric risk of less than 1% for hepatoblastoma can be cited to persons with FAP for their children.
与普通人群相比,家族性腺瘤性息肉病(FAP)基因杂合子人群中婴幼儿和儿童期肝母细胞瘤的发病率更高。这一观察结果基于大量病例报告以及对FAP登记处的国际调查结果。然而,FAP患者中这种罕见肿瘤的发病率尚不清楚。在对我们家族史数据的回顾性研究中,241名FAP患者所生的470名儿童中有2名(0.42%)患肝母细胞瘤。这一数字显著高于普通人群中1/100,000的肝母细胞瘤发病率。然而,出于遗传咨询的目的,对于患有FAP的人,可向其告知其子女患肝母细胞瘤的经验风险低于1%。