Tsai S Y, Jeng Y M, Hwu W L, Ni Y H, Chang M H, Wang T R
Department of Pediatrics, National Taiwan University Hospital, Taipei.
J Formos Med Assoc. 1996 Feb;95(2):180-3.
A 3-month-old Chinese male infant with typical manifestations of Beckwith-Wiedemann Syndrome (BWS), such as macroglossia, hepatomegaly, umbilical hernia and hypoglycemia, presented with a large hepatic tumor. The tumor measured 7.6 x 8.0 x 7.5 cm. An open biopsy of the tumor revealed hepatoblastoma. The family refused chemotherapy, so only supportive care was given. The tumor grew very rapidly and the infant died 17 days after admission due to respiratory failure. To our knowledge, this is the first report of BWS associated with hepatoblastoma in a Chinese infant. This patient was a typical example of the association of BWS and hepatoblastoma, and the possible effect of growth factors on the rapid proliferation of the neoplasm in BWS.
一名3个月大的中国男婴,患有贝克威思-维德曼综合征(BWS)的典型表现,如巨舌、肝肿大、脐疝和低血糖,出现了一个巨大的肝脏肿瘤。肿瘤大小为7.6×8.0×7.5厘米。对肿瘤进行的开放性活检显示为肝母细胞瘤。家属拒绝化疗,因此仅给予支持性治疗。肿瘤生长非常迅速,婴儿入院17天后因呼吸衰竭死亡。据我们所知,这是中国婴儿中BWS合并肝母细胞瘤的首例报告。该患者是BWS与肝母细胞瘤关联的典型例子,以及生长因子对BWS中肿瘤快速增殖可能产生的影响。