Quak S H, Prabhakaran K, Kwok R, O'Reilly A P
Department of Paediatrics, National University Hospital, Singapore.
Aust Paediatr J. 1988 Feb;24(1):55-8. doi: 10.1111/j.1440-1754.1988.tb01334.x.
A 3 year old Chinese girl with watery diarrhoea, abdominal distension and hypokalaemia due to a thoracic paraspinal vasoactive intestinal peptide (VIP) secreting ganglioneuroma is reported. The pre-operative serum VIP was 314 pmol/l (normal less than 30). Her diarrhoea stopped after the removal of the tumour. The VIP was 14 pmol/l 6 months post-operatively. Review of the 19 reported cases in children with documented elevated serum VIP showed that many of the cases presented with watery diarrhoea for prolonged duration before the diagnosis was made. Earliest age of onset was 2 weeks of age. The male to female ratio was 9:10. Ganglioneuroma and ganglioneuroblastoma were the commonest tumours. Pancreatic non-beta cell hyperplasia and neurofibroma were also reported. Location of the tumour was variable: neck, chest or abdomen. Increased urinary catecholamine excretion was reported in 50% of the cases. Abdominal distension, flushing, episodic hypertension and failure to thrive were the other associated features.
报告了一名3岁中国女童,因胸段椎旁血管活性肠肽(VIP)分泌性神经节神经瘤出现水样腹泻、腹胀和低钾血症。术前血清VIP为314 pmol/l(正常低于30)。肿瘤切除后腹泻停止。术后6个月VIP为14 pmol/l。对19例有血清VIP升高记录的儿童病例回顾显示,许多病例在确诊前出现长时间水样腹泻。最早发病年龄为2周龄。男女比例为9:10。神经节神经瘤和神经节神经母细胞瘤是最常见的肿瘤。也有胰腺非β细胞增生和神经纤维瘤的报告。肿瘤位置各异:颈部、胸部或腹部。50%的病例报告尿儿茶酚胺排泄增加。腹胀、潮红、发作性高血压和发育不良是其他相关特征。