Weidner N, Flanders D J, Mitros F A
Am J Surg Pathol. 1984 Oct;8(10):779-86. doi: 10.1097/00000478-198410000-00007.
Two unusual cases of colonic ganglioneuromatosis are described. One case was associated with multiple adenomatous polyps in a 74-year-old man and the second case was associated with juvenile polyps in a 16-year-old boy. To our knowledge this is the first report of colonic mucosal ganglioneuromatosis associated with multiple adenomatous polyps and the second report associated with multiple juvenile polyps. In the first case the ganglioneuromatosis was found in colonic mucosa without adenomatous changes, while in the second case ganglioneuromatosis was found both within normal mucosa and within juvenile polyps. The relationship between mucosal ganglioneuromatosis and multiple colonic polyps remains unclear. Neither patient has yet developed features to suggest either multiple endocrine neoplasia type 2b or von Recklinghausen's disease.
本文描述了两例罕见的结肠神经节瘤病病例。一例发生在一名74岁男性身上,与多发性腺瘤性息肉有关;另一例发生在一名16岁男孩身上,与幼年性息肉有关。据我们所知,这是首例与多发性腺瘤性息肉相关的结肠黏膜神经节瘤病报告,也是第二例与多发性幼年性息肉相关的报告。在第一例中,神经节瘤病发现于无腺瘤样改变的结肠黏膜;而在第二例中,神经节瘤病在正常黏膜和幼年性息肉中均有发现。黏膜神经节瘤病与多发性结肠息肉之间的关系仍不清楚。两名患者均未出现提示2b型多发性内分泌肿瘤或冯·雷克林豪森病的特征。