Mahaffey S M, Martin L W, McAdams A J, Ryckman F C, Torres M
Department of Pediatric Surgery, Children's Hospital Medical Center, Cincinnati, OH 45229.
J Pediatr Surg. 1990 Jan;25(1):101-3. doi: 10.1016/s0022-3468(05)80172-1.
A 3-year-old child was referred with a tentative diagnosis of Hirschsprung's disease because of life-long constipation and "megacolon" demonstrated radiographically. Our rectal biopsy revealed hyperganglionosis suggestive of multiple endocrine neoplasia (MEN) type II B. This, in addition to an elevated serum calcitonin level, prompted surgical removal of her thyroid, which appeared grossly normal but on sectioning, contained a medullary carcinoma in each lobe. She remains disease-free 5 years later. Gastrointestinal symptoms are a significant component of the MEN type II B syndrome, and often antedate the full phenotypic expression of the syndrome and the development of potentially lethal endocrine neoplasms. On the basis of this experience, it is recommended that MEN II B be included in the differential diagnosis of chronic constipation.
一名3岁儿童因终生便秘及影像学显示的“巨结肠”被转诊,初步诊断为先天性巨结肠症。我们的直肠活检显示神经节细胞增多,提示为II B型多发性内分泌腺瘤病(MEN)。此外,血清降钙素水平升高,促使对其甲状腺进行手术切除,甲状腺外观大体正常,但切片检查发现每个叶均有髓样癌。5年后她仍无疾病迹象。胃肠道症状是II B型MEN综合征的重要组成部分,且往往先于该综合征的完全表型表达以及潜在致命性内分泌肿瘤的发生。基于这一经验,建议将II B型MEN纳入慢性便秘的鉴别诊断。