Wen Xiao-Heng, Wang Lan, Wang Yu-Xuan, Qian Jia-Ming
Xiao-Heng Wen, Lan Wang, Jia-Ming Qian, Department of Gastroenterology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences, Beijing 100730, China.
World J Gastroenterol. 2014 Jun 21;20(23):7518-22. doi: 10.3748/wjg.v20.i23.7518.
Cronkhite-Canada syndrome (CCS) is a rare nonfamilial polyposis syndrome characterized by epithelial disturbances in the gastrointestinal tract and skin. The aim of this study was to investigate the clinical features and potential therapies for CCS. Six patients with CCS admitted from December 1992 to July 2008 to Peking Union Medical College Hospital were evaluated. All patients had clinical manifestation of nonhereditary gastrointestinal polyposis with diarrhea, skin hyperpigmentation, alopecia, and nail dystrophy. Fecal occult blood was positive in all six cases. Serum hemoglobin, potassium, calcium and protein were below the normal range in two cases. Anti-Saccharomyces cerevisiae and antinuclear antibodies were present in three cases. Multiple polyps were found in all patients by gastroscopy and colonoscopy, with only one in the esophagus. Histologically, there were hyperplastic polyps in five cases, tubular adenoma in three, and juvenile polyp in one with chronic inflammation and mucosal edema. Comprehensive treatment led by corticosteroids can result in partial remission of clinical symptoms, and long-term follow-up is necessary.
克朗凯特-加拿大综合征(CCS)是一种罕见的非家族性息肉病综合征,其特征为胃肠道和皮肤的上皮紊乱。本研究的目的是调查CCS的临床特征和潜在治疗方法。对1992年12月至2008年7月入住北京协和医院的6例CCS患者进行了评估。所有患者均有非遗传性胃肠道息肉病的临床表现,伴有腹泻、皮肤色素沉着、脱发和指甲营养不良。6例患者粪便潜血均为阳性。2例患者血清血红蛋白、钾、钙和蛋白质低于正常范围。3例患者存在抗酿酒酵母抗体和抗核抗体。所有患者经胃镜和结肠镜检查均发现多发息肉,仅1例位于食管。组织学检查,5例为增生性息肉,3例为管状腺瘤,1例为幼年性息肉伴慢性炎症和黏膜水肿。以糖皮质激素为主的综合治疗可使临床症状部分缓解,需要进行长期随访。