We report a 50-year-old Japanese woman with typical clinical manifestations of Cronkhite-Canada Syndrome (CCS) and possible novel treatment modality for this disease. The patient was diagnosed as CCS based on the presence of several clinical manifestations, such as a diffuse alopecia, nail deformities, hypogeusia, pigmentation of skin, and abdominal discomfort combined with diarrhea and wasting. In addition, she also had multiple polypoid lesions in the gastrointestinal (GI) tract. She was first treated with hyperalimentation and corticosteroid. While this combination therapy seemed to reduce several clinical manifestations, abdominal symptoms and diarrhea recurred with the beginning of oral nutrition. Endoscopy and histology showed that inflammatory changes remained, especially in the lower intestine. Therefore, mesalazine was started. A few days after this therapy, her clinical symptoms disappeared and the polypoid lesions in the large bowel completely resolved. It was therefore possible to restart oral nutrition. We predict that the administration of mesalazine might be one of the useful therapies for CCS.
We describe a case of ulcerative colitis (UC) where clinical symptoms began abruptly within a few weeks after colon resection. The patient, a 44-year-old woman, was first referred to our hospital for the treatment of colon cancer. During the past several years, she had not had any inflammatory bowel disease-like clinical symptoms, such as frequent diarrhea or abdominal discomfort. Before the operation, both macroscopic and microscopic examination revealed that no remarkable inflammatory change was associated with the cancer in any area of her colon. At 10 days after the operation, she started to complain of frequent watery diarrhea. Two weeks after the operation, she was readmitted to our hospital because of frequent bloody diarrhea, fever, and abdominal discomfort. Based on endoscopic and histological examinations, she was diagnosed as having severe UC and was treated with hyperalimentation, predonisolone, mesalazine, and granulocyte apheresis. However, she did not respond to this combination therapy. At 45 days after the first operation, owing to sudden onset hemorrhagic shock, she underwent a second colectomy. The resected specimen of the entire colon showed severe pancolitis, and histological examination revealed severe inflammatory changes in the lamina propria together with crypt distortion, all of which were consistent with UC.
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