Cho C, Linscheer W G, Bell R, Smith R
Gastroenterology. 1982 Jul;83(1 Pt 1):121-6.
Alpha-chain disease with involvement of small intestine-resulting in characteristic villus atrophy and malabsorption has not been reported in this country. We studied a 57-yr-old male who presented with a polypoid tumor of the hepatic flexure of the colon. There was no evidence of malabsorption as manifested by a normal fat balance, serum carotene, and D-xylose absorption studies and the small bowel biopsy did not show villus atrophy. The tumor in the colon was surgically removed and diagnosed as a malignant lymphoma of lymphocytic type. Tumor tissue infiltrated in the mesentery could not be excised. Alpha-chain disease protein was demonstrated in serum and urine, and also in tumor tissue by immunoperoxidase techniques. The alpha-chain disease protein was further purified and classified as subclass 1. The patient had a good clinical response to cyclophosphamide and prednisone, but still has intraabdominal lymphoma with gastric involvement, and his serum alpha-chain protein persists. This case report may represent a distinct variant of alpha-chain disease.
国内尚未报道过累及小肠导致特征性绒毛萎缩和吸收不良的α链病。我们研究了一名57岁男性,他表现为结肠肝曲的息肉样肿瘤。脂肪平衡正常、血清胡萝卜素及D-木糖吸收试验均表明无吸收不良证据,小肠活检也未显示绒毛萎缩。结肠肿瘤经手术切除,诊断为淋巴细胞型恶性淋巴瘤。肠系膜浸润的肿瘤组织无法切除。通过免疫过氧化物酶技术在血清、尿液及肿瘤组织中均证实存在α链病蛋白。α链病蛋白进一步纯化并归类为1亚类。患者对环磷酰胺和泼尼松有良好的临床反应,但仍有累及胃部的腹内淋巴瘤,其血清α链蛋白持续存在。本病例报告可能代表α链病的一种独特变异型。