UNRESPONSIVE ENTEROPATHY ASSOCIATED WITH CIRCULATING ENTEROCYTE AUTOANTIBODIES IN A BOY WITH COMMON VARIABLE HYPOGAMMAGLOBULINEMIA AND TYPE-I DIABETES
UNRESPONSIVE ENTEROPATHY ASSOCIATED WITH CIRCULATING ENTEROCYTE AUTOANTIBODIES IN A BOY WITH COMMON VARIABLE HYPOGAMMAGLOBULINEMIA AND TYPE-I DIABETES
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DOI：
10.1097/00005176-198807000-00022
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发表时间：
1988-07-01
影响因子：
2.9
通讯作者：
GIORGI, PL
中科院分区：
文献类型：
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作者：
CATASSI, C;MIRAKIAN, R;GIORGI, PL
1例13岁男孩，患有常见可变性低丙种球蛋白血症和I型糖尿病，发生严重肠病，经证明对任何治疗（包括全肠外营养）均无反应。在该受试者中未发现吸收不良和/或分泌性腹泻已知病因的证据。高滴度的补体结合肠上皮细胞自身抗体持续发现在患者的血清中。这些特征表明，肠病的主要免疫缺陷受试者有自身免疫性起源。一个周期的环磷酰胺未能显示腹泻的任何改善或自身抗体滴度的显著降低。
A 13-year-old boy with common variable hypogammaglobulinemia and type I diabetes developed a severe enteropathy that proved to be unresponsive to any treatment, including total parenteral nutrition. No evidence of known etiologies of malabsorption and/or secretory diarrhea was found in this subject. A high titer of complement-fixing enterocyte autoantibody was persistently found in the patient's serum. These features suggest that the enteropathy of this primarily immunodeficient subject had an autoimmune origin. A cycle of cyclophosphamide failed to show any amelioration of the diarrhea or a significant decrease in the autoantibody titer.