主要组织相容性复合体II类缺乏需要早期诊断:1例报告。

D. Canioni, N. Patey, B. Cuenod, M. Benkerrou, N. Brousse
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引用次数: 7

摘要

主要组织相容性复合体(MHC) II类缺乏症是一种罕见的原发性免疫缺陷疾病,其特征是人白细胞抗原II类表达缺陷,人白细胞I类分子表达不一致,缺乏对外来抗原的细胞和体液免疫反应。临床发病发生在生命早期,伴有反复感染和慢性腹泻。预后差,死亡时平均年龄为4岁。唯一的治疗方法是骨髓移植(BMT),它可以让免疫系统重建。BMT应该在生命早期进行,因为长期生存似乎取决于以前病毒感染的数量。我们报告的MHC II类缺陷的情况下发现晚在一个4岁的女孩通过免疫组织化学小肠活检显示MHC II类表达缺失。这名儿童接受了两次骨髓移植,但由于严重的病毒感染而死亡。本病例强调有必要探索以感染和慢性腹泻为表现的儿童,以发现MHC II类缺乏。通常,诊断是通过细胞自旋进行的,但当临床上遗漏时,可以通过免疫组织化学在小肠活检中进行诊断。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Major histocompatibility complex class II deficiency needs an early diagnosis: report of a case.
Major histocompatibility complex (MHC) class II deficiency is a rare primary immunodeficiency disorder characterized by defects in human leukocyte antigen class II expression, inconsistent expression of human leukocyte class I molecules, and a lack of cellular and humoral immune responses to foreign antigens. Clinical onset occurs early in life with recurrent infections and chronic diarrhea. The prognosis is poor, and mean age at the time of death is 4 years. The only curative treatment is bone marrow transplantation (BMT), which allows the immune system's reconstitution. BMT should be done early in life, because long-term survival seems to depend on the number of previous viral infections. We report the case of an MHC class II deficiency discovered late in a 4-year-old girl by means of immunohistochemistry of small bowel biopsy revealing the absence of MHC class II expression. The child received a BMT twice but died because of a overwhelming viral infection. This case underlines the necessity to explore children presenting with infections and chronic diarrhea in order to find MHC class II deficiency. Usually, diagnosis is performed on cytospins, but when it has been missed clinically, it can be performed by using immunohistochemistry on small bowel biopsies.
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