Canioni D, Patey N, Cuenod B, Benkerrou M, Brousse N
Department of Pathology, Hôpital Necker Enfants-Malades, Paris, France.
Pediatr Pathol Lab Med. 1997 Jul-Aug;17(4):645-51.
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.
主要组织相容性复合体(MHC)II类缺陷是一种罕见的原发性免疫缺陷疾病,其特征在于人类白细胞抗原II类表达缺陷、人类白细胞I类分子表达不一致以及对外来抗原缺乏细胞免疫和体液免疫反应。临床发病于生命早期,表现为反复感染和慢性腹泻。预后较差,死亡时的平均年龄为4岁。唯一的治愈性治疗方法是骨髓移植(BMT),它能使免疫系统得以重建。BMT应在生命早期进行,因为长期生存似乎取决于既往病毒感染的次数。我们报告了一例4岁女童通过小肠活检免疫组化发现MHC II类缺陷较晚的病例,该检查显示不存在MHC II类表达。该患儿接受了两次BMT,但因严重的病毒感染而死亡。该病例强调了对出现感染和慢性腹泻的儿童进行检查以发现MHC II类缺陷的必要性。通常,诊断通过细胞涂片进行,但如果临床上漏诊,可通过小肠活检的免疫组化来进行诊断。