主要组织相容性复合体II类缺陷超过婴儿期。

IF 1.5 Q4 IMMUNOLOGY
Case Reports in Immunology Pub Date : 2025-07-27 eCollection Date: 2025-01-01 DOI:10.1155/crii/8570051
Aziza Bachir Kattra, Ibtihal Benhsaien, Asmaa Drissi Bourhanbour, Zahra Aadam, Abderrahmane Errami, Fatima Ailal, Ahmed Aziz Bousfiha, Jalila El Bakkouri
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引用次数: 0

摘要

主要组织相容性复合体类(MHC)-II缺乏症是一种罕见的常染色体隐性联合免疫缺陷,占北非和中东先天性免疫缺陷(IEI)病例的4.1%。大多数患者活不过10岁。本研究中描述的MHC-II缺乏症罕见且不寻常。我们报告了四例摩洛哥血统的MHC-II缺乏症无关患者。采用流式细胞术对淋巴细胞亚群进行免疫分型和人白细胞抗原dr (HLA-DR)表达分析。通过直接测序进行遗传分析。患者的平均年龄为18.75岁(16-26岁);平均诊断年龄14.07岁,平均发病年龄5.25个月。临床表现为复发性肺部感染,以支气管扩张和出血性直肠结肠炎为主。所有患者均无HLA-DR表达,并在RFXANK中检测到c.338-25_338del突变。我们的3名患者(75%)仍然活着,并每月接受静脉注射免疫球蛋白(IVIG)治疗。在所有年龄组的联合免疫缺陷的鉴别诊断中考虑MHC-II缺乏是很重要的。需要进一步的研究来阐明与这种情况相关的各种表型。
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 Beyond Infancy.

Major Histocompatibility Complex Class II Deficiency Beyond Infancy.

Major Histocompatibility Complex Class II Deficiency Beyond Infancy.

Major Histocompatibility Complex Class II Deficiency Beyond Infancy.

Major histocompatibility complex class (MHC)-II deficiency is a rare autosomal recessive combined immunodeficiency, accounting for 4.1% of inborn errors of immunity (IEI) cases in North Africa and the Middle East. Most patients do not survive beyond the age of 10 years. The cases described in this study are rare and unusual for MHC-II deficiency. We report the cases of four unrelated patients of Moroccan origin with MHC-II deficiency. Immunophenotyping of lymphocyte subpopulations and analysis of human leukocyte antigen-DR (HLA-DR) expression were performed using flow cytometry. Genetic analysis was conducted through direct sequencing. The mean age of our patients was 18.75 years (range 16-26 years); the mean age at diagnosis was 14.07 years, and the mean age of onset of symptoms was 5.25 months. The clinical presentation is characterized by recurrent pulmonary infections with predominant bronchial dilatation and hemorrhagic rectocolitis. The diagnosis was confirmed in all patients by absence of HLA-DR expression and detection of the c.338-25_338del mutation in RFXANK. Three (75%) of our patients are still alive and are on monthly intravenous immunoglobulin (IVIG) therapy. It is important to consider MHC-II deficiency in the differential diagnosis of combined immunodeficiencies across all age groups. Further studies are needed to elucidate the various phenotypes associated with this condition.

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来源期刊
CiteScore
1.90
自引率
0.00%
发文量
14
审稿时长
15 weeks
期刊介绍: Case Reports in Immunology is a peer-reviewed, Open Access journal that publishes case reports and case series related to allergies, immunodeficiencies, autoimmune diseases, immune disorders, cancer immunology and transplantation immunology.
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