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一名年轻女性患2型高IgM综合征,伴有罕见的AICDA基因突变变体。

Type 2 hyper-IgM syndrome with a rare variant of AICDA gene mutation in a young woman.

作者信息

Prakash Prithivi Raaj, Gupta Gaurav, Aggarwal Mukul, Baitha Upendra

机构信息

Medicine, All India Institute of Medical Sciences, New Delhi, Delhi, India.

Medicine, All India Institute of Medical Sciences, New Delhi, Delhi, India

出版信息

BMJ Case Rep. 2023 Mar 17;16(3):e253878. doi: 10.1136/bcr-2022-253878.

Abstract

We report the case of a woman in her early 20s with a history of recurrent infection, atopic dermatitis, filariasis and bilateral purulent ear discharge since childhood with tonsillar enlargement on examination. She was started on supportive care and evaluated for primary immunodeficiency disease. Blood investigations revealed increased IgM levels with reduced IgG, IgA and IgE levels. Radiological imaging of the chest revealed bilateral bronchiectasis. Otoscopic examination showed features suggestive of chronic suppurative otitis media. Next-generation sequencing identified homozygous single base pair deletion in exon 2 of the activation-induced cytidine deaminase gene. Thus, a diagnosis of hyper-IgM syndrome type 2 was confirmed. The patient was started on monthly intravenous immunoglobulin replacement therapy and is currently symptomatically better, and she remains under regular follow-up.

摘要

我们报告了一名20岁出头女性的病例,她自幼有反复感染、特应性皮炎、丝虫病和双侧脓性耳漏病史,检查发现扁桃体肿大。她开始接受支持性治疗,并接受原发性免疫缺陷病评估。血液检查显示IgM水平升高,IgG、IgA和IgE水平降低。胸部影像学检查显示双侧支气管扩张。耳镜检查显示有慢性化脓性中耳炎的特征。下一代测序在活化诱导胞苷脱氨酶基因外显子2中发现纯合单碱基对缺失。因此,确诊为2型高IgM综合征。患者开始每月接受静脉注射免疫球蛋白替代治疗,目前症状有所改善,仍在定期随访中。

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