[Update on the diagnosis and management of severe combined immunodeficiency].

Ryota Komori, Fumiaki Sakura, Takaki Asano, Satoshi Okada
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引用次数: 0

Abstract

Severe combined immunodeficiency (SCID) is one of the most severe forms of inborn errors of immunity (IEI), characterized by profound defects in T-cell development and function. Without treatment, affected infants typically develop life-threatening infections and often die within the first year of life. Early diagnosis before the onset of infection is critical because timely curative treatment, such as hematopoietic cell transplantation, can markedly improve survival. In recent years, newborn screening (NBS) using T-cell receptor excision circles (TRECs) has enabled presymptomatic identification of SCID, leading to substantial changes in the clinical management of this disease. However, several challenges have emerged with the widespread implementation of NBS, including false-negative results in adenosine deaminase (ADA) deficiency and difficulties in interpreting TRECs-low results in patients with underlying conditions associated with impaired thymic function. In this review, we summarize the clinical features and pathophysiology of SCID and outline the principles and current status of NBS. We also present a case of X-linked SCID associated with Down syndrome to highlight diagnostic pitfalls and the importance of comprehensive evaluation for early diagnosis.

[严重联合免疫缺陷的诊断和治疗的最新进展]。
严重联合免疫缺陷(SCID)是一种最严重的先天性免疫缺陷(IEI),其特征是t细胞发育和功能的严重缺陷。如果不进行治疗,受影响的婴儿通常会发生危及生命的感染,并往往在出生后的第一年死亡。感染发生前的早期诊断至关重要,因为及时的治疗,如造血细胞移植,可以显著提高生存率。近年来,使用t细胞受体切除环(trec)的新生儿筛查(NBS)使SCID的症状前识别成为可能,从而导致该疾病的临床管理发生实质性变化。然而,随着NBS的广泛实施,出现了一些挑战,包括腺苷脱氨酶(ADA)缺乏的假阴性结果,以及在与胸腺功能受损相关的潜在疾病患者中解释trec -低结果的困难。本文综述了SCID的临床特点和病理生理,概述了NBS的原理和现状。我们还报告了一个与唐氏综合征相关的x连锁SCID病例,以强调诊断缺陷和综合评估对早期诊断的重要性。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
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