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新生儿筛查后尽早治疗对于提高重症联合免疫缺陷症(SCID)患者的生存率至关重要。

Newborn Screening Followed By Early Treatment is Essential to Improve Survival in SCID.

作者信息

Goebel Gabriela Assunção, Cunha Luciana Araújo Oliveira, Minafra Fernanda Gontijo, Pinto Jorge Andrade

机构信息

Hospital das Clínicas da Universidade Federal de Minas Gerais, Av. Professor Alfredo Balena, 110, Belo Horizonte, Minas Gerais, 30.130-100, Brazil.

Department of Pediatrics, Universidade Federal de Minas Gerais, Belo Horizonte, Minas Gerais, Brazil.

出版信息

J Clin Immunol. 2025 May 15;45(1):94. doi: 10.1007/s10875-025-01887-x.

Abstract

Severe combined immunodeficiency (SCID) is a heterogeneous genetic disease characterized by severe T-cell lymphopenia with a profound impairment of T- and B-cells' function and, in some types, also NK cells. Hematopoietic cell transplantation (HCT) is the only curative treatment currently available in Brazil. Late diagnosis and treatment are the main factors affecting the survival of these children. This study aims to describe the demographic, phenotypic, genotypic, and clinical characteristics of twenty SCID patients (including typical SCID, leaky-SCID, and Omenn Syndrome) followed at a Brazilian referral center and correlate these data with their clinical outcome. The children were analyzed into two groups: patients diagnosed early by newborn screening (NBS) or family history, n = 7, and patients with late diagnosis, by clinical presentation, n = 13. The 2-year overall survival (OS) of the late group was 29.2%, in contrast to the 2-year OS of the early diagnosis group of 71.4% (p = 0.053). However, despite early diagnosis in the first group, timely access to HCT was delayed, with a median of 11 months. This research reveals that survival depends not only on timely diagnosis but also on early definitive treatment. To improve SCID survival rates, developing countries need public policies that allow rapid access to curative treatment for these patients.

摘要

重症联合免疫缺陷病(SCID)是一种异质性遗传疾病,其特征为严重的T细胞淋巴细胞减少,T细胞和B细胞功能严重受损,在某些类型中,NK细胞也受损。造血细胞移植(HCT)是巴西目前唯一可用的治愈性治疗方法。诊断和治疗延迟是影响这些儿童生存的主要因素。本研究旨在描述在巴西一家转诊中心随访的20例SCID患者(包括典型SCID、渗漏型SCID和奥门综合征)的人口统计学、表型、基因型和临床特征,并将这些数据与其临床结果相关联。将这些儿童分为两组:通过新生儿筛查(NBS)或家族史早期诊断的患者,n = 7,以及通过临床表现晚期诊断的患者,n = 13。晚期组的2年总生存率(OS)为29.2%,而早期诊断组的2年OS为71.4%(p = 0.053)。然而,尽管第一组进行了早期诊断,但获得HCT的时间延迟了,中位数为11个月。本研究表明,生存不仅取决于及时诊断,还取决于早期确定性治疗。为了提高SCID的生存率,发展中国家需要制定公共政策,使这些患者能够迅速获得治愈性治疗。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/4e78/12081553/cf6083a8cc81/10875_2025_1887_Fig1_HTML.jpg

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