探索景观先天性和特发性中性粒细胞减少在摩洛哥儿童:一个全面的回顾性分析。

IF 3.3 4区 医学 Q3 IMMUNOLOGY Immunologic Research Pub Date : 2025-01-09 DOI:10.1007/s12026-024-09581-7
Zakaria Kasmi, Milouda Chihi, Zahra Aadam, Hind Ouair, Asmaa Drissi Bourhanbour, Jalila El Bakkouri, Ibtihal Benhsaien, Ahmed Aziz Bousfiha, Fatima Ailal
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引用次数: 0

摘要

先天性中性粒细胞减少症(CoN)是一种异质性的先天免疫缺陷(IEI),以复发性感染和早发性中性粒细胞减少症(NP)为特征。本研究旨在调查摩洛哥CoN和特发性中性粒细胞减少症(IN)儿童的人口统计学和临床数据。我们对CoN患者进行了回顾性研究,并分析了1999年至2018年在一家大型儿科医院临床免疫学部门诊断的CoN和IN患者的临床和实验室结果。我们确定了88例患者,51例IN, 37例con。57%为男性,43%为女性,年龄从1个月到19岁不等。发病时中位年龄为8个月,诊断时中位年龄为36个月。57%的病例有血缘关系,27.3%的病例有兄弟姐妹感染复发史。最常见的感染并发症是耳鼻喉(ENT)感染、皮肤和软组织感染以及肺部感染。CoN患者分为7种综合征:重度先天性中性粒细胞减少9例,循环中性粒细胞减少11例,糖原积存病1b型6例,嗜中性粒细胞减少斑千皮病5例,Griscelli综合征3例,Hermansky-Pudlak综合征II型2例,Cohen综合征1例。本研究提供了摩洛哥儿童队列CoN和IN的全面概述,代表了该国对这些疾病最重要的单中心调查。我们的研究结果强调了CoN的重大负担,占摩洛哥登记的IEI的5%,这一比例高于一些邻国。该研究强调了CoN患者细菌感染的早期发病和严重程度,强调了及时准确诊断的迫切需要。
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Exploring the landscape of congenital and idiopathic neutropenia in Moroccan children: a comprehensive retrospective analysis.

Congenital neutropenia (CoN) is a heterogeneous group of inborn errors of immunity (IEI) characterized by recurrent infections and early onset of neutropenia (NP). This study aimed to investigate the demographic and clinical data of children with CoN and idiopathic neutropenia (IN) in Morocco. We performed a retrospective study of patients with CoN and analyzed the clinical and laboratory findings of patients with CoN and IN diagnosed between 1999 and 2018 in a clinical immunology unit of a large pediatric hospital. We identified 88 patients, 51 with IN and 37 with CoN. Fifty-seven percent were males, and 43% were females, ranging from 1 month to 19 years. The median age at onset was 8 months, and the median at diagnosis was 36 months. Consanguinity was observed in 57% of the cases, and a history of recurrent infections in the siblings was found in 27.3%. The most common infectious complications were ear, nose, and throat (ENT) infections, skin and soft tissue infections, and lung infections. Patients with CoN were classified into seven syndromes: 9 with severe congenital neutropenia, 11 with cyclic neutropenia, 6 with glycogen storage disease type 1b, 5 with poikiloderma with neutropenia, 3 with Griscelli syndrome, 2 with Hermansky-Pudlak syndrome type II, and 1 with Cohen syndrome. This study provides a comprehensive overview of CoN and IN in a pediatric cohort from Morocco, representing the country's most considerable single-center investigation of these conditions. Our findings highlight the significant burden of CoN, accounting for 5% of IEI in the Moroccan registry, a proportion higher than in some neighboring countries. The study emphasizes the early onset and severity of bacterial infections in CoN patients, underlining the critical need for timely and accurate diagnosis.

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来源期刊
Immunologic Research
Immunologic Research 医学-免疫学
CiteScore
6.90
自引率
0.00%
发文量
83
审稿时长
6-12 weeks
期刊介绍: IMMUNOLOGIC RESEARCH represents a unique medium for the presentation, interpretation, and clarification of complex scientific data. Information is presented in the form of interpretive synthesis reviews, original research articles, symposia, editorials, and theoretical essays. The scope of coverage extends to cellular immunology, immunogenetics, molecular and structural immunology, immunoregulation and autoimmunity, immunopathology, tumor immunology, host defense and microbial immunity, including viral immunology, immunohematology, mucosal immunity, complement, transplantation immunology, clinical immunology, neuroimmunology, immunoendocrinology, immunotoxicology, translational immunology, and history of immunology.
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