SOCS1缺陷——自身免疫和自身炎症的交叉点——2例报告

IF 2 3区 医学 Q2 PEDIATRICS Frontiers in Pediatrics Pub Date : 2025-01-07 eCollection Date: 2024-01-01 DOI:10.3389/fped.2024.1516017
Kajetan Trojovsky, Maximilian Seidl, Florian Babor, Stephan Ehl, Min Ae Lee-Kirsch, Michael Friedt, Hans-Juergen Laws, Nibras Naami, Prasad Thomas Oommen, Sujal Ghosh
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引用次数: 0

摘要

细胞因子信号传导抑制因子(SOCS)蛋白在调节免疫信号通路中起着关键作用。缺乏SOCS1可导致各种自身免疫性病理。我们提出两个不相关的患者有不同的临床表现。患者1,一名来自几内亚的16岁男性,表现为埃文斯综合征、肌肉骨骼疼痛和肝酶升高。患者2,一名6岁的德国男孩,出现复发性口腔溃疡、轻度炎症性肠病和慢性复发性多灶性骨髓炎。两例患者均经基因检测诊断为SOCS1缺乏症。治疗策略包括类固醇、JAK抑制和秋水仙碱。这些病例强调了在自身免疫性或自身炎症性疾病患者以及不明原因的IgE水平升高患者中考虑SOCS1缺乏的重要性。他们强调需要对正在进行的多中心登记进行进一步研究,以更好地了解这种情况。
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SOCS1 deficiency-crossroads of autoimmunity and autoinflammation-two case reports.

Suppressors of cytokine signaling (SOCS) proteins play a critical role in regulating immune signaling pathways. Deficiency of SOCS1 leads to various autoimmune pathologies. We present two unrelated patients with distinct clinical manifestations. Patient 1, a 16-year-old male from Guinea, presented with Evans Syndrome, musculoskeletal pain and elevated liver enzymes. Patient 2, a 6-year-old German boy, developed recurrent oral aphthous ulcers, mild inflammatory bowel disease and chronic recurrent multifocal osteomyelitis. Both patients were diagnosed with SOCS1 deficiency by genetic testing. Treatment strategies included steroids, JAK inhibition and colchicine. These cases emphasize the importance of considering SOCS1 deficiency in patients with autoimmune or autoinflammatory diseases but also in patients with unexplained elevated IgE levels. They highlight the need for further research in ongoing multicenter registries to better understand this condition.

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来源期刊
Frontiers in Pediatrics
Frontiers in Pediatrics Medicine-Pediatrics, Perinatology and Child Health
CiteScore
3.60
自引率
7.70%
发文量
2132
审稿时长
14 weeks
期刊介绍: Frontiers in Pediatrics (Impact Factor 2.33) publishes rigorously peer-reviewed research broadly across the field, from basic to clinical research that meets ongoing challenges in pediatric patient care and child health. Field Chief Editors Arjan Te Pas at Leiden University and Michael L. Moritz at the Children''s Hospital of Pittsburgh are supported by an outstanding Editorial Board of international experts. This multidisciplinary open-access journal is at the forefront of disseminating and communicating scientific knowledge and impactful discoveries to researchers, academics, clinicians and the public worldwide. Frontiers in Pediatrics also features Research Topics, Frontiers special theme-focused issues managed by Guest Associate Editors, addressing important areas in pediatrics. In this fashion, Frontiers serves as an outlet to publish the broadest aspects of pediatrics in both basic and clinical research, including high-quality reviews, case reports, editorials and commentaries related to all aspects of pediatrics.
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