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引用次数: 0
摘要
幼年皮肌炎是一种罕见的全身性炎症性自身免疫性疾病,涉及肌肉、皮肤和血管。大多数患者对初始治疗没有完全反应,而是出现慢性难治性或多循环病程。发病机制尚不完全清楚,但已描述了免疫细胞失调(尤其是 B 细胞)、线粒体功能障碍、中性粒细胞和中性粒细胞胞外捕获物(NET)的变化以及 I 型和 II 型干扰素(IFN)信号的增加。针对幼年皮肌炎(JDM)的药物随机对照试验非常有限,治疗主要基于质量较低的数据,如病例系列、回顾性研究和开放标签的前瞻性研究。这些数据被汇编成专家建议或共识治疗方案,有助于指导治疗。虽然初始治疗较为标准,大多数包括皮质类固醇(大剂量口服和/或脉冲静脉注射甲基强的松龙)和甲氨蝶呤,但对于难治性患者,指导方针则更加多样化,有多种选择或组合,包括生物疗法。显然,JDM 需要更有效的个性化疗法。值得进一步研究的JDM新兴治疗方案包括靶向IFN信号(JAK、IFNAR1、IFN beta)、B细胞(CD20、CD19、BAFF、TACI、CD38、BCMA),包括嵌合抗原受体(CAR)-T细胞疗法、线粒体功能障碍和NET。
Juvenile Dermatomyositis: Updates in Pathogenesis and Biomarkers, Current Treatment, and Emerging Targeted Therapies.
Juvenile dermatomyositis is a rare systemic inflammatory autoimmune disease involving muscle, skin, and vessels. Most patients do not fully respond to initial therapy, instead having a chronic refractory or polycyclic disease course. Pathogenesis is not completely understood, but immune cell dysregulation, particularly of B cells, mitochondrial dysfunction, changes in neutrophils and neutrophil extracellular traps (NETs), and increased type I and type II interferon (IFN) signaling have been described. There are limited randomized controlled trials of drugs in juvenile dermatomyositis (JDM), and treatment is largely based on lower-quality data such as case series, retrospective studies, and open-label prospective studies. These data have been compiled into expert recommendations or consensus treatment plans, which help guide therapy. While initial therapy is more standard with most including corticosteroids (high-dose oral and/or pulse intravenous methylprednisolone) and methotrexate, for refractory patients, guidelines are more varied with multiple options or combinations, including biologic therapies. There is a clear need for more efficacious and personalized therapy in JDM. Emerging treatment options worthy of further study in JDM include targeting IFN-signaling (JAK, IFNAR1, IFN beta), B-cells (CD20, CD19, BAFF, TACI, CD38, BCMA) including Chimeric Antigen Receptor (CAR)-T cell therapy, mitochondrial dysfunction, and NETs.
期刊介绍:
Pediatric Drugs promotes the optimization and advancement of all aspects of pharmacotherapy for healthcare professionals interested in pediatric drug therapy (including vaccines). The program of review and original research articles provides healthcare decision makers with clinically applicable knowledge on issues relevant to drug therapy in all areas of neonatology and the care of children and adolescents. The Journal includes:
-overviews of contentious or emerging issues.
-comprehensive narrative reviews of topics relating to the effective and safe management of drug therapy through all stages of pediatric development.
-practical reviews covering optimum drug management of specific clinical situations.
-systematic reviews that collate empirical evidence to answer a specific research question, using explicit, systematic methods as outlined by the PRISMA statement.
-Adis Drug Reviews of the properties and place in therapy of both newer and established drugs in the pediatric population.
-original research articles reporting the results of well-designed studies with a strong link to clinical practice, such as clinical pharmacodynamic and pharmacokinetic studies, clinical trials, meta-analyses, outcomes research, and pharmacoeconomic and pharmacoepidemiological studies.
Additional digital features (including animated abstracts, video abstracts, slide decks, audio slides, instructional videos, infographics, podcasts and animations) can be published with articles; these are designed to increase the visibility, readership and educational value of the journal’s content. In addition, articles published in Pediatric Drugs may be accompanied by plain language summaries to assist readers who have some knowledge of, but not in-depth expertise in, the area to understand important medical advances.