Treatment strategies for Sjögren's syndrome with childhood onset: a systematic review of the literature.
Treatment strategies for Sjögren's syndrome with childhood onset: a systematic review of the literature.
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DOI：
10.1093/rheumatology/keab579
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发表时间：
2022-03-02
期刊：
影响因子：
--
通讯作者：
Ciurtin C
中科院分区：
文献类型：
--
作者：
Doolan G;Faizal NM;Foley C;Al-Obaidi M;Jury EC;Price E;Ramanan AV;Lieberman SM;Ciurtin C
儿童期发病的 SS 是一种罕见的自身免疫性疾病，其特征为异质性表现。缺乏经过验证的分类标准使得诊断变得困难。由于青少年 SS 疾病罕见且针对该患者群体的研究很少，因此尚无基于证据的治疗指南。本系统综述旨在总结和评价目前关注儿童期发病 SS 的药理学策略的文献。使用 2009 年系统评价和荟萃分析报告清单的首选报告项目，筛选了截至 2020 年 12 月的 PubMed 和 MEDLINE/Scopus 数据库，以寻找合适的报告，重点介绍儿童期发病的 SS 的药物治疗。动物研究被排除在外。共有 43 项研究（34 项病例报告、8 项小型病例系列和 1 项试点研究）符合分析资格。检索到的研究中 88% (120/137) 的案例中包括女孩，且置信度非常低。 HCQ 被用于治疗腮腺肿胀，以及与 MTX 和 NSAID 联合治疗关节炎和关节痛患者。皮质类固醇，例如长期口服泼尼松和静脉注射。甲基泼尼松龙通常用于治疗病情严重的儿童。利妥昔单抗主要用于治疗粘膜相关淋巴组织淋巴瘤以及肾脏和神经系统并发症。其他常规 DMARD 被用于治疗有腺外表现的特定病例。青少年 SS 的治疗可采用多种疗法，并根据临床专家的意见制定。目前尚无高质量的研究为儿童期发病的 SS 的治疗提供临床建议。
SS with childhood onset is a rare autoimmune disease characterized by heterogeneous presentation. The lack of validated classification criteria makes it challenging to diagnose. Evidence-based guidelines for treatment of juvenile SS are not available due to the rarity of disease and the paucity of research in this patient population. This systematic review aims to summarize and appraise the current literature focused on pharmacological strategies for management of SS with childhood onset. PubMed and MEDLINE/Scopus databases up to December 2020 were screened for suitable reports highlighting pharmacological treatment of SS with childhood onset using the Preferred Reporting Items for Systematic Reviews and Meta-Analyses 2009 reporting checklist. Animal studies were excluded. A total of 43 studies (34 case reports, 8 mini case series and 1 pilot study) were eligible for analysis. The studies retrieved included girls in 88% (120/137) of cases and had very low confidence levels. HCQ was prescribed for parotid swelling, as well as in association with MTX and NSAIDs in patients with arthritis and arthralgia. Corticosteroids such as long courses of oral prednisone and i.v. methylprednisolone were commonly prescribed for children with severe disease presentations. Rituximab was mainly indicated for mucosa-associated lymphoid tissue lymphoma and renal and nervous system complications. Other conventional DMARDs were prescribed in selected cases with extraglandular manifestations. Various therapies are used for the management of juvenile SS and are prescribed based on expert clinician’s opinion. There are currently no good-quality studies that allow clinical recommendations for treatment of SS with childhood onset.
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影响因子：
4.7
作者：
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