出现非典型溶血性尿毒症综合征的系统性红斑狼疮:病例报告和文献综述。

IF 3.2 3区 医学 Q2 RHEUMATOLOGY Rheumatology International Pub Date : 2024-10-01 Epub Date: 2024-03-19 DOI:10.1007/s00296-024-05558-9
Justin Smith, Varinder Hans, Elaine Yacyshyn, Azin Rouhi, Monika Oliver
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引用次数: 0

摘要

系统性红斑狼疮(SLE)可出现多种血液学表现,其中非典型溶血性尿毒症综合征(aHUS)较为罕见。如果不及时干预,系统性红斑狼疮诱发的 aHUS 会导致严重的发病率和死亡率,但其发病率仍不确定,补体导向疗法的最佳策略也主要由专家决定。我们进行了一次全面的文献综述,并介绍了一例新诊断为系统性红斑狼疮/四级狼疮肾炎的 23 岁女性患者的病例,她出现了 aHUS,并对 C5 拮抗剂 eculizumab 迅速产生了反应。通过对现有文献的回顾,我们发现了49个已发表的并发aHUS的系统性红斑狼疮病例,并发现年轻的系统性红斑狼疮患者更容易出现aHUS,同时伴有狼疮性肾炎、抗dsDNA阳性和补体系统异常。超过70%的病例使用依库珠单抗作为补体导向疗法,尽管报告的结果差异很大,但实验室指标改善的时间有加快的趋势。早期识别系统性红斑狼疮患者的 aHUS 对指导适当的治疗干预至关重要,及时开始使用依库珠单抗可降低浆细胞清除术和额外免疫抑制可能带来的发病率。虽然依库珠单抗显示出了良好的效果,但其最佳治疗时机和持续时间仍然难以确定。了解患者的补体遗传学有助于制定治疗策略,目前正在进行的补体靶向疗法研究为系统性红斑狼疮和 aHUS 的治疗提供了前景广阔的途径。
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Systemic lupus erythematosus presenting with atypical hemolytic uremic syndrome: a case report and review of the literature.

Systemic lupus erythematosus (SLE) can present with a diverse array of hematologic manifestations, among which atypical hemolytic uremic syndrome (aHUS) is a rare entity. SLE-triggered aHUS has significant morbidity and mortality without timely intervention, yet its frequency remains uncertain and optimal strategies for complement-directed therapies are largely expert-driven. We performed a comprehensive literature review and present a case of a 23-year-old female newly diagnosed with SLE/class IV lupus nephritis who developed aHUS that rapidly responded to the C5 antagonist, eculizumab. Review of the current literature identified forty-nine published cases of SLE with concurrent aHUS and revealed a predilection for aHUS in younger SLE patients, concurrent presentation with lupus nephritis, anti-dsDNA positivity, and complement system abnormalities. Over seventy percent of cases used eculizumab as complement-directed therapy with a trend towards faster time to improvement in laboratory parameters, though reported outcomes were highly variable. Early recognition of aHUS in SLE is pivotal in guiding appropriate therapeutic interventions, and prompt initiation of eculizumab may reduce the potential morbidity associated with plasmapheresis and additional immunosuppression. While eculizumab showcases promising results, its optimal timing and duration remain elusive. An understanding of a patients' complement genetics could aid management strategies, and ongoing research into complement-targeted therapies offers promising avenues for both SLE and aHUS treatment.

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来源期刊
Rheumatology International
Rheumatology International 医学-风湿病学
CiteScore
7.30
自引率
5.00%
发文量
191
审稿时长
16. months
期刊介绍: RHEUMATOLOGY INTERNATIONAL is an independent journal reflecting world-wide progress in the research, diagnosis and treatment of the various rheumatic diseases. It is designed to serve researchers and clinicians in the field of rheumatology. RHEUMATOLOGY INTERNATIONAL will cover all modern trends in clinical research as well as in the management of rheumatic diseases. Special emphasis will be given to public health issues related to rheumatic diseases, applying rheumatology research to clinical practice, epidemiology of rheumatic diseases, diagnostic tests for rheumatic diseases, patient reported outcomes (PROs) in rheumatology and evidence on education of rheumatology. Contributions to these topics will appear in the form of original publications, short communications, editorials, and reviews. "Letters to the editor" will be welcome as an enhancement to discussion. Basic science research, including in vitro or animal studies, is discouraged to submit, as we will only review studies on humans with an epidemological or clinical perspective. Case reports without a proper review of the literatura (Case-based Reviews) will not be published. Every effort will be made to ensure speed of publication while maintaining a high standard of contents and production. Manuscripts submitted for publication must contain a statement to the effect that all human studies have been reviewed by the appropriate ethics committee and have therefore been performed in accordance with the ethical standards laid down in an appropriate version of the 1964 Declaration of Helsinki. It should also be stated clearly in the text that all persons gave their informed consent prior to their inclusion in the study. Details that might disclose the identity of the subjects under study should be omitted.
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