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成人 IgA 血管炎患者接受系统性免疫抑制治疗的病例系列研究。

A Case Series of Adult Patients Diagnosed with IgA Vasculitis Requiring Systemic Immunosuppression.

机构信息

Department of Clinical Immunology, Westmead Hospital, Westmead, New South Wales, Australia.

Department of Renal Medicine, Orange Health Service, Orange, New South Wales, Australia.

出版信息

Am J Case Rep. 2021 Dec 8;22:e933407. doi: 10.12659/AJCR.933407.

Abstract

BACKGROUND IgA vasculitis (IgAV) is a rare and potentially life-threatening small-vessel vasculitis in adults. The disease course is often more severe than its childhood counterpart. The disease is noted for its heterogeneous presentation with varying severity. There are no current treatment guidelines for severe multi-organ involvement of IgAV. The treatment approaches based on the clinical discretion of treating doctors remain controversial, especially regarding the role, duration, and type of immunosuppression. CASE REPORT We present 3 cases of severe multi-organ IgAV encountered at our tertiary referral center between 2016 and 2021, which were treated with different immunosuppression regimens, including combination of systemic corticosteroids, oral immunosuppressants (azathioprine, mycophenolate, and sirolimus), rituximab, and cyclophosphamide. In these patients, IgAV presented differently but were all organ-threatening or life-threatening in nature. IgAV in all patients responded to therapies; however, infection complicating underlying comorbidities was the cause of death in 1 patient and the cause of comorbidities in the other 2. Other treatment-related complications included weight gain, adrenal insufficiency, and secondary hypogammaglobulinemia. CONCLUSIONS IgAV can be a polyphasic and a potentially challenging severe organ-threatening disease to treat in adults. The outcomes presented here highlight the morbidity and substantial risks involved in treating complex IgAV patients. Early use of biologics may have a role in preventing treatment-related toxicity. Further studies on IgAV in adults are urgently needed.

摘要

背景

IgA 血管炎(IgAV)是一种罕见且可能危及生命的成人小血管血管炎。其疾病过程通常比儿童期更为严重。该疾病以不同严重程度的异质性表现为特征。目前尚无针对 IgAV 严重多器官受累的治疗指南。基于治疗医生临床判断的治疗方法仍存在争议,尤其是关于免疫抑制的作用、持续时间和类型。

病例报告

我们在 2016 年至 2021 年间于三级转诊中心遇到了 3 例严重多器官 IgAV 病例,这些病例采用了不同的免疫抑制方案治疗,包括全身皮质类固醇、口服免疫抑制剂(硫唑嘌呤、霉酚酸酯和西罗莫司)、利妥昔单抗和环磷酰胺。在这些患者中,IgAV 的表现不同,但均具有威胁器官或危及生命的性质。所有患者的 IgAV 均对治疗有反应;然而,感染合并潜在合并症是 1 例患者死亡的原因,也是其他 2 例患者合并症的原因。其他与治疗相关的并发症包括体重增加、肾上腺功能不全和继发性低丙种球蛋白血症。

结论

IgAV 可能是一种多相且具有挑战性的严重威胁器官疾病,在成人中治疗较为困难。这里呈现的结果强调了治疗复杂 IgAV 患者所涉及的发病率和巨大风险。早期使用生物制剂可能在预防治疗相关毒性方面发挥作用。迫切需要对成人 IgAV 进行进一步研究。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/78e0/8669519/c356273820ee/amjcaserep-22-e933407-g001.jpg

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