Suppr超能文献

血浆置换成功治疗DiGeorge综合征患者的难治性自身免疫性溶血性贫血:病例报告及文献复习

Refractory autoimmune hemolytic anemia in a patient with DiGeorge syndrome treated successfully with plasma exchange: a case report and review of the literature.

作者信息

Damlaj Moussab, Séguin Chantal

机构信息

Division of Hematology, Department of Medicine, McGill University Health Centre (MUHC), Montreal General Hospital, Room A7.117.1, 1650 Cedar Avenue, Montreal, QC, H3G 1A4, Canada.

出版信息

Int J Hematol. 2014 Nov;100(5):494-7. doi: 10.1007/s12185-014-1648-1. Epub 2014 Jul 31.

Abstract

Warm antibody autoimmune hemolytic anemia (AIHA) results from targeted antibodies towards the red blood cells (RBCs) and can be either idiopathic or secondary to certain diseases, such as autoimmune disorders or malignancy, drugs, or infection. Patients with DiGeorge syndrome are particularly vulnerable to autoimmune conditions secondary to thymic hypoplasia and dysfunction of the immune system. First-line therapy for AIHA consists of corticosteroids, with most patients showing signs of response. Relapses are not uncommon and are treated with splenectomy or rituximab. There is a paucity of reports in the literature regarding treatment options beyond this stage. Herein, we describe an unusual case of a 20-year-old female affected by DiGeorge syndrome with a history of immune thrombocytopenia (ITP), who presented with life-threatening AIHA. Standard first- and second-line therapeutic modalities were ineffective in controlling her disease and she ultimately underwent plasma exchange therapy with successful resolution of hemolysis. At her last follow-up, one year after her initial presentation, she remains clinically well without signs of hemolysis. We conclude that in refractory cases of warm AIHA, plasma exchange therapy can be a valuable tool in the therapeutic armamentarium.

摘要

温抗体型自身免疫性溶血性贫血(AIHA)是由针对红细胞(RBC)的靶向抗体引起的,可分为特发性或继发于某些疾病,如自身免疫性疾病、恶性肿瘤、药物或感染。患有DiGeorge综合征的患者尤其容易因胸腺发育不全和免疫系统功能障碍而出现自身免疫性疾病。AIHA的一线治疗包括使用皮质类固醇,大多数患者会出现反应迹象。复发并不罕见,可通过脾切除术或利妥昔单抗进行治疗。关于此阶段之后的治疗选择,文献报道较少。在此,我们描述了一例不寻常的病例,一名20岁患有DiGeorge综合征且有免疫性血小板减少症(ITP)病史的女性,出现了危及生命的AIHA。标准的一线和二线治疗方法在控制她的病情方面均无效,她最终接受了血浆置换治疗,溶血得以成功缓解。在她首次就诊一年后的最后一次随访中,她临床状况良好,没有溶血迹象。我们得出结论,在温抗体型AIHA的难治性病例中,血浆置换治疗可以成为治疗手段中的一种有价值的工具。

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验