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泰国一名女性患者出现复发性播散性 :抗干扰素-γ自身抗体治疗方法的困境。

Recurrent disseminated in a female patient from Thailand with anti-interferon-gamma autoantibodies: dilemma on treatment approach.

机构信息

Pharmacy, Tampa General Hospital, Tampa, Florida, USA.

Pediatric Allergy/Immunology, University of South Florida College of Medicine, Tampa, Florida, USA.

出版信息

BMJ Case Rep. 2021 Jan 28;14(1):e237909. doi: 10.1136/bcr-2020-237909.

Abstract

Anti-interferon-gamma (IFN-γ) autoantibodies has been recognised as an adult-onset immunodeficiency in the past decade in people who originate from Southeast Asia. These patients are susceptible to particular opportunistic infections, especially non-tuberculous mycobacteria (NTM). We present the case of a woman whom originally came from Thailand with disseminated complex infection (pleural, pericardium, bloodstream and lung parenchymal involvement). Her infection continued to progress while receiving proper antibiotic treatment. Once high titre neutralising anti-IFN-γ autoantibodies were detected, rituximab was added as adjunctive treatment. The patient had remarkable clinical improvement against persistence of anti-IFN-γ autoantibodies. Although her lung disease has improved, the patient continues on triple therapy for NTM. The kinetics of anti-IFN-γ autoantibodies in the context of clinical progression, indication and length for rituximab and triple therapy is discussed in view of the current literature.

摘要

在过去的十年中,人们认识到源自东南亚的成年人免疫缺陷与抗干扰素-γ(IFN-γ)自身抗体有关。这些患者易患特定的机会性感染,特别是非结核分枝杆菌(NTM)。我们介绍了一位来自泰国的女性患者,她患有播散性复杂感染(累及胸膜、心包、血流和肺实质)。尽管接受了适当的抗生素治疗,但她的感染仍在继续恶化。一旦检测到高滴度中和抗 IFN-γ 自身抗体,就添加利妥昔单抗作为辅助治疗。尽管抗 IFN-γ 自身抗体持续存在,但患者的临床状况显著改善。尽管她的肺部疾病有所改善,但患者仍继续接受 NTM 的三联疗法。根据当前文献,讨论了抗 IFN-γ 自身抗体在临床进展、利妥昔单抗和三联疗法的适应证和疗程方面的动力学。

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