Rujirachun Pongprueth, Sangwongwanich Jirath, Chayakulkeeree Methee
Department of Microbiology, Faculty of Medicine Siriraj Hospital, Mahidol University, Bangkok, Thailand.
Faculty of Medicine Siriraj Hospital, Mahidol University, Bangkok, Thailand.
BMC Infect Dis. 2020 Mar 19;20(1):232. doi: 10.1186/s12879-020-4949-4.
The most common infection in patients positive for anti-interferon-gamma autoantibodies (anti-IFN-γ AAbs) is disseminated nontuberculous mycobacterial (dNTM) infection. Here, we report a rare case of triple infection caused by Cryptococcus, varicella-zoster virus (VZV), and nontuberculous mycobacterium in a patient with anti-IFN-γ AAbs.
A 53-year-old Thai man presented with a progressively enlarging right cervical mass with low-grade fever and significant weight loss for 4 months. He also developed a lesion at his left index finger. A biopsy of that lesion showed granulomatous inflammation with yeast-like organisms morphologically consistent with cryptococcosis. Serum cryptococcal antigen was positive. Histopathology of a right cervical lymph node revealed chronic granulomatous lymphadenitis, and the lymph node culture grew Mycobacterium abscessus. One month later, he complained of vision loss in his left eye and subsequently developed a group of painful vesicles at the right popliteal area of S1 dermatome. Lumbar puncture was performed and his cerebrospinal fluid was positive for VZV DNA. His blood test for anti-HIV antibody was negative. Anti-IFN-γ AAbs was positive, but test for anti-granulocyte-macrophage colony-stimulating factor autoantibodies (anti-GM-CSF AAbs) was negative. He was treated with amphotericin B plus fluconazole for cryptococcosis; a combination of amikacin, imipenem, azithromycin, and levofloxacin for dNTM infection; and, intravenous acyclovir for disseminated VZV infection. After treatment, our patient's fever and cervical lymphadenopathy were subsided, and his vision and visual acuity were both improved.
This is the first case of triple infection with cryptococcosis, VZV, and dNTM in a patient who tested positive for anti-IFN-γ AAbs and negative for anti-GM-CSF AAbs. This case will increase awareness and heighten suspicion of these infections in patients with the described presentations and clinical characteristics, and this will accelerate diagnosis and treatment.
抗干扰素-γ自身抗体(抗IFN-γ AAbs)阳性患者中最常见的感染是播散性非结核分枝杆菌(dNTM)感染。在此,我们报告1例抗IFN-γ AAbs患者发生新型隐球菌、水痘-带状疱疹病毒(VZV)和非结核分枝杆菌三重感染的罕见病例。
一名53岁泰国男性,右侧颈部肿块逐渐增大,伴有低热和显著体重减轻4个月。其左手食指也出现一处皮损。该皮损活检显示肉芽肿性炎症,有形态上符合隐球菌病的酵母样生物体。血清隐球菌抗原呈阳性。右侧颈部淋巴结组织病理学检查显示慢性肉芽肿性淋巴结炎,淋巴结培养分离出脓肿分枝杆菌。1个月后,他主诉左眼视力丧失,随后在S1皮节的右腘窝区域出现一群疼痛性水疱。进行了腰椎穿刺,其脑脊液VZV DNA检测呈阳性。其抗HIV抗体血液检测为阴性。抗IFN-γ AAbs呈阳性,但抗粒细胞-巨噬细胞集落刺激因子自身抗体(抗GM-CSF AAbs)检测为阴性。他接受了两性霉素B加氟康唑治疗隐球菌病;阿米卡星、亚胺培南、阿奇霉素和左氧氟沙星联合治疗dNTM感染;静脉注射阿昔洛韦治疗播散性VZV感染。治疗后,患者的发热和颈部淋巴结病消退,视力和视敏度均有所改善。
这是首例抗IFN-γ AAbs阳性且抗GM-CSF AAbs阴性患者发生隐球菌病、VZV和dNTM三重感染的病例。该病例将提高对具有所述表现和临床特征患者中这些感染的认识并增强怀疑,这将加速诊断和治疗。