Disseminated Talaromyces marneffei And Mycobacterium avium Infection Accompanied Sweet’s Syndrome In A Patient With Anti-Interferon-γ Autoantibodies: A Case Report

Abstract

Shan-Shan Su,1,* Sheng-Nan Zhang,1,* Jun-Ru Ye,1 Ling-Na Xu,1 Peng-Cheng Lin,1 Han-Yan Xu,1 Qing Wu,2 Yu-Ping Li1 1Department of Respiratory and Critical Care Medicine, The First Affiliated Hospital of Wenzhou Medical University, Wenzhou, Zhejiang 325015, People’s Republic of China; 2The Center of Laboratory and Diagnosis, The First Affiliated Hospital of Wenzhou Medical University, Wenzhou, Zhejiang 325015, People’s Republic of China*These authors contributed equally to this workCorrespondence: Yu-Ping LiDepartment of Respiratory and Critical Care Medicine, The First Affiliated Hospital of Wenzhou Medical University, South Baixiang, Ouhai District, Wenzhou, Zhejiang Province 325015, People’s Republic of ChinaTel/fax [phone]Email [email protected]: Patients with high-titer anti-IFN-γ autoantibodies present disseminated non-tuberculous mycobacterial (NTM) and other opportunistic infections. Due to its rare occurrence and non-specific symptoms, this syndrome is difficult to diagnose during early disease stages. Here, we report a case with high-concentrations of serum anti-IFN-γ autoantibodies who presented with disseminated Talaromyces marneffei and NTM disease accompanied Sweet’s syndrome.Case presentation: A 62-year-old Chinese woman with no previous history was admitted to our hospital in August 2016 due to intermittent fever for 2 years, left chest wall redness, and swelling for 3 months. During hospitalization, the patient was confirmed with disseminated T. marneffei and successfully treated with antifungal therapy. In July 2017, upon second admission, Mycobacterium avium intracellular (MAC) pulmonary infection was established after positive cultures from the right lung tissue. The patient failed treatment after 1 month of anti-NTM therapy due to side effects. In May 2018, she was confirmed as having disseminated MAC disease accompanied by hand rashes, which was considered as Sweet’s syndrome. High-level anti-IFN-γ antibodies in the patient serum were detected upon comparison with normal controls (2.85-fold increase). Following anti-NTM therapy, both symptoms and pulmonary infiltration gradually improved, and joint destruction and lymphadenitis remained.Conclusions: Patients with anti-interferon-γ autoantibodies should be considered for severe, recurrent infections in adults in the absence of other known risk factors. Sweet’s syndrome is a common skin manifestation of the syndrome.Keywords: Talaromyces marneffei, Mycobacterium avium, Sweet’s syndrome, autoantibodies, interferon-gamma

Keywords

• Talaromyces marneffei
• Mycobacterium avium
• Sweet syndrome
• Autoantibodies
• Interferon-gamma.