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

Abstract

Background

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

Acknowledgments

We thank the patients and their families and all staff in the department for their support. This study was supported by the Wenzhou Municipal Science and Technology Bureau (CN) (ZH2017001).

Abbreviations

NTM, non-tuberculous mycobacteriosis; IFN-γ, interferon-gamma; CT, computed tomography; CRP, C-reactive protein; Ig G, Immunoglobin G; G test, (1, 3)-D glucan; GM, galactomannan test; T-SPOT.TB, tuberculosis infection T cell spot test; PCT, pro-calcitonin; ESR, erythrocyte sedimentation rate; MAC, Mycobacterium avium intracellular; APCs, antigen-presenting cells.

Availability of Data And Material

All the information supporting our conclusions and relevant references are included in the manuscript. There are no datasets related to this case report.

Consent For Publication

Written informed consent was obtained from the patient for the publication of this case report and any accompanying images.

Author Contributions

All authors contributed to data analysis, drafting or revising the article, gave final approval of the version to be published, and agree to be accountable for all aspects of the work.

Disclosure

The authors report no conflicts of interest in this work.