Sweet’s Syndrome Accompanied by Coinfection with Multiple Pathogens and Disseminated Mycobacterium phlei Infection Presenting with Osteolytic Destruction During 12 Years of Follow-Up: A Rare Case Report

Abstract

Background Anti-IFN-γ autoantibodies (AIGAs) are closely related to the disseminated infection of multiple pathogens. Mycobacterium phlei (M. phlei) is a nonpathogenic nontuberculous mycobacteria (NTM), and M. phlei infection of the bone is extremely rare. We report a rare case of high-titer AIGAs presenting with Sweet’s syndrome (SS) accompanied by opportunistic coinfection with multiple pathogens during 12 years of follow-up. The patient in this case also developed disseminated M. phlei infection with osteolytic destruction after treatment for SS. Case Presentation A 68-year-old Chinese woman was admitted to our hospital in August 2009 due to fever and cough with expectoration for 3 months. The patient was successively infected with Klebsiella pneumoniae , herpes zoster virus and Candida . Chest computed tomography (CT) showed recurrent consolidations in different lung fields. After 15 months of antimicrobial treatment, the patient experienced partial recovery. In September 2010, the patient was pathologically diagnosed with SS due to the presence of multiple rashes. After prednisone and thalidomide treatment, the rashes subsided, and the pulmonary lesions had completely absorbed. In May 2011, the patient was diagnosed with disseminated tuberculosis and was administered anti-tuberculosis therapy for 3 months without improvement. NTM was subsequently cultured from her sputum and chest wall pus, and she improved after 20 months of anti-NTM therapy. In March 2016, the patient developed osteolytic destruction of the C7-T2 vertebral bodies with a back abscess. NTM was eventually cultured from the dorsal abscess pus and further identified as M. phlei . High-titer AIGAs were detected in the patient’s serum. After another round of aggressive anti-NTM therapy, the patient was finally cured. Conclusion Patients with AIGA-associated anti-cytokine autoantibody disease can present with multiple opportunistic infections and SS involving the lung. AIGA-associated immunodeficiency leads to infection with nonpathogenic M. phlei , which is refractory, can cause relapse, and even leads to osteolytic destruction.

Anticytokine Autoantibodies in Infectious Diseases: A Practical Overview

“…Anti-IFNα and IFNω auto-antibodies were also described in an 18-year-old female with common variable immune deficiency and severe facial herpes vegetans with recurrent herpes simplex viremia [ 73 ]. A 68-year-old Chinese woman with anti-IFNγ auto-antibodies suffered from recurrent herpes zoster infections in addition to an NTM infection [ 74 ]. In a group of 83 patients with postherpetic neuralgia, 12% showed high-titer neutralizing anti-IFNγ auto-antibodies [ 75 ].…”