[Pubmed] Refractory <em>Mycobacterium genavense</em> infection secondary to thymoma-associated endogenous IL-12 inhibito
Posté : 06 juin 2022 12:00
BMJ Neurol Open. 2022 May 18;4(1):e000285. doi: 10.1136/bmjno-2022-000285. eCollection 2022.
ABSTRACT
CASE: A 39-year-old man with thymoma-associated acetylcholine receptor antibody myasthenia gravis (MG) presented with fevers, night sweats, abdominal pain and weight loss. Marked splenomegaly and intra-abdominal lymphadenopathy were found. Biopsies confirmed disseminated Mycobacterium genavense infection. Despite antimicrobials and reduced immunosuppressive medications, he worsened. We suspected a thymoma-associated cytokine inhibitory antibody. The addition of subcutaneous interferon-gamma (IFN-γ) induced clinical and radiological improvement. His antimicrobials were able to be ceased. MG remained stable. Subsequent testing demonstrated an endogenous interleukin-12 (IL-12) inhibitor, likely inhibiting the IL-12/IFN-γ axis crucial for defence against mycobacterial infections.
DISCUSSION: This case illustrates the autoimmune manifestations that can occur with thymoma. It illustrates the benefit of exogenous IFN-γ in overcoming the immune deficit. In this case, its use did not exacerbate existing autoimmune disease or trigger others. We raise awareness of the need to consider cytokine pathway defects as a contributing factor to refractory atypical infections in patients with thymoma-associated MG.
PMID:35663588 | PMC:PMC9119138 | DOI:10.1136/bmjno-2022-000285
Source: https://pubmed.ncbi.nlm.nih.gov/3566358 ... 7&v=2.17.6