Refractory Mycobacterium genavense infection secondary to thymoma-associated endogenous IL-12 inhibitor
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 infec...
| Main Authors: | , , , , , |
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| Format: | Article |
| Language: | English |
| Published: |
BMJ Publishing Group
2022-05-01
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| Series: | BMJ Neurology Open |
| Online Access: | https://neurologyopen.bmj.com/content/4/1/e000285.full |
| _version_ | 1797984758351790080 |
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| author | MaiAnh Nguyen Jessie Chen Stephen Reddel Elaine Cheong D Sean Riminton Hannah Hu |
| author_facet | MaiAnh Nguyen Jessie Chen Stephen Reddel Elaine Cheong D Sean Riminton Hannah Hu |
| author_sort | MaiAnh Nguyen |
| collection | DOAJ |
| description | 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. |
| first_indexed | 2024-04-11T07:07:36Z |
| format | Article |
| id | doaj.art-437740eac3a6475c91745ad97441b311 |
| institution | Directory Open Access Journal |
| issn | 2632-6140 |
| language | English |
| last_indexed | 2024-04-11T07:07:36Z |
| publishDate | 2022-05-01 |
| publisher | BMJ Publishing Group |
| record_format | Article |
| series | BMJ Neurology Open |
| spelling | doaj.art-437740eac3a6475c91745ad97441b3112022-12-22T04:38:21ZengBMJ Publishing GroupBMJ Neurology Open2632-61402022-05-014110.1136/bmjno-2022-000285Refractory Mycobacterium genavense infection secondary to thymoma-associated endogenous IL-12 inhibitorMaiAnh Nguyen0Jessie Chen1Stephen Reddel2Elaine Cheong3D Sean Riminton4Hannah Hu5Department of Immunology, Concord Repatriation General Hospital, Concord, New South Wales, AustraliaDepartment of Neurology, Concord Repatriation General Hospital, Concord, New South Wales, AustraliaDepartment of Neurology, Concord Repatriation General Hospital, Concord, New South Wales, AustraliaDepartment of Infectious Diseases and Microbiology, Concord Repatriation General Hospital, Concord, New South Wales, AustraliaDepartment of Immunology, Concord Repatriation General Hospital, Concord, New South Wales, AustraliaDepartment of Immunology, Concord Repatriation General Hospital, Concord, New South Wales, AustraliaCase 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.https://neurologyopen.bmj.com/content/4/1/e000285.full |
| spellingShingle | MaiAnh Nguyen Jessie Chen Stephen Reddel Elaine Cheong D Sean Riminton Hannah Hu Refractory Mycobacterium genavense infection secondary to thymoma-associated endogenous IL-12 inhibitor BMJ Neurology Open |
| title | Refractory Mycobacterium genavense infection secondary to thymoma-associated endogenous IL-12 inhibitor |
| title_full | Refractory Mycobacterium genavense infection secondary to thymoma-associated endogenous IL-12 inhibitor |
| title_fullStr | Refractory Mycobacterium genavense infection secondary to thymoma-associated endogenous IL-12 inhibitor |
| title_full_unstemmed | Refractory Mycobacterium genavense infection secondary to thymoma-associated endogenous IL-12 inhibitor |
| title_short | Refractory Mycobacterium genavense infection secondary to thymoma-associated endogenous IL-12 inhibitor |
| title_sort | refractory mycobacterium genavense infection secondary to thymoma associated endogenous il 12 inhibitor |
| url | https://neurologyopen.bmj.com/content/4/1/e000285.full |
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