Tuberculous meningitis and hemophagocytic lymphohistiocytosis in a patient on adalimumab: diagnostic challenges in the setting of suspected non-tuberculous mycobacteria co-infection

Authors

• Dimitrios Dimitriadis Second University Department of Internal Medicine, University Hospital of Alexandroupolis, Democritus University of Thrace, Alexandroupoli, Greece
• Irene Terzi Second University Department of Internal Medicine, University Hospital of Alexandroupolis, Democritus University of Thrace, Alexandroupoli, Greece
• Ioulia Dragoumani Second University Department of Internal Medicine, University Hospital of Alexandroupolis, Democritus University of Thrace, Alexandroupoli, Greece
• Konstantinos Liapis Department of Haematology, University Hospital of Alexandroupolis, Democritus University of Thrace, Alexandroupoli, Greece
• Dimitrios Papazoglou Second University Department of Internal Medicine, University Hospital of Alexandroupolis, Democritus University of Thrace, Alexandroupoli, Greece

dimijim@yahoo.gr

Keywords

Abstract

Background: Tumour necrosis factor-alpha (TNF-alpha) inhibitors increase susceptibility to granulomatous infections, including both Mycobacterium tuberculosis (MTB) and nontuberculous mycobacteria. We describe a complex case of sequential disseminated Mycobacterium avium complex (MAC) and central nervous system MTB infection in a patient treated with adalimumab, complicated by hemophagocytic lymphohistiocytosis (HLH).
Case report: A 65-year-old man on long-term adalimumab for psoriasis presented with prolonged fever, hepatosplenomegaly, cytopenia and elevated inflammatory markers. Bone marrow aspiration revealed hemophagocytosis and liver and bone marrow biopsy revealed granulomatous inflammation. Polymerase chain reaction (PCR) testing of bronchoalveolar lavage (BAL) fluid identified MAC, supporting a diagnosis of disseminated MAC-associated HLH. The patient responded to triple MAC therapy (azithromycin, ethambutol, rifampicin), intravenous immunoglobulin and low-dose corticosteroids, with rapid clinical improvement. Three months later, he was readmitted with fever and altered mental status. Brain magnetic resonance imaging showed meningeal thickening. Lumbar puncture revealed cerebrospinal fluid pleocytosis, hypoglycorrhachia and elevated protein. PCR detected MTB complex deoxyribonucleic acid and a rifampicin resistance gene, prompting the initiation of a four-drug antituberculosis regimen (isoniazid, pyrazinamide, levofloxacin, ethambutol) and high-dose dexamethasone. The patient improved and was discharged after a month of hospitalization, remaining clinically stable at 1-year follow-up.
Conclusion: This case highlights the risk of sequential or overlapping MAC and MTB infections in patients receiving TNF-alpha inhibitors, the potential for HLH as a serious complication, and the diagnostic value and limitations of BAL PCR testing. Vigilant screening and multidisciplinary management are essential in such high-risk populations.

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