Excess manganese in the human body – mainly from chronic or occupational exposure – is associated with numerous toxic effects, predominantly neurological, resembling Parkinson’s disease (rigidity, cognitive alterations, bradykinesia). Manganism results from manganese accumulation in the central nervous system, especially the basal ganglia, globus pallidus, subthalamic nucleus, and substantia nigra. M.C.M, female, 45 years old, diagnosed with AIDS in 2015 during neurotoxoplasmosis treatment. In 2021, met criteria for systemic lupus erythematosus and rheumatoid arthritis. On regular ART. In 2019, after methotrexate and azathioprine use for rheumatologic activity, she developed elevated transaminases without prior liver disease. In November 2024, after partial hepatic improvement, she developed confusion, vertigo, and tremor with bradykinesia, unresponsive to levodopa. Brain MRI showed bilateral T1 hyperintensity in the globus pallidus and hypointensity in supratentorial and capsular white matter, suggesting manganese deposition. CSF analysis was normal; serum manganese at the upper limit of normal, urinary within normal range. Liver biopsy showed drug-induced hepatitis. Four months later, she had a seizure followed by aspiration, requiring intubation, progressing to dialysis-dependent acute renal failure and death. Occupational exposure is the best-established risk factor for manganism; however, hepatic dysfunction also increases risk, regardless of metal overload. This report highlights the need to consider manganism in the differential diagnosis of neurological diseases in PLHIV with liver dysfunction.
Primo et al. (Sun,) studied this question.