M.J. Vos, MD, Y.J. Debets-Ossenkopp, MD, PhD, F.A.P. Claessen, MD, G.J. Hazenberg, MD, J.J. Heimans, MD, PhD, Amsterdam, the Netherlands
A 16-year-old Surinam boy presented with a 3-month history of progressive paraparesis and periods of fever and neck stiffness. Neurologic examination demonstrated a severe spastic paraparesis, Babinski signs, and sensory disturbance under level T4. The patient was not taking immunosuppressants, was HIV-seronegative, and had normal CD4+ lymphocyte counts. Postcontrast T1-weighted MRI of brain and spinal cord revealed a ring-enhancing left cerebellar lesion, meningeal contrast enhancement, and multiple intramedullary lesions (figure). CSF examination showed a lymphocytic pleocytosis, elevated total protein, lowered glucose, and negative Gram staining and cultures. Biopsy of one intramedullary lesion disclosed granulomatous lymphohistiocytic inflammation with giant cells and necrosis; cultures were positive for Histoplasma capsulatum. Some neurologic improvement occurred after sequential treatment with amphotericin B and itraconazole.
Figure. MRI at admission (postcontrast T1-weighted). (A) Ring-enhancing left cerebellar lesion and meningeal contrast enhancement. (B) Multiple intramedullary lesions.
Primary neurologic presentation of histoplasmosis is extremely rare, especially in young, nonimmunocompromised hosts. Tan et al. reported a case of disseminated histoplasmosis in a 72-year-old man, which was the first report of histoplasmosis presenting as a myelopathy.1 Tabbal and Harik recently reported cerebral histoplasmosis in a 60-year-old man, presenting as a bilateral pyramidal syndrome.2
To our knowledge, this is the first case of cerebellar, meningeal, and medullar histoplasmosis reported in a young, nonimmunocompromised host. The severity of the neurologic presentation, lack of systemic manifestations, and extensiveness of CNS lesions on MRI are exceptional.
1. Tan V, Wilkins P, Badve S, et al. Histoplasmosis of the central nervous system. JNNP 1992;55:619–622.
2. Tabbal SD, Harik SI. Cerebral histoplasmosis. N Engl J Med 1999;340:1176.
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