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Infection of the central nervous system (CNS) is a rare but devastating complication of invasive aspergillosis. We report a case of invasive aspergillosis with spinal involvement in a human immunodeficiency virus (HIV)-infected patient without neutropenia. A 42-year-old, antiretroviral-naïve, HIV-infected man presented with progressive weakness in the lower limbs and urinary and faecal incontinence for 2 weeks. The patient had been prescribed broad-spectrum antibiotics and prednisone. He had upper motor neuron signs and a sensory level at T1, with accompanying neck stiffness on flexion. Magnetic resonance imaging revealed diffuse abnormal signals of the vertebral bodies in the lower cervical and thoracic areas, with cord compression in the C2 and C3 region and signal distortions of the T2 and T3 vertebral bodies. Chest X-ray and computerized tomography demonstrated post-tuberculous apical cavities with suspected fungal colonization. Histopathology of an extradural spinal lesion at T1/T2 suggested invasive aspergillosis. The patient was started on fluconazole in response to the histopathological evidence of Aspergillus infection, but died within 3 weeks. Post-mortem analysis of the biopsy sample by PCR identified the infectious agent as Aspergillus fumigatus. Atypically, his CD4(+) T-cell count was 239 cells mm(-3) and he had no evidence of neutropenia. Invasive aspergillosis should be considered as part of the differential diagnosis among HIV-infected patients with non-specific, focal CNS symptoms, even among those without classical risk factors such as neutropenia, and aggressive antifungal therapy should be instituted as early as possible. more...