Background: Opportunistic infections (OIs) are clinical clues for clinicians to suspect an HIV or AIDS diagnosis and often patients contact healthcare due to OI, not because of HIV infection itself. OIs come with various clinical presentations which pose difficult challenges for clinicians to diagnose and treat. Case description: A young male was referred due to progressive weakness of both limbs in the last two months prior to admission. Physical examination showed loss of sensation in thoracal segment level VII-VIII. Laboratory tests showed positive HIV tests, low CD4 and detectable HIV RNA load. MRI showed tuberculomatous lesion in spinal cord thoracal vertebrae level I-X and syringomyelia. Patient received cotrimoxazole, antituberculous drugs and antiretroviral drugs but he died before anti-tuberculous treatment complete. Conclusion: Intramedullary spinal tuberculosis is very rare, and its clinical presentation is unspecified. Neuroimaging such as MRI should be a mandatory to be performed since histopathological tests sometimes are unable to undergo. Pharmacological treatment is based on clinical judgement and surgical intervention should be offered if any indications exist.