To describe the clinical presentation, management and outcome of a rare case of isolated oculomotor nerve palsy in an immunocompromised adult secondary to a central nervous system (CNS) tuberculoma.

This is a case report.

A Filipino male in his 30s developed severe throbbing headache followed by binocular diplopia and drooping of the right upper eyelid. Findings were compatible with a neurologically-isolated pupil-involving, complete oculomotor nerve palsy on the right. Brain magnetic resonance imaging demonstrated enlargement and contrast enhancement of the cisternal portion of the right oculomotor nerve. Serologic testing was positive for the human immunodeficiency virus (HIV) and syphilis. Cerebrospinal fluid (CSF) analysis showed lymphocytic pleocytosis and elevated protein. CSF polymerase chain reaction was positive for Mycobacterium tuberculosis (TB). The patient was treated with penicillin, quadruple anti-Koch’s, and anti-retrovirals. Eyelid position and ocular motility improved after treatment. Aberrant regeneration of the right oculomotor nerve was observed with elevation of the right eyelid on downgaze (pseudo-Graefe sign).

CNS tuberculoma may present as a neurologically-isolated oculomotor nerve palsy, particularly in immunocompromised individuals. In TB-endemic countries, like the Philippines, it should be considered in the differential diagnosis. Early recognition and appropriate antimicrobial therapy can lead to neurologic improvement.