Background: Acute transverse myelitis is a rare manifestation of dengue viral infection. Four cases have been previously reported in the literature. Objective: To report a case of a 61-year-old woman who developed acute transverse myelitis 6 days after the onset of a dengue viral infection. Findings: Magnetic resonance imaging of spinal cord showed hypersignal intensity on T2W at T9-T10. Laboratories studies revealed a high titer of hemagglutination inhibition of dengue virus. Treatment with intravenous pulse methylprednisolone and physiotherapy yielded a partial recovery, followed by complete resolution at 1 year postinfection. Conclusion: Acute transverse myelitis is a rare manifestation of dengue infection that can occur in either the peri-infectious or postinfectious phases.