"Paradoxical deterioration" during antituberculous therapy is generally defined as the clinical or radiological worsening of pre-existing tuberculous lesions or the development of new lesions in a patient who initially improves. The phenomenon of paradoxical deterioration in intramedullary tuberculoma of the spinal cord is rare and is a less established entity. The authors present an unusual case of paraparesis as a result of paradoxical deterioration of intramedullary tuberculoma despite adequate antituberculous therapy. Here, we review the relevant literatures and discuss its possible pathogenic mechanisms.