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Immune reconstitution inflammatory syndrome (IRIS) is an exaggerated immune response which can occur with various coinfections in human immunodeficiency virus (HIV) infected patients, of which the most commonly implicated in central nervous system (CNS)-IRIS are progressive multifocal leukoencephalopathy (PML), cryptococcosis, and tuberculosis (TB). TB-IRIS is a known complication of pulmonary TB or TB lymphadenitis coinfection in HIV infected patients who are on antituberculosis treatment (ATT) after the initiation of antiretroviral therapy (ART). However, development of IRIS in extrapulmonary TB such as CNS TB is very rare. Our case is that of an isolated CNS-TB-IRIS, presenting as increase in the size and perilesional edema of the ring enhancing lesions in the brain, which was observed in two sequential magnetic resonance imaging done over a period of 2 months in a retropositive patient who presented with clinical deterioration after commencement of ART. As prompt diagnosis was made and specific management aimed at IRIS was started without delay, the patient improved symptomatically.