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Cycloserine (CS) is an important part of the tools against multidrug-resistant tuberculosis (MDR-TB). Similar to most of the second line drugs, CS has many adverse drug reactions (ADR). The ADR due to CS related to the central nervous system (CNS) are very rare and are reported in few isolated case reports. We herein, present a first case series from the national capital of India of three MDR-TB patients with CS-induced ADR´s of the CNS. In all the three cases the drug was withdrawn and the symptoms were relieved. The present case series will definitely serve as a tool to spread awareness about the rare ADR´s associated with CS.