Cyclosporine is an imperative part of GvHD prophylaxis post-transplant. It acts as an immunosuppressive agent by targeting T-cells and inhibiting the activation cascade for lymphokine production. Among much toxicity, Cyclosporine induced neurotoxicity remains slightly daunting owing to inadequate number of studies elucidating the mechanism and implication behind it. The toxicities present with varying symptoms such as visual disturbances, ataxia, seizure episodes and signs of encephalopathy. Herein We report a case of AML, FLT3-ITD mutated with low allelic ratio, C-KIT mutated and AML 1-ETO positive, Post Allogenic stem cell transplant on cyclosporine for immunosuppression which induced Mesial temporal sclerosis. Cyclosporine induced Mesial Temporal Sclerosis is inadequately reported and therefore briefly understood. The availability of case reports with similar clinical picture encourages further study to understand the risk factors and consequent management of the toxicity.