Sirolimus is currently used as an immunosuppressive agent in kidney transplantation due to its lack of nephrotoxicity and antiproliferative properties. However, a large number of side effects has been described with the use of m-Tor inhibitors. Most are reversible when treatment is withdrawn. Hepatotoxicity is one of these side effects, considered as a benign condition and resulting generally in a transitory and small increase in transaminase levels. We report here, to the best of our knowledge, the first case of severe sirolimus-induced acute hepatitis confirmed by liver biopsy, in a renal transplant recipient. This condition was completely cured in few weeks after sirolimus withdrawal.