Levetiracetam (LEV) is one of the newest antiepileptic drugs available on the market and is frequently used in neurosurgical patients requiring antiepileptic assistance. LEV is mainly excreted by the kidney with minimal hepatic metabolism, so it is considered to have a low liver toxicity. Drug-induced liver injury (DILI) associated with LEV administration is extremely rare, with only eight reported cases. In this report, we describe the case of a 44-year-old man who was admitted because of generalized convulsion, and LEV administration at a dose of 3000 mg/day was started following a diagnosis of status epilepticus. Laboratory values before LEV administration were as follows: alanine aminotransferase (ALT), 17 IU/L; aspartate transaminase (AST), 41 IU/L; and total bilirubin, 0.59 mg/dL. Viral serology tests for hepatitis B and hepatitis C yielded negative results. Several hours after LEV administration, the patient developed high-grade fever and his liver enzyme levels were found to be elevated. LEV administration was stopped immediately; the peak laboratory values were as follows: ALT, 1,192 IU/L; AST, 3,150 IU/L; and total bilirubin, 2.02 mg/dL. After conservative treatment, the patient's laboratory values were normalized. A drug-induced lymphocyte stimulation test (DLST) was performed and showed a positive response, indicating that the administration of LEV was responsible for DILI in this patient. Clearly, LEV can provoke DILI despite its low liver metabolism profile. Therefore, readministration of the drug should be avoided in such cases. An
in vitro
examination, such as a DLST, can be useful for ensuring a definitive diagnosis of DILI.