Herein we present a case of a 9-month-old boy admitted to our hospital for acute femoral osteomyelitis and hip septic arthritis treatment. Acute hepatitis with concomitant skin rash developed on the sixth day of intravenous oxacillin therapy. No other known virus- or drug-related skin eruption or hepatitis was suspected. The boy recovered completely after antibiotics were shifted to teicoplanin, and no sequelae were noted. To our knowledge, this boy is the youngest case of reported oxacillin-related hepatitis in the literatures.