Hypokalemia can result in life-threatening complications if not treated appropriately. Although hypokalemia is a frequent adverse effect of amphotericin B therapy, there are no reports in the pediatric literature on hypokalemia-associated rhabdomyolysis induced by this drug. A ten-year-old boy with a history of one week amphotericin B treatment was admitted with weakness of the lower extremities, inability to walk and calf pain. Laboratory tests showed a serum potassium of 1.7 mEq/L and a serum creatinine phosphokinase of 3937 U/L plus myoglobulinuria. Following fluid expansion and intravenous potassium replacement, the patient progressed to achieve full regression of muscular weakness after one week. This report highlights hypokalemia as a rare cause of rhabdomyolysis. Patients on amphotericin B should be checked for this rare yet potentially life-threatening complication.