Rationale: Terbinafine is an antimicrobial agent commonly prescribed for fungal infections. Its side effect profile is generally benign, but there is limited evidence that it has the potential to cause rhabdomyolysis. Rhabdomyolysis is a potentially life-threatening condition caused by profound muscle injury. It has characteristic findings of muscle pain, weakness, and dark urine. When recognized early, patients with rhabdomyolysis can be managed conservatively with hydration and watchful monitoring. However, if treatments are delayed, or in severe cases of rhabdomyolysis, complications such as electrolyte abnormalities, acute kidney injury, and disseminated intravascular coagulation can develop. Presenting concerns of the patient: A previously healthy 22-year-old male presented with nausea, vomiting, and dark urine after taking terbinafine 250 mg daily for a tinea infection for 9 days. He developed severe rhabdomyolysis with a serum creatine kinase (CK) of >100 000 U/L as well as anuric acute kidney injury. Diagnosis: The clinical history combined with the diagnostic findings suggest acute kidney injury and rhabdomyolysis associated with terbinafine use. Interventions: Terbinafine use was stopped immediately. The patient was started on intravenous fluids and bicarbonate drip. Hemodialysis was initiated to prevent further complications. After his CK level decreased and his clinical status stabilized, he was discharged home and continued to receive outpatient hemodialysis treatments. Outcome: The patient’s kidney function returned to baseline after 1 month of outpatient hemodialysis treatments. Novel finding: In this report, we present a case of rhabdomyolysis associated with terbinafine use that progressed to acute kidney injury requiring dialysis. Our case highlights a less known and severe side effect of this medication and emphasizes the importance of early recognition and treatment of rhabdomyolysis.