Penile hair tourniquet syndrome (PHTS) is an unusual phenomenon. A physician should have a high index of suspicion when a circumcised child presents with glans swelling and inflammation. It must be considered a surgical emergency, as early diagnosis and treatment can prevent complications (e.g., urethra-cutaneous fistula, complete urethral transection, penile gangrene, and penile amputation). We report a case of two-year-old boy to highlight the importance of early diagnosis and prompt treatment.