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Patient: Male, 13-year-old Final Diagnosis: Tourniquet-related nerve injury Symptoms: Muscle weakness • numbness Clinical Procedure: — Specialty: Neurology • Orthopedics and Traumatology • Rehabilitation Objective: Rare disease Background: Congenital thumb duplication comes under the Wassel type IV classification is the hypoplastic variety, with the extra digit growing from the dominant thumb’s soft tissue alone. Excising the hypoplastic finger while reconstructing for the retained one has been the most adopted approach. Tourniquets are commonly utilized tools in orthopedic surgeries to reduce the amount of blood, thus enhancing the visibility. Unfortunately, tourniquet-related nerve injury (TNRI) is gaining more attention as a serious complication of tourniquet use in surgery. Case Report: A 13-year-old Asian boy with preaxial polydactyly Wassel type IV of the right hand underwent reconstruction surgery. A pneumatic tourniquet was applied at 200 mmHg on the right mid-upper arm and maintained for 90 min. After the surgery, the patient had total weakness with numbness, tingling, and burning sensation from his right upper arm to his fingertips. The neurological examination and nerve conduction studies (NCS) results were consistent with axonotmesis lesions. Pharmacological and physical rehabilitation therapy had successfully restored full motoric and sensory function after 6 months. Conclusions: Nerve injury should be acknowledged as a possible complication from routinely-utilized tourniquets in orthopedic surgeries. Our cases may expand the need for further studies to establish a guideline for tourniquet use and TRNI management.