Background: In some patients with neuropathic pain (NP), such as complex regional pain syndrome (CRPS), itching rather than pain is the main symptom making diagnosis and treatment difficult.Case: We report a case of a 23-year-old man with a history of hypoxic brain damage who presented with pruritus of the left foot and ankle. His left foot was fractured, and he underwent surgery 6 months previously. After the operation and cast application, he developed uncontrolled pruritus, swelling, sweating, and flushing of the left foot skin with limping. On examination, he showed well-known features of CRPS without pain. He was diagnosed with an atypical CRPS with neuropathic itching (NI). With treatment modalities used for NP and CRPS, his pruritus subsided gradually, and the his ankle mobility improved. Conclusions Unexplained itching can be the main symptom in some CRPS patients. Treatment according to NP can improve symptoms of NI in CRPS patients.