Pain after hip replacement is one of the most common problems during rehabilitation and is often the main obstacle in rehabilitation, even though it can often be controlled by localized cryotherapy and/or administration of analgesics. However, patients with positive anamnesis for hip arthritis and long-lasting pain may report persistence of symptoms for months after surgical intervention; often, in these patients, contractures and muscle retraction in the pelvic region are observed. The present study reports the case of a female patient who suffered from complications after total hip replacement (THR) for osteoarthritis. Due to severe pain in the gluteal region not responding to standard treatments the patient was unable to stand in an upright position or walk, so she was forced to stop the rehabilitation program. Treatment by injection of botulinum toxin type A (BTX-A) in the gluteus maximus muscle brought about the complete resolution of pain and functional recovery. The follow-up visits, carried out after 6 and 16 months, confirmed the complete healing of the patient. BTX-A has been shown to be effective in the treatment of painful localized contractures even in the absence of neurological lesions. Therefore, BTX-A could be a feasible option to treat painful localized contractures that do not respond to standard treatments. Further investigations are suggested to better identify appropriate dosages and the best inoculation schedule.