LAUSR

Introduction and importance Heterotopic ossification (HO) associated with Anti-N-methyl-d-aspartate (anti-NMDA) receptor encephalitis is rare and the treatment strategy is unclear. Case presentation We report the case of a 31-year-old female diagnosed with anti-NMDA receptor encephalitis from Osaka General Medical Center, Osaka, JAPAN that developed heterotopic ossification of the knees during prolonged coma. She was unable to walk because of pain and limited range of motion in both knees. Thirty months after the onset of the coma, surgical excision of the heterotopic bone in both knees was performed. The range of motion of both the knees improved markedly and she had no recurrence of heterotopic ossification on a three-year follow-up. Thus, this case can be used as a guide for surgeons with such patients. Conclusions We reported a case of heterotopic bone formation in the periarticular region of both bilateral knees in a patient who suffered a 2-month coma following anti-NMDA receptor encephalitis. Surgical excision of the heterotopic bones significantly improved the passive range of motion in both knees. Three years after the operation, the patient had no complaints about her knees, and there was no recurrence of HO.