We report a rare case of osteochondromatosis of the posterior ankle extra-articular space with a longitudinal tear of flexor hallucis longus (FHL). A 77-year-old woman was referred to our hospital with an approximately 4-year history of pain and swelling in the right posterior ankle joint without obvious trauma. The pain had worsened in the previous 2 years. On presentation, she had tenderness at the posteromedial and posterolateral ankle. Imaging revealed several ossified loose bodies in the posterior ankle extra-articular space. We removed the loose bodies, performed tenosynovectomy around the FHL, and released the FHL tendon using a posterior arthroscopic technique via standard posterolateral and posteromedial portals. A longitudinal tear and fibrillation were detected in the FHL. The patient was able to return to her daily activities approximately 3 weeks after surgery. At the 1-year follow-up visit, she continued to have minor discomfort and slight swelling on the posteromedial aspect of the right ankle but had no recurrence of the ossified loose bodies. To our knowledge, this is the first report of osteochondromatosis of the posterior ankle extra-articular space with a longitudinal tear of the FHL that was treated by removal of loose bodies, tenosynovectomy around the FHL, and release of the FHL tendon via posterior ankle arthroscopy.