The painful os trigonum syndrome is one cause of posterolateral ankle pain. This syndrome is most prevalent in athletes who perform frequent and/or forceful plantar flexion. The painful os trigonum may be misdiagnosed as Achilles and/or peroneal tendinitis. In this case, the patient was misdiagnosed for 15 months and treated for tendinitis. The appropriate clinical tests to evaluate the os trigonum as a source of posterolateral ankle pain are outlined. The surgical and postoperative management for the patient are discussed. Clinicians should be aware of the painful os trigonum syndrome as a possible source of posterolateral ankle pain.