PT - JOURNAL ARTICLE AU - Naoki Kanda AU - Koichi Takeda AU - Shuji Hatakeyama AU - Masami Matsumura TI - Ankylosing spondylitis presenting with enthesitis at an uncommon site and fever of unknown origin AID - 10.1136/bcr-2019-230113 DP - 2019 Aug 01 TA - BMJ Case Reports PG - e230113 VI - 12 IP - 8 4099 - http://casereports.bmj.com/content/12/8/e230113.short 4100 - http://casereports.bmj.com/content/12/8/e230113.full SO - BMJ Case Reports2019 Aug 01; 12 AB - A 58-year-old man presented with a 2-month history of arthralgia and bilateral temporal region pain, and a 1-month history of fever. He had had refractory neck pain since his 20s. Reduced cervical and lumbar mobility was observed. Radiographs of cervical and thoracic vertebrae disclosed syndesmophytes. Pelvic radiographs showed sclerosis in the right sacroiliac joint and ankylosis in the left sacroiliac joint. MRI with contrast enhancement showed enthesitis in the upper extremities and enhancement in the bilateral temporal muscle, which indicated enthesitis of temporal muscle. He was diagnosed with ankylosing spondylitis based on the limitation in mobility of the lumbar spine and radiographic findings. To the best of our knowledge, this is the first report describing enthesitis of the temporal muscle. This case highlights that ankylosing spondylitis can be accompanied with enthesitis at the temporal muscle and fever of unknown origin at the initial presentation.