Ankylosing spondylitis is a debilitating disease afflicting the joints. This disease is a major cause of functional impairment and disability in younger population with the mean age of onset around 26 years. The disease is characterized by chronic back pain and limited spinal mobility due to slowly occurring inflammation and later ankylosis of joints. Temporomandibular joint (TMJ) involvement is one such presentation of this disease with the incidence reported 4–35% among different case series. We describe a case of severe ankylosing spondylitis with TMJ involvement in a 27-year-old female patient.