Bilateral pedicle stress fracture is a rare entity and few cases have been reported in the literature. Furthermore, the majority of these reports concern previous spine surgery or stress-related activities. Here, the authors report ankylosing spondylitis as a new cause of bilateral pedicle stress fractures accompanying spondylolysis. The reported case adds to the literature on bilateral pedicle stress fracture and spondylolysis by documenting that ankylosing spondylitis is another cause of this condition.