Piriformis syndrome is a neuromuscular disorder characterized by hip, buttock, and leg pain. Axial spondyloarthritis is a rheumatic disease primarily affecting the sacroiliac joint and the spine. Due to their anatomical proximity, the potential relationship between piriformis syndrome and sacroiliitis has been discussed for some time. However, literature review revealed that there is no study on piriformis syndrome in individuals with axial spondyloarthritis. Here, we present the case of a 30-year-old female with axial spondyloarthritis who developed severe low back, hip, and buttock pain that persisted despite initial treatment for axial spondyloarthritis. We first re-evaluated her condition through physical examination, magnetic resonance imaging, and an injection test for piriformis syndrome. Following a comprehensive assessment, the patient was diagnosed with both axial spondyloarthritis and piriformis syndrome. Subsequently, a tailored treatment plan was devised, addressing both conditions, and after a 3-month course of treatment, we obtained significant reduction in pain of the patient. This is the first case report in literature, where we used injection test to confirm the diagnosis of the piriformis syndrome in a patient with axial spondyloarthritis. We therefore strongly advocate considering piriformis syndrome as a potential etiology for pain in individuals with axial spondyloarthritis consistently. This recognition is important as piriformis syndrome does not respond adequately to non-steroidal anti-inflammatory drugs and may lead to unnecessary use of biological disease-modifying antirheumatic drugs. Timely identification and intervention are imperative in ensuring optimal patient care.