We report a very rare case of a unilaterally absent piriformis muscle in a 60 year old woman. Accompanying variations comprised a common gluteal artery (instead of two distinct superior and inferior gluteal arteries), and an absent gemellus inferior muscle. The contralateral left side showed a normally developed piriformis muscle. In hominoids, the piriformis is constant, but is regularly missing in several other vertebrates. The piriformis muscle is an anatomical landmark for ultrasound investigations and ultrasound-guided interventions in the deep gluteal region such as a superior gluteal nerve block or even a sacral plexus block, also for any surgical approach such as total hip arthroplasty. A missing piriformis muscle therefore affects the orientation in the deep gluteal region and therefore the identification of the targeted structures.