Anatomical variations at the origin of the biceps tendon have been described by several authors, but occurrences of an origin in the supraspinatus are rare. It is unclear whether this variation might contribute toward pathological conditions of the shoulder. Our objective here was to describe a case of an anatomical variation in the origin of the tendon of the long head of the biceps. The clinical information, preoperative images and arthroscopic images relating to a patient with an aberrant origin of the long head of the biceps, which was observed during shoulder arthroscopy, were reviewed. In this case study, the origin of the biceps was found in the rotator cuff, without any origin from the supraglenoid tubercle or upper labrum. This variant did not seem to contribute toward the pathological condition of the shoulder, and standard treatment for the concomitant condition was sufficient for treating it.