Atrophy of both the supraspinatus and infraspinatus muscles is usually caused by chronic rotator cuff tear, but may also derive from suprascapular nerve entrapment at the spinoglenoid notch. Isolated infraspinatus muscle atrophy is uncommon, and typically associates with suprascapular nerve entrapment occurring distal to the spinoglenoid notch. However, isolated atrophy of the infraspinatus muscle due to insertional tear of the infraspinatus tendon may also occur. We present a case of a 43-year-old male with isolated infraspinatus muscle atrophy and fatty degeneration following an isolated full-thickness infraspinatus tendon tear at the insertion site on the humerus. While it is important to rule out other causes of infraspinatus muscle atrophy, such as concomitant rotator cuff tendon/muscle pathology or suprascapular nerve palsy, we present this case to increase awareness of this uncommon clinical presentation and the potential implications for treatment.