Arthroscopic revision of rotator cuff lesions is an increasingly popular procedure with a relatively safe profile. However, associated deep articular infection has been described, with potentially destructive joint sequelae. When occurring, it poses the double challenge of eradicating the infectious agent while preserving the articulation and its function. Experience remains scarce and is mostly based on case reports and small series. These also rely on the evidence from the better-described lower extremity joint infections. Through a complex case, the following report addresses this exceptional situation and offers an unusual solution, taking into consideration the peculiarities of the shoulder joint. With the consent of the patient, a single-stage resection arthroplasty with the implantation of an antibiotic-impregnated cement spacer was performed as a long-lasting—if not definite—treatment. After 4 years, the patient maintains excellent function with no radiological signs of wear or loosening.