Pneumomediastinum (PNM) and pneumothorax (PNX) are documented complications of arthroscopic shoulder surgery (ATS). Plexus anesthetic block and tracheal lesions during endotracheal intubation are hypothesized to be the underlying risk factors; however, the actual evidence supporting this hypothesis is scarce.A case of bilateral laterocervical emphysema, subcutaneous edema, and signs of PNM after ATS performed under general anesthesia and supra-scapular nerve block is presented. An up-to-date systematic review of PNM/PNX during orthopedic surgery was performed, involving six databases: PubMed (1996–present), Embase (1974–present), Scopus (2004–present), SpringerLink (1950–present), Ovid Emcare (1995–present), and Google Scholar (2004–present).Twenty-five case studies met the eligibility criteria. In 24 cases, the patient underwent general anesthesia and orotracheal intubation; in 9 of these, a plexus anesthetic block was also performed. One case involved ATS under plexus anesthetic block only. In 10 cases, the diagnostic finding was PNM. In 5 cases, the diagnostic finding was associated with PNX. PNX was detected in 17 cases. In 2 cases, SE was found in the absence of any evidence of either PNM or PNX. A tracheal lesion was identified in 3 cases.Endotracheal intubation and loco-regional anesthesia are not the only predisposing risk factors at play in the pathogenesis of PNM/PNX. Rather, multi-factorial pathogenesis seems more probable, necessitating that specific attention is paid during ATS to the change in patient position on the operating bed, to any slipping of the endotracheal tube, to patient monitoring whilst under the drapes, and to the cuff pressure. PROSPERO registration number: CRD42021260370.