Eyelids emphysema is a rare condition due to air trapping in subcutaneous tissue of the orbit. It has been clinically and radiologically documented, but histologic evaluation has not been noted. We report a case of a middle-aged woman with periorbital swelling due to self-induced Valsalva maneuver, persisting after decompressive therapy. A fine-needle biopsy was performed and showed a mixed population of osteoclastic-like giant cells and mononuclear epithelioid cells, which were suspected of pathological proliferative disease. Specimens from blepharoplasty indicated pseudocystic empty spaces surrounded by epithelioid cells and giant cell granulomas. Immunohistochemical stains favored diagnosis of benign granulomatous disease, such as subcutis cystic pneumatosis. Histologic examination represented an important tool for differential diagnosis with soft tissue neoplasms of the orbit in subcutis emphysema with unusual clinical presentation and abnormal course.