Patients with silent sinus syndrome typically present for investigation of facial asymmetry. Unilateral, spontaneous enophthalmos and hypoglobus are the prominent findings at examination. Imaging of the orbit and sinuses characteristically show unilateral maxillary sinus opacification and collapse with inferior bowing of the orbital floor. It has been suggested that SSS is due to hypoventilation of the maxillary sinus secondary to ostial obstruction and sinus atelectasis with chronic negative pressure within the sinus. Treatment involves functional endoscopic sinus surgery for reestablishing a functional drainage passage, and a reconstructive procedure of the floor of the orbit for repairing the hypoglobus and cosmetic deformity. Ophthalmologists, otorhinolaryngologists, and radiologists must be familiarized with this relatively newly reported disease.