Purpose: Spontaneous enophthalmos and hypoglobus, in the absence of ot
her symptoms and unrelated to trauma or surgery, may be alarming to bo
th physician and patient. The authors describe the clinicopathologic f
eatures of a benign syndrome (''silent sinus syndrome'') with this con
stellation of features and discuss the possible pathophysiology. Metho
ds: A multicenter retrospective search for similar clinical cases was
performed. All clinical records, computed tomographs, and pathology re
ports for each case were reviewed at one center. A literature search f
or similar cases also was conducted. Results: Nineteen cases of a new
syndrome are presented. This syndrome affects individuals at approxima
tely the fourth decade of life (average age, 36 years; range, 29-46 ye
ars); is characterized by bone resorption and remodeling of the orbita
l floor due to otherwise asymptomatic maxillary sinus disease; is asso
ciated with ipsilateral maxillary sinus hypoplasia; and is not fully e
xplained by any previously described, classic cystic lesion of the max
illary antrum. Conclusion: Enophthalmos and hypoglobus unassociated wi
th prior trauma, surgery, or other symptoms may represent ''silent sin
us syndrome,'' which is ipsilateral maxillary sinus hypoplasia and orb
ital floor resorption.