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Show ORIGINAL CONTRIBUTION Silent Sinus Syndrome Presenting as Enophthalmos Long After Orbital Trauma Sandra R. Montezuma, MD, Harsha Gopal, MD, Aaron Savar, MD, Angela Turalba, MD, Dean M. Cestari, MD, and Nurhan Torun, MD, FRCSC Abstract: Late enophthalmos is a well- known consequence of large orbital floor fractures. In rare cases, late enophthalmos can occur after direct trauma to the maxillary ostiomeatal complex and present as silent sinus syndrome ( SSS). We report two cases of SSS manifesting as enophthalmos years after facial trauma. The first patient developed SSS 4 years after a minimally displaced orbital floor fracture. The second patient had progressive enophthalmos as a result of atelectasis of the maxillary sinus years after facial trauma and surgical repair of nasal fractures. There have been two prior reports of SSS presenting after orbital trauma. Our patients differ from these prior reports in that the enophthalmos was discovered years after the initial facial trauma. In the first patient, surgery addressing the blockage of the ostiomeatal complex arrested the enophthalmos; in the second patient, it reversed the enophthalmos. (/ Neuro- Ophthalmol 2008; 28: 107- 110) The most common form of enophthalmos after orbital trauma is seen in large orbital floor ( blowout) fractures ( 1). In such cases, the enophthalmos develops as a consequence of enlargement of the orbital cavity due to the displacement of the orbital floor with changes in soft tissue shape and position ( 2- 6). We report two cases of posttraumatic enophthalmos based not on a major fracture but on retraction of the floor caused by contraction of the maxillary sinus that was due to damage of the ostiomeatal complex. This phenomenon, first described Department of Ophthalmology ( SRM, AS, AT, DMC), Massachusetts Eye and Ear Infirmary, Harvard Medical School, Boston, Massachusetts; and Divisions of Otolaryngology ( HG); and Ophthalmology ( NT), Beth Israel Deaconess Medical Center, Harvard Medical School, Boston, Massachusetts. Address correspondence to Sandra R. Montezuma, Massachusetts Eye and Ear Infirmary and the Department of Ophthalmology, 243 Charles Street, Boston, MA 02114; E- mail: sandra_ montezuma@ meei. harvard. edu after subclinical chronic maxillary sinusitis without trauma, has been called the silent sinus syndrome ( SSS) ( 7- 9). SSS is characterized by progressive enophthalmos, hypoglobus, and pseudoptosis with deepening of the superior sulcus due to progressive collapse of the orbital floor. SSS is thought to be due to atelectasis of the maxillary sinus in the presence of subclinical chronic maxillary sinusitis with obstruction of the ostiomeatal complex ( 8). The first cases were reported by Montgomery ( 7) and Soparkar et al ( 9), who described a series of patients presenting with enophthalmos and hypoglobus in the absence of signs or symptoms of intrinsic sinonasal inflammatory disease or trauma. Enophthalmos after minimal orbital trauma was first recognized by Ross and Kersten ( 10) and Gagnon et al ( 11), who each reported a single case with minimally displaced orbital floor fractures. They attributed the SSS to traumatic damage of the ostiomeatal complex. We report two additional cases with a more delayed presentation than described previously. CASE REPORTS Case 1 A 27- year- old woman was referred to the Neuro-ophthalmology Service of Beth Israel Deaconess Medical Center complaining of a chronic progressive droopy right upper eyelid for an unclear period of time over the last 6 weeks. She described no variability in the lid position during the day. Her past medical history was unremarkable except for a history of being hit with a ball in the right eye while playing lacrosse 4 years earlier. She had not been wearing eye protection, and no imaging had been performed. Examination disclosed deepening of the right superior sulcus ( Fig. 1A). The vertical interpalpebral fissure height, levator function, and marginal reflex distance were normal and symmetric in both eyes. Exophthalmometry measurements were 16 mm on the right and 18 mm on the left ( Fig. IB). The globes were nontender to palpation, and there was no resistance to retropulsion. Best- corrected visual acuity was 20/ 25 in both J Neuro- Ophthalmol, Vol. 28, No. 2, 2008 107 J Neuro- Ophthalmol, Vol. 28, No. 2, 2008 FIG. 1. Case 1. External photographs show a deep right superior sulcus ( A) and right enophthalmos ( B). eyes and extraocular motility was full. Results of the eye examination were otherwise normal. CT of the orbits revealed complete opacification of the right maxillary sinus with mucosal thickening and a central area of high attenuation material consistent with inspissated secretions. The right ostiomeatal unit was opacified suggesting that it was blocked. The left ostiomeatal unit was patent. There was a 4 mm depressed fracture of the right orbital floor with 1 mm of displacement ( Fig. 2). MRI of the brain and orbits revealed similar findings with regard to the ostiomeatal complex. The patient underwent endoscopic right anterior ethmoidectomy and maxillary antrostomy Intraoperatively, the uncinate process was found to be displaced laterally and adherent to the medial orbital wall. This was carefully removed avoiding orbital trauma. The inferior wall fracture was found to extend into the medial wall, causing collapse of the right ethmoidal infundibulum. Anteriorly, in the region of the natural ostium of the maxillary sinus, there was very thick overlying bone. This was removed revealing the natural ostial area anteriorly and superiorly within the ethmoidal infundibulum. A large antrostomy was performed with microdebrider- assisted removal of the thickened mucosal tissue and polypoid tissue from within the maxillary sinus. There were no complications, and the patient tolerated the procedure well. Cultures from the mucus within the maxillary sinus revealed rare growth of oropharyngeal flora. Histopathology revealed thickening of the basement membrane and Montezuma et al FIG. 2. Case 1. Coronal CT of the maxillary sinus shows complete opacification of the right maxillary sinus, mucosal thickening, a 4 mm depressed fracture of the orbital floor with 1 mm displacement ( black arrow), and closure of the ostiomeatal complex ( white arrow). inflammatory changes of the sinus mucosa with a prominent eosinophilic component. Six months after surgery, there was mild asymmetry of the lids; exophthalmometric measurements and the rest of the examination remained unchanged. Case 2 A 34- year- old woman was referred to the Neuro-ophthalmology Service of the Massachusetts Eye and Ear Infirmary because of a history of progressive right eyelid changes. The patient had noted a relatively prominent right upper lid crease. She also complained that her right eye had become deeper- set for an uncertain period. She had sustained facial trauma at age 7, and underwent three subsequent operations to repair a nasal fracture. The first operation was done at the time of the trauma; the second and third operations were done 17 years later. There was no detailed information about these operations. Her medical and ocular history was otherwise unremarkable. High upper lid sulci were evident on both sides, but the right was more prominent than the left ( Fig. 3 A). There was no ptosis, lid lag, or scleral show. Levator function was intact on both sides, and the vertical palpebral fissure and marginal reflex distance measurements were symmetric. Exophthalmometry measurements were 13 mm in the right eye and 16 mm in the left eye ( Fig. 3B). Best- corrected visual acuity was 20/ 20 in both eyes and extraocular motility was full. The eye examination was otherwise normal. 108 © 2008 Lippincott Williams & Wilkins Silent Sinus Syndrome Presenting as Enophthalmos J Neuro- Ophthalmol, Vol. 28, No. 2, 2008 FIG. 3. Case 2. External photographs show a deep right superior sulcus ( A) and right enophthalmos ( B). CT of the orbits revealed attenuation of the right maxillary sinus with an inferiorly bowed right orbital floor and closure of the ostiomeatal complex. There was no opacification and only mild mucosal thickening of the sinuses ( Fig. 4). Based on these findings, the patient was believed to have maxillary atelectasis with increased orbital volume causing enophthalmos on the right side. She underwent a right maxillary sinus antrostomy in conjunction with right orbital floor augmentation. Intra-operatively, there was mild edema of the anterior portion of the right middle turbinate. The right uncinate process was found in an extreme lateral position very close to the medial orbital wall. There was no mucosal thickening or fluid noted in the maxillary sinus. The right uncinate process was removed at its inferior portion and the natural ostiomeatal complex of the maxillary sinus was widened. Right orbital augmentation was subsequently performed using an orbital wedge implant. Five months after the surgery, there was mild asymmetry of the eyelids with deepening of the left superior sulcus. Exophthalmometry measurements were 16.5 mm in the right eye and 16 mm in the left eye. The rest of the examination was unchanged. Orbital CT showed a right augmentation implant in good position. DISCUSSION Our patients differ from prior case reports of SSS after facial trauma with respect to the time of presentation. Our patients had a later presentation, many years after trauma, compared with 2 months ( 11) and 6 months ( 10) as reported previously. The delay in presentation demonstrates the fact that the natural history of SSS can be protracted. In our patients, the manifestations probably developed gradually and were so minimal that the patients waited a long time before seeking medical attention. The enophthalmos caused by large blowout orbital fractures is diagnosed within 4 weeks in most patients ( 12). The enophthalmos in our patients developed years after facial and orbital trauma and probably resulted from SSS, in which direct damage to the ostiomeatal complex leads to chronic ostiomeatal obstruction, accumulation of secretions with chronic subclinical inflammation, and hypoventilation of the maxillary sinus. As a consequence, there is increased negative pressure that causes maxillary sinus atelectasis ( 9,13- 15). Stenosis of the ostiomeatal complex can also occur from altered anatomy in the absence of accumulated secretions in the sinus, as demonstrated in our Case 2. The resulting presentation is a slow progressive enophthalmos and hypoglobus due to retraction of the orbital floor ( 16). In our Case 1, CT showed characteristics described in SSS, including total or near total opacification of the maxillary sinus, infundibular obstruction, lateral retraction of the uncinate process, apposition of the uncinate process against the inferomedial portion of the orbital wall, and a relatively enlarged middle meatus ( 17). In our Case 2, CT did not reveal opacification or fluid in the sinuses, but it 109 J Neuro- Ophthalmol, Vol. 28, No. 2, 2008 Montezuma et al demonstrated lateral retraction of the uncinate process with closure of the ostiomeatal complex and atelectasis of the right maxillary sinus. Illner et al ( 18) described these radiologic findings of increased orbital volume on the affected side as a result of downward retraction of the orbital floor into the maxillary sinus and a variable degree of inward bowing of all of the maxillary sinus walls. In cases of enophthalmos, particularly if there is a recent history of trauma, we suggest a careful radiologic evaluation of the ostiomeatal complex, as well as assessment of the sinus volume, wall configuration, appearance of the sinus infundibulum, position of the uncinate process, and evaluation of the adjacent structures including the orbit, middle meatus, and middle turbinate ( 17). If obstruction of the ostiomeatal complex is demonstrated, treatment should consist of surgically restoring the patency of the ostiomeatal complex and performing an endoscopic maxillary antrostomy with or without orbital floor reconstruction depending on the severity of the enophthalmos. The lack of surgical repair may lead to progressive worsening of the condition. Our Case 1 did not require surgical augmentation of the orbital floor because she had mild enophthalmos. In Case 1 surgery arrested the enophthalmos, and in Case 2 it eliminated the enophthalmos. Both patients, however, had persistent mild eyelid asymmetry. A two- stage operation with delayed repair of the orbital floor has been recommended to minimize the risk of infection with an orbital implant and to ensure that the obstruction has been successfully cleared. Endoscopic antrostomy alone may allow for spontaneous resolution of the enophthalmos ( 19- 21). REFERENCES 1. Pearl RM, Vistnes LM. 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