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| CASE REPORT |
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| Year : 2023 | Volume
: 16
| Issue : 1 | Page : 133-135 |
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Silent sinus syndrome: A missed entity?
Pratheeba Devi Nivean1, TS Mohammed Sayee2, M Nivean1
1 Consultant M N, Chennai, Tamil Nadu, India
2 Medical Superindentant, Arunai Medical College and Hospital, Vellore, Tamil Nadu, India
| Date of Submission | 14-Nov-2021 |
| Date of Decision | 17-Apr-2022 |
| Date of Acceptance | 04-Aug-2022 |
| Date of Web Publication | 21-Feb-2023 |
Correspondence Address:
Pratheeba Devi Nivean
M N Eye Hospital, Chennai, Tamil Nadu
India
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/ojo.ojo_332_21
 Abstract | | |
Silent sinus syndrome is a rare anomaly caused due to hypoventilation in the maxillary sinus. This is mostly unilateral and asymptomatic in majority of patients. Some patients develop complications due to this such as hypoglobus and enophthalmos. The usual age of occurrence is after 30 years. We report this unique case as our patient was very young to develop this.
Keywords: Maxillary atelectasis, maxillary sinus, silent sinus syndrome
How to cite this article:
Nivean PD, Mohammed Sayee T S, Nivean M. Silent sinus syndrome: A missed entity?. Oman J Ophthalmol 2023;16:133-5 |
| Introduction | |  |
Silent sinus syndrome (SSS) is a silent entity which comprises painless enophthalmos, hypoglobus, and facial asymmetry.[1] It is a part of chronic maxillary atelectasis (CMA). The term SSS was coined by Soparkar et al with the observation in a series of 14 patients.[1]
SSS and CMA are different entities but they share certain common findings.[2] Rose et al. have defined it as “imploding antrum syndrome” to describe the more acute and symptomatic event of sinus implosion that happens rapidly after long periods of chronic underlying atelectasis.[3]
| Case Report | | |
An 18-year-old boy came with complaints of acute lagophthalmos and deviation of the mouth. He was apparently normal before the onset of symptoms which were very acute. He noticed the symptoms when he woke up in the morning. On examination his best corrected vision was 6/6, n6 in both eyes. Extraocular motility was normal in both eyes. His anterior and posterior segment examination was within normal limits. There was lagophthalmos, and he was diagnosed to have Bell's palsy. There was no corneal exposure as his Bell's phenomenon was good. Baseline blood tests were normal. Magnetic resonance imaging brain was advised to rule out any pathology in the brain. Incidentally, we noted SSS in his right maxillary sinus [Figure 1]. We observed mild hypoglobus and slight facial asymmetry in the right side. Hertel's exophthalmometry showed a 2-mm difference between both eyes. He did not give any history of trauma or surgery in the right eye. We reassured the person and treated him with electrostimulation, oral steroids, and vitamin supplements. We advised an ENT opinion, but the patient did not undergo any procedure as he was asymptomatic. We present this case due to the rarity of this disease. | Figure 1: (a) External photograph of the boy with the linear line showing the hypoglobus and the arrow showing the right midfacial hypoplasia. (b) MRI coronal section showing attenuated right maxillary sinus suggesting silent sinus syndrome (arrow). MRI: Magnetic resonance imaging
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| Discussion | | |
It is usually a unilateral disorder affecting the maxillary sinus, and is found at the age of 30–60, with no gender predisposition.[4] Our patient was very young. (average age reported is 26 years).[1]
SSS occurs due to hypoventilation of the maxillary sinus due to obstruction of the osteomeatal complex. Occlusion of the sinus opening and hypoventilation causes atelectasis.[5] Another theory is mechanical theory where there is contraction and relaxation of masticatory muscles leading to negative pressure within the sinus and causes collapse.[6]
The common eye findings are upper lid retraction, deepened upper lid sulcus, malar depression, facial asymmetry, hypoglobus, and enophthalmos. Vertical diplopia is a common presenting complaint in many patients.
Nasal septum deviation, laterally deviated middle turbinate, narrowed infundibular passage, and aberrant nasal anatomy are thought to be risk factors for SSS. These variations predispose to a “flap-valve” occlusion of the natural maxillary sinus ostium and development of negative pressure in the sinus antrum.[7] Chronic sinusitis, osteomyelitis, malignant sinus infiltration, orbital trauma, scleroderma, pseudoenophthalmos and atrophy of orbital contents, secondary granulomatous polyangiitis, irradiation, or sclerosing pseudotumor could be the differentials of SSS.[8]
Williams syndrome (WS), also referred to as Williams–Beuren syndrome, is a rare genetic, multisystem disorder where there are typical facial features, cardiovascular anomalies, growth failure, skeletal abnormalities, hypercalcemia, endocrine disorders, and distinct neurodevelopmental and behavioral profile. The prevalence of WS has been reported to be around 1 in 10,000 live births. Petraroli et al. have reported SSS with Williams syndrome in a 7-year-old patient.[9] According to the authors, the peculiar facial features typical of WS could be responsible for the development of SSS in the early part of life.
Endoscopic antrostomy to widen the sinus opening will help in sinus aeration and prevent further collapse. In symptomatic patients with severe hypoglobus, orbital floor has to be repaired. Surgical modality is employed to treat orbital floor weakness and thereby prevent the collapse of the maxillary sinus. Some authors propose that pathogenesis is self-reversing by natural orbital floor and reconstruction, whereas others debate that the maxillary opened should be done endoscopically as a secondary procedure.[10]
Nonsurgical methods of injecting fillers in the intraconal and extraconal compartments have been reported by Mavrikakis et al.[11] This is to correct the residual enophthalmos after sinus surgery.
| Conclusion | | |
Although the finding was incidental, we report this case as it is a rare syndrome and can be missed easily. Our patient was very young compared to the reports in the literature. Facial asymmetry could also be caused by SSS, and this stresses the importance of holistic examination.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form, the legal guardian has given her consent for images and other clinical information to be reported in the journal. The guardian understands that her names and initials will not be published and due efforts will be made to conceal the patient's identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
| References | | |
| 1. | Soparkar CN, Patrinely JR, Cuaycong MJ, Dailey RA, Kersten RC, Rubin PA, et al. The silent sinus syndrome. A cause of spontaneous enophthalmos. Ophthalmology 1994;101:772-8.  |
| 2. | Kass ES, Salman S, Rubin PA, Weber AL, Montgomery WW. Chronic maxillary atelectasis. Ann Otol Rhinol Laryngol 1997;106:109-16. |
| 3. | Rose GE, Sandy C, Hallberg L, Moseley I. Clinical and radiologic characteristics of the imploding antrum, or “silent sinus,” syndrome. Ophthalmology 2003;110:811-8. |
| 4. | Sesenna E, Oretti G, Anghinoni ML, Ferri A. Simultaneous management of the enophthalmos and sinus pathology in silent sinus syndrome: A report of three cases. J Craniomaxillofac Surg 2010;38:469-72. |
| 5. | Davidson JK, Soparkar CN, Williams JB, Patrinely JR. Negative sinus pressure and normal predisease imaging in silent sinus syndrome. Arch Ophthalmol 1999;117:1653-4. |
| 6. | Canzi G, Morganti V, Novelli G, Bozzetti A, Sozzi D. Posttraumatic delayed enophthalmos: Analogies with silent sinus syndrome? Case report and literature review. Craniomaxillofac Trauma Reconstr 2015;8:251-6. |
| 7. | Babar-Craig H, Kayhanian H, De Silva DJ, Rose GE, Lund VJ. Spontaneous silent sinus syndrome (imploding antrum syndrome): Case series of 16 patients. Rhinology 2011;49:315-7. |
| 8. | Burroughs JR, Hernández Cospín JR, Soparkar CN, Patrinely JR. Misdiagnosis of silent sinus syndrome. Ophthalmic Plast Reconstr Surg 2003;19:449-54. |
| 9. | Petraroli M, Riscassi S, Panigari A, Maltese M, Esposito S. Silent sinus syndrome and williams syndrome: Two rare diseases found in a pediatric patient. Front Pediatr 2020;8:211. |
| 10. | Thomas RD, Graham SM, Carter KD, Nerad JA. Management of the orbital floor in silent sinus syndrome. Am J Rhinol 2003;17:97-100. |
| 11. | Mavrikakis I, Detorakis ET, Yiotakis I, Kandiloros D. Nonsurgical management of silent sinus syndrome with hyaluronic acid gel. Ophthalmic Plast Reconstr Surg 2012;28:e6-7. |
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