Introduction: Bilateral silent sinus syndrome (SSS) is a very rare pathology reported only in few papers in literature. Most of the described cases are simultaneous, and only one had a metachronous presentation. The evolutionary phases of the disease have yet to be well demonstrated and a complete radiological evaluation is needed to demonstrate the pathogenetic mechanisms that cause the disease.
Case report: A 45-year-old male presented with a left SSS and a bilateral concha bullosa. He developed a contralateral SSS two years after an endoscopic uncinectomy and re-ventilation of the diseased maxillary sinus. This case is the second reported in literature with a metachronous presentation. A pure endoscopic approach has led to the resolution of symptomatology and the full restoration of the ventilation of the maxillary sinuses. The key role of the uncinate process in the genesis of the pathology has been well demonstrated by the onset of a contralateral SSS in a normally developed maxillary sinus thanks to a complete radiological follow-up.
Conclusion: Bilateral presentation is a rare entity; however, it should be considered in patients with SSS. A minimal endoscopic uncinectomy could also prevent the onset of the disease on the healthy side.
Keywords: Diplopia; Endoscopy; Enophthalmos; Maxillary Sinus; Orbit; Paranasal Sinuses.