Granulomatosis With Polyangiitis as an Etiology of Silent Sinus Syndrome: A Case Report.

Silent sinus syndrome (SSS) is a rare condition characterized by the collapse of the maxillary sinus and the sinking of the eye socket (enophthalmos). Only around 100 cases of SSS have been reported so far. The underlying cause of this condition is the chronic obstruction of the osteomeatal complex, which leads to sinus contraction. In this case, we present a novel finding linking SSS with granulomatosis with polyangiitis (GPA). The patient described is a 39-year-old male who was diagnosed with SSS after a prolonged period of sinus pressure, headaches, epistaxis, and generalized congestion. Additionally, the patient reported a significant autoimmune history, including a previous occurrence of ANCA-mediated glomerulonephritis. Surgical intervention revealed the presence of significant granulation tissue, while histopathological examination identified areas of necrosis, vasculitis, and multinucleated giant cells consistent with GPA. This finding was further supported by the detection of positive blood c-ANCA. This case is particularly noteworthy as it is the first reported instance of GPA causing SSS. It serves as an excellent example to illustrate the underlying pathophysiology of SSS.

Contemporary Treatment of Silent Sinus Syndrome: A Case Report and Literature Review.

Silent sinus syndrome is a rare clinical entity affecting the maxillary sinus, characterized by ipsilateral enophthalmos and hypoglobus. Its etiology and pathophysiology are still debated. It is diagnosed by clinical examination and confirmed with computed tomography. It is commonly managed surgically. We present the case of a 34-year-old woman with silent sinus syndrome treated with a patient-specific implant for orbital reconstruction, functional endoscopic sinus surgery approach, intraoperative scan, and surgical navigation, successfully restoring orbital volume and sinus ventilation.

Post-traumatic Sinus Syndrome, Proposal for a New Clinical Entity (CDR Syndrome) as Variant of the Silent Sinus Syndrome: Systematic Review and Case Series.

The diagnostic criteria for silent sinus syndrome (SSS) are still controversial, especially for the post-traumatic/surgery cases that are, nowadays, excluded from the diagnosis of SSS because lacking of spontaneously. We present a systematic review of the last 10 years and our case series of SSS associated to previous trauma/surgery, proposing a new interpretation of SSS. In this work, following the PRISMA guide lines for systematic reviews, we collected 86 articles published on PubMed, Cochrane Library and Medline Plus since 2013 to 2023 about SSS. We divided them in six groups forming the structure of the review: (1) epidemiology, (2) clinical presentation, (3) imaging, (4) etiopathogenesis, (5) sss and craniofacial trauma and (6) treatment. We reported two explicative clinical cases: two men of 34 and 37 years old, involved in motorcycle accident in 2020 and 2014, respectively, and underwent surgery. They came back in 2023 referring diplopia documented by Hess-Lancaster test. CT-scan reported two clear cases of SSS. Basing on what is reported in literature, and basing on our experience, the post-traumatic/surgery SSS are more frequent than the idiopathic ones. Our proposal is to considered them as two individual entities. We propose to adopt the name of Post-traumatic sinus syndrome, or CDR syndrome (Catalfamo-De Rinaldis), for all cases that respect four specific diagnostic criteria reported into the text.

A novel staging system to consolidate silent sinus syndrome and chronic maxillary atelectasis: A systematic review and case series.

KEY POINTS: Silent sinus syndrome (SSS) and chronic maxillary atelectasis (CMA) represent an overlapping clinical entity, both likely lying on the spectrum of one disease process. There is widespread inconsistency of diagnosis in the literature of reported cases of SSS and CMA. We propose a novel, comprehensive staging system to simplify diagnosis and inform management.

Bilateral maxillary silent sinus syndrome: A case report and literature review.

Silent sinus syndrome refers to a spontaneous enophthalmos caused by maxillary sinus collapse without any symptomatic sinonasal illnesses. Its prevalence is almost entirely unilateral. The authors report a patient with a bilateral silent sinus syndrome managed successfully by middle meatal antrostomies. This case brings attention to recognizing bilateral silent sinus syndrome. Because of its bilateral involvement, the facial disfigurement might be recognized late due to the symmetrical presentation. This article highlights the pathophysiology and reports the detailed course of such a rare disease. The balloon dilatation is promising as a newly described treatment modality in a patient with silent sinus syndrome, although more long-term data on its outcome is needed.

Silent Sinus Syndrome Post Trauma: A Case Report and Literature Review.

Silent sinus syndrome (SSS) is a relatively rare clinical condition occurring in the maxillary sinus exhibiting progressive enophthalmos and hypoglobus. The communication between the orbit and maxillary antrum due to trauma further leads to changes in the maxillary sinus. This could lead to the development of negative pressure within the maxillary sinus, collapse of antrum walls, and sucking in of orbital contents. Here, we present a case of a delayed orbital trauma, which was not treated initially. The patient's complaint was a constant feeling of a sunken right eye along with restriction in eye movements. On examination, the clinical and radiographic features were consistent with SSS. Orbital floor reconstruction was performed with the help of a titanium mesh for the correction of hypoglobus and enophthalmos. During follow-up, the patient showed no recurrence of the presenting functional and esthetic complaints. This clinical entity needs prompt diagnosis and early intervention so as to prevent further complications.

Silent sinus syndrome: A case report.

A 65-year-old patient was referred to the Faculty of Dentistry, Mashhad University of Medical Sciences, Mashhad, Iran, with left facial pain and numbness in the upper lip. Based on clinical examinations and radiographic investigations, the patient was diagnosed with silent sinus syndrome. This study discusses oral findings associated with silent sinus syndrome.

Silent sinus syndrome with interfrontal sinus retraction: A 3-case series using CARE methodology.

INTRODUCTION: Silent sinus syndrome (SSS) is a rare entity, almost exclusively involving the maxillary sinus, frontal location being very rarely reported. The aim of the present study was to describe clinical and radiological characteristics and surgical treatment using the CARE methodology. RESULTS: One woman and 2 men were referred for chronic unilateral frontal pain with imagery showing silent sinus syndrome. All showed partial or complete liquid opacification of the affected sinus associated with a thin interfrontal sinus (IFS) retracted toward the affected sinus. Functional endoscopic sinus surgery was performed in all cases, with good functional results. DISCUSSION: We describe 3 cases of SSS with IFS involvement. The frontal sinus wall seemed most vulnerable, probably most liable to be weakened by atelectasis. The study suggests that frontal SSS can be an etiology in chronic frontal sinusitis. Preoperative findings of IFS retraction are useful for surgical restoration of frontal sinus ventilation, relieving chronic pain and preventing complications.

Silent sinus syndrome: A missed entity?

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.

Bilateral pneumatic blowout fractures in a patient with silent sinus syndrome while diving.

A 37-year-old scuba diver developed sudden severe headache, mid-facial pressure/pain and diplopia while diving. Upon examination, he had signs and symptoms consistent with silent sinus syndrome (SSS), including bilateral enophthalmos, hypoglobus, and diplopia/strabismus in conjunction with CT findings of occluded maxillary sinus ostia, atrophic/collapsed maxillary sinuses, and bilateral orbital floor fractures with fat herniation. As there was no history of trauma or chronic sinusitis, this rare case of bilateral SSS was deemed the cause of the spontaneous fractures (i.e. barotrauma secondary to a lack of equalization to ambient surrounding pressure). Transconjunctival repair of the fractures was successful and maxillary sinus antrostomies re-established aeration of the sinuses. We present this case and its management, as well as review the literature concerning sinus barotrauma as a result of diving and SSS. There have been no reports of orbital fractures primarily caused by scuba diving, nor secondarily from diving with the rare entity, SSS.

Volumetric Analysis of the Sinus and Orbit in Silent Sinus Syndrome After Endoscopic Sinus Surgery.

OBJECTIVE: The term "silent sinus syndrome" (SSS) describes spontaneous enophthalmos secondary to subclinical maxillary sinus atelectasis. Debate remains on whether treatment with endoscopic maxillary antrostomy alone is adequate in reversing atelectasis and globe displacement. This study aims to determine the degree of volume change of the diseased sinus and orbit as well as the change in orbital height in patients treated with endoscopic antrostomy without orbital floor augmentation. STUDY DESIGN: Retrospective review with image analysis. SETTING: Single tertiary care institution. METHODS: Three-dimensional (3D) analysis of computed tomographic imaging data was performed using 3D Slicer. 3D models of the maxillary sinus and orbit of the diseased and normal sides were created, and volume measurements were calculated using the segmentation program. RESULTS: Thirteen patients with SSS who underwent endoscopic sinus surgery (ESS) and had follow-up computed tomographic imaging were analyzed. After endoscopic antrostomy, the mean volume of the diseased maxillary sinus significantly increased by 9.82%, from 6.37 to 7.00 cm3 (p = .0302). There was no significant change in mean orbital volume; however, the mean orbital height decreased by 5.67%, from 38.09 to 35.93 mm from pretreatment to posttreatment samples (p = .0101). All patients had resolution of clinical or radiographic enophthalmos and orbital displacement with ESS alone. CONCLUSION: Endoscopic maxillary antrostomy alone in the treatment of SSS significantly increased maxillary sinus volume and decreased diseased orbital height. These changes were associated with clinical and radiographic improvement in globe displacement. These findings support performing ESS alone, reserving orbital augmentation for patients who do not exhibit adequate clinical improvement.

What may surprise a rhinologist in everyday clinical practice: silent sinus syndrome or pneumosinus dilatans/pneumocele? Literature review and own experience.

BACKGROUND: The aim of the study was to present rare sinus syndromes known as silent sinus syndrome (SSS) and frontal sinus syndrome with excessive pneumatization and bone defects in the wall (pneumocele). The available literature describing pneumocele cases was reviewed. METHODOLOGY: PubMed and Science Direct databases were searched by two independent reviewers. The primary outcome was finding descriptions of the sinus pneumocele. In the end, papers on frontal sinus pneumocele that was not the result of trauma, congenital defects or comorbidities were selected. Moreover, the authors presented their own cases of SSS and pneumocele. RESULTS: Twelve case reports of frontal sinus pneumocele were found, one own case was presented. In addition, 8 subjects with SSS, diagnosed and treated in the period from September 2017 to May 2022, were described. CONCLUSIONS: With the increasing number of patients suffering from sinus diseases and the growing number of endoscopic surgeries, the knowledge of rare sinus syndromes will increase the safety of the procedures performed.

Prevalence of chronic maxillary atelectasis: a radiological study.

OBJECTIVES: Chronic maxillary atelectasis is an infrequent entity and data on its prevalence are lacking. This study investigated the prevalence of chronic maxillary atelectasis and aimed to determine the bilaterality of this entity. METHODS: The data for 5835 patients who underwent paranasal sinus computed tomography from 2016 to 2020 were retrospectively analysed. RESULTS: Fifty-four patients were diagnosed with chronic maxillary atelectasis; its prevalence was 0.92 per cent. The mean age of these 54 patients was 42.98 ± 18.89 years (range, 18-85 years); 17 of the patients were female and 37 were male. Chronic maxillary atelectasis was unilateral in 42 patients and bilateral in 12 patients (22.2 per cent). Eight patients were found to have enophthalmos with apparent facial asymmetry. CONCLUSION: The prevalence of bilateral chronic maxillary atelectasis may be higher than previously reported and bilaterality may increase as the number of diagnosed cases increases. A unified classification is also proposed, which describes the silent sinus syndrome as chronic maxillary atelectasis IIIS.

Chronic Maxillary Atelectasis without Obstruction of the Maxillary Ostium - A Case Report.

Rationale: Chronic maxillary atelectasis (CMA) is assumed to be caused by negative pressure in the maxillary sinus secondary to the obstruction of the ostiomeatal complex. Patient Concerns: A 49-year-old female patient first presented to our hospital complaining of right nasal congestion, rhinorrhoea and cheek pain. Diagnosis: Computed tomography (CT) accidentally revealed the inward bowing of the left maxillary sinus, which is a typical sign of CMA or silent sinus syndrome despite the maxillary ostium being potent. Treatment: We did not consider any intervention for CMA because she had no symptoms related to it. Outcomes and Take-Away Lessons: No progression was noted clinically or on CT at the 6-month follow-up. The pathogenesis of CMA in our patient was not explainable by the commonly accepted theory. Apparent hypertrophy of the left maxillary bone was confirmed on CT, thus osteitis with chronic rhinosinusitis might be the potential cause of CMA in the open maxillary sinus.

Silent Sinus Syndrome Secondary to Lymphoma: An Unusual Case With Radiological Evidence of Rapid Progression.

OBJECTIVE: To describe a case of silent sinus syndrome secondary to malignancy and discuss the pertinent clinical findings. Silent Sinus Syndrome (SSS) refers to a rare, asymptomatic condition whereby occlusion of the maxillary sinus ostium results in gradual resorption of air, creation of negative pressure and collapse of the maxillary walls. METHODS: Review of medical records and literature review using NCBI/PubMed. RESULTS: We describe a case of a 54-year-old gentleman presenting solely with enophthalmos. He had been diagnosed with stage IVa small lymphocytic lymphoma (SLL) 1.5 years prior to this, which was being managed with active surveillance. CT demonstrated severe bowing of the anterior and posterolateral wall, inferior displacement of the floor of the orbit and right enophthalmos, thus supporting a diagnosis of silent sinus syndrome. Compared to previous staging CT at the time of the lymphoma diagnosis these findings were entirely new, and soft tissue in the pterygomaxillary fissure was found to be enlarged. The patient underwent endoscopic sinus surgery and a right maxillary mega-antrostomy was performed to ventilate the maxillary sinus and prevent progression of eye symptoms. A biopsy was taken from the pterygopalatine fossa, which was confirmed to be chronic lymphocytic leukemia (CLL). CONCLUSION: This case is unique both in being secondary to malignancy, as well as being rapidly progressive given the presence of radiologically normal appearances 1.5 years prior to presentation. Although a rare condition, prompt recognition of SSS is vital to prevent ophthalmological complications. This report highlights malignancy as a potential cause in cases with focal bony remodeling.

Ethmoidal silent sinus syndrome after nasal swab test.

This study reported the case of a healthy male in his 40s who presented with a 3-month history of frontal headache and post-nasal drip, which did not improve with oral antibiotics. One month prior to the onset of the symptoms, he underwent a nasopharyngeal swab testing for SARS-CoV-2 (which yielded a negative result) for a history of malaise and cough. The patient claimed that the swab insertion into the nasal cavity was particularly painful on the left side. Sinus computed tomography (CT) scan showed deformity of the left middle nasal turbinate with occlusion of the osteomeatal complex, resulting in ethmoid silent sinus syndrome. This study makes a significant contribution to the literature because nasopharyngeal, midturbinate and anterior nasal swabs have been recommended as initial diagnostic specimen collection methods by the US Centers for Disease Control and Prevention (CDC) for the coronavirus disease 2019. These methods require introducing an instrument into the nasal cavity, potentially leading to adverse effects due to the delicate and complex nasal anatomy. However, complications related to swab testing for respiratory pathogens have not yet been fully discussed in the literature.

Patient specific implants in orbital reconstruction: A pilot study.

PURPOSE: Successful repair of the orbital skeleton restores function and cosmesis by normalizing globe position and allowing full motility of the extraocular muscles. Routine repairs are successful with standard implants. However, defects that are irregular or cause volume deficiency can be challenging to repair. The development of patient specific implants (PSI) offers an additional tool in complex cases. Herein, we report our experience using PSI for orbital reconstruction. METHODS: An IRB-approved review was conducted of consecutive patients who received PSI from 8/2016-9/2018. Demographic and examination findings were recorded. PSI was designed using high-density porous polyethylene or polyetheretherketone (PEEK) and implanted for repair. The postoperative course was reviewed for outcomes and complications. RESULTS: Eight patients were identified. Two had silent sinus syndrome, 3 were complex facial fracture revisions, and 3 were post-oncologic reconstruction. Seven received porous polyethylene implants, and 1 had a PEEK implant. Mean follow up time was 10.2 months (3.3-28.3). All had an improved functional and aesthetic result. Diplopia and enophthalmos completely resolved in 60% of fracture and silent sinus patients. All fracture and silent sinus patients were orthotropic without diplopia in primary gaze at last follow up. Tumor patients had improvement in symmetry and functionality. There were no complications. CONCLUSION AND IMPORTANCE: Complex orbital skeleton derangements can be difficult to repair and standard implants may incompletely resolve the anatomic problem. In challenging cases, PSI may better achieve an aesthetically and anatomically successful outcome and improve functionality.

Microbiology, Histopathology, and Radiographic Findings in Silent Sinus Syndrome.

BACKGROUND: The histopathology and microbiology associated with silent sinus syndrome (SSS) have not been well described. OBJECTIVE: This study details the histopathological and microbiological characteristics in addition to radiographic findings of SSS in comparison to those of chronic maxillary sinusitis (CRS). METHODS: 42 patients diagnosed with SSS at Mayo Clinic Hospital in Arizona were identified. Paranasal computed tomography scans of the 42 SSS patients as well as 42 matched CRS patients were analyzed in order to assess differences in the prevalence of septal spurs/deviation. 20 of the SSS patients and 19 of the matched CRS patients also had histopathology and microbiology reports, which were compiled and summarized. Additionally, 19 SSS and 19 matched CRS patients were contacted via phone survey for a more complete patient history regarding maxillary dental disease/surgery. RESULTS: SSS patients have a significantly higher prevalence of septal spurs/deviation than CRS patients. The microbiomes of SSS patients more closely resemble those of healthy controls than those of CRS patients. Analysis of the histopathology of SSS reveals chronic, non-specific inflammation similar to that seen in non-eosinophilic CRS without polyps. SSS patients were significantly more likely to have a history of maxillary dental disease requiring surgery. CONCLUSION: These data support the hypothesis that the pathogenesis of SSS is more likely due to anatomical/mechanical factors than inflammatory/microbiological factors.