Abstract
Objective
Unilateral sphenoid sinus opacification (SSO) on imaging is a common incidental radiologic finding. Inflammatory sinus disease is rarely isolated to one sinus cavity therefore SSO raises the potential for neoplastic etiology. The clinical significance of SSO was evaluated and compared to maxillary sinus opacification (MSO).
Methods
A systematic review of unilateral sinus opacification was performed via Medline (1966–January 12th, 2015) and Embase (1980–January 12th, 2015), limited to English literature and human subjects. Case series of patients treated with radiologic evidence of unilateral sinus opacification either from maxillary or sphenoid sinuses and with pathology results were included. Individual cases were classified as neoplastic, malignant, or a condition requiring surgical intervention (i.e. fungal ball). Exclusion criteria were single case reports, lack of primary data, series of complications, or single pathology series. Case-by-case analysis was performed for both SSO and MSO.
Results
Search strategy revealed 3264 studies. A total of 31 studies including 1581 patients met the inclusion criteria. In these studies, SSO was described in n = 1215 (76.9%) and MSO in n = 366 (23.1%). For SSO, the final diagnosis was neoplasia 18%, (malignancy in 10.9%). 58.3% of cases required surgical intervention and 13% were inflammatory. For MSO, neoplasia represented 18.3% (malignancy 7.1%), surgical intervention required in 47% of cases and 27.6%. were inflammatory.
Conclusion
Isolated MSO and SSO is a marker of neoplasia in 18% and malignancy in 7–10% of patients presenting with these radiologic findings. Clinicians should be wary of conservative management given the high incidence of neoplasia and consider a lower threshold for early surgical intervention.
1
Introduction
The finding of isolated sphenoid sinus opacification (ISSO) is an increasingly common clinical problem with the growing utilization of computed tomography (CT) and magnetic resonance imaging (MRI). ISSO is found both incidentally and in the investigation of symptomatic patients, and can be caused by a broad range of pathologic processes.
There is a tendency for some clinicians to treat ISSO conservatively, especially when it is an incidental finding in an asymptomatic patient. While neoplasia and malignancy clearly warrant operative intervention, and infectious etiologies may resolve with medical therapy, unfortunately there are often few clues to suggest a diagnosis and guide decision making.
The clinical diagnosis of ISSO is difficult as it is often insidious, with non-specific symptoms . The most common presenting complaint is headache in various locations and intensities , reported to be present in between 33 and 81% of cases . Visual disturbances including decreased visual acuity and diplopia are the second most common symptom in 24–50% of patients .
Physical examination of sphenoid disease is difficult due to its location deep within the skull base, and nasal endoscopy is normal in a third of cases . When findings are present, they often represent edematous change and are non-specific. Unless pathology protrudes beyond the sphenoethmoid recess, only discharge may be seen.
Radiologic findings on CT and MRI may be suggestive of etiology. Generally, features of bone expansion, thinning, and remodeling suggest inflammatory or benign pathology, whereas bony erosion is more common in malignant tumors but again is not specific .
Given the difficulty in diagnosing ISSO, there is little consensus in the otolaryngology literature on how isolated lesions within the sphenoid sinus should be addressed. Some advocate for surgical intervention regardless of etiology because of the sphenoid’s location in the skull base and potential for serious complications due to proximity to adjacent neurovascular structures.
The aim of this study was to systematically review published studies defining the pathologic findings of ISSO. The reported rates of neoplasia, malignancy, and the proportion of cases that require operative intervention were sought in order to guide management of this condition. Anecdotally it is thought ISSO has a higher rate of neoplasia than other isolated paranasal sinus disease, quoted to be around 15–16% in case series . We performed a simultaneous analysis of isolated maxillary sinus opacification (IMSO) cases in order to compare these conditions and provide further clinical context. Understanding the likelihood of neoplasia, malignancy and the proportion that require operative intervention is critical in determining the management of these patients.
2
Materials and methods
2.1
Search strategy and selection of studies
A systematic review of the literature was performed via Embase and Medline to identify studies containing any published case series, case-control or cohort studies in which the pathology of isolated sphenoid and maxillary opacification was investigated.
Isolated disease was considered as opacification that did not extend beyond the sphenoethmoidal recess, or in the case of maxillary opacification, beyond the maxillary osteum. Studies in which there were cases involving extra-sinus extension were excluded, as the focus of this study was to guide management of disease isolated to the sphenoid sinus.
Published case series, case-control studies, cohort studies and randomized control trials were included. Eligible studies reported the pathologic and radiologic findings for patients with isolated sphenoid sinus disease. Single case reports, single pathology series, series of complications and descriptive/expert opinion papers or those that lacked primary data were excluded in order to minimize selection bias. No unpublished trials were included.
2.2
Data extraction and risk of bias assessment
PRISMA guidelines were followed and this research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors. Embase® (1974-present) and Medline® (1946-present) was searched on January 12th, 2015 using a comprehensive strategy with a combination of MESH terms and keywords. This was limited to English and human subjects and the results verified by a second reviewer. The studies identified were evaluated against the inclusion criteria for eligibility.
Once studies were selected, case-by-case data was collected. Case data was extracted from graphs, tables, images and text, and each case was assessed for inclusion. These primary data were then collated into a structured data form and categorized by cavity involved. Cases were excluded where a pathologic diagnosis was not reported without follow up for resolution, and where cases lacked pathology results AND did not resolve with medical management (and could not therefore be presumed inflammatory or infectious).
Each case was further categorized as neoplastic or non-neoplastic, malignant or benign and labeled with a pathologic diagnosis. Pathologic diagnosis groups were benign neoplasms (such as inverting papilloma, pituitary adenoma), malignant neoplasms (squamous cell carcinoma, adenocarcinoma, sinonasal undifferentiated carcinoma, metastases), fungal diseases, antrochoanal/sphenochoanal polyps, simple cysts, mucocoeles, acute and chronic inflammatory rhinosinusitis, fibroosseous diseases, meningoencephalocoeles, cerebrospinal fluid leaks, foreign bodies, and vascular aneurysms. The diagnosis was used to define cases as neoplastic or non-neoplastic, benign or malignant, and whether or not it required surgical intervention for resolution.
Among the included studies, there was a large range of measurement methods and study objectives. Studies and the cases included were deemed suitable if they contained clear and appropriate inclusion and exclusion criteria and reported pathology. Cases that resolved with medical management without pathology results were classified as inflammatory/infectious.
2.3
Outcomes
The primary outcomes were the frequency of neoplasia (both benign and malignant), and the proportion of cases that required operative intervention. A “condition requiring operative intervention” was defined as pathology that would not resolve with medical management. For example, this includes most non-inflammatory/infectious conditions (mucocoeles, fibroosseous diseases, foreign bodies, and meningocephaloceles), but also some inflammatory/infective conditions (like fungal balls or allergic fungal sinusitis) that are known to require operative intervention for resolution.
2.4
Statistical analysis
Statistical analyses were performed using SPSS v 22 (IBM SPSS Statistics for Windows, Version 22.0 Armonk, NY). Descriptive data are presented as percentages and chi-squared analysis was used for relationships of nominal variables. Student’s t -tests (2-tailed) were used for comparisons of parametric data. Results were deemed significant with a p value of < 0.05.
2
Materials and methods
2.1
Search strategy and selection of studies
A systematic review of the literature was performed via Embase and Medline to identify studies containing any published case series, case-control or cohort studies in which the pathology of isolated sphenoid and maxillary opacification was investigated.
Isolated disease was considered as opacification that did not extend beyond the sphenoethmoidal recess, or in the case of maxillary opacification, beyond the maxillary osteum. Studies in which there were cases involving extra-sinus extension were excluded, as the focus of this study was to guide management of disease isolated to the sphenoid sinus.
Published case series, case-control studies, cohort studies and randomized control trials were included. Eligible studies reported the pathologic and radiologic findings for patients with isolated sphenoid sinus disease. Single case reports, single pathology series, series of complications and descriptive/expert opinion papers or those that lacked primary data were excluded in order to minimize selection bias. No unpublished trials were included.
2.2
Data extraction and risk of bias assessment
PRISMA guidelines were followed and this research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors. Embase® (1974-present) and Medline® (1946-present) was searched on January 12th, 2015 using a comprehensive strategy with a combination of MESH terms and keywords. This was limited to English and human subjects and the results verified by a second reviewer. The studies identified were evaluated against the inclusion criteria for eligibility.
Once studies were selected, case-by-case data was collected. Case data was extracted from graphs, tables, images and text, and each case was assessed for inclusion. These primary data were then collated into a structured data form and categorized by cavity involved. Cases were excluded where a pathologic diagnosis was not reported without follow up for resolution, and where cases lacked pathology results AND did not resolve with medical management (and could not therefore be presumed inflammatory or infectious).
Each case was further categorized as neoplastic or non-neoplastic, malignant or benign and labeled with a pathologic diagnosis. Pathologic diagnosis groups were benign neoplasms (such as inverting papilloma, pituitary adenoma), malignant neoplasms (squamous cell carcinoma, adenocarcinoma, sinonasal undifferentiated carcinoma, metastases), fungal diseases, antrochoanal/sphenochoanal polyps, simple cysts, mucocoeles, acute and chronic inflammatory rhinosinusitis, fibroosseous diseases, meningoencephalocoeles, cerebrospinal fluid leaks, foreign bodies, and vascular aneurysms. The diagnosis was used to define cases as neoplastic or non-neoplastic, benign or malignant, and whether or not it required surgical intervention for resolution.
Among the included studies, there was a large range of measurement methods and study objectives. Studies and the cases included were deemed suitable if they contained clear and appropriate inclusion and exclusion criteria and reported pathology. Cases that resolved with medical management without pathology results were classified as inflammatory/infectious.
2.3
Outcomes
The primary outcomes were the frequency of neoplasia (both benign and malignant), and the proportion of cases that required operative intervention. A “condition requiring operative intervention” was defined as pathology that would not resolve with medical management. For example, this includes most non-inflammatory/infectious conditions (mucocoeles, fibroosseous diseases, foreign bodies, and meningocephaloceles), but also some inflammatory/infective conditions (like fungal balls or allergic fungal sinusitis) that are known to require operative intervention for resolution.
2.4
Statistical analysis
Statistical analyses were performed using SPSS v 22 (IBM SPSS Statistics for Windows, Version 22.0 Armonk, NY). Descriptive data are presented as percentages and chi-squared analysis was used for relationships of nominal variables. Student’s t -tests (2-tailed) were used for comparisons of parametric data. Results were deemed significant with a p value of < 0.05.
3
Results
The search produced n = 1846 EMBASE and n = 1418 MEDLINE manuscripts. When duplicates were removed 1947 studies remained (Fig. 2 – PRISMA in Section A.2 ). Title review identified 274 relevant studies, and a review of abstracts produced 41 studies. Full text review of these yielded 25 studies of isolated sphenoid sinus disease and 6 studies of maxillary sinus disease which were included.
Case-by-case analysis was performed, and one study’s cases were excluded as patients’ data was reported in a later study . In total, 1215 patients with isolated sphenoid disease, and 366 cases of isolated maxillary disease met inclusion criteria . Table 1 in Section A.3 details the characteristics of included studies.
Neoplasia was present in 18.4% (n = 224) of isolated SSO and 18.3% (n = 67) of MSO (p = 0.165). Malignancy was present in 10.9% (n = 132) of isolated SSO and 7.1% (n = 26) of all MSO cases (p = 0.035). The proportion of isolated sphenoid sinus opacities (ISSO) requiring surgical intervention was 58.3% (n = 709) vs 47.0% (n = 172) of MSO (p = 0.0001).
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