Maxillary antral mucocele with sphenoidal sinus empyema—a rare case report

Introduction

Maxillary antrum cysts are frequently misdiagnosed because of their variable presentation, unknown etiology, and rarity in occurrence (1). These lesions most often expand and progress to involve the adjacent alveolar bone, prompting the clinician to classify them as odontogenic lesions. A sinus mucocele (SM) is a benign cyst lined by epithelium that develops within one of the paranasal sinuses. The most common sites for occurrence are frontal and ethmoidal with maxillary sinus involvement being rare (2). SM is reported to have a higher incidence in the third and fourth decades of life, with no gender predominance (2,3). Mucocele formation occurs due to the gradual accumulation of mucous in the maxillary sinus caused by blockage of osteomeatal opening. The cause of most cases is unknown, although infection or trauma, which may be due to chronic sinusitis, polyps, bone tumors, and surgical intervention in the paranasal sinuses are prevalent factors (3). Mucoceles usually are asymptomatic slow-growing lesions, but if it is symptomatic, manifest as nasal congestion, facial pain, headache, and fever (3,4). There may be facial asymmetry due to swelling, respiratory obstruction due to medial extension, superior extension resulting in diplopia, and inferior extension resulting in tooth mobility (4).

An expanding mucocele may cause obstruction of drainage or when infected, can spread the infection to other paranasal sinuses (5). The ethmoid sinus, where they are known as ethmoidoceles, is the most common site of such involvement (6).

SMs are usually diagnosed based on a patient’s history and physical exam, but sometimes can be accidentally diagnosed on routine radiographs. Computed tomography (CT) scan or magnetic resonance imaging (MRI) showing fluid-filled cysts within the bone that surrounds each paranasal sinus is more definitive (7).

We present a case of large maxillary SM leading to a sphenoidal sinus infection which was asymptomatic except for facial asymmetry. This rare and unusual presentation was mimicking as odontogenic in origin posing a diagnostic challenge. The uniqueness of this case lies in its spread and extent of involvement, where involvement of maxillary sinus led to an early obstruction and secondary infection of the sphenoid sinus. The existing literature supports the evidence that infection or infection secondary to obstruction from the maxillary sinus has resulted in the spread to the ethmoidal and frontal sinuses. After thorough and deeper radiological and endoscopic evaluations, a treatment plan was developed involving a team of specialists. Timely diagnosis and prompt treatment have resulted in reduced morbidity. We present this case in accordance with the CARE reporting checklist (https://amj.amegroups.com/article/view/10.21037/amj-23-61/rc).

Case presentation

A 21-year-old Dravidian female patient presented to the Department of Oral and Maxillofacial Surgery on 5^th May 2020 with the chief complaint of swelling in the right cheek region for the past month that gradually progressed with no other associated symptoms. The patient had no relevant medical or dental history. Extraoral examination revealed a solitary diffuse swelling of 3×2 cm in size, extending supero-inferiorly from 2 cm below the lower eyelid up to 2 cm above a line drawn from the corner of the mouth to ear lobule and anteroposteriorly 1 cm away from the lateral aspect of the nose to 3 cm in front of the tragus, with obliteration of nasolabial fold with no change in color and texture of the overlying skin. The swelling was firm in consistency with no local rise in temperature.

Intraorally a single 2×3 cm ovoid swelling extending from mesial aspect of the upper right second premolar to the third molar region, with vestibular obliteration and erythematous mucosa overlying was observed. The swelling was firm and non-tender to palpation. On hard tissue examination right upper first molar was grossly decayed, second and third molars were Grade II mobile (Figure 1). A differential diagnosis of radicular cyst, dentigerous cyst, unicystic ameloblastoma was made based on the above findings.

Figure 1 Vestibular obliteration in the right maxilla.

CT revealed a unilocular, well-defined hypodense area extending from the distal aspect of upper right second premolar to posterior border of maxilla with floating appearance of posterior teeth indicating extensive destruction of right maxilla (Figure 2). Contrast-enhanced computed tomography (CECT) coronal view revealed an expansile cystic collection in the right maxillary sinus with air fluid levels, maxillary sinus wall erosions, right osteomeatal unit obliteration, displacement of the medial wall of the antrum obliterating the nasal cavity and opacification in the right sphenoidal sinus (Figures 3,4).

Figure 2 Panoramic view showing extensive involvement of right maxillary sinus with floating teeth appearance.

Figure 3 CECT coronal view showing the air-fluid spaces, obliteration of osteomeatal unit, and bony wall erosions. CECT, contrast-enhanced computed tomography.

Figure 4 CECT coronal view showing opacification of right sphenoidal sinus. CECT, contrast-enhanced computed tomography.

To evaluate the sphenoidal sinus involvement, nasal endoscopy was performed, where obliteration of the sphenoidal recess was observed due to medial expansion of the lesion which resulted in obstruction of drainage and secondary infection and was evident by the accumulation of pus in the posterior aspect of nasal cavity. Neurologic and ophthalmic consultations revealed no abnormality.

A complete hemogram had no abnormality. Fine needle aspiration cytology had chocolate brown fluid, with a protein content of 4.94 g/dL. Incisional biopsy and simultaneous marsupialization were planned due to its size and extent, and the report showed fibro cellular connective tissue with irregularly arranged collagen fibers, focal areas showing pseudostratified columnar epithelium with goblet cells, mild to moderate infiltrate with predominance of lymphocytes, plasma cells and few macrophages suggesting an inflammatory lesion.

Surgical excision of the lesion was planned after considering all the above investigations. Under general anesthesia, the lesion was completely enucleated from right maxillary antrum through an intraoral crevicular incision. Mucopurulent discharge was observed during the surgical evacuation. An intraoperative nasal endoscopy was performed to confirm the clearance of obstruction for sphenoidal recess. After the complete excision of the lesion (Figure 5), nasal antrostomy was performed. Post-operative recovery was uneventful, and the nasal pack was removed after 48 h. Pus was sent for culture and sensitivity, and the patient was given amoxycillin and clavulanic acid 1.2 g twice daily, along with metronidazole 500 mg thrice daily intravenously for 5 days.

Figure 5 Intraoperative picture showing eroded lateral wall through which mucocele was enucleated.

Histopathological report of the lesion showed pseudostratified ciliated columnar epithelium with underlying fibro-cellular connective tissue and mucous-filled areas, dense chronic inflammatory infiltrate with predominantly lymphocytes, plasma cells, and numerous dilated blood vessels are also evident. The diagnosis was rendered as mucocele of maxillary antrum correlating clinically and radiographically (Figures 6,7). Healing was satisfactory at one month. At the end of 3 years, the patient was satisfied with the treatment rendered and had no signs of recurrence clinically and radiographically. Timeline of treatment is described in Figure 8.

Figure 6 Gross specimen after fixation and small bits of sinus lining enucleated along with the lesion.

Figure 7 JenyH&E-stained histopathology showing (×20) pseudostratified ciliated columnar epithelium with mucous-filled areas and proliferating blood capillaries along with flat plump endothelial cells and dense chronic inflammatory cells. H&E, hematoxylin and eosin.

Figure 8 Timeline and duration of treatment. (A) Description of events through the timeline. (B) Illustration of events through the timeline: [1] history, [2] clinical and radiographic evaluation, with Figures 1,2, [3] computed tomography evaluation, with Figures 3,4, [4] nasal endoscopy, [5] complete enucleation, with Figure 5, [6] gross specimen and histopathological section with Figures 6,7. (C) Evaluation and treatment timeline in months.

The surgical procedures performed in the current report are in accordance with the Helsinki Declaration (as revised in 2013). Institutional ethics committee approval was obtained (IECVDC/22/PG01/OMFS/IVV/119) from the Institutional review board, Vishnu Dental College. Written informed consent was obtained from the patient for the publication of this case report and accompanying images. A copy of the written consent is available for review by the editorial office of this journal.

Discussion

The most common sites for occurrence of mucocele are the frontal (89%), ethmoid (8%) sinuses than sphenoidal and maxillary (2%) (2). Prevalence of maxillary SMs is approximately 1 in 100,000 people. The clinical presentation of maxillary SMs can vary from asymptomatic to highly symptomatic depending on the extent of involvement. The most common presenting symptoms are headache, unilateral epistaxis, swallowing pain, and cheek swelling. Maxillary antral mucoceles generally involve the lateral wall of the sinus first, the hard palate, the orbital floor, and less frequently the medial (nasal) wall (3,8). Expansion occurs mainly through the direct effect of positive pressure within the mucocele and the production of bone resorption factors like prostaglandins, interleukin, and tumor necrosis factor (TNF) which may lead to infraorbital or intracranial extension (9).

The pathogenesis of maxillary SMs is unknown; however, trauma to the paranasal sinuses or poor drainage from infection or inflammation has been proposed as causes. Sinusitis-associated mucoceles are frequently accompanied by sinus pain and nasal congestion, and they are known to recur at a higher rate (50%) than non-sinusitis (10.2%) associated mucoceles (5). Therefore, it is necessary to determine the etiology behind mucocele formation and plan the treatment accordingly. This can be done by reviewing the patient’s history, symptoms and radiographs.

Involvement of fronto ethmoidal sinuses is common, however our case had involvement of maxillary sinus with an obstruction and secondary infection of sphenoidal sinus. Sphenoidal sinus infections are rare but frequently associated with chronic rhinosinusitis with nasal polyps. Earache, neck pain, pain behind the eyes or in the temples is the most common symptoms. If not diagnosed and treated promptly, can result in potentially fatal complications like cranial nerve involvement, acute pansinusitis with bacteremia, brain abscess, and meningitis (10).

The main stay for the management of SMs remains surgery. The goal of treatment is to remove the mucocele tissue, restore normal anatomy, function and establish drainage pathways for future control of infection (8). The most common surgical treatment for maxillary SM is endoscopic evacuation with wide antrostomy of the middle meatus (EEWAM), or the traditional Caldwell-Luc approach, which involves total removal of the mucocele followed by nasal antrostomy (11-13). When the mucoceles are small and asymptomatic, non-surgical management by means of aspiration, steroid injections and use of sclerosing agents may be appropriate (13). However, when a patient suffers from recurrent infections or when the mucocele enlarges and obstructs sinus drainage pathways, surgery should be considered (14).

Other surgical modalities include enucleation, marsupialization followed by enucleation, endoscopic guided marsupialization and enucleation (15). The role of marsupialization and open surgery in the management of maxillary SM is debatable. This may be attributed to the recurrence reported with the presence of an underlying obstructive sinonasal disease (50%) or poor compliance with follow-up examinations (14). With the wide use of endoscopic-guided surgeries, the recurrence rate has come down to 0.9% to 2.2% (14-16). It is important to carefully evaluate patients before surgery and exclude those who are unsuitable for marsupialization. The following guidelines are recommended:

• Maxillary SM should be treated only if there is a risk of airway compromise, infection, or ocular symptoms due to extension of the lesion.
• Patients with underlying obstructive sinonasal disease or poor compliance are unsuitable for marsupialization.
• When surgery is performed, complete removal of any inflammatory tissue is crucial.

In our case, we opted for open surgery due to the presence of acute infection as well as secondary sphenoidal empyema which if not treated at the earliest could have resulted in fatal complications.

The strength of this case report lies in its rare presentation of an abnormally huge maxillary SM eroding the walls of the antrum breaching the adjacent anatomic boundaries thereby leading to the sphenoid sinus empyema, which was managed through a traditional open surgical approach along with a close long-term follow-up for 3 years. Lack of systematic reviews suggests that case reports on mucocele of the maxillary sinus with erosive cavity formation are scarce. As a result, this case report adds to the rare pathology and diagnosis. The limitation of this report lies in, not providing a comprehensive review of the relevant literature, despite citing the publications that provided the most support.

Conclusions

Maxillary bone cysts present a diagnostic dilemma for clinicians. Cystic lesions involving the paranasal sinuses should be treated appropriately aided with antrostomy, unlike lesions of odontogenic origin. Management of the lesion in present case may have required extensive surgical procedures like resection because of the substantial bone destruction and clinical manifestations that resemble an odontogenic tumor. On the other hand, failure to perform an antrostomy could result in fatal complications due to the spread and involvement of other adjacent vital structures, which could cause morbidity. An accurate and timely understanding of this rare involvement can help clinicians tailor an appropriate treatment plan with minimal complications.

Acknowledgments

We thank the Department of Oral and Maxillofacial Surgery/Department of Oral and Maxillofacial Pathology, Vishnu Dental College, Bhimavaram for the support.

Funding: None.

Footnote

Reporting Checklist: The authors have completed the CARE reporting checklist. Available at https://amj.amegroups.com/article/view/10.21037/amj-23-61/rc

Peer Review File: Available at https://amj.amegroups.com/article/view/10.21037/amj-23-61/prf

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://amj.amegroups.com/article/view/10.21037/amj-23-61/coif). The authors have no conflicts of interest to declare.

Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. All procedures performed in this study were in accordance with the ethical standards of the institutional ethical committee (IECVDC/22/PG01/OMFS/IVV/119) and with the Helsinki Declaration (as revised in 2013). Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the editorial office of this journal.

Open Access Statement: This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the non-commercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). See: https://creativecommons.org/licenses/by-nc-nd/4.0/.

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