Middle Turbinate Pyocele, an Uncommon Differential for Nasal Mass: A Case report

Abstract

Mucoceles are respiratory epithelium-lined cystic lesions found in the paranasal sinuses. Pyocele occurs when a mucocele becomes infected. Mucoceles develop as a result of obstruction of the normal sinus drainage tract with superadded infection. Inflammatory mediators are secreted, which lead to bone resorption and expansion of the mucocele. Concha bullosa is one of the most common anatomical variations in the nasal cavity. It refers to an air-filled cavity within the middle turbinate. Obstruction of the concha bullosa can rarely lead to the formation of a mucocele which may be secondarily infected forming a mucopyocele. The mucocele of the middle turbinate may present as an uncommon cause of nasal obstruction, headache, and other nasal or ocular symptoms, which, if correctly diagnosed, can be completely reversed by endoscopic surgery.

Introduction

Mucocele is usually defined as a cystic lesion lined by epithelial. All sinus mucoceles are true mucus retention cysts with an epithelial lining principally caused by obstruction. Different etiologic factors may lead to such obstruction, including nasal polyps, surgery, trauma, and benign tumors. Mucoceles are a serious condition due to their capacity to expand and erode overlying bone [1]. Here we present a case of mucopyocele of the middle turbinate presenting as an uncommon cause of nasal obstruction which was removed completely under endoscopic guidance. In the formation of a true mucocele, there is local bone destruction, remodeling, and expansion. There is a postulate that stimulation of fibroblasts by local lymphocytes may result in the release of bone-resorbing factors which subsequently lead to thinning and expansion of local bone to facilitate enlargement of the mucocele.

Case Report

A 9-years-old female child, presented with complaints of (R)sided nasal obstruction for the last 1–2 months which was insidious in onset, persistent, and more during the episodes of URTI associated with the history of mouth breathing and snoring. There was no history of recurrent sneezing/bleeding/discharge from the nose. No other ENT complaints.

On examination, there was no obvious external deformity. Anterior rhinoscopy revealed a deviated nasal septum to (L) with a pinkish mass completely filling the (R) nasal cavity (Fig. 1a). The nasal mucosa was normal, and no discharge was seen. The rest of the ENT examination was within normal limits.

Fig. 1.

a Swelling over the right side of the nose filling the (R) nasal cavity [arrow]

Diagnostic Nasal Endoscopy revealed deviated nasal septum to (L) with a pinkish mass completely filling the (R) nasal cavity. Mass was firm in consistency, not bleeding on touch, and was able to pass the probe all around the mass except on the lateral wall.

CECT nose and PNS revealed a well-defined, non-enhancing, soft tissue density lesion measuring 28 × 10 × 14 mm filling the right nasal cavity (Fig. 2a, b), causing deviation of the nasal septum to (L) likely expanded right middle turbinate (Fig. 2c). There was a non-enhancing soft tissue thickening involving expansion of the right maxillary sinus and ostium with thinning of the medial and posterior wall of the right maxillary sinus. No bony erosions noted.

Fig. 2.

a CECT Nose and PNS—a well-defined non-enhancing soft tissue density measuring 28 × 10 × 14 mm filling (R) with expanded middle turbinate (R) [Marked arrow]. b CECT Nose and PNS—a well-defined non-enhancing soft tissue density (R) nasal cavity with expanded middle turbinate (R) [Marked arrow]. c CECT Nose and PNS—a well-defined non-enhancing soft tissue density (R) nasal cavity with expanded middle turbinate (R)and deviation of nasal septum to (L) [Marked arrow]

The child was admitted as a case of (R) nasal mass (Inv). Intraoperatively fleshy mass arising from (R) middle turbinate filling (R) nasal cavity, pushing septum to (L) was seen. (R) nasal mass covered with bony covering suggestive of concha bullosa filled with pus, mucoid and cheesy material was identified and suctioned out (Fig. 3). Endoscopically (R) nasal mass with its covering was excised in toto using microdebrider along with (R) middle meatus antrostomy and widening of (R) maxillary sinus ostium.

Fig. 3.

The cavity of middle meatus antrostomy (R) after drainage of pus [arrow]

A biopsy of the mass revealed an inflammatory polyp lined by respiratory epithelium (Fig. 4).

Fig. 4.

Inflammatory polyp lined by respiratory epithelium

No organisms were isolated from the aspirates on microbiologic examination.

The postoperative hospital stay was uneventful and the child was discharged on the 3rd post-op day. The child recovered well after the surgery and was continued on intranasal steroid spray with symptomatic improvement.

Discussion

Mucoceles are cystic lesions found in paranasal sinuses which are lined by respiratory epithelium [2]. When infected they are called mucopyocele. Pneumatization of the middle turbinate is variable and categorized into three types according to its location—Lamellar, Bullous, and Concha bullosa. It is termed as lamellar when it involves vertical lamella of the concha, whereas inferior bulbous segment involvement is termed as ‘‘bullous’’. If the entire concha (both the lamellar and bullous portions) is pneumatised it is called a ‘‘true concha bullosa.’’ The incidence of concha bullosa varies from 14 to 53% [3]. Concha bullosa was first described by Zuckerkandl in 1893 [4]. Drainage commonly occurs through the conchal ostium, which is located near the frontal recess. Rarely, the concha bullosa drains along the basal lamella to open directly into the adjacent air cells [5].

Mucopyocele of the middle turbinate was first described, in 1994, by Badia et al. in adults and congenital mucocele of the middle turbinate had been reported by Toledano et al. [2]. The frontoethmoidal complex is the most common site of mucocele formation owing to the anatomy of its outflow tract. Less frequently, mucoceles of the maxillary sinus, sphenoethmoidal area, concha bullosa, clinoid process, pterygoid process, and in areas of abnormally displaced mucosa [4] and isolated sphenoid sinus are also mentioned in literature. Underlying pathophysiology explains these rare locations of mucoceles.

Concha bullosa mucocele(CBM) is exceptionally rare. There are very few reported cases of concha bullosa pyocele in the literature [5]. Several hypotheses have been proposed regarding the pathogenesis and etiology of this condition, such as Pressure-induced osteolysis, mediated by potent osteolytic mediators like the eicosanoids (products of the action of cyclo-oxygenase and lipoxygenase on arachidonic acid) and a range of inflammatory cytokines including interleukin-1 (IL-l), IL-6 and tumor necrosis factor (TNF) [1]. However, none of the proposed hypotheses can clearly explain the pathogenesis of the middle turbinate concha bullosa mucocele. Chronic blockage of the ostium of concha bullosa leads to impaired ventilation of the pneumatized cavity and its originating compartment, usually the anterior ethmoid and frontal recess. This obstructive mechanism also explains the intact epithelial contour of the sinonasal mucocele. Studies indicated that CBM is due to, either a mechanical obstruction(trauma, surgery, nasal polyposis, or benign tumors) or an inflammatory etiology(infection, allergy, cystic fibrosis), whereas few studies still debate. So, the cause-effect mechanism is still not clearly understood [6].

A mucocele or mucopyocele can cause local bone erosion, diplopia, and nasal obstruction [5]. Concha bullosa mucopyoceles should be noted in patients with a large hyperemic nasal mass [6].

Computed tomography plays an important role in delineating concha bullosa mucoceles as well as demonstrating surrounding bony destruction. It demonstrates the expanded middle turbinate, thinning of the compact bone at its margins, and eventual deviation of the nasal septum to the contralateral side. The finding of a bony margin on CT evaluation allows for differentiating a conchal mucocele from other nasal masses [5]. Further, When the normal sinus tract is obstructed associated with superadded infection, inflammatory mediators are secreted. This leads to bone resorption and bone expansion, ultimately resulting in thinning of bone in the direction of the natural flow of secretions of sinuses, which was observed in this case [2].

The Standard of care for mucopyocele in concha bullosa is resection and drainage by Endoscopic sinus surgery(ESS). The mucocele recurrence rate reported in the literature is as low as 0% after ESS [7].

Conclusion

Concha bullosa mucopyoceles are rare, especially in children [8]. Although uncommon, a concha bullosa mucocele or mucopyocele should be considered in the case of nasal obstruction with an enlarged middle turbinate. The peculiarity of the middle turbinate mucocele lies more in the diagnosis than in the management as most of these can be completely resected or marsupialized endoscopically.

Funding

No funding was received to assist with the preparation of this manuscript.

Declarations

Conflict of interest

The authors have no relevant financial or non-financial interests to disclose.

Human and Animal Rights

None.

Informed Consent

Informed consent was taken from the patient for the case report.