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An Unusual Case of Headache: Isolated Fungus Ball in Concha Bullosa

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Copyright © 2018 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.

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An Unusual Case of Headache:

Isolated Fungus Ball in Concha Bullosa

Fuat Bulut, MDand Kadir Cagdas Kazikdas, MDy

Abstract: A concha bullosa forms when the middle turbinate becomes pneumatized, which is a common anatomic variation;

however, fungus ball in concha bullosa is rather rare. An otherwise healthy 52-year-old man presented to our ear, nose and throat clinic with the complaints of midfacial pressure headache and malodorous postnasal drip. Computed tomography of the para- nasal sinuses demonstrated a polypoid hyperdense lesion with slight microcalcifications in the right nasal cavity, accompanied with a complicated fluid collection in the right frontoethmoideal recess seen as hypodensity in contrast to this hyperdensity. The histopathological examination reported a fungal infection. We present an extremely rare case of isolated fungal mass in the right middle concha detected in an early stage without any evidence of fungal infection of the other paranasal sinuses and discuss the importance of reevaluation of the computed tomography scans in suspicion of a fungal sinusitis.

Key Words:Computed tomography, concha bullosa, fungus ball, sinusitis

F

ungal diseases of the paranasal sinuses are categorized as either invasive or noninvasive based on the presence or absence of tissue invasion. Noninvasive fungus balls can be found most frequently in the maxillary sinus and occasionally in the sphenoid sinus.1Stavrakas et al2have pointed out that even the suspicion of fungal sinusitis requires early specialist referral and aggressive treatment. General practitioners and ear, nose and throat (ENT) surgeons should be familiar with the signs, symptoms, and early radiological findings.3 Herein we present the 3rd case of fungal sinusitis involving the concha bullosa solely without any evidence of fungal infection of the other paranasal sinuses in the English literature1and the differential diagnosis was rather difficult to make preoperatively due to expansile nature of the lesion and unconven- tional radiological signs.

From the Department of Otorhinolaryngology, Rumeli University REYAP Hospital, Istanbul, Turkey; and yDepartment of Otorhinolar- yngology, Near East University, Faculty of Medicine, Nicosia, Cyprus.

Received October 25, 2017.

Accepted for publication February 9, 2018.

Address correspondence and reprint requests to Kadir Cagdas Kazikdas, MD, Department of Otorhinolaryngology, Near East University Faculty of Medicine, Near East Blv, 99138 Nicosia, Cyprus;

E-mail: ckazikdas@gmail.com The authors report no conflicts of interest.

Copyright#2018 by Mutaz B. Habal, MD ISSN: 1049-2275

DOI: 10.1097/SCS.0000000000004529

The Journal of Craniofacial Surgery  Volume 29, Number 6, September 2018 Brief Clinical Studies

#2018 Mutaz B. Habal, MD

e551

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Copyright © 2018 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.

CLINICAL REPORT

An otherwise healthy 52-year-old man presented to our ENT clinic with the complaints of midfacial pressure headache and malodorous postnasal drip. The patient had a history of recurrent sinusitis and seasonal allergy symptoms. He had been treated medically numerous times by his family physician. Computed tomography (CT) of the paranasal sinuses demonstrated a polypoid hyperdense lesion with slight microcalcifications in the right nasal cavity. The lesion was expansile with no bone lysis or demineralization. Also there was complicated fluid collection in the right frontoethmoideal recess seen as a lower density in contrast to this hyperdensity (Fig. 1A).

Nasal endoscopic examination revealed a rather sticky purulent nasal discharge in right nasal cavity, left-sided septal deviation, and a hypertrophic right middle concha. The patient then underwent sep- toplasty followed by endoscopic conchaplasty surgery under general anesthesia. After removing the lateral portion, a dark gray, cheese- like material (Fig. 1B) was aspirated from within the right concha bullosa and the histopathologic analysis revealed that this mass was consistent with the diagnosis of an aspergilloma or mucormycosis.

Fungal hyphae masses stained with periodic acid schiff were positive for fungi. The material sent for fungal culture was reported as negative. The postoperative course was uneventful and the patient experienced no further symptoms (Fig. 1C).

DISCUSSION

There is an increasing trend of fungal infections of the paranasal sinuses in recent years and this has been attributed to improved diagnostic approaches to investigating the paranasal sinuses, and also to the increase in conditions that favour fungal infections, such as diabetes, abuse of broad-spectrum antibiotics, and diseases- causing immunodeficiency.1Increased dental procedures such as endodontic treatments have been postulated as a theory to explain how the sinus promotes the growth of fungal elements.4However, this theory does not explain the occurrence of a fungus ball solely in one-sided concha bullosa and noninvasive fungus balls can even typically affect otherwise healthy individuals with no history of endodontic treatment as in this presented case.

Like all other aerated cells, concha bullosa possesses a muco- ciliary transport system, with the ostium connecting the airy cell lumen to the frontal recess.5The pathogenesis of fungal infections of the paranasal region is still unclear. Development of the fungus ball inside the paranasal sinuses is a very slow process. Hypoven- tilation could be important in trapping fungal spores and hyphae, thus providing further oedema and anaerobic conditions for fungus ball development inside concha bullosa. This entrapment will impair the normal sinus drainage and clearance. Alternatively, allergic mucosa might be the triggering factor (the chicken or the egg dilemma). The nutrition provided by the pus from fron- toetmoidal region and the narrowness of the passage of frontoet- moidal recess seemed to provide anaerobic conditions, aiding in the

development of the fungus ball in this early detected case with sparing of the rest of the other sinuses.6Based upon these findings, the frontoethmoid recess might be considered as a potential site of developing fungus balls, gradually spreading to the neighbouring sinuses as it would be observed in advanced fungal infections.

The density differences in between the lesion in nasal cavity and the fluid in frontoethmoidal recess could easily be noted on coronal paranasal CT images. An opacified sinus or concha bullosa includ- ing hyperdense contents is usually a sign of benign disease. The

‘‘hyperdensity’’ representing slight calcification on unenhanced CT is because of one or a combination of the following: entrapped proteinaceous secretions, blood products, or fungus as in this present case. A sinus fungus ball may not show specific symptoms;

thus, it will present a clinical diagnostic challenge before surgery. It is thought that sinus fungus balls mostly show areas of mineraliz- ation hyperattenuation and macrocalcification because of calcium and magnesium salts deposits. However high-density secretion features rather than classical calcifications, observed in a small percentage of sinuses using CT scans are also quite important for diagnosing a sinus fungus ball preoperatively.7Our case illustrates this finding clearly with the reevaluation the CT scans showing areas of hyperdensity representing a fungal infection and a lower density designating a frontoethmoidal blockage.

Isolated fungal mass in concha bullosa is a rather rare condition with only a few cases published in the literature.1,8A differential diagnosis includes bacterial sinusitis, mucocele, pyocele, inverted papilloma, or squamous cell carcinoma. Fungus balls should be detected in an early stage and treated surgically.8Untreated fungal ball in the concha bullosa can cause bacterial superinfection, bacterial sinusitis, meningitis, and even brain empyema.

REFERENCES

1. Ciger E, Demiray U, Onal K, et al. An unusual location for a fungus ball:

the concha bullosa. J Laryngol Otol 2012;126:844–846

2. Stravrakas M, Karkos PD, Dova S, et al. Unilateral fungal sphenoiditis presenting with diplopia and ptosis. Indian J Otolaryngol Head Neck Surg 2017;69:428–429

3. Ozkiris M, Kapusuz Z, Sec¸kin S, et al. Fungus ball in concha bullosa: a rare case with anosmia. Case Rep Otolaryngol 2013;2013:920406 4. Kim JS, So SS, Kwon SH. The increasing incidence of paranasal sinus

fungus ball: a retrospective cohort study in two hundred forty-five patients for fifteen years. Clin Otolaryngol 2017;42:175–179 5. Kazikdas KC. Polyps in bilateral conchae bullosa. Ear Nose Throat J

2011;90:12

6. Lee JH, Jeong HM. A rare occurrence of a fungus ball in the sphenoethmoid recess. Ear Nose Throat J 2014;93:E47–E48

7. Ng TY, Wang JY, Tsai MH, et al. Hyperdense findings in sinus computed tomography of chronic rhinosinusitis. Int Forum Allergy Rhinol 2015;5:1181–1184

8. Toplu Y, Toplu SA, Can S, et al. Fungus ball in concha bullosa: an unusual cause of retro-orbital pain. J Craniofac Surg 2014;25:e138–e140

Comparison of Incidence and Severity of Chronic Postsurgical Pain Following Ear Surgery

Mehmet Gu¨ven, MD,yAhmet Kara, MD,y Mahmut Sinan Yilmaz, MD,yDeniz Demir, MD,y and Ebru Mihriban Gu¨ven, MDy

Objectives: Literature review shows a limited number of studies investigating chronic pain following ear surgeries. The effect of

FIGURE 1. (A) Coronal computed tomography (CT) scan showing hyperdensity with slight microcalcifications (star) in right concha bullosa and the presence of a complicated fluid collection in the right frontoethmoideal recess characterized by a lower density (white arrow). (B) The concha bullosa cavity filled with a dark gray, cheese-like material. (C) Postoperative CT scan in coronal section.

Brief Clinical Studies The Journal of Craniofacial Surgery  Volume 29, Number 6, September 2018

e552

#2018 Mutaz B. Habal, MD

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