Cholesteatoma of the concha bullosa: a case report
© Cukurova et al; licensee BioMed Central Ltd. 2010
Received: 10 January 2010
Accepted: 17 December 2010
Published: 17 December 2010
Cholesteatoma is a relatively common disease within the middle ear cavity, but rarely it manifests in the paranasal sinuses. There is, to the best of our knowledge, only one other published case of cholesteatoma inside the concha bullosa in the English language literature.
An 81-year-old Caucasian woman was admitted to our hospital complaining of nasal obstruction, headache and diplopia. After endoscopic and radiological evaluation a transnasal endoscopic approach was chosen. The diagnosis of cholesteatoma was established by histopathological evaluation of the mass inside the concha bullosa.
Although it is rarely seen, cholesteatoma should be considered in the differential diagnosis of slow-growing and destructive paranasal masses.
Concha bullosa (CB) is the pneumatization of the middle turbinate and is one of the most common variations of the sinonasal anatomy. A 14% to 53.6% frequency of CB has been reported by various studies . Many pathological entities were described in the concha bullosa such as polyps, pyocele, and mycosis [2–4]. Cholesteatoma is a relatively common disease within the middle ear cavity and temporal bone, whereas cholesteatoma of the nasal and paranasal region is an exceptionally rare entity. Cholesteatoma can be seen in the frontal, ethmoid and maxillary sinuses [5–7]. However, to the best of our knowledge, there is only one other published case of cholesteatoma inside the CB in the English language literature .
An 81-year-old Caucasian woman presented to the outpatient clinic of our hospital with nasal obstruction, headache and diplopia. She had experienced nasal obstruction for over ten years and her headache had worsened for three months. She did not have epistaxis or epiphora. She had been treated medically with antibiotics, nasal decongestants, and nasal steroids. However, her complaints were not alleviated. Her otorhinolaryngological history was unremarkable.
An endoscopic examination revealed a massively large middle turbinate on the left side. The mucosa was normal and no infection signs were detected in the nasal passage. The enlarged middle turbinate contacting the lateral nasal wall and the septum was the only endoscopic finding. Our patient has proptosis, but her ophthalmic examination revealed 20/20 visual acuity in both eyes and normal intraocular pressures.
Four theories on the pathogenesis of paranasal sinus cholesteatomas have been put forward . The most widely accepted theory is the congenital (primary) cholesteatoma theory. Congenital (primary) cholesteatoma is believed to be a result of misplaced ectodermal, epithelial cell remnants during the formation of the face in the third to fifth week of embryogenesis . The development of acquired (secondary) cholesteatomas can be explained by three theories. One theory is the implantation of epidermoid cells after surgery or trauma. Immigration and metaplasia theories are the other proposals. As in most other cases, in our patient the congenital cholesteatoma theory is the most likely explanation. The implantation theory cannot be accepted because our patient has no history of previous trauma or surgery to the head and neck region. Immigration of epithelium from the nasal vestibule to the intranasal region has never been reported and we also could not show such an intranasal tract in our case . The metaplasia theory has been rejected by many authors because squamous epithelium deriving from metaplasia due to chronic rhinosinusitis is of a non-keratinizing type .
Due to the rarity of this lesion, cholesteatoma in the paranasal sinuses is seldom pre-operatively diagnosed. Cholesteatoma has a slow-growing pattern and it has an erosive effect on bone. In our patient, we initially considered the possibility of a neoplastic lesion or pyocele during the differential diagnosis. However, as shown in this case, cholesteatoma can cause similar findings.
Although it is rarely seen, cholesteatoma should be considered in the differential diagnosis of slow-growing and destructive paranasal masses. Total excision is the treatment of choice for paranasal cholesteatomas. Our patient was treated successfully with a transnasal endoscopic approach.
Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
- Zinreich S, Albayram S, Benson M, Oliverio P: The osteometal complex and functional endoscopic surgery. Head and Neck Imaging. Edited by: Som P. 2003, St Louis, MO: Mosby, 149-173. 4Google Scholar
- Mirante JP, Christmas DA, Yanagisawa E: Polyps arising in a concha bullosa of the middle turbinate. Ear Nose Throat J. 2003, 82: 241-242.PubMedGoogle Scholar
- Okuyucu S, Akoglu E, Dagli AS: Concha bullosa pyocele. Eur Arch Otorhinolaryngol. 2008, 265: 373-375. 10.1007/s00405-007-0448-0.View ArticlePubMedGoogle Scholar
- Massegur H, Adema JM, Lluansi J: The middle turbinate as the cause of sinusal pathology. Acta Otorhinolaryngol Esp. 1995, 46: 27-29.Google Scholar
- Chandra RK, Palmer JN: Epidermoids of the paranasal sinuses and beyond: endoscopic management. Am J Rhinol. 2006, 20: 441-444. 10.2500/ajr.2006.20.2895.View ArticlePubMedGoogle Scholar
- Hansen S, Sørensen CH, Stage J, Mouritzen A, Cayé-Thomasen P: Massive cholesteatoma of the frontal sinus: case report and review of the literature. Auris Nasus Larynx. 2007, 34: 387-392. 10.1016/j.anl.2006.11.011.View ArticlePubMedGoogle Scholar
- Palacios E, Robertson H: Cholesteatoma of the maxillary sinus. Ear Nose Throat J. 2005, 84: 758-PubMedGoogle Scholar
- Cukurova I, Demirhan E, Karaman Y, Yigitbasi OG: Extraordinary pathologic entities within the concha bullosa. Saudi Med J. 2009, 30: 937-941.PubMedGoogle Scholar
- Michaels L: Origin of congenital cholesteatoma from a normally occurring epidermoid rest in the developing middle ear. Int J Pediatr Otorhinolaryngol. 1988, 15: 51-65. 10.1016/0165-5876(88)90050-X.View ArticlePubMedGoogle Scholar
- Storper IS, Newman AN: Cholesteatoma of the maxillary sinus. Arch Otolaryngol Head Neck Surg. 1992, 118: 975-977.View ArticlePubMedGoogle Scholar
This article is published under license to BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (<url>http://creativecommons.org/licenses/by/2.0</url>), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.