Respiratory Epithelial Adenomatoid Hamartoma Originating from Nasal Septum

Clinical and Experimental Otorhinolaryngology Vol. 6, No. 1: 45-47, March 2013 http://dx.doi.org/10.3342/ceo.2013.6.1.45 pISSN 1976-8710 eISSN 2005-0720 Case Report Respiratory Epithelial Adenomatoid Hamartoma Originating from Nasal Septum Il-Ho Park·Hak Chun Lee·Heung-Man Lee Department of Otorhinolaryngology-Head and Neck Surgery, Korea University College of Medicine, Seoul, Korea Respiratory epithelial adenomatoid hamartoma (REAH) is a rare benign lesion in nasal cavity. We report two cases of REAH of the nasal cavity arising from nasal septum. The etiology of REAH is unknown although inflammation may induce gland proliferation observed in hamartomas. One of our cases was associated with nasal polyposis. REAH is a self-limiting disease, so it is important to differentiate REAH from other pathologic process, including inverted papilloma and low-grade adenocarcinoma. The treatment of choice is complete excision through a conservative approach. Keywords: Hamartoma, Nasal septum, Differential diagnosis, Nasal cavity, Respiratory tract INTRODUCTION CASE REPORTS Case 1 Hamartomas are non-neoplastic malformations or inborn errors of tissue development first described in 1904. Hamartomas can occur in any area of the body, but commonly originate from the spleen, intestine, and lung [1]. However, hamartomas in the upper aerodigestive tract are very rare [2]. In 1995, Birt and KnightJones [3] reported the fisrt case of a harmatoma in the upper aerodigestive tract. In 1995, Wenig and Heffner [4] documented the clinicopathologic features of 31 cases of hamartoma and introduced the term respiratory epithelial adenomatoid hamartoma (REAH) as one particular type of hamartoma. Approximately 70% of REAHs occur in the nasal cavity, most often localized to the posterior part of the nasal septum. These lesions also have been reported to occur in paranasal sinuses and nasopharynx [5,6]. We experienced two cases of REAH originating from the nasal septum, which were treated by endoscopic surgery, and present their clinical findings and histopathologic features. A 50-year-old woman presented with left nasal obstruction for 3 years and had no other associated sinonasal symptoms. The patient denied any past history including chronic rhinosinusitis, alleric rhinitis, and sinonasal operation. Examination with a nasal endoscope revealed a smooth, mucosa-covered, and polypoid mass on the posterior aspect of nasal septum in the left nasal cavity; the mass originated from the posterior part of the septum (Fig. 1A). The mass extended to the right side through the nasopharynx (Fig. 1B). Computed tomography (CT) coronal view scans of paranasal sinuses revealed a left nasopharyngeal mass (Fig. 2A). Axial view CT revealed that the mass was attached to the posterior aspect of the nasal septum (Fig. 2B). A pre-operative punch biopsy of the mass suggested the diagnosis of REAH. Under general anesthesia, intranasal endoscopic excision was performed. The excised specimen measured 23 mm× 16 mm×13 mm. On histological analysis, the surface of the lesion was found to comprise pseudostratified, ciliated, columnar epithelium and was composed of proliferated glands lined by ciliated respiratory epithelium. No atypical cells were seen and there was no evidence of metaplasia (Fig. 3A). The pathologic finding was consistent with REAH. The patient tolerated the operation well and there were no complications during the postoperative period. At 1-year follow-up, there was no evidence of recurrence. ••Received December 31, 2009 Revision January 26, 2010 Accepted February 25, 2010 ••Corresponding author: Heung-Man Lee Department of Otorhinolaryngology-Head and Neck Surgery, Korea University College of Medicine, 148 Gurodong-ro, Guro-gu, Seoul 152-703, Korea Tel : +82-2-2626-3185, Fax : +82-2-868-0475 E-mail: Copyright © 2013 by Korean Society of Otorhinolaryngology-Head and Neck Surgery. This is an open-access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited. 45 46 Clinical and Experimental Otorhinolaryngology Vol. 6, No. 1: 45-47, March 2013 A B 200 µm A C D 50 µm C Fig. 1. Nasal endoscopy details of case 1 (A, B) and case 2 (C, D). (A, B) Nasal endoscopic finding revealed a smooth mucosa covered, polypoid mass on the posterior aspect of nasal septum in the left nasal cavity. It was originated from posterior part of septum. The mass extended to right side through the nasopharynx. (C, D) Nasal endoscopy shows bilateral nasal polyposis in middle meatus. 200 µm B Fig. 3. Microscopy details of case 1 (A) and case 2 (B, C). (A) Proliferated gland was lined by ciliated respiratory epithelium. No atypical cell was seen and there was no evidence of metaplasia (H&E, ×100). (B) Photomicroscopic finding shows elongated invaginations into the lamina propria. Glandular proliferation originating from the invaginating surface epithelium was seen (H&E, ×100). (C) The lining of gland was ciliated respiratory epithelium (H&E, ×400). Case 2 A B C D Fig. 2. Computed tomography (CT) details of case 1 (A, B) and case 2 (C, D). (A) CT scan of paranasal sinuses coronal view shows left nasophryngeal mass (white arrow). (B) Axial view reveals the mass is attached to posterior aspect of nasal septum (white arrow head). (C) Coronal view and (D) axial view of CT scan of paranasal sinus showed soft tissue density in maxillary and ethmoid sinus and both nasal cavity. A 23-year-old man with a history of obstruction of both nasal passages visited our office. Symptoms first developed about 10 years previously. The patient had undergone a nasal polypectomy 7 years before the current presentation. Nasal endoscopy revealed bilateral nasal polyposis in the middle meatus (Fig. 1C, D). A CT scan of the paranasal sinus showed soft tissue density in all sinus cavity and both middle meatus (Fig. 2C, D). A preoperative punch biopsy of the mass suggested the diagnosis of REAH. Under general anesthesia, functional endoscopic sinus surgery was performed. Left nasal cavity mass originated from posterior aspect of nasal septum and mass was removed without remnant. Pathology of left nasal cavity mass revealed REAH associated with nasal polyposis and right nasal cavity polyp revealed chronic inflammation. There was a glandular proliferation originating from the surface epithelium (Fig. 3B). The gland was lined by ciliated respiratory epithelium (Fig. 3C). There were no postoperative complications. At 1-year follow-up, there was no evidence of recurrence. DISCUSSION REAHs are very rare in the sinonasal tract and were first recognized as a distinctive entity by Wenig and Heffner in 1995 [4]. To date, a very limited number of cases have been reported. In Park I-H et al.: REAH in Nose the report by the afore (...truncated)