Sinonasal respiratory epithelial adenomatoid hamartoma: a rare clinical entity masked as nasal polyposis
DOI:
https://doi.org/10.18203/issn.2454-5929.ijohns20262396Keywords:
Case report, Hamartoma, REAH, Polyposis, Nasal massAbstract
A hamartoma is a benign malformation or congenital error of tissue development indigenous to a specific part of the body, common in the lung, kidney, liver, spleen, and intestine but rare in the head and neck region, particularly nasal cavity and paranasal sinuses. A 50-year-old female presented with complaints of decreased smell sensation for 7 years; snoring, bilateral nasal obstruction, serous nasal discharge which is bloodstained occasionally and mouth breathing for 2 years. On Diagnostic nasal endoscopy, on right side, a single, pedunculated, globular mass originating from roof extending up to the floor of nasal cavity, seen between septum and inferior turbinate. Bilateral full house functional endoscopic sinus surgery with posterior septectomy was done and excised mass was sent for biopsy. Histopathological examination shows fragments of polypoidal tissue lined predominantly by pseudostratified ciliated columnar epithelium with intervening goblet-like cells at some areas, underlying stroma is loose, edematous. Irregularly branched, dilated glands lined by respiratory epithelium with thickened basement membrane, abundant intraluminal mucinous secretory material along with foamy macrophages and intervening stroma appears hyalinised with mixed inflammatory infiltrate with no evidence of malignancy and diagnosis was confirmed as REAH with inflammatory nasal polyposis. REAH is very rare and self-limiting lesions which do not regress spontaneously with majority of cases presenting as polypoidal mass causing nasal obstruction, nasal stuffiness, and hyposmia, resembling chronic rhinosinusitis.
References
Wenig BM, Heffner CDK. Respiratory Epithelial Adenomatoid Hamartomas of the Sinonasal Tract and Nasopharynx: A Clinicopathologic Study of 31 Cases. Ann Otol Rhinol Laryngol. 1995;104(8):639-45.
Lin YW, Lai KJ, Shen KH. A case report of adolescent respiratory epithelial adenomatoid hamartoma. Ear Nose Throat J. 2022;1-5.
Agrawal SR, Joshi AA, Dhorje N, Bradoo R. Respiratory epithelial adenomatoid hamartoma: a diagnostic challenge in sinonasal lesions. BMJ Case Rep. 2019;12(8):e230082.
Lee JT, Garg R, Brunworth J, Keschner DB, Thompson LDR. Sinonasal respiratory epithelial adenomatoid hamartomas: Series of 51 cases and literature review. Am J Rhinol Allergy. 2013;27:322-8.
Kumar D, Handa KK, Handa A, Gautam P. Recurrent respiratory epithelial adenomatoid hamartoma of the nasal cavity. Proc (Bayl Univ Med Cent). 2022;35(5):668-9.