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Case Report

ENT Updates 2017;7(1):53–56 doi:10.2399/jmu.2017001007

Giant angiomatous choanal polyp originating from the middle turbinate: a case report

Aleksandar Perić1, Aleksandar Jovanovski2, Biserka Vukomanović Durdević3

1Department of Otorhinolaryngology, Faculty of Medicine, Military Medical Academy, Belgrade, Serbia 2Institute for Radiology, Faculty of Medicine, Military Medical Academy, Belgrade, Serbia 3Institute for Pathology, Faculty of Medicine, Military Medical Academy, Belgrade, Serbia

Abstract Özet: Orta konkadan kaynaklanan dev anjiyomatöz koanal polip: Olgu sunumu Choanal polyps (CPs) can be defined as histologically benign, solitary, Koanal polip (KP) histolojik olarak benign, nazal kavite ile nazofarenks soft tissue lesions extending towards the junction between the nasal cavi- aras› birleflme noktas›na koana yoluyla uzanan, soliter yumuflak doku ty and the nasopharynx through the . They usually originate from lezyonu olarak tan›mlan›r. Genellikle maksiller sinüsten köken al›r. Bu the . In this report, we present an unusual case of a giant olgu sunumunda orta konkan›n alt bölümünden ç›kan ve nazofarenksi angiomatous CP arising from the inferior part of the middle turbinate tamamen dolduran ola¤and›fl› bir dev anjiyomatöz KP’yi sunmaktay›z. that completely filled the nasopharynx. A 24-year-old man presented with Burnun sol taraf›nda t›kanma, burun ak›nt›s› ve hafif-orta derecede bu- five-year history of left-sided nasal obstruction, nasal discharge and mild- run kanamas› üzerine 5 y›ll›k öyküsü olan 24 yafl›ndaki erkek hasta kli- to-moderate epistaxis. The diagnosis was supported by contrast- ni¤imize baflvurdu. Tan›, paranazal sinüslerin kontrastl› bilgisayarl› to- enhanced computed tomography scan of the with mografi taramas›yla kombine anjiyografiyle desteklendi ve histopatolo- angiography and confirmed by histopathological examination. The lesion jik incelemeyle do¤ruland›. Lezyon kombine endoskopik ve transoral was removed by combined endoscopic and transoral approach. In addi- yaklafl›mla ç›kart›ld›. Sunumumuzda ayr›ca, anjiyomatöz KP’lerin pato- tion, we discuss the pathogenesis, clinical, radiological and pathological genezini, klinik, radyolojik ve patolojik özelliklerini ve ay›r›c› tan›lar›n› characteristics of angiomatous CPs, and their differential diagnosis. tart›flmaktay›z. Keywords: High altitude, allergic , bronchial hyperreactivi- Anahtar sözcükler: Epistaksis, nazal polipler, nazal cerrahi prosedür, ty. konka.

Choanal polyps (CPs) are solitary, benign soft tissue lesions nate from maxillary sinus. We present an unusual case of a which originate from the or paranasal sinuses giant angiomatous polyp arising from the inferior part of and extend into the nasopharynx through the choana. the middle turbinate and, to our knowledge, this is the first Although CPs most commonly originate from the maxillary such case reported. antrum, named antrochoanal polyps (ACPs), other sites of origin may be sphenoid, ethmoid, , cribriform Case Report plate, inferior and middle turbinate.[1] CP of middle turbinate is extremely rare and, according to the English lit- A 24-year-old male patient presented with a five-year his- erature, there are only six cases reported previously.[1–5] tory of bilateral nasal obstruction, snoring, left-sided nasal Angiomatous nasal polyps are rare subtypes of CPs, charac- discharge and recurrent mild-to-moderate nasal bleeding. terized by extensive vascular proliferation and ectasia.[6] As He had no significant past medical history. Anterior well as other CPs, angiomatous nasal polyps usually origi- rhinoscopy and endoscopic examination identified a big,

Correspondence: Aleksandar Perić, MD, PhD. Department of Otorhinolaryngology, Faculty of Medicine, Online available at: Military Medical Academy, Crnotravska 17, 11000 Belgrade, Serbia. www.entupdates.org e-mail: [email protected] doi:10.2399/jmu.2017001007 QR code: Received: November 19, 2016; Accepted: December 27, 2016

©2017 Continuous Education and Scientific Research Association (CESRA) Perić A, Jovanovski A, Vukomanović Durdević B

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Fig. 1. (a) Coronal CT scan shows soft tissue lesion arising from the inferior part of the left middle turbinate extending to the choana. Note that the left maxil- lary and is clear. (b) Sagittal contrast-enhanced CT scan shows a giant soft tissue mass involving nasal cavity and extending to the nasopharynx which is completely filled by lesion. Note the presence of small feeding vessels and the absence of rich, irregular vascular supply characterized for angiofibroma. reddish-grey polypoid mass originating from the inferior middle turbinate was 5.5 cm in length, with maximum diam- portion of the left middle concha, obliterating the left nasal eter of 1.4 cm. The dimension of nasopharyngeal part of the cavity and extending posteriorly to the choana. Posterior mass was 4.5×4.2×2.6 cm (Fig. 2). A nasal pack consisting of rhinoscopy revealed a huge polypoid mass which complete- cotton gauze with ointment was removed on the fourth post- ly filled the nasopharynx. Endoscopic finding of the right operative day. Histopathological analysis confirmed the diag- nasal cavity was normal. Computed tomography (CT) of nosis of an angiomatous CP. In the nasal part, the lesion was the paranasal sinuses demonstrated a soft-tissue mass com- covered with ciliated pseudostratified respiratory . pletely filling the left nasal cavity and nasopharynx. The stroma was strongly infiltrated by plasma cells, neu- However, all paranasal sinuses were clear (Fig. 1a). As we trophils and macrophages (Fig. 3a). In the nasopharyngeal supposed to diagnose an angiofibroma with extranasopha- part of the polyp, the metaplasia of the ryngeal origin, we performed a contrast-enhanced CT scan in stratified epithelium of the transitional type was found. A with angiography. We found a vessel-like marked enhance- ment on contrast-enhanced CT scans of the mass in both early and delayed phase scans. These findings suggested that vessel-like marked enhancement is attributed to dilat- ed neovascularization. On angiography, we found only a few demonstrable feeding vessels, without significant enlargement, and small areas of neovascularity are also seen within the mass in the late capillary-venous phase (Fig. 1b). So, CT angiography did not confirm a vascular lesion, and the diagnosis of angiofibroma was excluded. At surgery, the nasal part of the lesion was debulked endo- scopically. The nasopharyngeal part was removed transoral- ly. This was followed by profuse bleeding, which was con- trolled by anterior nasal packing. The gross appearance of the excised mass demonstrated reddish-grey, soft, translucent areas, alternating with firm, black necrotic areas which were associated with a strong, offensive odor. The nasal part of the polyp with stalk arising from the anteroinferior part of the left Fig. 2. The nasopharyngeal part of mass, excised transorally.

54 ENT Updates Giant angiomatous choanal polyp originating from the middle turbinate

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Fig. 3. (a) Photomicrograph of an angiomatous CP shows pseudostratified respiratory ciliated epithelium and strong plasma cell and macrophage infiltration of the polyp stroma. (b) Large, dilated, thin-walled capillary-like blood vessels in the polyp stroma. (Hematoxylin & Eosin, magnification ×100).

small number of goblet cells and a paucity of submucous esized that the initial vascular dilatation, stasis, edema, and were also seen in all sections of the excised mass. The infarction occur in these sites following compression of examination revealed large, dilated, but thin-walled capillary- blood vessels. Reactive and repetitive changes lead to neo- like blood vessels in the polyp stroma (Fig. 3b). The postop- vascularization and fibrosis.[8] In the case of our patient, erative course of the patient was uneventful and 12 months of instead of the paranasal sinus ostium, the first site of vascu- follow-up showed no recurrence. lar compromise could be the inferior portion of the middle turbinate. Discussion On the other hand, Sayed and Abu-Dief[9] suggest that The etiology of CPs, first described by Killian in 1906, angiomatous polyp is a distinct type of nasal polyp and not a remains unclear. However, chronic is consid- derivate of the ordinary CP. They found that only angioma- ered to play a role in the etiology of CPs.[7] CPs of the mid- tous variant of nasal polyps presents with epistaxis. One of dle turbinate are extremely rare and there have been only six the main hystological characteristics of angiomatous nasal reported in the literature, of which one arose from the infe- polyps is strong plasma cell infiltration of the polyp stroma. rior, three arose from the medial and two arose from the This finding could be linked with angiogenesis and vascular posterior region.[1–5] One of the main characteristics of CPs endothelial proliferation within the polyp stroma suggesting is the tendency for rapid growth, resulting in their impres- that significantly increased number of plasma cells may be sive dimensions. This could be due to the fact that expres- the main cause of histological changes found in this variant sions of basic fibroblast growth factor (bFGF) and trans- of inflammatory nasal polyps. Blood flow in ordinary CPs is forming growth factor beta (TGF-β) are significantly high- decreased with smaller number of blood vessels compared to er in tissue of CPs than in bilateral nasal polyposis and, espe- with healthy . The stroma of angiomatous cially, in healthy nasal mucosa.[4] Angiomatous nasal polyps polyps have large numbers of dilated capillary spaces and are an uncommon subtype of CPs, which are characterized numerous large hemosiderin-laden macrophages suggesting by large numbers of dilated capillary spaces, with strong the presence of two parallel processes: blood extravasation [6,9] inflammatory infiltrates and abundant extracellular fibrin.[6] and stromal tissue reparation. Batsakis and Sneige[8] suggests that angiomatous polyp most Clinically and radiologically, angiomatous CPs may be often develop secondary to change in a CP because of vas- confused with vascular neoplasms, including nasopharyn- cular compromise in four sites of the upper airway: the geal angiofibroma. The early identification of cases similar ostium of the paranasal sinus, the posterior end of the infe- to angiofibroma is of crucial importance. This finding can rior turbinate, the choana and the nasopharynx. It is hypoth- have important implications for pre-surgical and surgical

Volume 7 | Issue 1 | April 2017 55 Perić A, Jovanovski A, Vukomanović Durdević B

management. A contrast-enhanced CT scan of the paranasal the benign nature of these lesions, they may be confused sinuses with angiography should be used to differentiate with neoplastic processes and vascular malformations. The between nasopharyngeal angiofibroma and angiomatous main differential diagnostic problem is nasopharyngeal CP. Regarding our case, it is particularly important in cases angiofibroma which can be excluded by contrast-enhanced of angiofibroma with extranasopharyngeal primary localiza- CT scan with angiography. tion. This is the rare form of angiofibroma originating from the paranasal sinuses, inferior and middle turbinate, and Conflict of Interest: No conflicts declared. from the nasal septum.[10] On angiography, angiomatous CPs have only a few demonstrable feeding vessels compared References to the rich irregular vascular supply of a nasopharyngeal 1. ‹la K, Topda¤ M, Öztürk M, ‹fleri M, Ayd›n Ö, Keskin G. angiofibroma. This is explained by the fact that angiomatous Retrospective analysis of surgical treatment of choanal polyps. CPs do not have a normal arborizing vascularity pattern but Kulak Burun Bogaz Ihtis Derg 2015;25:144–51. rather irregular arrangements of dilated capillary-type ves- 2. Prasad U, Sagar PC, Shahul Mameed OAN. Choanal polyp. J Laryngol Otol 1970;85:951–4. sels and newly endothelialized spaces with endoluminal [6] 3. Özkan C, Duce MN, Görür K. Choanal polyp originating from thrombosis. Thus, preoperative embolization is not neces- the middle turbinate. Eur Arch Otorhinolaryngol 2004;261:184–6. sary for management of angiomatous CPs. In contrast, the 4. Özk›r›fl M. Choanal polyp originating from the middle turbinate: resection of a nasopharyngeal angiofibroma requires preop- a case report. Ac›badem Üniversitesi Sa¤l›k Bilimleri Dergisi erative embolization due to the possible severe intraopera- 2012;3:187–9. tive bleeding. In patients with angiomatous CP, there was 5. Kim KS. Choanal polyp originating from the middle turbinate. no finding on paranasal CT scan indicating bony destruc- Otolaryngol Head Neck Surg 2013;148:525–6. tion, which is a frequent radiological characteristic of 6. Ceylan A, Asal K, Çelenk F, Uslu S. An angiomatous nasal polyp: angiofibroma. So, the excision of an angiomatous CPs, sim- a very rare variant of sinochoanal nasal polyps. B-ENT 2007;3:145–7. ilarly to ordinary CPs, is a relatively simple surgical proce- 7. Lopatin A, Bykova V, Piskunov G. Choanal polyps: one entity, one dure and polyp recurrence is relatively rare. surgical approach? Rhinology 1997;35:79–83. 8. Batsakis JG, Sneige N. Choanal and angiomatous polyps of the Conclusion sinonasal tract. Ann Otol Rhinol Laryngol 1992;101:623–5. We present an extremely rare case of an angiomatous CP 9. Sayed RH, Abu-Dief EE. Does antrochoanal polyp present with epistaxis? J Laryngol Otol 2010;124:505–9. originating from the with its clinical, 10. Perić A, Sotirović J, Cerović S, Živić L. Immunohistochemistry in radiological and pathological characteristics. Angiomatous diagnosis of extranasopharyngeal angiofibroma originating from CPs should be considered as a subtype of CPs with unilater- nasal cavity: case presentation and review of the literature. Acta al nasal obstruction and epistaxis as main symptoms. Despite Medica (Hradec Kralove) 2013;56:133–41.

This is an open access article distributed under the terms of the Creative Commons Attribution-NonCommercial-NoDerivs 3.0 Unported (CC BY- NC-ND3.0) Licence (http://creativecommons.org/licenses/by-nc-nd/3.0/) which permits unrestricted noncommercial use, distribution, and reproduc- tion in any medium, provided the original work is properly cited. Please cite this article as: Perić A, Jovanovski A, VukomanovićDurdević B. Associations among high altitude, , and bronchial hyperreactivity. ENT Updates 2017;7(1): 53–56.

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