Extranasopharyngeal angiofibroma mimicking choanal polyp in patients with chronic paranasal sinusitis

Extranasopharyngeal angiofibroma mimicking choanal polyp in patients with chronic paranasal sinusitis

G Model ANL-2465; No. of Pages 4 Auris Nasus Larynx xxx (2018) xxx–xxx Contents lists available at ScienceDirect Auris Nasus Larynx journal homepage...

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ANL-2465; No. of Pages 4 Auris Nasus Larynx xxx (2018) xxx–xxx Contents lists available at ScienceDirect

Auris Nasus Larynx journal homepage: www.elsevier.com/locate/anl

Two cases of extranasopharyngeal angiofibroma mimicking choanal polyp in patients with chronic paranasal sinusitis Hae Dong Kim, Ick Soo Choi * Department of Otorhinolaryngology—Head and Neck Surgery, Ilsan Paik Hospital, Inje University College of Medicine, Goyang, Republic of Korea

A R T I C L E I N F O

A B S T R A C T

Article history: Received 28 November 2017 Accepted 28 May 2018 Available online xxx

Extranasopharyngeal angiofibroma (ENA) is an angiofibroma that occurs in the extranasopharynx. It shows pathologic findings, such as juvenile angiofibroma (JNA), which accounts for 0.5% of head and neck mass. However, compared with JNA, the prevalence, affected site, and clinical characteristics are completely different, which leads some physicians to classify ENA as a disease different from JNA. ENA of the nasal turbinate origin are rarely reported in the literature. In addition, choanal polyp originating from the posterior part of the nasal turbinate is uncommonly reported. Recently, we encountered two cases of ENA, which were not diagnosed by intraoperative frozen section examination, but were histopathologically diagnosed post-operatively. Although we were unable to diagnose and perform embolization pre-operatively, ENA has been successfully treated by endoscopic surgery, without profuse bleeding. © 2018 Elsevier B.V. All rights reserved.

Keywords: Angiofibroma Choanal polyp Turbinate Chronic rhinosinusitis

1. Introduction Extranasopharyngeal angiofibroma (ENA), unlike juvenile nasopharyngeal angiofibroma (JNA), occurs outside the nasopharyngeal pterygopalatine fossa and surrounding tissue. In general, JNA accounts for approximately 0.5% of head and neck neoplasm [1], and the prevalence of ENA is even lower. According to de Vincentiis, only 13 of 704 angiofibroma cases were reported as ENA [2]. From the report based on the location of ENA by Windfuhr and Vent, ENA occurs most frequently at the nasal septum, followed by the maxillary sinus. However, ENA originating from the nasal turbinate is rarely reported [3]. Also, no previous study on ENA originating from the nasal turbinate that

* Corresponding author at: Department of Otorhinolaryngology—Head and Neck Surgery, Ilsan Paik Hospital, Inje University College of Medicine, Goyang, Republic of Korea 170, Juhwa-ro, Ilsanseo-gu, Goyang-si, Gyeonggido 10380, Republic of Korea. E-mail address: [email protected] (I.S. Choi).

mimicked a choanal polyp and concurrently had sinusitis was not reported. In this study, we presented two cases of patients with chronic rhinosinusitis who underwent endoscopic sinus surgery without pre-surgical vascular embolization for the treatment of ENA that mimicked a choanal polyp. Although the initial diagnosis from examination of frozen sections during the surgery was not ENA, post-surgical histopathologic examination identified the lesion as ENA, and the patient received appropriate treatment. 2. Case 1 A 41-year-old male patient was admitted because of right nasal bleeding. He had a history of endoscopic sinus surgery for rhinosinusitis 15 years prior, but he had no abnormalities or underlying diseases. Nasal endoscopy showed a choanal mass or polyp on the previous surgical site. Using contrast-enhanced sinus computed tomography (CT) (Fig. 1-A), we identified a choanal polyp on the right and mucocele of the ethmoidal sinus. We considered angiomatous polyp as a preoperative diagnosis.

https://doi.org/10.1016/j.anl.2018.05.015 0385-8146/© 2018 Elsevier B.V. All rights reserved.

Please cite this article in press as: Kim HD, Choi IS. Two cases of extranasopharyngeal angiofibroma mimicking choanal polyp in patients with chronic paranasal sinusitis. Auris Nasus Larynx (2018), https://doi.org/10.1016/j.anl.2018.05.015

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Fig. 1. (A) A contrast-enhanced paranasal sinus computed tomography scan showing the not contrasted enhancing mass (arrow) in the nasal cavity and choana with sinusitis (arrow head). (B) During the surgery, endoscopy shows a smooth surface and a round mass with proliferation of abundant blood vessels. (C) The vessels are lined by thin endothelial cells and are supported by a collagenous stroma containing numerous spindle- to stellate-shaped fibroblasts (H&E 100).

Because the patient had underlying chronic rhinosinusitis, we planned and performed endoscopic sinus surgery under general anesthesia. No abnormal findings were observed during the surgery, and we performed endoscopic paranasal sinus surgery on both sides and choanal polyp removal on the right. A neoplasm that appeared to be a choanal polyp, which resembled a long, thin line at the posterior end of the middle turbinate, was filling the nasopharynx and choana. The surface was relatively smooth, and the boundary was clearly observed (Fig. 1-B). We have cauterized and resected the polyp, and we did not observe excessive bleeding. Frozen section examination indicated that it was a regular polyp, but the final histopathologic examination showed that it was ENA (Fig. 1-C). The patient did not present with nasal bleeding, and he had no other complications postoperatively. He was followed-up for 1.6 years, and no signs of recurrence were observed.

choanal neoplasm that appeared to have originated from the right inferior turbinate. We considered inflammatory polyp or lymphoma as a preoperative diagnosis. We performed endoscopic sinus surgery under general anesthesia. Surgical findings showed a neoplasm with an exophytic pattern with proliferation of blood vessels. (Fig. 2-B). We believed that additional differential diagnosis was required for the neoplasm, and intraoperative frozen section examination was performed. The neoplasm was benign, and we performed endoscopic paranasal sinus surgery on both sides and removal of choanal polyp originating from the right inferior turbinate. We also removed a polyp of the left inferior turbinate, and we did not observe excessive bleeding or abnormal findings during the surgery. However, the polyp was diagnosed as ENA from the final histopathologic examination (Fig. 2-C), and the patient was discharged without remarkable complications. He is regularly visiting the outpatient clinic for 15 months, without signs of recurrence.

3. Case 2 4. Discussion A 22-year-old male patient with continuous nasal stuffiness and rhinorrhea visited our center after being diagnosed with a polyp within the nasal cavity and chronic rhinosinusitis. Endoscopic sinus examination showed a polyp within the nasal cavity and choanal neoplasm. Using pre-surgical contrast-enhanced sinus CT (Fig. 2-A), we observed paranasal sinusitis on both sides and

Angiofibroma is a histologically benign but locally aggressive vascular neoplasm. They are unencapsulated neoplasms of the vascular network within the fibrous matrix [3]. It usually occurs in nasopharynx and is commonly referred to as nasopharyngeal angiofibroma (NAF). NAF is also called

Please cite this article in press as: Kim HD, Choi IS. Two cases of extranasopharyngeal angiofibroma mimicking choanal polyp in patients with chronic paranasal sinusitis. Auris Nasus Larynx (2018), https://doi.org/10.1016/j.anl.2018.05.015

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Fig. 2. (A) A non-enhanced paranasal sinus computed tomography scan showing the mass (arrow) in the nasal cavity and choana with sinusitis (arrow head). (B) During the surgery, endoscopy shows a hyperemic polyp, and lymphoma or malignancy was suspected. (C) The specimen showed fibrocollagenous stroma and vessels of varying arrangement and caliber (H&E 100).

Juvenile angiofibroma(JNA) because it occurs mainly in adolescent males [1]. The main symptoms of JNA is nasal obstruction and epistaxis. The JNA can readily be identified during nasal endoscopy as a hypervacular mass in the nasopharynx. It exhibit an homogenous contrast enhancement on computed tomography(CT) and T1/T2-weighted magnetic resonance imaging(MRI) [4]. Because of these typical clinical findings, most of the JNA can be diagnosed clinically before the histological diagnosis. It is also known that there is a possibility that JNA develops a feeding vessel and causes many bleeding during surgery. Therefore, prior to clinical diagnosis, we can prevent massive hemorrhage during surgery by preoperative angiography and feeding vessel embolization [5]. However, in the case of ENA, it has same pathological feature as JNA but have different origins. It is known to occur in any part of the upper respiratory digestive tract. There are some different from JNA in age and gender of occurrence. Even, It does not show the typical imaging features seen in JNA, so it can be regarded as a disease with very different clinical features from JNA [3]. Because of these clinical differences, ENA is known that it is very difficult to perform the same procedure as preoperative angiogram for clinical diagnosis before the histological examination. According to Windfuhr and Vent, 174 cases of ENA were analysed. Biopsies were taken before therapy in 43 patients,

confirming the correct diagnosis in 32 patients. In 10 patients, different diagnoses were made such as benign tumor, granulation tissue, vascular tumour, etc. respectively. Only 24 of them had pre-operative angiogram, and only 12 required embolization due to hypervascularity. Furthermore, in 11 cases, severe hemorrhage was observed as biopsy or surgery was performed without clinical diagnosis. Among this report, There were 23 case reports of ENA from nasal turbinate (Table 1) [3]. Of 23 cases, 3 and 20 originated from the middle and inferior turbinate, respectively. Of 23 cases, 7 had preoperative histological examination, but only 1 had confirmed histological diagnosis, and had no excessive bleeding from histological examination. Therefore, angiography is selected to determine the vascular pattern and blood flow dynamics of angiofibromas prior to selective embolization, which helps reducing the amount of intraoperative bleeding significantly [3]. However, exclusion of hypervascularity with arteriography does not exclude ENA [6,7]. In most cases of ENA occurring in the nasal cavity, depending on the location and size of the tumor, the surgeon could use the endoscopic technique to completely remove the tumor, with or without previous selective angiography or embolization. Ultimately, the surgical approach to these tumors is determined by the location, blood supply, and size of the lesion as well as by the experience and technical skills of the surgeon.

Please cite this article in press as: Kim HD, Choi IS. Two cases of extranasopharyngeal angiofibroma mimicking choanal polyp in patients with chronic paranasal sinusitis. Auris Nasus Larynx (2018), https://doi.org/10.1016/j.anl.2018.05.015

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Table 1 Extranasopharyngeal angiofibroma—cases in the literature. Case no. Year Sex Age Site 1 2 3

1982 F 1996 F 1997 M

26 78 9

6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21 22 23

2000 2004 2004 2005 2005 2005 2005 2006 2009 2012 2012 2013 2013 2013 2014 2015 2015 2015 2016

14 60 30 60 33 41 57 62 63 56 57 52 9 16 8 26 48 12 28

M F M M M F M M F F F F M M F F M F F

Half of IT Anterior tip of IT Medial surface of IT MT (expanding nasal cavity mass) Medial surface of MT IT IT Anterior of IT IT MT IT Anterior part of IT MT Posterior of IT Anterioir part IT Anterior half of IT Head of IT Laser turbinoplasty site of IT Anterior of IT Tail of IT Posterior of IT IT Anterior of IT

Symptom Epistaxis Nasal obstruction Epistaxis Epistaxis Nasal obstruction, Nasal obstruction, Nasal obstruction, Nasal obstruction Nasal obstruction Nasal obstruction, Epistaxis Nasal obstruction Nasal obstruction, Facial swelling Nasal pain Nasal obstruction, Epistaxis Nasal obstruction Nasal obstruction, Epistaxis Nasal obstruction Epistaxis Nasal obstruction,

Biopsy Angiography Embolization Therapy –

+

epistaxis epistaxis epistaxis

+ + +

+ + +

epistaxis

+

+

+ epistaxis +

+ 9

epistaxis

+ +

epistaxis + epistaxis

+

Endoscopic approach Transnasal resection Subperiosteal resection En bloc excision Partial medial maxillectomy Endoscopic approach Endoscopic approach Transnasal resection Medial maxillectomy Endoscopic approach Endoscopic approach Transnasal resection Endoscopic approach Endoscopic approach Endoscopic approach Endoscopic approach Endoscopic approach Endoscopic approach Endoscopic approach Endoscopic approach Endoscopic approach Endoscopic approach Endoscopic approach

IT = Inferior turbinate; MT = Middle turbinate.

Hitherto, a case of ENA has not been reported to occur in the turbinate accompanied with chronic rhinosinusitis. We also do not believe that ENA was the cause for chronic rhinosinusitis in these cases. Because, In the case 1, patients had previous surgery and In the case 2, the size is not enough large to drainage of secretory fluid from sinuses. Therefore, we did not think that two cases were secondary to sinusitis by ENA. For cases similar to Case 1 of the report, Choanal neoplasm or vascular polyp can be considered as a differential diagnosis, which is histologically confirmed. The presence or absence of inflammatory infiltration within the tissue can assist in distinguishing these conditions [8]. The patient in Case 1 in this report had two previous rhinosinusitis surgeries, and he may have developed ENA due to changes after the surgery. Another previous report had shown the development of ENA on the surgical site after laser turbinate surgery [9], and therefore the patient in Case 1 may have developed ENA from surgical trauma. Moreover, there has not been a case report on ENA case that had concurrent chronic rhinosinusitis, or the association between intraoperative frozen section and final histopathological examinations. Although there has not been a clear explanation, the cases in this report did not have histopathological diagnosis even after performing intraoperative frozen section examination. We believe that since typical histological findings of angiofibroma are only found within the tumor itself, histological examination of a small section of the surface is not sufficient to provide a clear diagnosis. Moreover, Surface biopsy can be misleading, as stated for JAFs and found in ten patients with ENA (5.8%), as typical histologic appearance is only seen internally [3]. Finally, We have successfully treated the these cases, but many of the clinical and biologic characteristics of the ENA are

poorly defined. Also, Guidelines to the management of ENA have not been established clearly, and no standard classification system exists. Although it is a clinically rare disease, We suggest that ENA should be considered as a differential diagnosis in patients with an choanal polyp pattern originating from the turbinate. In conclusion, the otolaryngologist can benefit from this study when choosing an appropriate treatment for patients with chronic rhinosinusitis suspected of ENA originating from the turbinate and have further insight to differential diagnosis. References [1] Gullane PJ, Davidson J, O’Dwyer T, Forte V. Juvenile angiofibroma: a review of the literature and a case series report. Laryngoscope 1992;102:928–33. [2] De Vincentiis G, Pinelli V. Rhinopharyngeal angiofibroma in the pediatric age group. Clinical-statistical contribution. Int J Pediatr Otorhinolaryngol 1980;2:99–122. [3] Windfuhr JP, Vent J. Extranasopharyngeal angiofibroma revisited. Clin Otolaryngol 2018;43:199–222. [4] Nicolai P, Schreiber A, Bolzoni Villaret A. Juvenile angiofibroma: evolution of management. Int J Pediatr 2012;2012:412545. [5] Katsiotis P, Tzortzis G, Karaminis C. Transcatheter arterial embolisation in nasopharyngeal angiofibroma. Acta Radiol [Diagn] 1979;20:433–8. [6] Celik B, Erisen L, Saraydaroglu O, Coskun H. Atypical angiofibromas: a report of four cases. Int J Pediatr Otorhinolaryngol 2005;69:415–21. [7] Debbarma S, Langstang AD, Kumar SJ. Extranasopharyngeal angiofibroma of inferior turbinate in a young female: a very rare presentation. J Otol Rhinol 2015;4:1–2. [8] Alves FRA, Granato L, Maia MS, Lambert E. Surgical approaches to juvenile nasopharyngeal angiofibroma — case report and literature review. Int Arch Otorhinolaryngol 2006;10:Error: FPage (162) is higher than LPage (–166)!. [9] Kang JW, Kim YH, Kim JH. Angiofibroma of inferior turbinate as an unusual complication of CO2 laser turbinoplasty. J Craniofac Surg 2013;24:e513–4.

Please cite this article in press as: Kim HD, Choi IS. Two cases of extranasopharyngeal angiofibroma mimicking choanal polyp in patients with chronic paranasal sinusitis. Auris Nasus Larynx (2018), https://doi.org/10.1016/j.anl.2018.05.015