Endoscopic removal of a silent middle turbinate angiofibroma

Case Report

Endoscopic removal of a silent middle turbinate angiofibroma －A Case Report Ping-Hung Shen1, William Lin Ho2, and Jen-Tsung Lai1* Department of Otolaryngology Head and Neck Surgery1 and Pathology2, Kuang-Tien General Hospital, Taichung, Taiwan

Abstract A 42-year-old male was found a right-side middle turbinate tumor incidentally during a routine sino-nasal surgery for hypertrophic rhinitis. Pathologist confirmed extranasopharyngeal angiofibroma. Due to its rarity of pathology and presentation, we report this case to remind otolaryngologists to be more careful about these highly vascular intranasal tumors, and this tumor can be excised under endoscope and without preoperative embolization. Key Words: angiofibroma, middle turbinate, extranasopharyngeal, embolization.

＊Corresponding author Received：19 Nov 2009；Accepted：01 Jan 2010

光田醫學雜誌第 5卷第 9期

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Ping-Hung Shen William Lin Ho and Jen-Tsung Lai1

Introduction

explained to the patient.

Angiofibroma is a rare nasopharyngeal tumor that primarily occurs in adolescent males . Extranasopharyngeal angiofibroma is relatively rare, and the most affected site is the maxillary sinus reported in the literature. We present an extremely rare case of extranasopharyngeal angiofibroma in a 42-year-old man arising from the right middle turbinate. The tumor was incidentally found during an operation for endoscopic inferior turbinoplasty. We report this case of an extranasopharyngeal

Figure 1. Axial CT scan showed that the tumor is fed from sphenopalatine area

angiofibroma and discuss the possibility of endoscopic resection of this tumor without preoperative embolization.

Case report A 42-year-old male of Tawainese, presented to our department due to nasal obstruction and postnasal dripping. Nasocopy revealed deviated nasal septum and bilateral hypertrophic inferior turbinates. Under the impression of nasal allergy and chronic

Figure 2.Coronal CT scan showed a limited lesion in the nasal cavity

hypertrophic rhinitis, endoscopic septoplasty and inferior turbinoplasty were suggested and then

The bleeding control for this operation was

performed. During the operation, a soft, gray-pinkish

under a hypotensive anesthesia via intravenous

colored mass originated from right middle turbinate

beta-blocker infusion, the mean arterial pressure is

was found incidentally and the tumor was incised for

maintained around 55 mmHg. The Wormald 17 cm

biopsy. The pathological report was an angiofiboma.

suction-bipolar is extensively used throughout the

There was no an episode of epistaxis during his

surgery, especially before each step of dissection to

lifetime, so it presented a silent tumor. After careful

ensure homeostasis. The tumor was found anterior-

preoperative endoscopic examination, CT scan and

medially from the inferior part of right middle

MRI study, the tumor showed to confine to the nasal

turbinate (Figure 3), extending laterally and attached

cavity, measuring 2 cm in diameter (Figures 1 and

to the posterior part of posterior maxillary fontanel,

2) .The possibility of endoscopic resection of this

posteriorly to the anterior ethmoid cell. During

tumor without preoperative embolization was well

carefully dissection from the maxillary fontanel,

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Endoscopic removal of a silent middle turbinate angiofibroma

the tumor seemed involved in the sphenopalatine fossa and partially in the pterygopalatine fossa. The

Discussion

majority of blood loss was seen in the manipulation

Juvenile nasopharyngeal angiofibromas account

of the pterygopalatine fossa. The pterygopalatine

for less than 0.5% of all head and neck tumors.

fossa was then packed with gelform, and no further

They are benign, highly vascular tumor but locally

nasal packing was needed after the surgery.

aggressive. They are most commonly found in young adolescent males, age range 7-19 yrs and rarely seen beyond 25 yrs(1). They usually arise from pterygopalatine fossa in the recess behind the sphenopalatine ganglion at the exit aperture of the pterygoid canal(2). The tumor then spreads mainly into nasal cavity and nasopharynx , or becoming large to adjacent areas, such as imfratemporal fossa, sphenoid sinus or middle cranial fossa. Primary extranasopharyngeal angiofibromas are rare, but have been reported sporadically in the literature and recently reviewed by Windfuhr et al(3). He

Figure 3. Endoscopic view of this tumor before excision Tumor can clearly seen laterally to the middle turbinate and attach to it. (NP: neuropatties, MT: middle turbinate, AF: angiofibroma, IT: inferior turbinate, S: nasal septum)

reviewed total of 65 patients ( 48males and 17 females) , the oldest patient being 78 years of age (mean 22.9 years). The maxilla was the most commonly affected site, only two cases from middle turbinate were reported(4,5); the top 3 symptoms of extranasopharyngeal angiofibromas were epistaxis alone (n=6) or combined with nasal obstruction (n=12) or painless facial swelling (n=3). In our case, despite of his nasal obstruction and postnasal dripping caused by nasal allergy, no any other specific symptoms was complained. If he did not receive the operation for inferior turbinoplasty, the tumor will not be found. Endoscopic removal of angiofibromas is becoming a main-stream treatment in nowadays.

Figure 4. T he tumor was composed primarily of dense fibrocollagenous tissue with interspersed vascular

Small nasal cavity tumors or with extension into the sinuses and pterygopalatine fossa and with limited

channels. The vascular channels were thin-walled

extension into the infratemporal fossa, can be

and lined by a single layer of endothelial cells.

removed endoscopically with a good success rate(6).


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Ping-Hung Shen William Lin Ho and Jen-Tsung Lai1

Minimal intracranial extension is also not an absolute

3. Windfuhr .IP, Remmert S. Extranasopharyngeal

contraindication if there is no clinical or radiological

angioftbroma: etiology. incidence and management.

involvement of the cavernous sinus(7-9).

Acta Otolaryngol. 2004; 124: 880-889.

Hyperselective preoperative embolization of

4.

Huang RY, Demrose EJ, Blackwell KE, Cohen AN,

the feeding arteries seemed to be of value in the

Calcaterra TC. Extranasopharyngeal angiofibroma.

endocopic surgery but some complications may

Int J Pediatr Otorhinolaryngol. 2000; 56:59-64

occur, such as blindness and brain ischemia(10).

5. P eloquin L, Klossek JM, Vasso-Brusa F. A rare

Considering our case, due to its small size and

case of nasopharyngeal angiofibroma in a pregnant

good surgical visualization, we try to manage it

woman. Otolaryngol Head Neck Surg. 1997; 117:

using hypotensive anesthesia without preoperative

S111-S114.

embolization. Our blood loss of this case is 250ml,

6.

Wormald PJ, Van Hasselt A. Endoscopic removal of

compared to the minimal blood loss described by

angiofibromas. Otolaryngol Head Neck Surg. 2003;

Borghei P et al(11), our result is by far acceptable.

129: 684-91.

To a c h i e v e t h i s g o o d b l e e d i n g c o n t r o l , t h e

7.

Nicolai P, Berlucchi M, Tomenzoli D, Cappiello J,

instrumentation is essential. A good suction-bipolar is

Trimarchi M, Maroldi R, Battaglia G, Antonelli AR.

undoubtedly helpful.

Endoscopic surgery for juvenile angiofibroma: when

Extranasopharyngeal angiofibromas are

and how. Laryngoscope. 2003; 113: 775-782.

extremely rare. They can occur in any age and

8. Hofmann T, Bernal-Sprekelsen M, Koele W, Reittner

gender. They however will be a diagnostic challenge

P, Klein E, Stammberger H. Endoscopic resection of

and form part of the differential diagnosis of

juvenile angiofibroma—long term results. Rhinology.

nasal tumors. With careful patient selection, the

2005; 43: 282-289.

operation will be done smoothly by endoscope and

9.

Douglas R, Wormald PJ. Endoscopic surgery for

achieve minimal blood loss without preoperative

juvenile nasopharyngeal angiofibroma: where are

embolization.

the limits? Curr Opin Otolaryngol Head Neck Surg. 2006; 14: 1-5.

References 1.

50

complication of embolization in juvenile

Antonelli AR, Cappiello J, Di Lorenzo D, Donajo C

nasopharyngeal angiofibroma. Int J Pediatr

A, Nicolai P, Orlandini A. Diagnosis, staging and

Otorhinolaryngol. 2005; 69: 423-428.

treatment of juvenile nasopharyngeal angiofibromas. 2.

10. Onerci M, Gumus K, Cil B, Eldem B. A rare

11. Borghei P, Baradaranfar MH, Borghei SH, Sokhandon

Laryngoscope. 1987; 97: 1319-1325.

F. Transnasal endoscopic resection of juvenile

Lloyd G, Howard D, Phelps P, Cheesman A. Juvenile

nasopharyngeal angiofibroma without preoperative

angiofibroma: the lessons of 20 yrs of modern

embolization. Ear Nose Throat J. 2006 ;85: 740-743,

imaging. J Laryngol Otol. 1999; 113: 127-134.

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個案報告

以鼻竇內視鏡切除潛藏的中鼻甲血管纖維瘤病例報告沈炳宏1 何霖2 賴仁淙1* 光田醫療社團法人光田綜合醫院耳鼻喉科1 病理科2

摘

要

　　鼻咽血管纖維瘤是很罕見的腫瘤，且大部份發生於年輕男性。鼻咽外血管纖維瘤更為少見，大部份的文獻報告中，發生的位置以上頜竇較為常見。本篇報告一位42歲成年男性病患，患有慢性肥厚性鼻炎接受下鼻甲部份切除手術時，意外發現右側中鼻甲有一異常的腫塊，經切片證實為血管纖維瘤。文中會針對此一罕見的鼻咽外血管纖維瘤，討論以鼻竇內視鏡手術的可能性，並說明如何在不用術前血管栓塞的方法，也可以順利的完成此手術。關鍵字：血管纖維瘤，中鼻甲，鼻咽外，血管栓塞

＊通訊作者收件日期：2009年11月19日；接受日期：2010年02月01日


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