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Transnasal endoscopic resection of juvenile nasopharyngeal angiofibroma without preoperative embolization.

Abstract

Juvenile nasopharyngeal angiofibroma (JNA) is a benign, highly vascular, and locally invasive tumor. Because the location of these tumors makes conventional surgery difficult, interest in endoscopic resection is increasing, particularly for the treatment of lesions that do not extend laterally into the infratemporal fossa. We report the results of our series of 23 patients with JNA (stage IIb or lower) who underwent transnasal endoscopic resection under hypotensive general anesthesia without preoperative embolization of the tumor. All tumors were successfully excised. The amount of intraoperative blood loss was acceptable. We observed only 1 recurrence, which was diagnosed 19 months postoperatively in a patient with a stage IIB primary tumor. We observed only 3 complications during follow-up--all synechia. We conclude that endoscopic resection of JNAs is safe and effective. The low incidence of recurrence and complications in this series indicates that preoperative embolization may not be necessary for lesions that have not undergone extensive spread: instead, intraoperative bleeding can be adequately controlled with good hypotensive general anesthesia.

Introduction

In 1906, Chaveau introduced the term juvenile nasopharyngeal angiofibroma (JNA). (1) This benign tumor is characterized by aggressive local invasiveness and a tendency toward local recurrence after incomplete resection: it occurs primarily in adolescent males. (2) JNA is a nonencapsulated, submucosal, spreading tumor made up of fibrous connective tissue and an abundance of endothelium-lined vascular spaces. (3)

Many studies have indicated that JNA originates in the pterygopalatine fossa at the aperture of the pterygoid canal. (4) From there, it can extend to surrounding structures, including the nasal cavity, sphenoid sinus and sella, infratemporal fossa, inferior orbital fissure, and intracranial area. (5)

A diagnosis of JNA is suggested by the classic triad of epistaxis, nasal obstruction, and a nasopharyngeal mass. The presence of other symptoms depends on the direction and extent of tumor spread. (6,7) The treatment of choice for localized primary tumors is surgical resection: for extensive masses and for recurrent tumors, radiotherapy is also recommended. (8,9)

Because the location of these tumors makes conventional surgery difficult, interest in endoscopic resection is increasing, particularly for lesions that do not extend laterally to the infratemporal fossa. (10,11) In this article, we report the results of our series of transnasal endoscopic resections without preoperative embolization of the tumor in patients with JNA that did not extend laterally into the infratemporal fossa.

Patients and methods

Our series included 23 patients with histopathologically proven JNA (stage IIB or lower) who underwent transnasal endoscopic resection without tumor embolization at our center from February 2000 through August 2004. All patients were male. Their ages ranged from 10 to 26 years (mean: 16.2). No patient underwent radiotherapy.

Staging. Tumor staging was based on the results of computed tomography (CT) and/or magnetic resonance imaging (MRI). Tumors were staged according to the system described by Radkowski et al (figure). (12) Five tumors were staged as IA, 9 as IB, 4 as IIA, and 5 as IIb (table 1).

Surgical technique. Hypotensive general anesthesia was administered (target mean arterial pressure: 55 to 65 mm Hg) with the patient in the reverse Trendelenburg position. Topical vasoconstriction was achieved by placing a cottonoid pledget soaked in 1:1,000 epinephrine solution for 10 minutes.

The surgical technique varied slightly from case to case, depending on the tumor's size and degree of extension, but all dissections followed a series of the same basic steps. The tumor was slowly pulled down with a retractor to expose the sphenopalatine foramen. If the sphenopalatine artery was evident through the sphenopalatine foramen, it was cauterized. We did not clip the artery, and we did not use cautery to shrink the tumor.

The tumor was detached from the nasal mucosa and sphenoid sinus and pushed medially and inferiorly. If extension to the lower part of the pterygopalatine fossa was present, the posterior portion of the inferior turbinate was resected. These maneuvers exposed the superior, inferior, and medial borders of the tumor. The posterior wall of the maxillary sinus was resected, and the retromaxillary periosteum was elevated, which provided access to the infratemporal fossa. The tumor and surrounding soft tissue were pulled inferomedially, and the pterygopalatine fossa was cleaned. The entire tumor was removed via the month. The nasal cavity was packed with Merocel for 48 hours.

The first follow-up endoscopic evaluation was performed 3 weeks postoperatively and repeated 2 months later. Subsequent follow-ups with CT or MRI were scheduled for the third month and then every 6 months up to 2 years and once yearly thereafter. Overall, the length of individual follow-ups ranged from 13 to 57 months (mean: 33).

Outcome variables included estimated perioperative blood loss, recurrences, and complications. Any patient whose symptoms returned or who exhibited an expanding tumor on imaging was considered to have experienced a recurrence and was therefore a candidate for reoperation.

Results

The estimated average amount of intraoperative blood loss per patient and the average number of blood transfusion packs required per patient were positively correlated with tumor stage (table 1):

* stage 1A: 666 ml blood loss; 0 packs for transfusion

* stage IB: 680 ml; 0.2 packs

* stage IIA: 1,068 ml; 0.75 packs

* stage IIB: 1,310 ml; 1.8 packs

During follow-up, 3 patients developed asymptomatic synechia between the nasal septum and one of the turbinates.

Only 1 patient (4.3%) experienced a recurrence. This patient's primary tumor had been staged as IIB. The recurrence was diagnosed 19 months postoperatively after the patient had complained of repeated episodes of epistaxis and occasional nasal obstruction. The patient underwent reoperation and remained disease-free at 28 months.

Discussion

A diagnosis of JNA requires close attention to symptoms. In most cases, the aforementioned triad of epistaxis, nasal obstruction, and a nasopharyngeal mass in a young male is suggestive of JNA. The presence of anterior bowing of the posterior wall of the maxillary sinus on imaging (Holman-Miller sign) is a known pathognomonic finding. (13)

Many surgical approaches to JNA have been used, including the transpalatal approach, medial maxillectomy, midfacial degloving, LeFort I surgery, lateral rhinotomy, and the infratemporal fossa approach. However, transnasal endoscopic resection is becoming an attractive alternative to these external approaches as our surgical expertise and our knowledge of the intranasal anatomy improve. Still, the proper role of endoscopic resection for the management of angiofibromas remains a subject of debate. (4)

Hypotensive general anesthesia. We did not perform preoperative embolization because we believe that intraoperative bleeding of lesions that have not spread extensively can be controlled by good hypotensive general anesthesia. Hypotensive general anesthesia may be appropriate for several types of operations, including head and neck surgery, neurosurgery, large orthopedic procedures, and a variety of plastic surgical procedures. The possible complications of hypotensive general anesthesia primarily involve the nervous system. The most common are dizziness and cerebral thrombosis. Less common complications include hemiplegia, retinal thrombosis, and even death.

During hypotensive general anesthesia, a target blood pressure of 50 to 65 mm Hg is safe for young, otherwise healthy patients. This type of anesthesia is probably more risky in the elderly and in those who have underlying organ dysfunction. (14) In our study, no patient experienced any anesthesia-related complication.

Preoperative embolization. Another reason to avoid embolization is that it has potential complications of its own. (15) They include nerve injury (e.g., facial nerve palsy) and devitalization of tissues such as the overlying skin and nasal mucosa. Intentional embolization of the branches of the internal carotid artery may result in accidental embolization of the brain and ophthalmic artery.

Even so, embolization does have its proponents, as several authors have described their favorable experiences with embolization during transnasal endoscopic excision of JNAs (table 2). For example, Li et al reported that patients with stage IIC or lower lesions who underwent preoperative embolization lost significantly less blood intraoperatively than did those who were not embolized (mean: 637 and 1.136 ml, respectively; p < 0.05). (15) Also, among patients who required transfusion, embolized patients required significantly less blood (mean: 400 and 836 ml; p < 0.01).

Likewise, Liu et al reported that preoperative embolization was associated with significantly less intraoperative bleeding in patients with stage IA tumors (mean: 275 and 840 ml, respectively). (16) They also compared the amount of blood loss in 17 patients with stage IB and higher tumors--8 of these patients had undergone embolization of feeding arteries and 9 had undergone ligation of an external carotid artery--but they found no significant difference.

Nicolai et al reported that the amount of intraoperative blood loss in 15 patients, all of whom underwent preoperative embolization, ranged from 80 to 600 ml (mean: 372). (17) Moulin et al also reported a significant decrease in intraoperative blood loss. (18)

Both Siniluoto et al (19) and Elasfour et al (20) reported that embolization not only reduced intraoperative blood loss, but it also contributed to improved surgical results.

Tranbahuy et al recommended intratumoral embolization. (21) Hazarika et al reported that preoperative embolization followed by KTP laser-assisted endoscopic excision of JNA is superior to radical approaches. (22)

Staging. As all of our patients were staged as IIB or lower, our findings support the effectiveness of the transnasal endoscopic approach for tumors that have extended to the nasal cavity, nasopharynx, paranasal sinuses, and pterygopalatine fossa. Other authors have recommended this procedure for tumors up to stage IIC, citing the benefits of fewer postoperative complications and a more rapid recovery and earlier discharge in addition to less bleeding. (12,23-25) However, involvement of the infratemporal fossa, anterior skull base, and orbit still requires a more invasive surgical approach, (26) although Onerci et al recommended endoscopic resection for lesions with minimal extension into the cranium. (27)

Recurrence. Scholtz et al performed transnasal endoscopic excision on 7 patients with JNAs that had extended into the nasal cavity, the nasopharynx, the pterygopalatine fossa, and the ethmoid, sphenoid, and maxillary sinuses. (10) They reported no recurrences.

Likewise, Wormald and Van Hasselt reported no recurrences in 7 patients with lesions that had extended into the nasal cavity, paranasal sinuses, and pterygopalatine fossa, including those with minimal invasion into the infratemporal fossa. (28)

Finally, Nicolai et al reported only 1 recurrence in 15 patients. (17) The recurrent lesion was small and did not extend laterally. That patient had undergone conservative surgery that had not included dissection of the pterygopalatine fossa; also, the middle turbinate was spared. The amount of intraoperative blood loss in these 15 patients, all of whom underwent preoperative embolization, ranged from 80 to 600 ml (mean: 372).

Complications. In our study, we observed only 3 complications--all synechia. Others have also reported a low incidence of complications. (10,24,25)

Conclusions

In summary, we conclude that the transnasal endoscopic technique is an acceptable approach to the resection of JNAs without extensive lateral extension. Our study demonstrated a low degree of bleeding, a low recurrence rate, and minimal complications.

Although we did not perform preoperative embolization of the feeding arteries, we did not encounter massive intraoperative bleeding. Therefore, we believe that intraoperative bleeding can be adequately controlled with good hypotensive general anesthesia in patients whose tumor does not extend into the infratemporal fossa or cranium.

References

(1.) Chaveau C. Histoire des Maladies du Pharynx. Paris: Balliere; 1906.

(2.) Iannetti G, Belli E, De Ponte F, et al. The surgical approaches to nasopharyngeal angiofibroma. J Craniomaxillofac Surg 1994;22: 311-16.

(3.) Ungkanont K, Byers RM, Weber RS, et al. Juvenile nasopharyngeal angiofibroma: An update of therapeutic management. Head Neck 1996;18:60-6.

(4.) Lloyd G, Howard D, Phelps P. Cheesman A. Juvenile angiofibroma: The lessons of 20 years of modern imaging. J Laryngol Otol 1999: 113:127-34.

(5.) Batsakis JG. Tumors of the Head and Neck: Clinical and Pathological Considerations. 2nd ed. Baltimore: Williams & Wilkins; 1979: 296-300.

(6.) Kennedy JD, Haines SJ. Review of skull base surgery approaches: With special reference to pediatric patients. J Neurooncol 1994;20: 291-312.

(7.) 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.

(8.) Wiatrak BJ, Koopmann CF, Turrisi AT. Radiation therapy as an alternative to surgery in the management of intracranial juvenile nasopharyngeal angiofibroma. Int J Pediatr Otorhinolaryngol 1993: 28:51-61.

(9.) Gudea F. Vega M, Canals E, et al. Role of radiation therapy for "juvenile" angiofibroma. J Laryngol Otol 1990:104:725-6.

(10.) Scholtz AW. Appenroth E, Kammen-Jolly K, et al. Juvenile nasopharyngeal angiofibroma: Management and therapy. Laryngoscope 2001:111:681-7.

(11.) Lim IR. Pang YT, Soh K. Juvenile angiofibroma: Case report and the role of endoscopic resection. Singapore Med J 2002;43: 208-10.

(12.) Radkowski D, McGill T, Healy GB, et al. Angiofibroma. Changes in staging and treatment. Arch Otolaryngol Head Neck Surg 1996; 122:122-9.

(13.) Cummings CW, ed. Otolaryngology-Head and Neck Surgery. 3rd ed. Vol. 2. St. Louis: Mosby: 1998:1514.

(14.) Cucchiara RF. Miller ED, Reves JG, et al. Anesthesia. 4th ed. Vol. 2. New York: Churchill Livingstone: 1994:1497-9.

(15.) Li JR, Qian J, Shan XZ, Wang L. Evaluation of the effectiveness of preoperative embolization in surgery for nasopharyngeal angiofibroma. Eur Arch Otorhinolaryngol 1998:255:430-2.

(16.) Liu L, Wang R, Huang D, et al. Analysis of intra-operative bleeding and recurrence of juvenile nasopharyngeal angiofibromas. Clin Otolaryngol Allied Sci 2002;27:536-40.

(17.) Nicolai P, Berlucchi M, Tomenzoli D, et al. Endoscopic surgery for juvenile angiofibroma: When and how. Laryngoscope 2003:113: 775-82.

(18.) Moulin G, Chagnaud C, Gras R, et al. Juvenile nasopharyngeal angiofibroma: Comparison of blood loss during removal in embolized group versus nonembolized group. Cardiovasc Intervent Radiol 1995;18:158-61.

(19.) Siniluoto TM, Luotonen JP, Tikkakoski TA, et al. Value of pre-operative embolization in surgery for nasopharyngeal angiofibroma. J Laryngol Otol 1993:107:514-21.

(20.) Elasfour A, Khafagy Y, Amer T, Tawfik A. Preoperative embolization of nasopharyngeal angiofibroma: A report of 34 cases. International Congress Series 2003:1240:1449-54.

(21.) Tranbahuy P, Borsik M, Herman P. et al. Direct intratumoral embolization of juvenile angiofibroma. Am J Otolaryngol 1994;15: 429-35.

(22.) Hazarika P, Nayak DR, Balakrishnan R, et al. Endoscopic and KTP laser-assisted surgery for juvenile nasopharyngeal angiofibroma. Am J Otolaryngol 2002:23:282-6.

(23.) Mann WJ, Jecker P, Amedee RG. Juvenile angiofibromas: Changing surgical concept over the last 20 years. Laryngoscope 2004:114: 291-3.

(24.) Jorissen M. Eloy P. Rombaux P. et al. Endoscopic sinus surgery for juvenile nasopharyngeal angiofibroma. Acta Otorhinolaryngol Belg 2000:54:201-19.

(25.) Bernal-Sprekelsen M, Vazquez AA, Pueyo J. Carbonell Casasus J. [Endoscopic resection of juvenile nasopharyngeal fibromas]. HNO 1998:46:172-4.

(26.) Naraghi M. Kashfi A. Endoscopic resection of nasopharyngeal angiofibromas by combined transnasal and transoral routes. Am J Otolaryngol 2003:24:149-54.

(27.) Onerci TM, Yucel OT, Ogretmenoglu O. Endoscopic surgery in treatment of juvenile nasopharyngeal angiofibroma. Int J Pediatr Otorhinolaryngol 2003:67:1219-25.

(28.) Wormald PJ, Van Hasselt A. Endoscopic removal of juvenile angiofibromas. Otolaryngol Head Neck Surg 2003:129:684-91.

Peyman Borghei, MD; Mohammad Hossein Baradaranfar, MD; Seyed Hebatodin Borghei, MD; Farnoosh Sokhandon, MD

From the Ear, Nose, Throat Research Center, Amir-Alam Hospital, Tehran University of Medical Sciences, Tehran, Iran (Dr. P. Borghei, Dr. Baradaranfar, and Dr. S.H. Borghei), and the Department of Diagnostic Radiology, University of Michigan Medical Center, Ann Arbor (Dr. Sokhandon).

Reprint requests: Peyman Borghei, MD, Ear, Nose, Throat Research Center, Amir-Alam Hospital, North Sa'di Ave., Tehran, 11457 Iran. Phone: 98-912-115-2868; fax: 98-21-6676-0245; e-mail: borgheii@sina.tums.ac.ir
Figure. The Radkowski staging systenn fbr juvenile nasopharyngeal
angiofibroma. (12)

Stage   Description

IA      Involvement limited to the nose and/or naso-
        pharynx

IB      Extension into one or more sinuses

IIA     Minimal extension into the pterygopalatine
        fossa

IIB     Occupation of the entire pterygopalatine
        fossa with or without erosion of the orbital
        apex

IIC     Involvement of the infratemporal fossa with
        or without extension to the cheek or posterior
        to the pterygoid plates

IIIA    Erosion of the skull base (the middle cranial
        fossa/base of the pterygoids); minimal intra-
        cranial extension

IIIB    Erosion of the skull base; extensive intra-
        cranial extension with or without cavernous
        sinus invasion

Table 1. Results of treatment with endoscopic transnasal technique

              No. units      Blood     Follow-up
Pt.   Stage   transfused   loss (ml)      (mo)

 1     IA         0            550         46
 2     IA         0            850         42
 3     IA         0            570         36
 4     IA         0            660         30
 5     IA         0            700         20
 6     IB         0            550         57
 7     IB         0            620         54
 8     IB         1            800         48
 9     IB         0            450         48
10     IB         1          1,150         46
11     IB         0            650         35
12     IB         0            750         22
13     IB         0            550         16
14     IB         0            600         14
15     IIA        0            820         44
16     IIA        0          1,100         17
17     IIA        1          1,050         47
18     IIA        2          1,300         13
19     IIB        2          1,200         47
20     IIB        2          1,300         24
21     IIB        1          1,600         22
22     IIB        2          1,350         18
23     IIB        2          1,100         16

Pt.   Recurrence    Complication

 1        --             --
 2        --             --
 3        --             --
 4        --             --
 5        --             --
 6        --             --
 7        --             --
 8        --             --
 9        --             --
10        --             --
11        --             --
12        --             --
13        --             --
14        --             --
15        --             --
16        --             --
17        --          Synechia
18        --             --
19         +          Synechia
20        --             --
21        --             --
22        --          Synechia
23        --             --

Table 2. Review of the results of other studies

                       Type of        Highest
Author                 surgery        stage          Recurrences

Li et al, (15) 1998    Conventional   IIC            1 of 21

Liu et al, (16) 2002   Conventional   I (see text)   0 of 10

Scholtz                Transnasal     IIB            0 of 7
et al, (10) 2001       endoscopic

Wormald and Van        Transnasal     IIC            0 of 7
Hasselt, (28) 2003     endoscopic

Nicolai                Transnasal     IIC            1 of 15
et al, (17) 2003       endoscopic

                       Mean blood              Mean blood
Author                 loss                    transfused

Li et al, (15) 1998    With preoperative       With preoperative
                       embolization: 637 ml;   embolization: 400 ml;
                       without: 1,136 ml       without: 836 ml

Liu et al, (16) 2002   With preoperative       Not reported
                       embolization: 275 ml;
                       without: 840 ml

Scholtz                Not reported            Not reported
et al, (10) 2001

Wormald and Van        Not reported            Not reported
Hasselt, (28) 2003

Nicolai                372 ml                  Not reported
et al, (17) 2003
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Author:Borghei, Peyman; Baradaranfar, Mohammad Hossein; Borghei, Seyed Hebatodin; Sokhandon, Farnoosh
Publication:Ear, Nose and Throat Journal
Article Type:Clinical report
Geographic Code:1USA
Date:Nov 1, 2006
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