The Laryngoscope C 2015 The American Laryngological, V

Rhinological and Otological Society, Inc.

Do Open and Endoscopic Resection Approaches to Juvenile Nasopharyngeal Angiofibroma Result in Similar Blood Loss and Recurrence Rates? John P. Dahl, MD, PhD, MBA; David A. Zopf, MD, MS; Sanjay R. Parikh, MD, FACS

BACKGROUND Juvenile nasopharyngeal angiofibromas (JNAs) are rare, slow-growing tumors of the nasal cavity and skull base that account for approximately 0.5% of all head and neck tumors. This benign, highly vascular tumor is almost exclusively found in adolescent males. JNAs classically present with nasal obstruction and/or recurrent epistaxis. More advanced tumors can present with facial asymmetry, facial swelling, and visual disturbances. Histologically, JNAs are nonencapsulated tumors comprised of an irregular network of blood vessels set in fibroblastic stroma. They are thought to originate from the sphenopalatine foramen and initially grow into the nasal cavity and pterygopalatine fossa. More advanced lesions are locally destructive and can extend into the infratemporal fossa, orbit, and middle cranial fossa. There are a number of staging systems for JNAs based on the size and extent of the tumor; however, there is no current consensus regarding the optimal classification. The treatment of JNAs is surgical excision, often in combination with preoperative angiography and embolization of the vessels supplying the tumor. JNAs were traditionally resected via open surgical approaches; however, with the advancement of endoscopic skull base techniques, an increasing number of these tumors are being removed endoscopically.

LITERATURE REVIEW Most literature regarding the treatment of JNAs are retrospective, single-institutional reviews. Such From the Department of Otolaryngology–Head & Neck Surgery, Seattle Children’s Hospital, University of Washington, Seattle, Washington, U.S.A. Editor’s Note: This Manuscript was accepted for publication June 8, 2015. The authors have no funding, financial relationships, or conflicts of interest to disclose. Send correspondence to John P. Dahl, MD, Seattle Children’s Hospital, 4800 Sand Point Way NE, OA.9.220, PO Box 53711, Seattle, WA 98145-5005. E-mail: [email protected] DOI: 10.1002/lary.25471

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studies limit clinical conclusions given the small sample sizes and inherent biases. Recent systematic reviews and meta-analyses have provided a means to look at outcomes of JNA surgery in larger populations. Boghani et al. (2013) published one such systematic review focusing on the outcomes of endoscopic, endoscopic-assisted, and open surgical approaches to JNA resection. A total of 85 studies comprising 1,047 surgical cases were identified.1 The studies were divided into groups by the authors depending on whether the data were presented as individual patients (IPD) (n 5 345) or the aggregate of all patients in the study (APD) (n 5 702). These groups were analyzed separately for recurrence rates, intraoperative blood loss, and the impact of preoperative embolization on intraoperative blood loss. For the IPD cohort, recurrence rates for the three approaches were 10.8% for endoscopic, 14.5% for open, and 46.6% for endoscopic assisted. There was no significant difference between open and endoscopic techniques. Regarding blood loss, there was a significant difference between the endoscopic group (average of 544 mL) and the open group (average of 1579.5 mL). In the endoscopic cases, patients with preoperative embolization had significantly lower blood loss (average of 406.7 mL) than nonembolized patients (average 828.3 mL). In the open group, preoperative embolization led to a significant increase in blood loss. This is an interesting finding that the authors argue is due to a limited number of cases in this group as well as selection bias with larger, more complex tumors undergoing embolization. In the APD cohort, the recurrence rate was significantly lower for the endoscopic approach (4.7%) compared to the open (20.6%) and endoscopic-assisted approaches (22.6%). The contrasting results were attributed to the differences between the studies comprising the IPD and APD groups. The authors also hypothesized that more experienced endoscopic surgeons published the larger series comprising the APD cohort and therefore had lower recurrence rates. This study did not examine the complication rates among the three surgical approaches to JNA resection. Dahl et al.: Resection Approaches to JNA

A second systematic review by Khoueir et al. (2014) focused solely on endoscopic resection of JNAs. This approach identified 92 individual studies and a total of 821 patients.2 The average blood loss was 564.2 mL; cases with preoperative embolization had an average blood loss of 414.6 mL, and those without preoperative embolization had an average blood loss of 774.2 mL. Although both of the systematic reviews include many of the same studies, the data support the concept that intraoperative blood loss is lower with the endonasal approach. The overall complication rate for endoscopic JNA resection was 9.3%, with intranasal synechiae being the most common, followed by neurologic disorders, bleeding/buccal hematoma, ocular disorders, facial edema, atrophic rhinitis, eustachian tube dysfunction, and cerebrospinal fluid leak. The study estimated residual tumor to be present in 7.7% of cases. Tumor recurrence rate was estimated to be 10%, which is consistent with the IPD cohort from Boghani et al. (2013).1 Although there is no parallel systematic review focusing on the outcomes of open resection of JNAs, Huang et al. (2014) recently reported a single intuitional case series that compared outcomes between open and endoscopic approaches.3 This study, which comprised 162 patients, found that endoscopic JNA resection had a lower median blood loss as well as a lower number of complications compared to transpalatal or transmaxillary approaches. Chan et al. (2013) reviewed their 18year experience with all three approaches to JNA resection and found that blood loss, not surgical approach, was the only clinical factor that was significantly associated with complications from JNA surgery.4 A growing body of evidence currently supports the concept that most JNAs can be resected via endonasal techniques; however, some question the feasibility of endoscopically resecting more extensive tumors, such as those with intracranial extension. In an attempt to address this question, Leong (2013) performed a systematic review focusing on the management of JNAs with intracranial extension, identifying 15 studies and 72 total patients.5 This study estimated the prevalence of JNA with intracranial extension to be 17%. In terms of surgical approach, 7% underwent endoscopic resection, 2% underwent combined endoscopic and transpalatal approach, and 90% underwent open resection. Seventyfive percent of patients had a gross total resection of the tumor. Average intraoperative blood loss was 1,709 mL—1,449 mL for those who underwent preoperative

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embolization and 4,250 mL for those who did not undergo preoperative embolization. The overall complication rate was 46% with facial paresthesia, ophthalmoplegia, intranasal crusting, and cerebrospinal fluid leak each occurring in greater than 10% of cases. The overall recurrence rate was 18%, which is consistent with the above studies. Given the limited number of cases, the recurrence and complication rates for the individual surgical approaches were not calculated.

BEST PRACTICE The question as to whether there is an optimal surgical approach for the resection of JNAs continues to be a source of controversy. There is a growing body of evidence supporting, at the very least, the equivalence of endoscopic and open surgical approaches in terms of tumor recurrence. It does appear that the endoscopic approach, either alone or in combination with preoperative embolization, has less intraoperative blood loss. The findings from the systematic reviews, though informative, highlight the need for prospective multiinstitutional trials, which employ validated tumor staging systems and clinical outcome measures. Although great strides have been made regarding evidence-based management of JNAs, the extent and location of the tumor, combined with the surgeon’s judgment and skills, should ultimately determine the optimal approach for each patient.

LEVEL OF EVIDENCE This review of the optimal surgical approach for the treatment of JNAs is based on level III studies.

BIBLIOGRAPHY 1. Boghani Z, Husain Q, Kanumuri VV, et al. Juvenile nasopharyngeal angiofibroma: a systematic review and comparison of endoscopic, endoscopicassisted, and open resection in 1047 cases. Laryngoscope 2013;123: 859–869. 2. Khoueir N, Nicolas N, Rohayem Z, Haddad A, Abou Hamad W. Exclusive endoscopic resection of juvenile nasopharyngeal angiofibroma: a systematic review of the literature. Otolaryngol Head Neck Surg 2014;150: 350–358. 3. Huang Y, Liu Z, Want J, Sun X, Yang L, Wang D. Surgical management of juvenile nasopharyngeal angiofibroma: analysis of 162 cases from 1995 to 2012. Laryngoscope 2014;124:1942–1946. 4. Chan KH, Gao D, Fernandez PG, Kingdom TT, Kumpe DA. Juvenile nasopharyngeal angiofibroma: vascular determinates for operative complications and tumor recurrence. Laryngoscope 2014;124:672–677. 5. Leong SC. A systematic review of surgical outcomes for advanced juvenile nasopharyngeal angiofibroma with intracranial involvement. Laryngoscope 2013;123:1125–1131.

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Do open and endoscopic resection approaches to juvenile nasopharyngeal angiofibroma result in similar blood loss and recurrence rates?

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