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Endoscopic Approach for Excision of Juvenile Nasopharyngeal
Angiofibroma: Outcome and Complications
Presented in 9th^ international congress of otolaryngology Tehran,IRAN , November
Mohammadi Ardehali, Mojtaba.MD; Samimi Ardestani,S.Hadyi.MD;Dabirmoghadam,peyman.MD; Yazdani, Nasrin.MD; Goodarzi, Hassan. MD.
From the Tehran University Medical Sciences Research Center of Otolaryngology, Head and Neck Surgery, Amir Alam Hospital, Tehran University of Medical Sciences, Tehran, Iran.
Summary objectives: The purpose of this study is to compare the efficacy of the endoscopic surgery of juvenile nasopharyngeal angiofibroma and its intra- and postoperative complications with similar studies and other approaches in the literature. Methods: This is a retrospective study of 47 cases of JNA who were treated with an endoscopic approach, since August 1998 till May 2005. After reviewing the medical files of the patients at Amir Alam hospital, all patients were followed by CT scan and endoscopic evaluation. Results : According to Radkowski et al. staging system, there were 21 patients in stages IA to IIB, 22 cases IIC, 3 cases IIIA, and one case IIIB. Mean age of patients was 17.1 years old (ranging from 7 to 37 years old). Five cases were embolized before surgery. Mean blood hemorrhage in all patients was 1336.2 cc, in stages IA-IIC 890 cc, in stage III 3450 cc, and in embolized patients 770 cc ( with 1403 cc in unembolized cases). Recurrence was found in 9 patients (19.1%) and mean time of recurrence was 17 months after surgery. Rupture of cavernous sinus occurred in 2 cases with no mortality, respectively. Mean hospital stay was 3.1 days in all cases and 1.8 days in embolized patients. Conclusions: According to this large number of cases, endoscopic resection of JNA is a safe and effective technique, because of less blood loss, decreased hospital stay and even recurrence rate, especially in tumors that are not intracranially expanded. Considering the advantage of endoscopic surgery, it should be accepted as first choice of treatment of JNA.
Keywords: Endoscopic surgery; Angiofibroma; Nasopharyngeal neoplasm
1. Introduction JNA is a histologically benign neoplasm that invades locally and is fully vascularized without any capsule. JNA is almost only seen in males, in childhood or adolescence. Although this tumor is the most common benign neoplasm of nasopharynx, some reports indicate the presence of angiofibroma in nasal septum, middle turbinate, hard palate and alveolar ridge [1, 2, 3, 4]. It is responsible of less than 0.05 percent of all head and neck tumors [5]. This tumor exactly originates from the superior border of sphenopalatine foramen which is formed by the junction of the trifurcation of Palatine bone, horizontal wing of vomer, and the roof of pterygoid plates [6]. The origin of the tumor is of great importance since it elucidates its pattern of distribution. In addition, it affects the surgery approach by which it is treated. The aforementioned junction
forms the superior margin of the sphenopalatine foramen. Initially, the tumor grows in the submucosa of the nasopharyngeal roof, reaching the septum and posterior aspect of nasal space, creating a mass effect that may cause nasal airway obstruction. As the tumor grows, the anterior face of sphenoid sinus is evolved and invaded. Angiofibroma may laterally extend to the Pterygomaxillary fossa and cause bowing of posterior wall of maxillary sinus. It may involve the infratemporal fossa and middle cranial fossa. Involvement of bilateral sphenopalatine foramen by JNA, separately, is also reported [7].The diagnosis of JNA is based essentially on clinical and radiologic examination[6].During initial evaluation, CT scan with and without contrast reliably assesses tumor extent. MRI studies, however, may be more accurate than CT in assessing intra cranial extension. Preoperative angiography is helpful for the evaluation of feeding vessels and allows embolization of these lesions. Although radiotherapy, hormone therapy, cryotherapy, electro coagulation, have all been described in the literature, surgery remains the treatment of choice for JNA [5]. Conventional surgical approaches including transpalatal, transzygomal, lateral rhinotomy, midface degloving, and transantral have been used. Kamel et al. in 1996, described transnasal endoscopic approach for the limited angiofibroma lesions [8].Tumor resection through the nose by means of an endoscope is ideally suitable for tumors limited to nasopharynx, nasal cavity, ethmoid, and sphenoid [8]. The goal of this study is to retrospectively assess the outcome of endoscopic surgery of nasopharyngeal angiofibroma and its complications in 47 cases.
2. Materials and method: Between August 1998 and May 2005, 47patients with JNA who had undergone endoscopic surgery at the Department of Otolaryngology, Amiralam referral ENT hospital, were studied. All the patients with an underlying disease other than angiofibroma and all the patients who had undergone surgery by approaches other than endoscopy were excluded. All the patients who underwent JNA endoscopic surgery evaluated endoscopically in two weeks after surgery, monthly in 6 months after surgery, and annually afterwards. An annual CT scan evaluation series have been commenced since the 3rd month post operation. The examination process of the patients took 2.5 years and all the patients were reassessed for an endoscopic evaluation, examination and if suspected for recurrence, underwent another CT scan. Among 47 patients who were according to the criteria included in the study, 31 cases (66%) were primary treated in this center by the endoscopic surgery approach. 16 cases (34%) received secondary treatments. they have history of previous conventional or Endoscopic surgery. Among 47 patients who underwent endoscopic surgery, 2 (4.2%) were operated by the combined approach of "Endoscopy and open surgery through the mouth"; 2 (4.2%) were operated by the combined surgical approach of “Endoscopy and sublabial surgery” and 43 (91.6%) were operated by the exclusively endoscopic approach. Diagnosis of JNA was based on the clinical manifestations and the data obtained from CT scan imaging with or without MRI. 46 patients (97.9%) had a CT scan image before the surgery and one patient (2.1%) had an MRI image, only; CT scan imaging and MRI were both performed for 2 patients (4.3%). Biopsies were performed in only two patients, one with the diagnosis of antrochoanal polyp and the other diagnosed as adenoid hypertrophy. The pathological examination after surgery revealed angiofibroma in all patients. The mean follow-up was 33.1 months with a range of 3 to 74 months. Two (4.3%) patients were followed for 3 months, 3 (6.4%) patients for 7-9 months, and 42 (89.3%) patients for 14-74 months. 3. Results:
Tumor recurrence: During the mean duration of 33.1 months follow-up, 9 patients (19.1%) had a relapse of the tumor and 38 patients (80.9%) were free of JNA.
4. Discussion: Endoscopic surgery in the nasal cavity and paranasal sinuses has been primarily used to cure non-neoplastic diseases. Along with the development of surgeons' skill in endoscopic operations, this approach was used for treatment of JNA. Seldom does this tumor have connections with its surrounding structures. Availability of the nutrient vessels of JNA and the ability to search for the places where the remnants of the tumor can potentially lead to a relapse are increased by endoscopic techniques [9]. This approach if meticulously carried out will be considered a very effective way to treat JNA, provided that it causes less disability in patients, decreases the duration of hospital stay, and lowers the intraoperative bleeding rate. Other advantages of endoscopic approach is the least morbidity including to avoiding of incision on the face, resecting the least amount of normal soft tissue, avoiding the destruction of facial bones and late facial deformity[10,11] In this report, a study of 47 patients with a mean follow-up period of 33.1 months has been carried out. The maximum number of patients reported thus far has been 20 cases with a mean follow-up period of 22 months [12]. In this section, general data and the comparison of primarily and secondarily treated patients will be discussed. In the end, a comparison will be made between this study and another one which has been carried out on 59 patients who'd undergone open surgery. The most common age at which JNA occurs is 10-25 years old. Even in ages over 25 and below 7, atypical JNA is considered.[13]In this study, the maximum number of patients (55.3%) are at stage IIC or higher, which shows that the tumors had already advanced to a great extent at the time of admission to the hospital. The most common clinical manifestation was nasal obstruction, with a frequency of 43 cases (91.5%); the second most common manifestation was epistaxis (63.8%). The most frequent number of hospitalization days was 2 days or less (83%). The number of hospitalization days for patients at stage III (4 cases / 8.5%) was 7.5 days, and for embolized patients (6 cases / 10.6%), was 1.8 days. This means a lowest hospitalization day for this group. There was no need for blood transfusion in 15 patients (31.9%). Since the intraoperative hemorrhage occurring in open surgeries is in large due to the incisions made in order to reach the tumor, rather than the extraction of the tumor itself [11] , endoscopic surgery plays a great role in reducing the intraoperative hemorrhage level. This study demonstrates that the intraoperative hemorrhage level won't be high in patients especially in whom the tumor has a low invasion inside the cranium. A mean blood transfusion in stages IA-IIC (890 cc) is acceptable, compared to 1000-1500cc in previous studies [14]. The role of embolization: In this study, a significant decrease of hemorrhage level (45.5%) in embolized cases is presented, when compared to the mean hemorrhage level of the remaining 42 cases (770cc vs. 1403.6cc). It also shows a 30.3% decrease when compared to the mean hemorrhage level of the 38 non-embolized patients up to stage IIC, i.e. 1104cc. The average intraoperative hemorrhage level in endoscopic surgeries performed on embolized patients is reported less than 770cc, i.e. 350cc and 372cc, in previous studies [11, 12]. However, embolizing patients have both less hospitalization days (1.8 days vs. 2.2 days) and less postoperative hemorrhage rate (0% vs.11.9%), significantly. Cranial and peripheral nerve injury: Among our series of patients, one case (Stage IIIA) of a serious damage to the cranial nerves II, III, and VI, two facial numbness (4.2%) and, two cases (4.2%) of decreased lacrimation were observed. However, in a long-term study on 44 JNA patients with open surgery for 11 years [15], one case
(2.3%) of meningitis, one case (2.3%) of amaurosis fugax were seen as early complications, and 5 cases (11.4%) of infraorbital nerve sensory dysfunction, 4 cases (9.1%) of lacrimal duct stenosis, 2 cases (4.5%) of secretory otitis that needed the application of VT, and 2 cases (4.5%) of diplopia that needed the manipulation of the floor of the orbit, as late complications. Dural injury and brain abscess: In this study, no cases of dural or meningeal injury, no cases of mucocele or brain abscess were detected. In previous studies, sphenoid mucocel has been one of the complications of endoscopic surgery, also [16]. One of the minor complications of all endoscopic surgeries is synechia, which occurs due to irritation and abrasion of the mucosa of the nasal cavity and fusion of its two face-to- face injured walls. In this study, there were 13 cases (27%) that experienced synechia, and recovered with endoscopic managements. Cavernous sinus injury: In our series of patients, 2 cases (4.3%) (Stages IIIA and IIIB) experienced a cavernous sinus damage (Fig.3). The operation was completed successfully, and both of the patients are alive. JNAs are tumors with a predilection for spread but rarely invade dura, acting instead to displace it. It is believed that surgery is the method of choice for treating these lesions and that an anterior surgical approach with microsurgical techniques should be used in the first instance [17]. We believe that the endoscopic approach to this stage of angiofibroma still is the preferred way because under the endoscopic magnification of the surgical field, meticulous dissection of tumor can be performed. Under this condition, if the surgeon decides to resect tumor completely from the cavernous sinus, in any way, with or without endoscope, he or she must ready to overcome a massive hemorrhage, so sufficient blood for transfusion must prepared. Recurrence of angipofibroma after endoscopic surgery: During the follow-up period, 9 patients (19.1%) experienced a relapse. The mean interval between the surgery and the relapse was 17 months. This percentage was 7% and 39.5% [16], in other studies. Comparison between the primary and secondary treatment of JNA: As mentioned before, 31 (66%) patients without a previous history of surgery and 16 (34%) patients with a history of open surgery which had led to relapse, underwent an endoscopic surgery. There is no valuable difference of the recurrence rates of JNA in the two groups: 19.3% in the primary treated patients vs. 18.7% in the secondary treated patients. There is a significant difference of the postoperative hemorrhage between the two groups (Table 1) with p= 0.027 in favour of less blood loss in the primary treated group. There is another significant difference in the hospitalization period between the two groups (Table 2) with p= 0.049 in favour of a shorter hospital stay of the primary treated group. Comparing endoscopic surgery versus open surgery of JNA Pryor SG, publish an article in July 2005 by the title of "Endoscopic versus Conventional Approaches of Resecting Juvenile Nasopharyngeal Angiofibroma" [18].Among the Pryor’s open surgeries, there were 34 cases (57.6%) with a need for blood transfusion ;7 cases (11.8%) of diplopia, 11 cases (18.6%) of facial paresthesia, 9 cases (15.25%) of lacrimal duct obstruction, 3 cases (5.1%) of serous otitis media, and 2 cases (3.4%) of wound infection, as the complications of the open surgery. There were 14(23.7%) cases of recurrence in this group of patients. The mean hospitalization days seem to be 3.1 days in our experience, versus 5.25 days in open surgery follow-up. The overall mean hemorrhage level was 1336.2 cc in endoscopic surgeries, and 1486.5cc in open surgeries. There was one case (2.1%) of injury to the cranial nerves II, III, and VI, while there were 7 cases (11.8%) of diplopia in open surgeries, probably due to injury to cranial nerve III, IV, VI. No case of serous otitis media was detected in endoscopic surgery; whereas 3 such cases (5.1%) were seen in
[14]. Onerci TM, Yucel OT, Ogretmenoglu O. Endoscopic surgery in treatment of juvenile nasopharyngeal angiofibroma_. Int J Pediatr Otorhinolaryngol_ 2003; 63 : 1219-1225. [15]. Herman P, Lot G, Chapot R, Salvan D, Huy PT. Long term follow-up of juvenile nasopharyngeal angiofibromas: analysis of recurrences. Laryngoscope. 1999; 109 : 140–147. [16]. Nicolai P, Berlucchi M, Tomenzoli D, Cappiello J, Trimarchi M, Maroldi R, et al.Endoscopic surgery for juvenile angiofibroma: When and how. Laryngoscope 2003; 113 : 775– 782. [17]. Danesi G, Panizza B, Mazzoni A, Calabrese V.Anterior approaches in juvenile nasopharyngeal angiofibromas with intracranial extension. Otolaryngol Head Neck Surg. 2000; 122 : 277–283. [18]. Pryor SG, Moore EJ, Kasperbauer JL. Endoscopic versus traditional approaches for excision of juvenile nasopharyngeal angiofibroma. Laryngoscope. 2005; 115 :1201-1207.
