Endoscopic excision of juvenile nasopharyngeal angiofibroma A case series Egyptian Journal of Ear, Nose, Throat and Allied Sciences xxx (2016) xxx–xxx Contents lists available at ScienceDirect Egyptia[.]
Egyptian Journal of Ear, Nose, Throat and Allied Sciences xxx (2016) xxx–xxx Contents lists available at ScienceDirect Egyptian Journal of Ear, Nose, Throat and Allied Sciences journal homepage: www.ejentas.com Case report Endoscopic excision of juvenile nasopharyngeal angiofibroma: A case series L.Y Lim a,c,⇑, Irfan Mohamad a, I.P Tang b,c a Department of Otorhinolaryngology-Head & Neck Surgery, School of Medical Sciences, Universiti Sains Malaysia Health Campus, Kota Bharu, Kelantan, Malaysia Department of ORL-HNS, Faculty of Medicine, Universiti Malaysia Sarawak, Sarawak, Malaysia c Sarawak General Hospital, Kuching, Sarawak, Malaysia b a r t i c l e i n f o Article history: Received 27 November 2016 Accepted 12 December 2016 Available online xxxx Keywords: Angiofibroma Endoscopy Nasal obstruction Retrospective studies a b s t r a c t Objectives: To present the outcome of excision of juvenile nasopharyngeal angiofibroma (JNA)-through endoscopic approach Method: Retrospective case series review of six cases of JNA patients, encountered between 2013 and 2015 All patients underwent endoscopic excision, preceded by embolisation Results: All patients were male, comprised of Chinese, Malay and Bidayuh ethnics, presenting with typical complaints of progressive unilateral nasal blockage, spontaneous painless epistaxis hyposmia and sleep disturbance The patients’ presentation comprised of all four stages based on Fisch classification, with one recurrence case Maxillary artery was the main blood supply to the lesion Surgical approach applied was endoscopic four-handed technique of excision All patients were discharged well with no major complications, nor cases of recurrence Conclusion: Endoscopic approach is possible for varies stages of JNA Preoperative angiographic embolisation has minimised the blood loss intraoperatively The endoscopic method has reduced the postoperative morbidity Ó 2016 Egyptian Society of Ear, Nose, Throat and Allied Sciences Production and hosting by Elsevier B.V This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-ncnd/4.0/) Introduction Juvenile nasopharyngeal angiofibroma (JNA) is a histologically benign but clinically invasive tumour It potentiates to bleed due to its high vascularity This tumour accounts for 0.05% of all head and neck tumours, mainly targeted to male adolescence.1 The tumour often arises from posterolateral aspect of the roof of nasal cavity in the region of sphenopalatine foramen Local invasion of this tumour includes involving the nasopharynx, paranasal sinuses, pterygopalatine fossa and the infratemporal fossa Larger tumour can also involve the orbit and cavernous sinus.2 The most common JNA presentation is recurrent epistaxis with progressive nasal blockage Invasive tumours may cause facial deformities, diplopia, proptosis, headache and blindness Computer Peer review under responsibility of Egyptian Society of Ear, Nose, Throat and Allied Sciences ⇑ Corresponding author at: Department of Otorhinolaryngology-Head & Neck Surgery, School of Medical Sciences, Universiti Sains Malaysia Health Campus, 16150 Kota Bharu, Kelantan, Malaysia E-mail address: nuyil@yahoo.com (L.Y Lim) tomographic (CT) imaging is important for diagnostic, often with angiogram,3 and embolisation of feeding vessels Several staging classification has been proposed, however Fisch classification (Table 1) has been widely used for its practicality Surgical resection remains the mainstay of treatment Extensive uncontrollable blood loss is the challenge faced by surgeons Thus, pre-operative angiographic embolisation is preferred Method This is a retrospective review of six patients with JNA cases presented to our centre from 2013 till 2015 The demographic data, clinical presentations, clinical findings, imaging investigations and treatment modalities of these patients were collected and reviewed based on medical records Fisch classification was used to classify all tumours based on the patients’ CT scan reports The modality of treatment for all six patients was transnasal endoscopic approach A small lateral rhinotomy, was combined at the end of endoscopic procedure, in one stage IV case, to assist the removal of tumour en bloc Five of these cases were primary resections except one, was a case of recurrence http://dx.doi.org/10.1016/j.ejenta.2016.12.007 2090-0740/Ó 2016 Egyptian Society of Ear, Nose, Throat and Allied Sciences Production and hosting by Elsevier B.V This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/) Please cite this article in press as: Lim L.Y., et al Endoscopic excision of juvenile nasopharyngeal angiofibroma: A case series Egypt J Ear Nose Throat Allied Sci (2016), http://dx.doi.org/10.1016/j.ejenta.2016.12.007 L.Y Lim et al / Egyptian Journal of Ear, Nose, Throat and Allied Sciences xxx (2016) xxx–xxx Table Fisch staging for juvenile nasopharyngeal angiofibroma (JNA) The surgical steps techniques are illustrated in the images (Figs 1–4) Stage Details I Tumour limited to the nasopharyngeal cavity; bone destruction negligible or limited to the sphenopalatine foramen Results II Tumour invading the pterygopalatine fossa or the maxillary, ethmoid or sphenoid sinus with bone destruction There were six patients diagnosed with JNA in our centre from 2013 till 2015 All patients were male The mean age of diagnosis was 16 (range 11–29) years old Among the six patients, two patients were Malay, two were Chinese and another two were local ethnic Bidayuh Four of these patients presented with spontaneous painless intermittent epistaxis (40%), while two patients presented with profuse bleeding requiring blood transfusion (20%) Other symptoms included progressive nasal blockage (20%), reduction in sense of smell (10%) and sleep disturbance (10%) (Fig 5) Nasal endoscopy examination of nasal cavity revealed a vascular nasal mass mainly occupying the nasopharynx in all patients No nasal biopsies were taken from any of these patients Contrast-enhanced CT imaging examination was ordered Fisch classification was used to stage the patients based on CT imaging result Two patients (33.3%) were at stage I, one patient (16.7%) was stage II, two patients (33.3%) were stage III and one patient (16.7%) was stage IV (Fig 6) Four patients (66.7%) presented with JNA arising from the left while two patients (33.3%) raised from the right All patients underwent angiography imaging and showed all tumours received blood supply mainly (80–90%) from ipsilateral maxillary artery with some blood supply from the contralateral Only one tumour had sole ipsilateral blood supply The feeding vessels were embolised 24 h before surgery with no complications The average duration of operating time was 2.5 (range 1–6) hours The operating time for stage I to stage III patients have minimal difference in operating time, with average (range 1–2.5) hours The longest operating time was on the stage IV patient that took h The mean blood loss was 1000 (range 700–1500) ml Only two patients from stage III and IV needed intraoperative blood transfusion (Table 2) Nasal packing remained for an average of 2.5 (range 2–4) days after surgery Only one patient needed intensive care unit care post-operatively Average duration of hospital stay for these patients was 6.3 (range 4–10) days All patients were followed up for minimal of one year and no recurrence were noted III Tumour invading the infratemporal fossa or orbital region: (a) without intracranial involvement (b) with intracranial extradural (parasellar) involvement IV Intracranial intradural tumour: without infiltration of the cavernous sinus, pituitary fossa or optic chiasm with infiltration of the cavernous sinus, pituitary fossa or optic chiasm 2.1 Operative technique Pre-operative angiography and embolisation was planned 24 h before surgery to assess tumour vascularity and to reduce intraoperative bleeding After anaesthesia induction, the nasal cavity was packed with Moffett’s solution4 (1 ml adrenalin 1:1000, ml 10% cocaine, ml of 8.4% sodium bicarbonate with 13 ml of water) for 20 Upon removal of nasal packing, the lateral nasal wall mucosa was infiltrated with 0.5% lidocaine solution with adrenalin 1/100,000 The extent of surgery depends on the extent of tumour invasion Inferior turbinectomy, posterior septectomy and sometimes, medial maxillectomy was performed for access and to facilitate better visualisation of the surgical field The ‘two-nostril four-hand’ technique was used in all these cases In case of stage IV, the nasoseptal rescue flap or HadadBassagasiteguy flap was raised to minimise bone exposure after an extensive resection.5 Tumour debulking begun from intranasal portion and proceeded beyond sinonasal structures assisted with cutting instruments, bipolar coagulation diathermy, large bore suctions and microdebrider For our patient in stage IV with involvement of orbit, the orbital part of tumour was accessed via the pterygopalatine fossa into the infraorbital fissure A small lateral rhinotomy was done to remove the tumour en bloc After the complete resection of tumour, the nasal cavity was irrigated with warm saline and residual bleeding was further cauterised Temporary nasal packing with SurgicalÒ held on by Foley’s catheter supported by MerocelÒ was applied at the end of procedure Figure Preoperative view of JNA tumour in a case of recurrence Note the polypoidal mucosa due to previous surgery Discussion The demographic data comparing to previous retrospective studies done shows a typical presentation of JNA usually affects male, adolescent age group with average age 16 years old, consistent with previous data collected6–8 with presentation of sponta- Figure The better view of the tumour after posterior septectomy Please cite this article in press as: Lim L.Y., et al Endoscopic excision of juvenile nasopharyngeal angiofibroma: A case series Egypt J Ear Nose Throat Allied Sci (2016), http://dx.doi.org/10.1016/j.ejenta.2016.12.007 L.Y Lim et al / Egyptian Journal of Ear, Nose, Throat and Allied Sciences xxx (2016) xxx–xxx Figure The ‘‘four-hand technique” whereby the assistant assist the surgeon in suction while the surgeon excise the tumour Figure The endoscopic view after the resection of the tumour neous painless intermittent epistaxis and nasal obstruction According to our data, the patients were equally distributed among Malay, Chinese and Bidayuh However, this data cannot be used to represent the distribution of JNA among ethnicity because the sample size is too small JNA diagnosis is essentially based on gender with high index of suspicion in adolescent age group, together with clinical history, clinical presentation, nasal endoscopic examinations, and subsequently assisted with imaging examination such as CT or magnetic resonance imaging (MRI) scans Nasal biopsies should be avoided Advancement in imaging techniques such as CT and magnetic resonance imaging (MRI), has helped surgeons in determining the precise invasion of JNA lesion and the supplying blood vessels, making possible for angiographic embolisation In our study, pre-operative angiography showed five tumours were supplied by bilateral maxillary artery, 80–90% from ipsilateral side Only one tumour received blood supply solely from ipsilateral artery We noticed that the intraoperative blood loss was relatively less in tumour with ipsilateral blood supply as compare to tumour receiving blood supply bilaterally This observation corresponds to a study on 13 patients by Tang et al.6 All our patients had embolisation preceded the surgery, average duration of operative time was 2.5 h, remained unchanged as compare to data collected by Iovanescu et al.7 with no pre-operative embolisation Pre-operative embolisation is also important in reduction of intra-operative blood loss A study by Antonelli et al.9, there is a 60% reduction in blood loss if pre-operative embolisation is performed Tumour excision remains the mainstay of treatment in JNA Various methods and approaches have been used to treat JNA previously The improvements of transnasal endoscopic route have vastly advanced the limitation of endoscopic surgery previously, owing to good visualisation and navigation system by high definition camera and neuro-navigation system These advantages have allowed tumours in various stages to be excised endoscopically, as seen in all six patients in this series Tumours invading pteryopalatine fossa and infratemporal fossa, can be visualised and access after medial maxillectomy and removal of posterior wall of maxilla.10 A collaboration between ENT surgeons and neurosurgeons, tumours invading intracranially into cavernous sinus and orbital apex can be excised entirely via transnasal endoscopic route McCombe et al.11 has reported a recurrence rate for JNA as high as 30–50% while Gullane et al.12 reported 36% recurrence after first operation Endoscopic approach for JNA resection has permits less post-operative morbidity, improve visualisation compare to traditional open methods, and lower recurrence rate (0–7%).13 Wormald and Van HAsselt14 and Pryor et al.15 have reported no case of recurrence in endoscopic method Our study also shows 0% recurrence rate Post-operative morbidity has also decreased following endoscopic approach Duration of hospital stay has been decreased.7,10,13,15 However, in our study, the average stay in hospital is 6.3 days as compare to 3.3 days as reviewed by Kopec´ et al.10, is attributed to geographical structure, whereby transportation and lodging need to be considered Figure The common clinical presentations showed by our patients Please cite this article in press as: Lim L.Y., et al Endoscopic excision of juvenile nasopharyngeal angiofibroma: A case series Egypt J Ear Nose Throat Allied Sci (2016), http://dx.doi.org/10.1016/j.ejenta.2016.12.007 L.Y Lim et al / Egyptian Journal of Ear, Nose, Throat and Allied Sciences xxx (2016) xxx–xxx Figure The number of patients in each staging Table The summary of surgical outcome of our patients * Patient No Fisch Staging Operative time (mins) Blood loss (ml) Duration of hospital stay I 90 1200 I 120 700 3* II 60 200 4 III 135 1100 III 150 1500 10 IV 360 1300 Patient No is the only patient the tumour showed sole blood supply from ipsilateral maxillary artery Conclusion JNA is a benign, highly vascular tumour and the capability of recurrence has made the management challenging for surgeons The improvement of endoscopic instruments and methods has enabled advance-staged tumours being excised endoscopically Proper selection of patients to the methods of excision and a good collaboration of multidisciplinary team approach between experienced surgeon, anaesthetist and neurosurgeon is much needed for a better outcome in treatment of JNA Radiation therapy should be reserved for inoperable tumours References Batsakis JG, ed Tumours of the head and neck: clinically and pathological considerations 2nd ed Baltimore: Williams & Wilkins; 1979 296-300 Gleeson M Juvenile angiofibroma In Gilbert RW, Gleeson M, Scott-Brown WG Scott-Brown’s Otorhinolaryngology Head and Neck Surgery Hodder Arnold 2008;187:2237-45 Watkinson J, Gilbert RW Stell & Maran’s textbook of head and neck surgery and oncology CRC Press; 2011 [Dec 30] Benjamin E, Wong DK, Choa D ‘Moffett’s’ solution: a review of the evidence and scientific basis for the topical preparation of the nose Clin Otolaryngol Allied Sci 2004;29:582–587 Tang IP, Carrau RL, Otto BA, et al Technical nuances of commonly used vascularised flaps for skull base reconstruction J Laryngol Otol 2015;129:752–761 Tang IP, Shashinder S, Gopala Krishnan G, Narayanan P Juvenile nasopharyngeal angiofibroma in a tertiary centre: ten-year experience Singapore Med J 2009;50:261–264 Iovanescu G, Ruja S, Cotulbea S Juvenile nasopharyngeal angiofibroma: timisoara ENT Department’s experience Int J 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Surg 2003;129:684–691 15 Pryor SG, Moore EJ, Kasperbauer JL Endoscopic versus traditional approaches for excision of juvenile nasopharyngeal angiofibroma Laryngoscope 2005;115:1201–1207 Please cite this article in press as: Lim L.Y., et al Endoscopic excision of juvenile nasopharyngeal angiofibroma: A case series Egypt J Ear Nose Throat Allied Sci (2016), http://dx.doi.org/10.1016/j.ejenta.2016.12.007 ... disturbance (10%) (Fig 5) Nasal endoscopy examination of nasal cavity revealed a vascular nasal mass mainly occupying the nasopharynx in all patients No nasal biopsies were taken from any of these... six patients in this series Tumours invading pteryopalatine fossa and infratemporal fossa, can be visualised and access after medial maxillectomy and removal of posterior wall of maxilla.10 A collaboration... clinical presentations showed by our patients Please cite this article in press as: Lim L.Y., et al Endoscopic excision of juvenile nasopharyngeal angiofibroma: A case series Egypt J Ear Nose Throat