panduan KOAS

Juvenile Juvenile Nasopharyngeal Nasopharyngeal Angiofibroma Angiofibroma

January 3, 2007January 3, 2007

JNAJNA

OverviewOverview AnatomyAnatomy DiagnosisDiagnosis RadiologyRadiology StagingStaging TreatmenTreatmen

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OverviewOverview

JNAJNA

Benign highly vascular tumorBenign highly vascular tumor

Locally invasive, submucosal spreadLocally invasive, submucosal spread

Vascular supply most commonly Vascular supply most commonly from internal maxillary arteryfrom internal maxillary artery Also: internal carotid, external carotid, Also: internal carotid, external carotid,

common carotid, ascending pharyngealcommon carotid, ascending pharyngeal

Up to 0.5% of head and neck tumorsUp to 0.5% of head and neck tumors

Occurring almost exclusively in malesOccurring almost exclusively in males

Average age of onset = 15 years oldAverage age of onset = 15 years old

Intracranial Extension between 10-20%Intracranial Extension between 10-20%

Recurrence Rates as high as 50%Recurrence Rates as high as 50%

JNA Facts and StatisticsJNA Facts and Statistics

AnatomyAnatomy

OriginOrigin

Considered to be posterolateral Considered to be posterolateral nasal wall at sphenopalatine nasal wall at sphenopalatine foramenforamen

Blood supplyBlood supply Primarily internal maxillary artery off of Primarily internal maxillary artery off of

external carotidexternal carotid

OriginOrigin Posterolateral nasal wall near Posterolateral nasal wall near

sphenopalatine foramensphenopalatine foramen

Routes of SpreadRoutes of Spread

Medial growthMedial growth Nasal cavityNasal cavity NasopharynxNasopharynx

Lateral growthLateral growth Pterygopalatine fossaPterygopalatine fossa

Vertical expansion through inferior orbital fissure to orbit Vertical expansion through inferior orbital fissure to orbit possiblepossible

Infratemporal fossaInfratemporal fossa Superior expansion through pterygoid process may involve Superior expansion through pterygoid process may involve

middle cranial fossamiddle cranial fossa Lateral and posterior walls of sphenoid sinus can be Lateral and posterior walls of sphenoid sinus can be

erodederoded Cavernous sinus may be involvedCavernous sinus may be involved Pituitary may be involvedPituitary may be involved

Sphenopalatine ForamenSphenopalatine Foramen

Sphenopalatine vesselsSphenopalatine vessels

NervesNerves NasopalatineNasopalatine Posterior superior nasalPosterior superior nasal

HistologyHistology Myofibroblast is cell of originMyofibroblast is cell of origin Fibrous connective tissue with abundant Fibrous connective tissue with abundant

endothelium-lined vascular spacesendothelium-lined vascular spaces Pseudocapsule of fibrous tissuePseudocapsule of fibrous tissue Blood vessels lack a complete muscular Blood vessels lack a complete muscular

layerlayer

DiagnosisDiagnosis

Midface and Anterior Skull Midface and Anterior Skull Base TumorsBase Tumors

Juvenile Nasopharyngeal Juvenile Nasopharyngeal AngiofibromaAngiofibroma

OsteomaOsteoma CraniopharyngiomaCraniopharyngioma Olfactory NeuroblastomaOlfactory Neuroblastoma ChordomaChordoma ChondrosarcomaChondrosarcoma RhabdomyosarcomaRhabdomyosarcoma Nasopharyngeal CarcinomaNasopharyngeal Carcinoma

DiagnosisDiagnosis

HistoryHistory Physical ExamPhysical Exam Radiological studyRadiological study

CT ScanCT Scan MRIMRI AngiogramAngiogram

Characteristic Characteristic PresentationPresentation

Teenage or young adult maleTeenage or young adult male

Recurrent epistaxisRecurrent epistaxis

Nasal obstructionNasal obstruction

Additional Findings at Additional Findings at PresentationPresentation

Conductive hearing lossConductive hearing loss RhinolaliaRhinolalia Hyposmia/AnosmiaHyposmia/Anosmia Swelling of cheekSwelling of cheek DacrocystitisDacrocystitis Deformity of hard and/or soft palateDeformity of hard and/or soft palate Orbital proptosisOrbital proptosis

AppearanceAppearance

Smooth lobulated mass in the Smooth lobulated mass in the nasopharynx or lateral nasal wall nasopharynx or lateral nasal wall

Pale, purplish, red-gray, or beefy redPale, purplish, red-gray, or beefy red

Compressible Compressible

RadiologyRadiology

Radiological StudiesRadiological Studies CT ScanCT Scan

Excellent for bone detailExcellent for bone detail Lesion enhances with contrast on CTLesion enhances with contrast on CT

MRIMRI Differentiate tumor from other soft tissue structuresDifferentiate tumor from other soft tissue structures

AngiogramAngiogram Evaluation of feeding blood vesselsEvaluation of feeding blood vessels

Holman-Miller SignHolman-Miller SignCharacteristic anterior bowing of posterior maxillary Characteristic anterior bowing of posterior maxillary

wallwall

Coronal CT: Bone Coronal CT: Bone WindowWindow

Widening of left Widening of left sphenopalatine sphenopalatine foramenforamen

Lesion fills left Lesion fills left choanae choanae

Extends into Extends into sphenoid sinussphenoid sinus

Axial CT: Soft Tissue Axial CT: Soft Tissue Window with ContrastWindow with Contrast

Homogenous Homogenous enhancementenhancement

Widening of left Widening of left sphenopalatine sphenopalatine foramenforamen

Extension intoExtension into NasopharynxNasopharynx Pterygopalatine fossaPterygopalatine fossa

Axial CT: Soft Tissue Axial CT: Soft Tissue Window with ContrastWindow with Contrast

Homogenous Homogenous enhancementenhancement

Widening of right Widening of right sphenopalatine sphenopalatine foramenforamen


Axial MRI: T1Axial MRI: T1

Heterogeneous Heterogeneous intermediate signalintermediate signal

Flow voids Flow voids represent enlarged represent enlarged vesselsvessels

Extension intoExtension into NasopharynxNasopharynx Masticator spaceMasticator space

Coronal MRI: T1 with Coronal MRI: T1 with ContrastContrast

Diffuse intense Diffuse intense enhancementenhancement

Multiple flow voids Multiple flow voids within within hypervascular masshypervascular mass

Extension intoExtension into NasopharynxNasopharynx Pterygopalatine Pterygopalatine

fossafossa

Axial MRI: T2Axial MRI: T2 Heterogeneous Heterogeneous

intermediate to high intermediate to high signal enhancementsignal enhancement

Multiple flow voids Multiple flow voids within hypervascular within hypervascular massmass


External Carotid External Carotid ArteriogramArteriogram

Feeding vessel = Internal Maxillary Artery

Radkowski Nasopharyngeal Radkowski Nasopharyngeal Angiofibroma Staging Angiofibroma Staging

SystemSystem

Radkowski et al. Arch. Radkowski et al. Arch. Otolaryngology, 1996.Otolaryngology, 1996.

Treatment OptionsTreatment Options

SurgerySurgery Gold standardGold standard

Radiation therapyRadiation therapy Reserved for unresectable, life-threatening Reserved for unresectable, life-threatening

tumorstumors ChemotherapyChemotherapy

Recurrent tumors with previous surgery and Recurrent tumors with previous surgery and radiationradiation

Hormone therapyHormone therapy Estrogens and antiandrogens used to decrease Estrogens and antiandrogens used to decrease

tumor size and vascularitytumor size and vascularity

Surgical ApproachesSurgical Approaches

Endoscopic transnasalEndoscopic transnasal TranspalatalTranspalatal Denker approachDenker approach Facial translocationFacial translocation Medial maxillectomyMedial maxillectomy Infratemporal fossa with or Infratemporal fossa with or

without craniotomywithout craniotomy

Preoperative Preoperative EmbolizationEmbolization

24 to 72 hours preoperatively24 to 72 hours preoperatively Gelfoam or polyvinyl alcohol foamGelfoam or polyvinyl alcohol foam

Gelfoam: resorbed in approximately 2 weeksGelfoam: resorbed in approximately 2 weeks Polyvinyl alcohol: more permanentPolyvinyl alcohol: more permanent

EfficacyEfficacy Stage I patients reduced from 840cc to 275cc Stage I patients reduced from 840cc to 275cc

blood lossblood loss ComplicationsComplications

Brain and ophthalmic artery embolizationBrain and ophthalmic artery embolization Facial nerve palsyFacial nerve palsy Skin and soft tissue necrosisSkin and soft tissue necrosis

Liu L et al. Analysis of intra-operative bleeding and recurrence of juvenile nasopharyngeal Liu L et al. Analysis of intra-operative bleeding and recurrence of juvenile nasopharyngeal angiofibromas. Clin Otolaryngol. 2002angiofibromas. Clin Otolaryngol. 2002

EmbolizationEmbolization

EmbolizationEmbolization




Endoscopic TransnasalEndoscopic Transnasal

Middle turbinectomy may be performed Middle turbinectomy may be performed for improved exposurefor improved exposure


Middle meatus antrostomyMiddle meatus antrostomy Resection of posterior maxillary wallResection of posterior maxillary wall


Sphenopalatine artery ligationSphenopalatine artery ligation Tumor resection from pterygopalatine fossaTumor resection from pterygopalatine fossa




TranspalatalTranspalatal

Soft palate is split and retractedSoft palate is split and retracted


Hard palate resection for enhanced exposureHard palate resection for enhanced exposure


Palatine bone and inferior aspect of pterygoid Palatine bone and inferior aspect of pterygoid plate resectedplate resected




Denker ApproachDenker Approach

Wide anterior antrostomyWide anterior antrostomy Removal of ascending process of maxillaRemoval of ascending process of maxilla Removal of inferior half of lateral nasal Removal of inferior half of lateral nasal

wallwall




Midface Degloving with Midface Degloving with Maxillary OsteotomiesMaxillary Osteotomies

Gingivobuccal incisionGingivobuccal incision Nasal intercartilaginous incisions with Nasal intercartilaginous incisions with

transfixion incisiontransfixion incision


Soft tissue elevationSoft tissue elevation


Le Fort I osteotemiesLe Fort I osteotemies




MaxillectomyMaxillectomy

Maxillary osteotomiesMaxillary osteotomies Sagittal osteotomySagittal osteotomy

MaxillectomyMaxillectomy

Alternative Approaches to Alternative Approaches to Nasal Cavities and Nasal Cavities and Paranasal SinusesParanasal Sinuses

Lateral RhinotomyLateral Rhinotomy Weber-Ferguson incisionWeber-Ferguson incision Weber-Ferguson with Lynch Weber-Ferguson with Lynch

extensionextension Weber-Ferguson with lateral Weber-Ferguson with lateral

subciliary extensionsubciliary extension Weber-Ferguson with subciliary Weber-Ferguson with subciliary

extension and supraciliary extensionextension and supraciliary extension




Infratemporal Fossa with or Infratemporal Fossa with or without Craniotomywithout Craniotomy

Choosing the Surgical Choosing the Surgical ApproachApproach

Retrospective chart review of Retrospective chart review of surgical intervention- 37 patientssurgical intervention- 37 patients

Staged using CT scan and/or MRIStaged using CT scan and/or MRI Follow-up CT scan or MRI: 3 Follow-up CT scan or MRI: 3

months, 6 months x 3 years, yearlymonths, 6 months x 3 years, yearly Recurrence rate = 27%Recurrence rate = 27%

Hosseini et al. Eur Arch Hosseini et al. Eur Arch Otorhinolaryngol. 2005.Otorhinolaryngol. 2005.

Surgical PlanningSurgical Planning Smaller tumors (IA, IB, IIA, IIB, IIC)Smaller tumors (IA, IB, IIA, IIB, IIC)

Trans-nasal endoscopicTrans-nasal endoscopic TranspalatalTranspalatal Transantral: lesions extending laterally up Transantral: lesions extending laterally up

to pterygopalatine fossato pterygopalatine fossa Larger tumors (IIIA, IIIB)Larger tumors (IIIA, IIIB)

Lateral rhinotomyLateral rhinotomy Midfacial deglovingMidfacial degloving

Extensive resection with higher morbidityExtensive resection with higher morbidity

Limited resection with higher recurrenceLimited resection with higher recurrence

Hosseini et al. Eur Arch Otorhinolaryngol, Hosseini et al. Eur Arch Otorhinolaryngol, 2005.2005.

Changing TechniqueChanging Technique

Retrospective chart review of Retrospective chart review of surgical intervention- 30 patientssurgical intervention- 30 patients

Marked shift towards endonasal Marked shift towards endonasal procedures while tumor stages procedures while tumor stages remained the sameremained the same

Endonasal approach contraindicated Endonasal approach contraindicated in Stage IV and some Stage III casesin Stage IV and some Stage III cases May be used in conjunction with other May be used in conjunction with other

approach in these casesapproach in these casesMann et al. Laryngoscope. Mann et al. Laryngoscope. 2004.2004.

Surgical ApproachSurgical Approach

Mann et al. Laryngoscope. 2004.Mann et al. Laryngoscope. 2004.

Pryor et al. Laryngoscope. 2005.Pryor et al. Laryngoscope. 2005.

Surgical TechniqueSurgical TechniqueApproach (65 Approach (65 pts)pts)

EndoscopEndoscopicic

OpenOpen

EBLEBL 225 ml 225 ml 1250 ml1250 ml

ComplicationsComplications 11 3030

Length of StayLength of Stay 2 days2 days 5 days5 days

Recurrence Recurrence RateRate

0 %0 % 24 %24 %

Surgical TechniqueSurgical Technique Retrospective study of 24 patients using Retrospective study of 24 patients using

Radkowski staging scaleRadkowski staging scale 10 patients IA through IIA had transpalatal 10 patients IA through IIA had transpalatal

approachapproach Before 1999Before 1999

9 patients IA through IIIA had transnasal 9 patients IA through IIIA had transnasal endoscopic approachendoscopic approach After 1999After 1999

5 patients IIA through IIIA had lateral 5 patients IIA through IIIA had lateral rhinotomy or degloving approachrhinotomy or degloving approach

Recurrence in 1 case with 12-56 month Recurrence in 1 case with 12-56 month follow-up rangefollow-up range Transpalatal approachTranspalatal approach

Tosun et al. J Craniofac Tosun et al. J Craniofac Surg. 2006Surg. 2006.

Surgical TechniqueSurgical Technique

Transnasal endoscopic approach can Transnasal endoscopic approach can replace transpalatal approachreplace transpalatal approach Less morbidityLess morbidity

Patients with IIA through IIIA previously Patients with IIA through IIIA previously treated with lateral rhinotomy may be treated with lateral rhinotomy may be treated with transnasal endoscopic treated with transnasal endoscopic approachapproach

Tumors extending to infratemporal fossa Tumors extending to infratemporal fossa require lateral rhinotomy and degloving require lateral rhinotomy and degloving for optimal exposurefor optimal exposure Greater morbidityGreater morbidity

Tosun et al. J Craniofac Surg. 2006Tosun et al. J Craniofac Surg. 2006.

Surgical TechniqueSurgical Technique

Surgical limitations of endoscopic Surgical limitations of endoscopic resection evaluated in literature resection evaluated in literature review review

Extremely limited IIIA and IIIB may Extremely limited IIIA and IIIB may be approached endoscopicallybe approached endoscopically

Preoperative embolization Preoperative embolization recommendedrecommended

Unlikely that limits on endoscopic Unlikely that limits on endoscopic resection of JNA have been reachedresection of JNA have been reached

Douglas et al. Curr Opin Otolaryngol Head Neck Surg. 2006.

Gamma Knife SurgeryGamma Knife Surgery

2 case reports used as booster 2 case reports used as booster treatment for residual tumor after treatment for residual tumor after surgerysurgery No change in tumor size of one patient, No change in tumor size of one patient,

regression in other patientregression in other patient

1 case report used as primary 1 case report used as primary treatment modality successfullytreatment modality successfully

Dare et al. Neurosurgery. 2003.

Park et al. J Korean Med Sci. 2006.

External Beam RadiationExternal Beam Radiation

Retrospective review of efficacy of Retrospective review of efficacy of radiation as primary treatment radiation as primary treatment modality for JNAmodality for JNA

15 patients received 3000-3500 cGy15 patients received 3000-3500 cGy Recurrence rate of 15%Recurrence rate of 15% External beam radiation is effective External beam radiation is effective

mode of treatment of advanced JNAmode of treatment of advanced JNA

Reddy et al. Am J Otolaryngol. 2001.

External Beam RadiationExternal Beam Radiation

Retrospective review of efficacy of Retrospective review of efficacy of radiation as primary treatment radiation as primary treatment modality for JNAmodality for JNA

27 patients received 3000-5500 cGy27 patients received 3000-5500 cGy Recurrence rate of 15% 2-5 years Recurrence rate of 15% 2-5 years

post-treatmentpost-treatment External beam radiation is effective External beam radiation is effective

mode of treatment of advanced JNAmode of treatment of advanced JNA

Lee JT et al. Laryngoscope. 2002.

External Beam RadiationExternal Beam Radiation Long-term sequelae of concernLong-term sequelae of concern

Growth retardation, panhypopituitarism, temporal Growth retardation, panhypopituitarism, temporal lobe necrosis, cataracts, radiation keratopathylobe necrosis, cataracts, radiation keratopathy

Retrospective review reported 2 cases out of Retrospective review reported 2 cases out of 55 patients developing secondary 55 patients developing secondary malignanciesmalignancies Thyroid carcinoma 13 years after receiving Thyroid carcinoma 13 years after receiving

3500cGy3500cGy Basal cell carcinoma of skin 14 years after Basal cell carcinoma of skin 14 years after

receiving 3500cGy initially, then 3000cGy for receiving 3500cGy initially, then 3000cGy for recurrencerecurrence

Cummings et al. Laryngoscope 1984.

ChemotherapyChemotherapy

Chemotherapy alternative therapyChemotherapy alternative therapy 1 unresectable tumor had 1 unresectable tumor had

chemotherapy for palliationchemotherapy for palliation Adriamycin and decarbazineAdriamycin and decarbazine Extensive regression of tumorExtensive regression of tumor Possible alternative to radiation?Possible alternative to radiation?

Shick et al. HNO. HNO. 1996.1996.

Hormonal TherapyHormonal Therapy

Estrogen, progesterone, and Estrogen, progesterone, and androgen receptors have been androgen receptors have been identified with varying frequencies in identified with varying frequencies in JNAsJNAs Some JNAs lack these receptorsSome JNAs lack these receptors

Limited utilityLimited utility Delays surgeryDelays surgery Feminizing side effectsFeminizing side effects Cardiovascular complicationsCardiovascular complications

Hormonal TherapyHormonal Therapy

Efficacy of treatment with flutamide Efficacy of treatment with flutamide evaluated in 7 patientsevaluated in 7 patients

Before and after measurement Before and after measurement comparison made using CT scancomparison made using CT scan No statistically significant difference in No statistically significant difference in

sizesize No difference in blood lossNo difference in blood loss

No advantage with treatmentNo advantage with treatment

Labra A et al. Otolaryngol Head Neck Surg. 2004.

SurveillanceSurveillance

Frequent physical examinationsFrequent physical examinations

CT Scan / MRICT Scan / MRI

Recurrence RatesRecurrence Rates

Post-operativePost-operative Stage I and II = 7%Stage I and II = 7% Stage III = 39.5%Stage III = 39.5%

Tumor stage – extracranial vs. Tumor stage – extracranial vs. intracranial tumorintracranial tumor Extracranial = 5%Extracranial = 5% Intracranial = 50%Intracranial = 50%

Herman F et al. Laryngoscope 1999.

Bremer JW et al. Laryngoscope 1986.

ConclusionsConclusions

Rare, benign, vascular tumor found Rare, benign, vascular tumor found almost exclusively in young malesalmost exclusively in young males

Surgery is the gold standard with a Surgery is the gold standard with a trend towards endoscopic approachestrend towards endoscopic approaches

Frequent follow-up after treatment is Frequent follow-up after treatment is necessarynecessary

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Laryngoscope 1986; 96: 1321-1329.Laryngoscope 1986; 96: 1321-1329. Cansiz H, Guvenc MG, Sekecioglu N. Surgical approaches to juvenile nasopharyngeal angiofibroma. J Craniomaxillofac Cansiz H, Guvenc MG, Sekecioglu N. Surgical approaches to juvenile nasopharyngeal angiofibroma. J Craniomaxillofac

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