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ARTICLE IN PRESS

+Model BrazJOtorhinolaryngol.2016;xxx(xx):xxx---xxx www.bjorl.org

Brazilian

Journal

of

OTORHINOLARYNGOLOGY

CASE

REPORT

Parotid

Masson’s

tumor:

case

report

Tumor

de

Masson

na

parótida:

relato

de

caso

Filippo

Carta

a,

,

Sara

Sionis

a

,

Valeria

Ledda

a

,

Clara

Gerosa

b

,

Roberto

Puxeddu

a

aDepartmentofOtorhinolaryngology,PoliclinicoD.Casula,AOU-UniversityofCagliari,Monserrato,Italy bDepartmentofPathology,P.O.SGiovannidiDio,AOU-UniversityofCagliari,Cagliari,Italy

Received5October2015;accepted8January2016

Introduction

Masson’stumor,alsoknownas‘Masson’svegetated intravas-cularhemangioendothelioma’,isapapillaryhyperplasiaof the endothelial vascular cells that generally develops in medium-sizedveins,butmayalsobeobservedinall-sized veinsand,lessfrequently,inarteries.1PierreMassonin1923

firstdescribedthistumor ininflamed hemorrhoidalplexus

andexplaineditspathogenesisasaprimaryendothelial

pro-liferation of endothelial cells into the vessel lumen due

to obstructive thrombosis, followed by degeneration and

necrosis.2Currently,itisconsideredareactivevascular

pro-liferationsecondarytovascularstasis3thatcandevelopin

tendons, head andneck, skin,intracranial, aero-digestive

tract, intra-abdominal areas, genital tract, and fallopian

canal;itisgenerallyendovascular,butPinsetal.4reported

13patientspresentingwitharareextravascularform.Corio

etal.in19825reported14casesofheadandneckMasson’s

tumor,onlyonefoundintheparotidgland;clinicalfeatures

werenotdetailed.Tothebestofourknowledge,thisisthe

first well documented reported case of a Masson’stumor

of theparotid gland.The authorswant ENTsurgeons and

pathologiststobeawareofthisentity.

Pleasecitethisarticleas:CartaF,SionisS,LeddaV,GerosaC,

PuxedduR.ParotidMasson’stumor:casereport.BrazJ Otorhino-laryngol.2016.http://dx.doi.org/10.1016/j.bjorl.2016.01.003

Correspondingauthor.

E-mail:filippocarta@unica.it(F.Carta).

Case

report

Thepublicationofclinicaldatawithrespectofthelaws

con-cerningprivacyandwasapprovedbytheEthicsCommittee

oftheUniversityHospital,andthepatientconsentedtothe

publicationoftheclinicaldataregardingthiscasereport.

A43year-oldwomanwasreferredtoourDepartment

pre-sentingwitha4yearshistoryofrightparotidswelling.The

tumorwassolidinconsistency,hadslowlyincreasedinsize

until2cminitsgreatestdimensionandwasmildlypainful.

Therestoftheclinicalexaminationwasunremarkable.

Contrast-enhanced magnetic resonance imaging (MRI)

showedamultilobulatedlesionofapproximately2.3cmin

diameterwithinthe superficiallobe of theparotid gland.

Thelesionwaselongatedandcomposedoftubular,tortuous

andectasicstructureswithsuggestive characteristics ofa

hypervascularizedbenignlesion(Fig.1).

Fineneedleaspirationcytologywasnotperformed.The

patientunderwentasubtotalparotidectomyundergeneral

anesthesia.The masswasresectedwithinthesovraneural

gland,preserving the facialnerve. The removal was

per-formed under microscopic view (Carl Zeiss® --- Germany)

withafocallengthof250mmandcontinuousintraoperative

facialnervemonitoring(N.I.M.Response3.0,Medtronic®

---UnitedStates).Thedeeperaspectofthemasswasstrictly

adheringtotheperipheralzygomaticbranchesofthefacial

nerveand required an extremelyprecise microscopic

dis-sectiontoavoidaccidentalruptureofthelesionandmajor

bleeding.Histologicalexaminationshowedamajorsalivary

gland (5cm×3cm×1cm) with a wide vascular cavity,

http://dx.doi.org/10.1016/j.bjorl.2016.01.003

1808-8694/©2016Associac¸ãoBrasileiradeOtorrinolaringologiaeCirurgiaCérvico-Facial.PublishedbyElsevierEditoraLtda.Thisisanopen accessarticleundertheCCBYlicense(http://creativecommons.org/licenses/by/4.0/).

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ARTICLE IN PRESS

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2 CartaFetal.

Figure1 (a)T2-weightedaxialsequenceshowsheterogeneoushypersignalintensity,duetothepresenceofareasofmarkedlow signalintensity.(b)T1-weightedFAT-SATaxialsequenceaftermediumcontrastintakeshowsanirregularenhancement,sincethe mediumcontrastuptakewasabsentinthecentralandinthecysticareas,butitwashigherinthesolidperipheralcomponentof thetumor.

a

b

Papillary proliferation

Figure 2 (a) Microscopic examination showed an organiz-ing thrombus (arrow). (b) Microscopic examination showed intravascularpapillaryproliferationlyingofendothelialcells.

containing an organizing thrombus and a small papilla

protruding into the lumen composed of a single layer of

endothelial cells; these cells appeared swollen, without

significant pleomorphicor mitotic figuresand surrounding

acorerichincollagen(Fig.2).Thesefindingsallowedthe

diagnosis of Masson’s tumor. Postoperative outcome was

uneventfulandthepatientwasdischargedthreedaysafter

the procedure. After30 monthsthe patientwaswell and

free from tumor-related symptoms, with an intact facial

nervefunction.

Discussion

Masson’stumoris an unusualformoforganizingthrombus

withexcessivepapillaryproliferationofendothelialcellsin

normalbloodvesselsorvascularmalformations.1 Although

thecasereportedwasnotrelatedtoanypredisposingfactor,

intheliteraturethislesionisusuallyrelatedtoirradiation

andchronictrauma,thatmaychangethevenousandarterial

flow3causingpapillaryendothelialproliferation.Thereisno

agepredilection(rangingfrom9monthsto80yearsold)and

itoccursequallyinmalesandfemales,butChangin20126

suggestedafemalepredilectionmainlyduetothetumor’s

responsetoestrogensandprogesterone.

Masson’s tumors are classified in three types: a pure

or primary form that arises de novo in dilated vascular

spaces,beingthemostfrequenttype(56%ofcases),amixed

type(secondaryorreactive)duetofocalchangesina

pre-existingvascularlesion(haemangioma,pyogenicgranuloma

orvascularmalformation)and,rarely,athirdtyperesulting

fromtheorganizationofahematomainan extravascular

location.4

Head and neck Masson’s tumors are found more

fre-quently inthe lateralregionofthe neck,wherethe most

commonsymptomisatenderorsolidslow-growingswelling

thatcanrapidlyincreaseinsize,duetothebleedinginside

thelesion.3Anotherfrequentsiteofthislesionisthe

sino-nasaltract,presentingwithrecurrentepistaxis.7

Parotid Masson’s tumors are extremely rare: In 1982,

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ARTICLE IN PRESS

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ParotidMasson’stumor 3

but noclinical or therapeutic data weredetailed in their

description5;Narwaletal.8describedin2013anintra-oral

Masson’stumorthatcouldbeoriginatedfromaminor

sali-varyglandbutnotfromtheparotid.

Differential diagnosisincludes allbenignandmalignant

soft-tissuetumorslocatedintheanatomicregionwherethe

Masson’stumordevelops;thecasereportedpresentedmore

similarlytotypicalbenignparotidtumors.

Radiologic diagnosis can be challenging: MRI is

equiv-ocal in the diagnosis because of the amount of blood

vesselsand thrombiusually present withinthe lesion and

a radiologic similarity to the low-grade angiosarcoma.5

Effectively Masson’s tumor typically appears on MRI as

minimally heterogeneous on T1-weighted images, mostly

isointensetomuscleandpostcontrastT1-weightedimages

show heterogeneous enhancement that may be

sugges-tive of malignantlesion.9 In the present case report, MRI

showedT1heterogeneoushyperintensity,T2heterogeneous

high signal intensity withcentral low signal intensity and

irregular medium contrast uptake in T1 weighted FAT-SAT

sequences,moresuggestiveofabenignhypervascularized

lesion.

Parotid Masson’stumor is aslow-growing benignlesion

adhering to the surrounding salivary gland parenchyma;

therefore the surgical removal throughsub-total or total

parotidectomyis consideredthe goldstandard treatment.

In ourcase the main challengewasdue tothe

inflamma-torybehaviorofthelesion,requiringafinedissectionofthe

branchesofthenerve;theuseoftheoperativemicroscope

associatedwiththeintraoperativenervemonitoringallowed

theanatomicpreservationofallthebranchesofthefacial

nerve,avoidingpostoperativeweaknessofthenerve.

Pathological analysis of the case demonstrated an

intravascular papillary growth composed of endothelial

hyperplasiaandacentralthromboticarea(Fig.2).

Endothe-lial proliferation was suspicious for angiosarcoma, but,

according to the specific diagnostic features reported by

Sarodeetal.,10 theproliferativeprocessentirelyconfined

totheintravascularspace, theregularaspect ofthecells

oftheendotheliallayer,andtheabsenceofnecrosisand/or

cellularatypiaintheendothelialproliferationallowedthe

diagnosisofMasson’stumor.

The prognosis of Masson’s tumor is excellent, with

extremelylowrecurrencerateaftercompleteexcision.3

Conflicts

of

interest

Theauthorsdeclarenoconflictsofinterest.

Acknowledgements

The authorgratefully acknowledges the Sardinia Regional

Governmentforthefinancialsupport(P.O.R.SardegnaF.S.E.

OperationalProgrammeof the AutonomousRegionof

Sar-dinia, European Social Fund 2007---2013 --- Axis IV Human

Resources,Objectivel.3,Line ofActivityl.3.1)‘‘Avvisodi

chiamataperilfinanziamentodiAssegnidiRicerca’’.

References

1.ElHusseiny M,Benhaiem N,VayssairatM,AllaireE.Masson’s

vegetant intravascular hemangioendothelioma inanexternal

jugularveinaneurysmwithrecurrentthrombosis.JVascSurg.

2011;53:1723---5.

2.MassonMP.AnnaAnatpathol.1923;93:517.

3.CrockerM,deSouzaR,EpaliyanageP,BodiI,DeasyN,Selway

R.Masson’stumorintherightparietallobeafterstereotactic

radiosurgeryfor cerebellarAVM:casereportandreview.Clin

NeurolNeurosurg.2007;109:811---5.

4.Pins MR, Rosenthal DI, Springfield DS, Rosenberg AE. Florid

extravascularpapillaryendothelialhyperplasia(Masson’s

pseu-doangiosarcoma) presenting as a soft tissue sarcoma. Arch

PatholLabMed.1993;17:259---63.

5.Corio RL, Brannon RB, Tarpley TM. Intravascular papillary

endothelialhyperplasiaoftheheadandneck.EarNoseThroat

J.1982;61:88---91.

6.Chang K, Barlaben A, Farley S. Masson’stumor in the ulnar

artery.JVascSurg.2012;56:223---5.

7.HoodaS,HumphreysMR,WongSW,EvansAS.Masson’s

pseudo-tumoroftheethmoidsinus---acasereport.JLaryngolOtol.

2008;122:990---2.

8.NarwalA,SenR,SinghV,GuptaA.Masson’shemangioma:arare

intraoralpresentation.ContempClinDent.2013;4:397---401.

9.Yerli H, AydinE, HaberalN, Harman A, Kaskati T, Alibek S.

Diagnosingcommon parotidtumorswithmagnetic resonance

imaging including diffusion-weighted imaging vs fine-needle

aspiration cytology: a comparative study. Dentomaxillofac

Radiol.2010;39:349---55.

10.SarodeGS,SarodeSC,KarmarkarSP.Oralintravascularpapillary

endothelialhyperplasia (Masson’stumor): areviewof

Riferimenti

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