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Intravascular papillary endothelial hyperplasia (Masson's tumor) of maxillary sinus
VORUZ, François Jean Albert, et al.
VORUZ, François Jean Albert, et al . Intravascular papillary endothelial hyperplasia (Masson's tumor) of maxillary sinus. Brazilian Journal of Otorhinolaryngology , 2020
DOI : 10.1016/j.bjorl.2020.11.007 PMID : 33349593
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http://archive-ouverte.unige.ch/unige:147406
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CASE REPORT
Intravascular papillary endothelial hyperplasia (Masson’s tumor) of maxillary sinus 夽 , 夽夽
Hiperplasia endotelial papilar intravascular (tumor de Masson) do seio maxilar
Franc ¸ois Voruz
a,∗,1, Grégoire Arnoux
b, Charles-Arnaud Serex
a,1, Claudio de Vito
b, Basile Nicolas Landis
aaGenevaUniversityHospitals,ClinicalNeurosciencesDepartment,OtorhinolaryngologyandHeadandNeckSurgery,Geneva, Switzerland
bGenevaUniversityHospitals,DiagnosticDepartment,Geneva,Switzerland
Received15September2020;accepted12November2020
Introduction
Intravascularpapillaryendothelial hyperplasia---knownas Masson’stumor---isararebenignentity.Itwasfirstdescribed in 1923 by a French pathologist under the name héman- gioendothéliome végétant intravasculaire.1 It is currently consideredareactiveintravascularproliferationthatdevel- ops either in a dilated vessel’s lumen, a hematoma or a preexisting vascular lesion.2,3 No malignant degeneration has been yet reported. We present three cases of Mas-
夽 Pleasecitethisarticleas:VoruzF,ArnouxG,SerexC-A,Vito C,LandisBN.Intravascularpapillaryendothelialhyperplasia(Mas- son’s tumor) of maxillary sinus. Braz J Otorhinolaryngol. 2020.
https://doi.org/10.1016/j.bjorl.2020.11.007
夽夽 PeerReviewundertheresponsibilityofAssociac¸ãoBrasileira deOtorrinolaringologiaeCirurgiaCérvico-Facial.
∗Correspondingauthor.
E-mail:francois.voruz@hcuge.ch(F.Voruz).
1 Franc¸ois Voruz and Charles-Arnaud Serex are the first two authors.
son’stumor ofthemaxillarysinus,whichwerealltreated withendoscopicsurgeryandinitiallyconsideredpotentially malignant.Aliterature reviewofother casesfoundinthe paranasalsinusesandnasalcavityispresented.
Case reports
1st caseA46-year-oldmanpresentedwithleftnasalobstructionand occasionalbloodyrhinorrheafor7months.Hehadahistory ofsubstitutedhypothyroidism.Nasalendoscopyrevealeda smallpolypintheleftmiddlemeatus.Computedtomogra- phy (CT)andmagnetic resonanceimaging (MRI)showeda leftmaxillarysinusfilledwithaheterogeneousfleshylesion (Fig.1A).Transnasal resectionwasproposedandrevealed an almost avascular soft tissue mass with noattachment within the maxillary sinus and surrounded by an inflam- matorymucosa.Thepostoperativecoursewasuneventful.
Histological analysis showed no signs of malignancy but spots of papillaryendothelial hyperplasia in an organized https://doi.org/10.1016/j.bjorl.2020.11.007
1808-8694/©2020Associac¸˜aoBrasileiradeOtorrinolaringologiaeCirurgiaC´ervico-Facial.PublishedbyElsevierEditoraLtda.Thisisanopen accessarticleundertheCCBY-NC-NDlicense(http://creativecommons.org/licenses/by-nc-nd/4.0/).
Figure1 (A)Patient1, coronalCT slicewithoutinjection: leftmaxillarysinus fullnesswith thickeningofsurroundingbone.
CoronalMRIsliceT2-weighted:Heterogeneoustumorinleftmaxillarysinuswithliquidretention.(B)Patient2,coronalCTslice withoutinjection: rightmaxillarysinusfullnesswith intersinusonasalwalldemineralizationfromtumorcompression,extending tothesurroundingethmoidcellsandnasalfossa.CoronalMRIsliceT2-weighted:rightmaxillarysinustumorinantrawithliquid retention.(C)Patient3,coronalCTslicewithoutinjection:partialmaxillarysinusesfullnesspredominantontherightwithintact surroundingbone.CoronalMRIsliceT2-weighted:rightantralmaxillarysinusheterogenoustumor.Thickenedmucosaintheleft maxillarysinus.
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BrazilianJournalofOtorhinolaryngologyxxx(xxxx)xxx---xxx Table1 Summaryofpublishedcasesofnasalintravascularendothelialpapillaryhyperplasia.
Cases Sex/Age Localization Symptoms
Stevens(1988) M/21 Rightinferiorturbinate Nasalobstruction,hyposmia,
rhinorrhea,frontalheadaches
Stern(1991) M/17 Rightmaxillarysinus,ethmoid
andnasalcavity
Frontalheadaches,cheekpain, exophthalmos
Safneck(1995) F/36 Rightinferiorturbinate Nasalobstruction,nasolabial
sulcusfullness Lancaster(1998) F/67 Leftmaxillarysinusand
ethmoid
Nasalobstruction,rhinorrhea
Moon(2000) M/35 Leftethmoid,sphenoidsinus
andsella
Decreasedvisualacuity,ptosis, exophthalmos,fronto-temporal headaches
Lombardi(2008) M/26 Leftethmoidandnasalcavity Nasalobstruction,purulent discharge,orbitalpain, epiphora
Hooda(2008) F/45 Rightethmoid Epistaxis
Wang(2009) M/42 Leftmaxillarysinus,ethmoid,
frontalsinus,andnasalcavity
Nasalobstruction,rhinorrhea, epistaxis,frontalheadaches
Tuna(2015) M/58 Leftethmoid,sphenoid,
maxillarysinus,nasalcavity, choana,pterygopalatinefossa andorbit
Nasalobstruction,epistaxis, exophthalmos
Al-Qahtani(2016) F/33 Rightpansinusal Nasalobstruction,epistaxis,
rhinorrhea,anosmia,frontal headaches,exophthalmos
Presentcase1 M/46 Leftmaxillarysinus Nasalobstruction,
bloody-serousrhinorrhea Presentcase2 M/76 Rightmaxillarysinusandnasal
cavity
Epistaxis,rhinorrhea
Presentcase3 F/33 Rightmaxillarysinus Rhinorrhea,orbitalpressure,
headaches
hematoma. The patient remained asymptomatic, withno endoscopicsignofrecurrence3yearsaftertheintervention.
2ndcase
A 76-year-old man consulted several times for repeated right-sided epistaxis associated with posterior rhinorrhea andcoughfor5months.Hehadahistoryofischemicheart disease, high blood pressure,high cholesterol, gout,obe- sity and active smoking(30 pack-years). Nasal endoscopy revealed a purplish translucent fleshy mass in the right nasal fossa, emerging from middle meatus (Fig. 2A). CT andMRI confirmed thepresence ofa heterogeneousmax- illary tissue mass extending into the nasal fossa, with partialdiffusionrestriction,associatedwithfluidretention in the surrounding ethmoid cells (Fig. 1B). Only minimal hemorrhagic material wasfound ontransnasal biopsy and a diagnostic resection was scheduled. The mass was not inserted, attached or adherent to surroundingstructures, allowingeasyandcompletetumorectomy.Adjacentmucosal andbonystructureswereintact,suggestingabenignprocess (Fig.2B).Histologicalanalysisrevealedapolypoidalteration of the respiratory mucosa withfoci of papillaryendothe- lialhyperplasiainahematomaintheprocessofadvanced organization,without malignancy(Fig. 3).The postopera-
tivefollow-upwasuncomplicated,andthepatientremained asymptomaticfromarhinologicalpointofview.Neverthe- less,due apersistentcough withhemoptysis,a CTofthe chestwasperformedanddemonstratedwhatwaslatercon- firmedasasmallcelllungcarcinoma.
3rd case
A 33-year-old woman presented right sided rhinorrhea for several years, associated with occasional ipsilateral increased intraorbital pressure and headache. She had a history of invasive ductal carcinoma with metastasis to lymph nodes and bones, treated with maintenance immunochemotherapy.Clinically,yellowishsecretionswere presentin therightmiddle meatus.MRI showeda hetero- geneoustumor in the right maxillary sinus surrounded by thickened mucosa. CT confirmed subtotal filling of right maxillary sinus with integrity of the surrounding bone (Fig.1C). In ordertoclarify a potential sinus metastasis, transnasal endoscopic resection was indicated. It showed the presence of a fleshy dark lesion enveloped in mucus without any infiltration to the surrounding mucosa. Post- operative follow-up was unremarkable. Histology showed multiple fibrohemorrhagic changes compatible with Mas- son’stumor,withoutmalignancy.
Figure2 Patient2(A),endoscopicviewoftherightnasalfossabeforeresection:whitearrow:tumor.Blackarrow:nasalseptum.
(B)Sameviewaftertotalendoscopicresection:onemustnoticetheintegrityofthemucosaandbone.Whiteasterisk:posterior wallofrightmaxillarysinus.Blackasterisk:rightmiddleturbinate.
Figure3 Patient2(A),histologicalanalysisshowingarespiratorymucosaoverhanginganold,organizedhemorrhage(framing) withevidenceofactiveresorption(siderophages).Thearrowindicatesthesurfaceepithelium.(B)Highermagnificationshowinga non-atypicalendothelialproliferationorganizedintheformofalabyrinthinenetworkandpapillarybuds.H&Ecoloring.Original magnification:(A)20×;(B)100×.
Discussion
Masson’stumorisarareentitythatmightbeunfamiliarto manyspecialists.Itmimicsmalignancyfromaclinical,endo- scopic but especiallyradiological, point of view. Sinceits first description in 1923, therehave been reported cases affectingalmostallpartsofthehumanbody,withonlyvery fewreportsinthesinonasalarea.Commonly,symptomsare not specific and related to the affected organ, with the appearanceofalumporableeding,rarelyassociatedwith pain. The radiological assessment and the biopsies often suggest malignancy.Accordingtooneof thelargestseries reported, this tumor is frequently subcutaneous and der- malwithapredilectionfortheheadandneck,digitaland trunkregions.2 Cases werealsodescribedinthe liver,the
orbit,intramuscularly,intheoralcavityandintracranially.
To thebestof ourknowledge, only 10cases, priortothe ourreportedones,involved theparanasalsinusesornasal cavity.3---12Areviewoftheexistingliteratureonthissubject issummarized in Table 1.The menversus women ratiois 1.6:1andthemeanageofpresentationis41±16yearsold.
Thesymptomsarethoseofanysinonasalpathologyanddo notshowspecificity.
Clinico-histologicalcorrelationisimportantwhendealing withthislesion. Inourexperience,thehighertheclinical suspicion,the higherthe chances tomatch proper histo- logical diagnosis. It remains speculation, but it could be thatMasson’stumorintheparanasalsinusesisperhapsless rarethan reported, due to alow clinical suspicionand a lackofcommunicationbetweenthesurgeonandthepathol-
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BrazilianJournalofOtorhinolaryngologyxxx(xxxx)xxx---xxx ogist. A similar phenomenon has been observed recently
withhamartomasoftheolfactoryclefs(REAH),13wherethe mentionof theentityin theclinical differentialdiagnosis addressedtothepathologistincreasesthenumberofcases found.
Theendoscopicaspectduringresectionmustalsoprompt thesurgeontospecificallyrequestthesearchforintravas- cular papillaryendothelial hyperplasia.Inthe threecases presented here, the clinical appearance was avascular, without the accompanying expected bleeding. The mass appeared to be poorly vascularized and easy to resect.
It was further surprising for the surgeon that the masses lacked a real attachment or insertion point to an intact mucosa,similartowhatisobservedwithremovalofafungus ball.
Radiologically,the threecasespresented similarlywith aheterogeneousmasssuggesting amalignantorat leasta solid and tissueprocess. The differentialdiagnosis for all threecaseswereinflammatory polyp,inverted papilloma, lymphoma, adenocarcinoma, squamous cell carcinoma or metastasis. Itisdifficult onthe basisofthesethreecases toconcludetowhatextentradiologycansuspectthisrare specificlesion.Ontheotherhand,itisclearthatMRIaddsa preoperativevalueincasesofunilateralopacitydiscovered onCT.Itisalsostrikingtoseethesimilarityonmagneticres- onanceimagingofallthreelesionspresentedandthecases reportedintheliterature,tothepointwhereweraisethe questionifthediagnosiscouldnotalreadybesuspectedin thebasisofMRI.
Histologically, Masson’s tumor must be differentiated fromangiosarcoma.14Histologicalanalysisshowedanorga- nizedhematomaassociatedwithnon-atypicalintraluminal endothelial proliferation, characteristic of intravascular papillary endothelial hyperplasia. A correct diagnosis will allow adequate treatment which consists of a complete resection, thus avoiding overtreatment. In our modest series,twopatients hadaconcomitant oncologicalcondi- tion,whichraises thequestionof asimplecoincidenceor of a potential cofactor promoting the formation of Mas- son’stumor.Theliterature,basedonsmallnumberofcases reported,cannotanswerthisquestion.
Conclusion
Three new cases of intravascular papillary endothelial hyperplasiaofthemaxillarysinusarepresented.Theendo- scopical, radiological and pathological characteristics are discussed. These are the 6th, 7th and 8th cases of maxil- lary sinus involvement reported in the literature. Due to itsclinicalandradiologicalpresentation,Masson’stumoris often mistaken for a malignant disease and the histology
analysisonlycanconfirmthebenigndiagnosis.Thedefinitive treatmentisthecompleteresectionofthelesion.
Conflicts of interest
Theauthorsdeclarenoconflictsofinterest.
References
1.MassonP.Hémangioendothéliomevégétantintravasculaire.Bull SocAnatParis.1923;93:517---23.
2.Hashimoto H, Daimaru Y, Enjoji M. Intravascular papillary endothelialhyperplasia.Aclinicopathologicstudyof91cases.
AmJDermatopathol.1983;5:539---46.
3.WangZH,HsinCH,ChenSY,LoCY,ChengPW.Sinonasalintravas- cularpapillaryendothelialhyperplasiasuccessfullytreatedby endoscopicexcision:acasereportandreviewoftheliterature.
AurisNasusLarynx.2009;36:363---6.
4.Stevens DJ. Papillary endothelial hyperplasia in the nose. J LaryngolOtol.1988;102:935---7.
5.SternY,BraslavskyD,SegalK,ShpitzerY,AbrahamA.Intravas- cularpapillaryendothelialhyperplasiainthemaxillary sinus.
Abenignlesionthatmaybemistakenforangiosarcoma.Arch OtolaryngolHeadNeckSurg.1991;117:1182---4.
6.Safneck JR, Alguacil-Garcia A, Dort JC. Intranasal papil- lary endothelial hyperplasia. Otolaryngol Head Neck Surg.
1995;113:766---70.
7.Lancaster JL, Alderson DJ, Sherman IW, Clark AH. Papillary endothelial hyperplasia (Masson’s tumour) of the maxillary sinus.JLaryngolOtol.1998;112:500---2.
8.MoonWS,ChungGH,HongKH.Intravascularpapillaryendothe- lialhyperplasiainavascularlesionoftheparanasalsinus.Arch PatholLabMed.2000;124:1224---7.
9.LombardiD,GaltelliC,KhraisT,MorassiML,NicolaiP.Giant hypervascularlesionofthesinonasaltractinvadingtheanterior skullbaseandorbit:apuzzlingcase.AnnOtolRhinolLaryngol.
2008;117:653---8.
10.HoodaS,HumphreysMR,WongSW,EvansAS.Masson’spseudo- tumouroftheethmoidsinus---acasereport.JLaryngolOtol.
2008;122:990---2.
11.TunaEEÜ,TürkayB,Kurukahvecio˘gluS,Atao˘gluÖ,EryılmazA.
Sinonasalintravascularpapillaryendothelialhyperplasia(Mas- son’stumor).ENTCase.2015.
12.Al-QahtaniKH.Intravascularpapillaryendothelialhyperplasia (Masson’stumor)asanasalmass:acasereportandreviewof theliterature.PanArabJRhinol.2016;6:33---5.
13.Lima NB, Jankowski R, Georgel T, Grignon B, Guillemin F, Vignaud JM. Respiratory adenomatoid hamartoma must be suspectedonCT-scanenlargementoftheolfactoryclefts.Rhi- nology.2006;44:264---9.
14.Kuo T, Sayers CP, Rosai J. Masson’s ‘‘vegetant intravascular hemangioendothelioma’’:alesionoftenmistakenforangiosar- coma:studyof seventeencaseslocated intheskinand soft tissues.Cancer.1976;38:1227---36.
