Unusual localization of glomus tumor of the knee

(1)

Pleasecitethis articleinpressas:ElHyaouiH,etal.Unusuallocalizationofglomustumoroftheknee. JointBoneSpine(2015), http://dx.doi.org/10.1016/j.jbspin.2015.07.001

ARTICLE IN PRESS

G Model

BONSOI-4197; No.ofPages3

JointBoneSpinexxx(2015)xxx–xxx

Availableonlineat

ScienceDirect

www.sciencedirect.com

Case report

Unusual localization of glomus tumor of the knee

Hicham El Hyaoui

^∗

, Abdeljabbar Messoudi , Mohamed Rafai , Abdelhak Garch

DepartmentofOrthopaedicsandTraumatology,IbnRochdUniversityHospitalCenter,1,ruedesHôpitaux,quartierdesHôpitaux,20360Casablanca, Morocco

a r t i c l e i n f o

Articlehistory:

Accepted5May2015 Availableonlinexxx

Keywords:

Glomustumor Extradigital Knee Pain

a b s t r a c t

Glomustumorisanuncommonbenignneuromyoarterialtumor.Theextradigitallocationattheknee isunusual.Ignoranceofthisdiseasecharacterizedbyatypicalclinicalsignsandtheabsenceofspeciﬁc imagingareresponsibleforasigniﬁcantdiagnosticdelayintheseformslocalizedintheknee.Complete resectionofthetumorresultsinanimmediateresolutionofthepain.Wereportthreerarelocationsofglo- mustumorinthekneewithanexceptionallocationinthequadricepstendonanddiscussepidemiological, diagnosticandtherapeuticaspectsofthesetumors.

1. Introduction

Glomustumorsarehamartomasdevelopingfromaneuromy- ovascularstructuresittingatthedermo-hypodermicjunction:the glomus.Theyarerare(1.6%ofsofttissuetumors)[1,2].

Theﬁrstdescriptionofaglomustumorwasmadein1872by Wood[3],whodescribeda“smallsubcutaneouspainfulnoduleof abenignnature”linkedin1924byMasson[4]asaglomusorigin.

Themostcommonlocationisthehandespeciallythefingers, butanatypicalanatomicallocalizationremainsrareoutsidethe fingerandusuallysmalltumorcanmakethedifficultanddelayed diagnosis.

Wereportauniquepresentationofthreeunusuallocalizations ofglomustumorintheknee.

2. Casereports 2.1. Case1

A 38-year-old man consulted with a 16-month history of exquisiteintermittentpaininhisrightknee,triggeredattheslight- estpressure.Hewasunabletotoleratethathistrousersorbed sheetstouchingtheanterioraspectofhisknee.Therewasnohis- toryoftraumaandnomechanicalsymptoms.Thesymptomswere notreducedwithmedicaltreatments.

Physicalexaminationrevealeda smallwhitishnoduleinthe anterior-uppersideofthepatella,measuring10mmindiameter,

∗ Correspondingauthor.

E-mailaddress:hicham.chu@gmail.com(H.ElHyaoui).

notattachedtodeepplanes.Itwasextremelysensitivetopalpa- tion,tothepointthatthepatientwastryingtoavoiditbecauseit triggeredverysharppain.Therangeofmotionoftheleftkneewas normalandtherestofthephysicalexaminationwasnormal.Plain radiographsofthekneeshowednopathology.Magneticresonance imagingshowedasmallsubcutaneousnodule,measuring7/3mm insize,round,well-deﬁnedandpre-patellar.Thelesionwashypo- intenseonT1-weightedimagesandhyperintenseonT2-weighted images(Fig.1).A2-cmskinincisionjustabovethemasswasper- formed.Asmallroundedmass,welldelineated,encapsulatedand purplishwasfoundinthesubcutaneoustissueandwasexcised completely.Histologicalexaminationconﬁrmedabenignglomus tumor.Postoperatively,theevolutionwasexcellentwithanimme- diateandcompleteresolutionofsymptomsandareturntodaily activities.

2.2. Case2

A40-year-oldwoman,withnoparticularmedicalhistory,con- sultedforparoxysmalpaininhisleftkneewhosebeginningdates backto14months.Shedidnotgiveanyhistoryoftrauma.

Thepainwasirregularinadiurnalcyclecausedbydirectcon- tactincludingthatofthebedsheetandbecomemoreintensefora monthpreventingthepatienttoperformdailyactivities.Thesymp- tomswerenotreducedwithconservativetreatmentsprovidedby hisdoctor(non-steroidalanti-inflammatorydrugs,analgesics).The patienthadalsoconsultedapsychiatristwhohadhimprescribe anxiolytics.Theclinicalexaminationrevealedasmallnoduleatthe anterolateralpartoftheknee,verysensitivetopalpationandmobi- lizationof theknee, firm andmobile,measuringapproximately 8mmwithoutinflammatorysignsnext.Thekneejointwasfree.

http://dx.doi.org/10.1016/j.jbspin.2015.07.001

(2)

Pleasecitethis articleinpressas:ElHyaouiH, etal.Unusuallocalizationofglomustumorof theknee. JointBoneSpine(2015), http://dx.doi.org/10.1016/j.jbspin.2015.07.001

ARTICLE IN PRESS

G Model

2 H.ElHyaouietal./JointBoneSpinexxx(2015)xxx–xxx

Fig.1.MRIshowingasmallsubcutaneousnodule,7/3mminsize,round,well-deﬁned,pre-patellar,hypo-intenseonT1-weightedimagesandhyperintenseonT2-weighted images.

Plainradiographsshowednoboneabnormalities.MRIguidedthe diagnosisbythe detectionof a very smallsubcutaneousround masslocated just forward of thepatellar tendon,hypo-intense onT1-weightedimagesandhyperintenseonT2-weightedimages withoutadjacenttissue reaction,measuring4mm,inrelatedto hypervascularprocessthatevokeaglomustumor.

Aftera longitudinal incision of 2-cm,a small whitish mass, well circumscribed had excised [Appendix A, Fig. S1; See the supplementarymaterialassociatedwiththisarticleonline].Histo- logicalanalysiswasinfavorofaglomustumor.Thepatientreported nopainpostoperatively.Afterthreeyearsfollow-up,shehadno pathologicalsymptomsorlocalrecurrence.

2.3. Case3

A22-year-oldmalewasreferredtoourorthopedicconsultation forchronicpaininthelowerendoftherightthighof4yearshistory.

Thepatientdoesnotreportspecialbackgroundorhistoryoftrauma.

Hispainwasgradualevolution,spontaneous,daytimeandnight- time,aggravatedbywalking.Hehadplainradiographsofthethigh andkneerequestedbyseveraldoctorsandinterpretedasnormal.A yearearlier,hehadconsultedanorthopedistwhohadrequestedan ultrasoundofthelowerendofthethighinsearchofamuscletear, buthadnotrevealedanyanomaliesorsofttissuemass.Thesymp- tomswerenotreducedwithconservativetreatmentsprovidedby previousphysicians(non-steroidalanti-inﬂammatorydrugs,analgesics,kneerehabilitationandphysiotherapy).

Examinationfoundapatientingoodgeneralcondition,with slightatrophyofthequadriceps,severepainintheanterioraspect ofthelowerthirdoftherightthighwelllocatedbytheﬁngerofthe patientnexttothequadricepstendon.Therewasnopalpablemass andthekneewasfree.

Plain radiography was normal. A Doppler ultrasound was requestedobjectifyingarounded swelling,well-deﬁned,hypoechoic,homogeneous,inthequadricepstendon,withintralesional hypersignalincolorDopplermode,measuring13mm/6mmwith- outassociatedanomalies.

Surgical exploration discovered a tumor mesur- ing18mm/10mm, brownish, encapsulated in the quadriceps tendon(AppendixA,Fig.S2).Histopathologicalﬁndingsconﬁrmed thediagnosisofaglomustumor.

The postoperative course was uneventful, with total and immediate relief of pain. At 7-year follow-up, the patient was asymptomaticandtherewasnolocalrecurrence.

3. Discussion

Glomustumorgrowsattheexpenseofneuromyoarterialtis- suefoundmainlyattheextremities.Theneuromyoarterialglomus composedofmany arterio-venousanastomosis,involved inthe regulationofcutaneousmicrocirculationandthermoregulation.

Solitary glomus tumors are usually seen in adults also fre- quentlyinbothsexes,exceptforsubungualglomustumorsthat show a female preponderance [5]. Their most frequent site is thehand,inparticulartheﬁngers[1].Theycanbedevelopedin diverseareasastheelbow,hipandfoot.Deeperlocationshave beendescribedinthevisceralorganssuchasthestomachorthe uterus,butalsoofsomebones,thesciaticnerveandtherotator cuff[6].

Thelocationofthekneeisauthenticandveryrare.Toourknowl- edge,18casesofglomustumorsinthekneehavebeenreportedin theliterature.

Thesetumorsaroundthekneehavebeenreportedinvariable locations;subcutaneous,sub-synovial,withinthepatellaligament, withinthefatpadandinthepoplitealarea.

Our cases had various locations around the knee: subcuta- neouslytotheanteriorsuperiorportionofthepatellainthecase1;

aheadofthepatellartendoninthecase2;onlythreecaseslocatedin thispre-patellarregionhavebeenreportedintheliterature[7–9];

thepatientofthethirdobservationhadanexceptionallocationin thequadricepstendon.Onlyonesimilarcaseintheliteraturewas reportedbyWahbietal.[10].

Glomus tumors are known to be painful. The characteris- tic triad “paroxysmal pain, exaggerated sensitivity to cold and shockandpointtenderness”isoftencitedasthekeytodiagno- sisindigitallocation.Painisanimportantsemiologicalelement butlittleevocativeinextradigitallocationsincludingaroundthe knee.Indeed,thetimebetweenonsetofsymptomsanddiagno- sisrangedfromafewmonthsto30yearsandcorrectdiagnosis is mentioned only in about 20% of cases [11]. It was diffuse, interestingthewholelegin thecasereportedbyMurphyetal.

[12].

In our third case, pain was somewhat evocative what was causing the delay in diagnosis for four years. No signs of the Triad“paroxysmalpain,exaggeratedsensitivitytocoldandshock and point tenderness” were present in this patient. The deep localizationthequadricepstendonmayalsoexplaintheabsence of palpable tumormass. Sometimes,these are consideredpsy- chogenicpainanddoconsultthepsychiatrist,asinoursecondcase.

(3)

Pleasecitethis articleinpressas:ElHyaouiH,etal.Unusuallocalizationofglomustumoroftheknee. JointBoneSpine(2015), http://dx.doi.org/10.1016/j.jbspin.2015.07.001

ARTICLE IN PRESS

G Model

H.ElHyaouietal./JointBoneSpinexxx(2015)xxx–xxx 3

Pintestistosearchthepainfulareausingasharpinstrument andischaracterizedbyitshighsensitivity(100%)[1].Hildreth’s sign,veryspeciﬁc(100%)butlesssensitivethanthepintest(71%), consistsinthereductionordisappearanceofalgicphenomenaafter performingaminuteischemia[1].Thistestwasnotperformedin ourpatients.

Therearenospecificimagingtechniquestoconfirmthediagno- sis.TheultrasonographycoupledwithDoppler,typicallyshowsa netintralesionalDopplersignalintensityinslowflowcontrol,ona well-demarcatedmass,roundedoroval,homogeneous,hypoechoic andnotcalcified[1,13].MRIbetterdefinestumorcharacteristics:

indeed,itisahomogeneouslesionalthoughlimited,hypo-intense onT1-weightedsequenceswithrapidenhancementaftergadolin- ium injection, and hyperintense on T2-weighted images. The deﬁnitivediagnosisismadebyhistology,whichshows aprolif- erationconsistsin varyingproportionsofglomuscells, vascular structures,andsmoothmuscletissues.Basedontheserelativepro- portions,therearethreetypes:solidglomustumor“classic”,the glomangiomeandglomangiomyome.

Anervouscomponent isalwaysassociated [1].Thedifferen- tial diagnosis is broad. It includes intra-articular lesions (such asmeniscal tears orcysts, synovitis, plica irritation,pigmented villinodular synovitis, foreign body and infection) and extra- articularlesions(ligamentousstrainsandsprains,neuropathicpain syndromes)[5].

Thestandard treatmentisa meticulousand completesurgi- calexcision.Thisexcisionshouldleadtoadramaticresolutionof symptomslikethecaseinourpatients.

Recurrences are not frequentlymentioned in the literature, perhaps because of insufﬁcient decline, as mentioned cases of recurrence,whilerare(7%)havethreetoﬁveyearsbeforemani- festingclinicallyorrelatedtomultipletumors[1,14].

Inallourthreecases,wehaveobservednorecurrencesduring afollow-upperiodofupto7yearsforthethirdobservation.To ourknowledge,thisseven-yearfollow-upisthelargestreported forglomustumorslocatedaroundtheknee.

Malignant sarcomatous transformation of glomus tumors is extremelyrare[15].

Disclosureofinterest

Theauthorsdeclarethattheyhavenoconﬂictsofinterestcon- cerningthisarticle.

AppendixA. Supplementarydata

Supplementarydata(Figs.S1andS2)associatedwiththisarticle canbefound,intheonlineversion,athttp://dx.doi.org/10.1016/

j.jbspin.2015.07.001.

References

[1]FrikhR,AliouaZ,HarketA,etal.Glomustumors:anatomoclinicalstudyof14 caseswithliteraturereview.AnnChirPlastEsthet2009;54:51–6.

[2]SouleEH,GhormleyRK,BulbulianAH.Primarytumorsofthesofttissuesof theextremitiesexclusiveofepithelialtumors:ananalysisofﬁvehundred consecutivecases.AMAArchSurg1955;70:462–74.

[3]WoodW.Painfulsubcutaneoustubercle.EdinbMedJ1812;8:283.

[4]MassonP.Leglomusneuromyoartérieldesrégionstactilesetsestumeurs.Lyon Chir1924;21:257–80.

[5]ClarkML,O’HaraC,DobsonPJ,etal.Glomustumorandkneepain:areportof fourcases.Knee2009;16:231–4.

[6]TuncaliD,YilmazAC,TerziogluA,etal.Multipleoccurrencesofdifferenthis- tologictypesoftheglomustumor.JHandSurg2005;30:161–4.

[7]DelBuonoF,FerrarioP,RoncaglioC.Acaseofglomustumoroftheprepatellar region.ChirItal1981;33:122–8[Abstract].

[8]PuttiE,TatòFB.Twocasesofglomustumorlocalizedintherightknee:removal andrecurrence.ChirOrganiMov1991;76:375–8[Abstract].

[9]AkgünRC,GülerUÖ,OnayU.Aglomustumoranteriortothepatellartendon:

acasereport.ActaOrthopTraumatolTurc2010;44:250–3.

[10]WahbiS,BelkourchiaE,Bouhouch A,etal.Tumeurglomique dutendon quadricipital :àproposd’uncas.RevChirOrthopReparatrice ApparMot 2005;91:272–5.

[11]TsuneyoshiM,EnjojiM.Glomustumor:aclinicopathologicandelectronmicro- scopicstudy.Cancer1982;50:1601–7.

[12]MurphyRX,RachmanRA.Extradigitalglomustumorasacauseofkneepain.

PlastReconstrSurg1993;92:1371–4.

[13]Gómez-SánchezME,Alfageme-RoldánF,Roustán-GullónG,etal.Theuseful- nessofultrasoundimagingindigitalandextradigitalglomustumors.Actas Dermosiﬁliogr2014;105:e45–9.

[14]FoucherG,LeVietD,DailianaZ,etal.Lestumeursglomiquesdelarégion unguéale.RevChirOrthop1999;85:362–6.

[15]FolpeAL,Fanburg-SmithJC,MiettinenM,etal.Atypicalandmalignantglomus tumors:analysisof52cases,withproposalforthereclassiﬁcationofglomus tumors.AmJSurgPathol2011;25:1–12.