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Tarsal tunnel syndrome masked by painful diabetic polyneuropathy

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InternationalJournalofSurgeryCaseReports15(2015)103–106

ContentslistsavailableatScienceDirect

International

Journal

of

Surgery

Case

Reports

j o u r n al ho m e p a g e :w w w . c a s e r e p o r t s . c o m

Tarsal

tunnel

syndrome

masked

by

painful

diabetic

polyneuropathy

Tugrul

Ormeci

a,∗

,

Mahir

Mahirogulları

b

,

Fikret

Aysal

c

aMedipolUniversity,FacultyofMedicine,DepartmentofRadiology, ˙Istanbul,Turkey

bMedipolUniversity,FacultyofMedicine,DepartmentofOrthopedicsandTraumatology, ˙Istanbul,Turkey cMedipolUniversity,FacultyofMedicine,DepartmentofNeurology, ˙Istanbul,Turkey

a

r

t

i

c

l

e

i

n

f

o

Articlehistory: Received3July2015

Receivedinrevisedform5August2015 Accepted21August2015

Availableonline28August2015 Keywords:

Tarsaltunnelsyndrome Diabeticpolyneuropathy Pain

Magneticresonance Superficialultrasound

a

b

s

t

r

a

c

t

INTRODUCTION:Variouscausesinfluencetheetiologyoftarsaltunnelsyndromeincludingsystemic

diseaseswithprogressiveneuropathy,suchasdiabetes.

PRESENTATIONOFCASE:Wedescribea52-year-oldmalepatientwithcomplaintsofnumbness,burning

sensationandpaininbothfeet.Thelaboratoryresultsshowedthatthepatienthaduncontrolleddiabetes,

andtheEMGshoweddistalsymmetricalsensory-motorneuropathyandnerveentrapmentattheright.

UltrasonographyandMRIshowedthecystinrelationtomedialplantarnerve,andedema-moderate

atrophywereobservedatthedistalmusclesofthefoot.

DISCUSSION:Footneuropathyindiabeticpatientsisacomplexprocess.So,inplanningtheinitial

treat-ment,medicalorsurgicaltherapyisselectedbasedonthelocationandtypeofthepathology.Foot

deformitiescanbecorrectedwithresting,anti-inflammatorytreatment,appropriateshoes,orthesisand

socks,andifrequired,anklestabilizationcanbeattempted.Ifthepatientisstillunresponsive,surgical

treatmentmaybeapplied.

CONCLUSION:Itisessentialtoinvestigatemorelocalizedreasonsliketarsaltunnelsyndromethatmay

mimicdiabeticneuropathy,shouldbetreatedprimarily.

©2015TheAuthors.PublishedbyElsevierLtd.onbehalfofSurgicalAssociatesLtd.Thisisanopen

accessarticleundertheCCBY-NC-NDlicense(http://creativecommons.org/licenses/by-nc-nd/4.0/).

1. Introduction

Thetarsaltunnelisa fibroosseousstructureofthefoot posi-tioned posteromedially, extending from the medial malleolus towardthenavicularbone.Itenclosestheposteriortibialtendon, flexorhallucislongustendonandflexordigitorumlongustendon, alongwiththeposteriortibialartery,veins,nervesandbranches

[1].

Tarsal tunnel syndrome is an entrapment neuropathic syn-dromethatdevelopsuponcompressionoftheposteriortibialnerve oritsbranches(medialandlateralplantarnerves)bytheflexor retinaculum.Systemicdiseaseprogressionwithneuropathysuch as diabetes should be considered during differential diagnosis. Elucidatingtheetiologyisthefirststepintreatment.Wereport a case withtarsaltunnel syndromemaskedby painful diabetic polyneuropathyinanefforttoraiseawarenessofthissyndrome anddescribetodiagnosticapproachtothepatient.

∗ Corresponding authorat:MedipolUniversity,FacultyofMedicine, Depart-ment of Radiology, Medipol Mega Hastaneler Kompleksi, Radyoloji Bölümü, TEM Avrupa Otoyolu Göztepe c¸ıkıs¸ı No: 1 Ba˘gcılar, 34214 ˙Istanbul, Turkey. Fax:+902124607050.

E-mailaddresses:tugormeci@yahoo.co.uk(T.Ormeci),

mmahirogullari@medipol.edu.tr(M.Mahirogulları),fikretaysal@yahoo.com

(F.Aysal).

2. Presentationofcase

A52-year-oldmalepatientwasadmitted withcomplaintsof numbness,burningsensationandpaininbothfeet(but predom-inantlyintherightfoot).Thesecomplaintswerepresentforthe preceding5 monthsbut increasedwithinthe last1 month. He hadbeenusingoralantidiabeticsduetotype2diabetes.He suf-feredfromthepaininhisrightfootincreaseduponwalking,which intensifiedthroughthefirsttoeandmedialfoot.

On neurological examination, bilateral hypoactive Achilles reflexes,hypoesthesiaandhypoalgesiawerepresentonbothfeet, moreprominentonrightfoot.Tineltestwaspositiveinrightankle. Noweaknesswasdetected.

Thelaboratoryresultswereasfollows:fastingplasmaglucose 243mg/dl,hemoglobinA1c9.2%,spoturinecreatinine299mg/dl andmicroalbumin232mg/dl.Thelaboratoryresultsshowedthat thepatienthad uncontrolleddiabetes,and hiscomplaintswere consideredtoberelatedtothisfinding.Hisparaestheticcomplaints improvedafterinsulintreatmentfordiabetes;however,hispain remained.

Nerve conduction studies revealed low compound muscle action potential (CMAP)amplitude in righttibialis nerve, non-detectablesensorynerveactionpotential(SNAP)ofmedialplantar nerveandchronicneurogenicchangesonneedleEMG(longterm, polyphasic,high-amplitudemotorunitpotentialswithdecreased recruitment)ofrightabductorhallucis(AH)andabductordigitiquinti

http://dx.doi.org/10.1016/j.ijscr.2015.08.033

2210-2612/©2015TheAuthors.PublishedbyElsevierLtd.onbehalfofSurgicalAssociatesLtd.ThisisanopenaccessarticleundertheCCBY-NC-NDlicense(http://

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104 T.Ormecietal./InternationalJournalofSurgeryCaseReports15(2015)103–106

Fig.1. Nerveconductionstudiesofbilateralposteriortibialnerves.Med.Malleo:medialmalleol,Pop.Fossa:poplitealfossa,ADQ:abductordigitiquinti,AH:abductorhallucis.

Fig.2. Wellcircumscribed,anechoiccysticlesion(asterisk)adjacenttotheposterior

tibialarteryandvein(arrows)inthemedialfoot.

(ADQ)muscles. These findings suggestedan entrapmentof the posteriortibialnerveattarsaltunnellevel(Fig.1).Amild sensory-motorpolyneuropathywasalsosuspected,becauseofslowingin motornervevelocityofexaminednervesandaslightdecreasein bothsuralnerves’SNAPamplitudes.Thiswasthoughttobe dia-betesrelated.Inaddition,resultscompatiblewithbilateralcarpal tunnelsyndromewerefound.

SuperficialUSimagingshowedahomogenouscysticlesionsized 15×10mmlocatedmediallytothemedialplantararteryandvein (Fig.2).

MRIofthefootandankle revealedahomogenous,T1 hypo-,T2hyperintense ganglioncystlocatedatthemedialcalcaneus, showingperipheralcontrastuptake.Thelocalizationofthecystin relationtotheflexorhallucislongustendon,posteriortibialartery andmedialplantarnervewasassessed(Fig.3).Edemaand mod-erateatrophywereobservedattheabductorhallucisandflexor hallucisbrevismuscles(Fig.4).

Tarsaltunnel syndromeinrelationwithmild sensory-motor polyneuropathyofdiabeticoriginwasdiagnosedbasedonclinical symptoms,electrophysiologicalfindingsandimagingofaganglion cystatthetarsaltunnellevelonUSandMRI.

Thepatientwasreferredtothedepartmentofsurgerywiththe diagnosisoftarsaltunnelsyndrome.However,thepatientrefused surgeryandregularlyusedhisanti-diabetesmedicationduringthe followingfourmonths,electrophysiologicalfindingsworsenedat controlinvestigation.

3. Discussion

Tarsal tunnel syndrome develops from compression of the posteriortibial nerve andits brancheswithinthetarsaltunnel. Compressionmayoccurduetotraumaticcausessuchasfracture, surgeryorscartissue[2],butitmayalsobeassociatedwith space-occupyinglesions includingtumors, ganglion, varicose vascular structuresandabnormalmuscletissue(accessoryflexordigitorum longusmuscleorhypertrophicadductorhallucis)[3].Inaddition, itmaydevelopinthefootinrelationtodeformitiessuchas

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T.Ormecietal./InternationalJournalofSurgeryCaseReports15(2015)103–106 105

Fig.3.(a)AxialT1turbospinecho(TSE),(b)axialshorttauinversionrecovery(STIR)TSE,(c)axialpostcontrastT1spectralpresaturationwithinversionrecovery(SPIR)

demonstrateT1hypointense,T2hyperintenseandperipherallycontrastedcysticlesion(ganglioncyst)(star)withclosedrelationsintheflexorretinaculum(smallarrow),

medialplantarnerve(blackarrow),flexorhallucislongustendon(arrowhead)andposteriortibialartery–vein(largearrow)positionedmediallyonthecalcaneus(C)inthe

tarsaltunnellocalization.

Fig.4.(a)coronalT1TSE,(b)coronalshorttauinversionrecovery(STIR)TSE.Moderatevolumelossinaandhyperintensityinbareseenintheabductorhallucis(small

arrow)andflexorhallucisbrevis(largearrows)musclesattheplantarsideofthedistalfoot.Thefindingsarecompatiblewithmuscleatrophy.(MT1):firstmetatarsal.

gus/varusdeformity,pesplanusandtarsalcoalition[4].Systemic diseasessuchasdiabetes,rheumatoidarthritisandperipheral arte-rialdiseasemayalsocausetarsaltunnelsyndrome[5].Despiteall possiblecauses,theexactcauseofthediseaseissometimesunclear. Thesymptomsdependonwhich branchoftheposterior tib-ialnerveisinvolvedandthelocationoftheinvolvement.Physical examinationgenerallyshowslossofsensationattheplantarface ofthefootandpositiveTinel’sfindings.Patientssufferfrompain withaburningsensationatthetoesandplantarregion.The condi-tionmaybeaccompaniedbyparaesthesiaanddysesthesia,andthe findingsincreasewithactivity.Motorfunctionimpairmentisalate findingthatisgenerallyunnoticeable,andweaknessmaydevelop rarelyatthetoeflexors.Inadditiontotheclinicalmanifestations, decreasedconductionrateintheposteriortibialnerveasseenon EMGconfirmsthediagnosis.

US examinations can show space-occupying solid or cystic lesions;however,itisdifficulttoassessmuscleinnervationsinthe absenceofatrophy.

MRIexaminationsareextremelyusefulinassessingthetarsal tunnel, defining theetiology of thesyndromeand determining thedifferentialdiagnosis.Flexorhallucislongustenosynovitismay becomethecauseofnerve compressioninsevereconditions.In somecases,ankleganglionscausethesesymptoms.Inthemajority ofcases,thenervecannotbevisualizedcompletelyalongitstrace. However,thenervevolume,signalalterationsanddistortionsin thenerve traceshouldbeconsidered[6].Theseevaluationscan helpeliminatetheabove-mentionedetiologiccauses.Somecases maypresentwithamildsignalalterationintheabductorhallucis andflexorhallucisbrevismusclesinnervedbythemedial

plan-tarnerve,andadvancedcasesmaydevelopatrophy.Asinthecase presentedhere,atrophygenerallyaffectsdistalmuscles.

Systemic disease progression with neuropathy should be consideredduring differentialdiagnosis.Diabetic neuropathyis diagnosed in diabetic patientswith complaintsand findings of peripheralnerve dysfunctionin the absenceof other causative conditions[7].Themostcommonsymptomsofpolyneuropathy indiabeticpatientsaretingling,coldness,painandparaesthesia inthelegs,particularlynotableatdistalsites.Examinationshows stocking-glovesensoryloss.Moderateatrophyandparesismaybe encounteredattheintrinsicmusclesofthefoot[8].

Carpaltunnel syndrome, another entrapmentneuropathy, is encounteredinthediabeticpopulationathigherratethanthatin thegeneralpopulation.Watanabeetal.reportedthattibialnervein tarsaltunnelandmediannerveincarpaltunnelincreaseinterms ofcrosssectionalareaindiabeticpatients[9].Thoseresultswere compatiblewithsystemiceffectsofdiabetesandourpatientalso hadcarpaltunnelsyndromeaccompanyingtarsaltunnelsyndrome. Whether the entrapment neuropathies that develop in dia-beticpatientsaredirectlyassociatedwithdiabetesiscontroversial

[10,11].Footneuropathyindiabeticpatientsisacomplexprocess.

So,inplanningtheinitialtreatment,medicalorsurgicaltherapy isselectedbasedonthelocationandtypeofthepathology.Foot deformitiescanbecorrectedwithresting,anti-inflammatory treat-ment,appropriateshoes,orthesisandsocks,andifrequired,ankle stabilizationcanbeattempted.Physicaltherapymodalitiesmight bepreferredinsuitablecases.Tricyclicantidepressantmedications andantiepilepticagentssuchasgabapentinandpregabalinmaybe useful.Lidocainandcortisonecanbeinjectedlocallytothenerve

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106 T.Ormecietal./InternationalJournalofSurgeryCaseReports15(2015)103–106

trace.Ifthepatientisstillunresponsive,surgicaltreatmentmaybe applied[1].

4. Conclusion

Successfultreatmentoftheinitialconditionofneuropathicfoot painindiabeticpatientsinvolveseliminationofthecausesof neu-ropathicpain,particularlytarsaltunnelsyndrome.Pathologiesthat maymimicdiabeticneuropathyshouldbetreatedprimarily. Funding None. Ethicalapproval N/A. Consent Obtained. Conflictofinterest None. References

[1]E.M.Delfaut,X.Demondion,A.Bieganski,M.C.Thiron,H.Mestdagh,A.Cotten,

Imagingoffootandanklenerveentrapmentsyndromes:from

well-demonstratedtounfamiliarsites,Radiographics23(2003)613–623.

[2]O.C.Aszmann,J.M.Ebmer,A.L.Dellon,Cutaneousinnervationofthemedial

ankle:ananatomicstudyofthesaphenous,sural,andtibialnervesandtheir

clinicalsignificance,FootAnkleInt.19(1998)753–756.

[3]G.J.Sammarco,S.F.Conti,Tarsaltunnelsyndromecausedbyananomalous

muscle,J.BoneJointSurg.Am.76(1994)1308–1314.

[4]E.Trepman,N.J.Kadel,K.Chisholm,L.Razzano,Effectoffootandankle

positionontarsaltunnelcompartmentpressure,FootAnkleInt.20(1999)

721–726.

[5]A.Donovan,Z.S.Rosenberg,C.F.Cavalcanti,MRimagingofentrapment

neuropathiesofthelowerextremity.Part2.Theknee,leg,ankle,andfoot,

Radiographics30(4)(2010)1001–1019.

[6]M.F.Lee,P.T.Chan,L.F.Chau,K.S.Yu,Tarsaltunnelsyndromecausedby

talocalcanealcoalition,Clin.Imaging26(2002)140–143.

[7]A.J.Boulton,F.A.Gries,J.A.Jervell,Guidelinesforthediagnosisandoutpatient

managementofdiabeticperipheralneuropathy,Diabet.Med.15(1998)

508–514.

[8]S.J.Bird,M.J.Brown,Diabeticneuropahhies,in:B.Katırjı,H.J.Kominski,D.C.

Preston,R.L.Ruff,B.E.Shapino(Eds.),NeuromuscularDisordersinClinical

Practice,ButterworthHeinemann,USA,2002,pp.598–621.

[9]T.Watanabe,H.Ito,A.Sekine,Y.Katano,T.Nishimura,Y.Kato,etal.,

Sonographicevaluationoftheperipheralnerveindiabeticpatients:the

relationshipbetweennerveconductionstudies,echointensity,and

cross-sectionalarea,J.UltrasoundMed.29(2010)697–708.

[10]D.M.Fraser,I.W.Campbell,D.J.Ewing,B.F.Clarke,Mononeuropathyin

diabetesmellitus,Diabetes28(1979)96–101.

[11]B.S.Boyd,A.Dilley,Alteredtibialnervebiomechanicsinpatientswith

diabetesmellitus,MuscleNerve.50(2014)216–223.

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Şekil

Fig. 2. Well circumscribed, anechoic cystic lesion (asterisk) adjacent to the posterior tibial artery and vein (arrows) in the medial foot.
Fig. 4. (a) coronal T1 TSE, (b) coronal short tau inversion recovery (STIR) TSE. Moderate volume loss in a and hyperintensity in b are seen in the abductor hallucis (small arrow) and flexor hallucis brevis (large arrows) muscles at the plantar side of the d

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