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© 2021 Journal of Orthopaedics, Traumatology and Rehabilitation | Published by Wolters Kluwer - Medknow 61
Abstract
Case Report
IntRoductIon
Complexregionalpainsyndrome(CRPS)Type I is a
chronicpaindisorderwhich occursafter somepredisposing
conditionsincludingtrauma,infection,surgery, cervical
radiculopathy,soft‑tissue contusions, fractures, tendon
ruptures,andmyocardialinfarction.[1,2]Symptomsandndings
arecharacterizedbypain,allodynia,edema,trophicand
vasomotorchangesintheaffectedlimb.Aswellasthepain
isnot limitedto aspecic nervedistributionordermatome,
itisnotassociatedwiththeseverityofinjuryeither.CRPSis
clinicallyseparatedintothreephasesasacute,dystrophic,and
atrophic.According to extremelyvariousdurationsofeach
period,separationofthesephasesisnotpossibleunlikely.
Thepathophysiologyisnotclear,and some theories are
suggestedsuchasirregularsympatheticsystem,neurogenic
inammation,andimmobilization.Furthermore,psychological
featuresareaccusedasapredisposingfactor.[3,4]CRPScanbe
reactivatedafteradisease‑freeperiodwithfurthertraumaor
anotheroperationinthesameoranotherlimb.[5]
Wereportacase,whohadCRPSTypeIhistoryinthelower
limbafterminortrauma,withrecurrentCRPSTypeIin
anotherlimbduetocoredecompressionandbonegraftingfor
osteonecrosisofthehip.
case RepoRt
A47‑year‑oldmalehadahistoryofCRPSTypeIafterleft
footfourthngersoft‑tissueinjury,duetothespreadofthe
complaintstothefeetandankle,andprolongedhissymptoms
throughout3months.Hehadbeenimplemented45sessions
ofphysicaltherapyprogramandmedicaltreatmentinour
departmentbecauseof leftlower‑limbCRPSTypeI.Then,
hehadcompletelyameliorated.Oneyearlater,thepatienthad
feltpaininhisrighthipandhehadappliedtoorthopaedist.
Inthesequel,osteonecrosisoftherightfemoralheadhad
beenidentifiedbymagneticresonanceimaging(MRI).
Havingbeenoperatedduetoosteonecrosisandperformed
coredecompressionandbonegraftingfortherighthipbyan
orthopedist,hehadbeenadvisedimmobilizationthroughout
2monthsbecauseofcastingaftersurgery.Alongwith
mobilization,thepatientbegantofeelpaininhisrightfoot.
Complexregionalpainsyndrome(CRPS)TypeI,achronicpaindisorder,occursinthesequelofcertainpredisposingconditions.Recurrence
maybeobservedin4%–10%ofcasesinthesameoranotherlimb.TherearenospecicparameterstopreventCRPSaftersurgeryortrauma.
However,someauthorshavereportedseveralrecommendationsaboutsurgicaltechniquesandVitaminCsupplementation.Wereportacase
whohadCRPSTypeIhistorybeforeanddevelopedrecurrentCRPSTypeIinthelowerlimbduetosurgeryforosteonecrosisofthehip.We
observedsignicantimprovementwithphysicaltherapyandmedication.
Keywords:Complexregionalpainsyndrome,hipsurgery,recurrentcomplexregionalpainsyndrome
Address for correspondence: Dr. Hüseyin Kaplan,
Department of Physical Medicine and Rehabilitation, School of Medicine,
Education and Research Hospital, Aksaray University, Aksaray, Turkey.
E‑mail: hkaplan_87@hotmail.com
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Recurrent Complex Regional Pain Syndrome after Minor Hip
Surgery
Hüseyin Kaplan, Emel Güler1, Mehmet Kırnap2
Department of Physical Medicine and Rehabilitation, School of Medicine, Education and Research Hospital, Aksaray University, Aksaray, 1Department of Physical
Medicine and Rehabilitation, Division of Algology, Kayseri City Hospital, 2Department of Physical Medicine and Rehabilitation, School of Medicine, Erciyes University,
Kayseri, Turkey
How to cite this article: KaplanH,GülerE,KirnapM.Recurrentcomplex
regionalpainsyndromeafterminorhipsurgery.JOrthopTraumatolRehabil
2021;13:61‑3.
Submission: 01.12.2019 Revision: 19.08.2020
Acceptance:11.04.2021 Web Publication:16.06.2021
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Kaplan, et al.: Recurrent CRPS
Journal of Orthopaedics, Traumatology and Rehabilitation ¦ Volume 13 ¦ Issue 1 ¦ January-June 2021
62
Ontheoccasionofprolongedcomplaints,hehadapplied
tohissurgeon. InconsequenceofX‑rays and MRI,hehad
beendiagnosedrightlower‑limbCRPSTypeI.Hehadbeen
performedphysicaltherapyinanotherhospitalthroughout
21sessions.However,he was consulted in our department
byhisorthopedistforcontinuingcomplaints.Thepatient
presentedwithcomplaintsofdifcultyinwalking,swelling
intherightfootandankle,paininthecourseof mobility,
andresting.Onphysicalexamination,ininspection,he
mobilizedwithtwocrutch,hisrightfootandanklewere
edematousandalittle red.Also, elongation andthickening
wereobservedinhairofrightfoot.Onpalpation,therewere
prominentlytenderness,allodynia,andparesthesiaonthe
rightankle.Furthermore,he had alimitedrangeofmotion
ontheankle.TheNumericRatingScalewas8outof10.In
laboratoryexamination,erythrocytesedimentationratewas7
mm/h(3–20),C‑reactiveprotein(CRP)was3.98mg/L(0–6),
andrheumatoidfactorwasnegative.Triple‑phasebonescan
wascompatiblewithCRPS[Figure1].Regionalosteoporosis
wasseeninhisankleonroutineX‑rayexamination[Figure2].
MRIoftherightfootshowedthat therewasacompression
fractureinthetalus[Figure3].Hewasusingsomedrugs
includingibuprofen1200mg/day, pregabalin 150 mg/day,
andcalciumcarbonate andVitamin D3.Hewasundergone
physicaltherapy(transcutaneouselectricalnervestimulation,
whirlpool,rangeofmotionexercises,andpartialweightingon
thelimb).Hismedicaltreatmentwasreadjusted(pregabalin
300mg/day,ibuprofen1600mg/day,andcalciumcarbonate
plusVitaminD3).Asignicantimprovementwasobserved
followingninesessionsofphysicaltherapyandmedication.
Inadditiontothis,theNumericRatingScaledecreasedtothe
levelof1efciently.
dIscussIon
CRPSTypeI,aposttraumaticdisorder,commonlyassociated
withsurgeryincludingextremities,isascarcelycomplex
clinicalentityforclinicians.Theincidencehasbeenreported
1%–37%afterdistalradiusfracture.Somesynonymsarealso
usedtodescribeCRPSsuchasreexsympatheticdystrophy
syndrome,Sudeck’satrophy,andalgodystrophy.[3]The
BudapestcriteriaforthediagnosisofCRPS,modiedversion
ofOrlandocriteria,haveagreaterspecicityandalsoinclude
motorfeaturesofthesyndrome[Table1].[1]
Whileithasagoodprognosiswithearlydiagnosisand
treatment,prolongedmisdiagnosisandlackoftreatment
resultinchronicdisability.Recurrenceisobservedin4%–
10%ofcases3monthsto20yearsaftertheinitialevent.[6]
Therearenospecicparameterswhichareknowntoprevent
CRPSaftersurgeryortrauma.Butstill,anumberofauthors
havereportedseveralrecommendations including careful
operativetechnique,knowledgeofanatomy, avoidance
ofnerve traction,andproperpostoperativecaretoreduce
thefrequencyofthisdisorder.Physical therapies, mirror
visualfeedback,medication(oral/topicalmedicationor
injections),andsurgery maybeeffectivesolutions inthe
processoftreatment.Vitamin C supplementationafter
surgeryisassociatedwithreducingtheriskofdeveloping
CRPS.Althoughoptimaldoseisnotclear,500–1000mg/
daydosesarecommonlytaughttobeeffectivethanlower
doses.Typically,a 500 mg/daydoseisused for 50 days.
Furthermore,earlymobilizationafterlimbsurgerieshasbeen
reportedtopreventCRPS.[5,7,8]Paralleltothisinformation,
inasequelof2‑monthimmobilizationperiod,ourcaseeven
developedCRPSTypeI.
Figure 2: Regional osteoporosis on X‑ray
Figure 1: Triple‑phase bone scan was compatible with complex regional
pain syndrome for the right ankle (anterior‑posterior and posterior‑anterior
images, respectively)
Figure 3: Compression fracture in the talus on magnetic resonance
imaging of the right ankle
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Kaplan, et al.: Recurrent CRPS
Journal of Orthopaedics, Traumatology and Rehabilitation ¦ Volume 13 ¦ Issue 1 ¦ January-June 2021 63
Spinalcordstimulationmaybeperformed,ifsymptomsstill
persistafter3–4months.[1]However,itisreportedthat74%
ofpatientswithCRPSTypeIachieveresolutionofsymptoms
withearlytreatment.[9]Inourpatient,weachievedgreat
improvementwithphysicaltherapyandmedication.
Inconclusion,itcanbesuggestedthat people who will be
performedanysurgicalproceduresshouldbeevaluatedwith
regardtoCRPShistoryaheadofoperation.Thismaybe
fairlyimportanttotakeprecautionseitherduringsurgeryor
inpostoperativecaretopreventrecurrence.
Declaration of patient consent
Theauthorscertify thattheyhaveobtained allappropriate
patientconsentforms.Intheformthepatient(s)has/have
givenhis/her/theirconsentforhis/her/theirimagesandother
clinicalinformationtobereportedinthejournal.Thepatients
understandthattheirnamesandinitialswillnotbepublished
anddueeffortswill be madetoconcealtheiridentity,but
anonymitycannotbeguaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
Therearenoconictsofinterest.
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8. ShibuyaN,HumphersJM,AgarwalMR,JupiterDC.Efcacyandsafety
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Table 1: Budapest clinical diagnostic criteria for complex
regional pain syndrome
Continuingpain,whichisdisproportionatetoanyincitingevent
Mustreportatleastonesymptominthreeofthefourfollowingcategories
Sensory:Reportsofhyperalgesiaand/orallodynia
Vasomotor:Reportsoftemperatureasymmetry,and/orskincolor
changes,and/orskincolorasymmetry
Sudomotor/edema:Reportsofedema,and/orsweatingchanges,and/or
sweatingasymmetry
Motor/trophic:Reportsofdecreasedrangeofmotion,and/ormotor
dysfunction(weakness,tremor,anddystonia),and/ortrophic
changes(hair,nail,andskin)
Mustdisplayatleastonesign*attimeofevaluationintwoormoreofthe
followingcategories
Sensory:Evidenceofhyperalgesia(topinprick)and/orallodynia(to
lighttouch,and/ordeepsomaticpressure,and/orjointmovement)
Vasomotor:Evidenceoftemperatureasymmetry,and/orskincolor
changes,and/orasymmetry
Sudomotor/edema:Evidenceofedema,and/orsweatingchanges,and/or
sweatingasymmetry
Motor/trophic:Evidenceofdecreasedrangeofmotionand/ortrophic
changes(hair,nail,andskin)
Thereisnootherdiagnosisthatbetterexplainsthesignsandsymptoms
*Asigniscountedonlyifitobservedatthetimeofdiagnosis
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