paediatric to adult care in patients with epilepsy Long-term effects of a multidisciplinary transition interventionfrom Seizure

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Long-term effects of a multidisciplinary transition intervention from paediatric to adult care in patients with epilepsy

R.P.J. Geerlings

a,

*, A.P. Aldenkamp

a,b,c,d

, L.M.C. Gottmer-Welschen

a

, A.L. van Staa

e,f

, A.J.A. de Louw

a,b

aEpilepsyCenterKempenhaeghe,Heeze,TheNetherlands

bFacultyofElectricalEngineering,UniversityofTechnology,Eindhoven,TheNetherlands

cDepartmentofNeurology,MaastrichtUniversityHospital,TheNetherlands

dDepartmentofNeurology,GhentUniversityHospital,Belgium

eInstituteofHealthPolicy&Management,ErasmusUniversityRotterdam,TheNetherlands

fResearchCentreInnovationsinCare,RotterdamUniversityofAppliedSciences,TheNetherlands

1. Introduction

Adolescenceisacriticalandvulnerableperiodinlifebecause adolescentshavetodeveloptheirownidentity,autonomy,peer relationshipsandtheirownsocialnetwork[1–3].Havingachronic

illness transferring into adulthood, such as epilepsy, makes adolescence even morecomplex. Several studiesindicated that patients with epilepsyare at risk of persistent long-termpoor psychosocial outcome on several transitional domains, e.g., educationandemployment[1,4–7].Therefore,age-specificissues often deserve special attention in adolescents with epilepsy.

Further,adolescentswith(chronic)epilepsyhavetotransferfrom paediatrictoadultmedicalcareatacertainpointinlife[3,4].If insufficientattentionisgiventothistransition,adolescentsand youngadultswithepilepsymaywithdrawfromnecessarymedical and psychosocial health care, and end up in a troublesome situation.Tocopewiththeseproblems,epilepsytransitionclinics have been set up for adolescents [8,9]. A transition clinic can ARTICLE INFO

Articlehistory:

Received14December2015

Receivedinrevisedform15February2016 Accepted10April2016

Keywords:

Transitiontoadultcare Epilepsy

Transitionclinic Psychosocialoutcome Transitionintervention

ABSTRACT

Purpose:Toevaluatethelong-termeffectsofamultidisciplinarytransitioninterventioncomparedtothe impactofpatient-relatedintrinsicfactorsontheimprovementinmedicalandpsychosocialoutcome.

Methods:All patients whovisited ourmultidisciplinary Epilepsy Transition Clinic between March 2012andSeptember2014wereinvitedtoparticipate(n=114).Patientsweresentonequestionnaire andinformedconsentwas obtained.Questionsincludedthepatient’s leveloffunctioningonthree transitionaldomainsandalistwithmedicalhealthcareworkers.Previouslydefinedscoresonthree transitional domains and the risk profile score were re-evaluated. Past and current patient characteristics were compared using descriptive statistics. Discriminant analyses were used to determinetheinfluenceofpatient-relatedintrinsicfactors(definedastheriskfactorsfromourprevious study)andamultidisciplinarytransitioninterventionontheimprovementofmedicalandpsychosocial outcome.

Results:Sixty-sixoutof114invitedparticipants(57.9%)completedthequestionnaire.Discriminant analysesshowed thatthepatient-relatedintrinsic factorscombined proveda strongpredictorfor improvementinmedicaloutcome(72.7%)andrelativelystrongforeducational/vocationaloutcome (51.5%).Thetransitioninterventionsarearelativestrongpredictorofimprovementinmedicaloutcome (56.1%),educational/vocationaloutcome(53.0%)andimprovementintheoverallriskscore(54.5%).

Conclusion:Based ontheoverall improvement of psychosocialoutcomein mostpatients, and the influenceofatransitioninterventiononmedical,educational/vocationaloutcomeandtheoverallrisk score,itis likely thatadolescents withepilepsy benefit fromvisiting amultidisciplinary epilepsy transitionclinic.

ß2016BritishEpilepsyAssociation.PublishedbyElsevierLtd.Allrightsreserved.

* Correspondingauthorat:DepartmentofResearch&Development,Epilepsy CenterKempenhaeghe,P.O.Box:61,NL-5590ABHeeze,TheNetherlands.

Tel.:+310402279022;fax:+310402265691.

E-mailaddresses:GeerlingsR@Kempenhaeghe.nl,rpjgeerlings@gmail.com (R.P.J.Geerlings),AldenkampB@Kempenhaeghe.nl(A.P.Aldenkamp), GottmerL@Kempenhaeghe.nl(L.M.C.Gottmer-Welschen),a.van.staa@hr.nl (A.L.vanStaa),LouwA@Kempenhaeghe.nl(A.J.A.deLouw).

ContentslistsavailableatScienceDirect

Seizure

j o urn a l hom e pa g e : ww w . e l se v i e r. c om / l oca t e / y se i z

http://dx.doi.org/10.1016/j.seizure.2016.04.004

1059-1311/ß2016BritishEpilepsyAssociation.PublishedbyElsevierLtd.Allrightsreserved.

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providehelpformedical,psychosocialanddevelopmentalissues during adolescence [8,10]. Although the main objective of an epilepsytransitionclinicistorevisethepreviouslymadeepilepsy diagnosisandtreatmentoptions,andtotransfertheadolescentto anadulthealthcaresystem,mosttransitionclinicsalsoprovide special attention for theabove mentioned developmental age- specificissuesofpatientswithepilepsy[4,8,9,11].

Severaldifferent modelsofepilepsytransition clinicstaffing have been reported, including both paediatricians and adult neurologists,ornursespecialist.Sometimesreferraltoapsychol- ogist,asocialworkeroracareeradviserismade[8,9,11–15].Only little evidenceis publishedabout the attempts that have been madetoevaluatetheeffectivenessoftransitioninterventionsin chronic disease [16]. Prior et al. [16] reviewed studies that described health care transition interventions in for example diabetes,kidneydiseaseandjuvenileidiopathicarthritis.However, tothebestofourknowledge,noinformationisavailableaboutthe true,i.e.long-termeffectsoftransitioninterventionsinpatients withepilepsy.Thereforetheobjectiveofthisstudywastoevaluate thelong-termeffectsofamultidisciplinarytransitionintervention comparedtopatient-relatedintrinsicfactorsontheimprovement inmedicalandpsychosocialoutcome.

2. Methods

2.1. Epilepsytransitionclinic

An Epilepsy Transition Clinic was set up in March 2012 in Epilepsy Center Kempenhaeghe, a tertiary referral center for patients with epilepsy. Our transition clinic is staffed with a neurologist,a clinicalneuropsychologist,asocial workerandan educationalist/vocationalcounselor,allwithadequateknowledge ofpaediatricandadolescentdevelopmentalissues,paediatricand adultmedicalcareandofepileptology.

Allpatientswhohadanappointmentforavisitatthetransition clinicwerebetween15and25yearsofage.Asdescribedinour previousstudy[11],this agelimitwaschosenbecausedevelop- mental milestones are often delayed in patients with epilepsy [11,17].Inouropinion,transitionisagradualprocess,andshould notbelimitedbyonlyreachingtheadultage.Togetanappointment atthemultidisciplinaryEpilepsyTransitionClinic,patientshadto haveadiagnosisofepilepsyandatleastonemedicalissue(e.g., problemswithtransitionfrompaediatrictoadultcare),psycho- logical issue (e.g.,in thedevelopment of self-management and independence)orpsychosocialissue(e.g.,careeradvice)relatedto thetransitionphase.Notallpatientshadhadanassessmentoftheir FullScaleIntelligenceQuotient(FSIQ)tomeasuretheirintelligence level inthe past,before their first visit tothetransition clinic.

Patients with severe mental disabilities (FS IQ<50) were not acceptedatthetransitionclinicbutreferredtoaspecialoutpatient clinic for patients with epilepsy and mental disabilities at our epilepsycenter.Allpatients weregiven appointmentswiththe above mentioned health care workers in three consecutive consultations on the same morning (the ‘carousel’) [11]. The neurologistandclinicalneuropsychologistworktogetherinone consultation,afterwhichallpatientshadappointmentswiththe social worker, and the educationalist/vocational counselor. All professionalsstimulate independenceand empowermentof the adolescent. After all three consecutive appointments, the four healthcareprofessionalsdiscusstheprogressoftransitiononthe medical,psychological,socialandeducational/vocationaldomain, inashortmultidisciplinarycase-meeting.Consequentlyaperson- alizedadviceisdiscussedwiththepatient.

Thetransitionclinic’sadvicemayincludeanew‘snap-shot’for adiagnosticwork-up,suchasamagneticresonanceimaging(MRI), electroencephalography(EEG),a neuropsychological test and/or

laboratorytestsorgeneticcounseling.Notallpatientshadastrict medical indication for a full diagnostic work-up. One or more diagnostic procedures(e.g.,MRI, EEG,neuropsychologicaltests) wereonlyconductedwhenconsiderednecessaryforrevisionofthe medicaldiagnosis(e.g.,EEG)orinoptimizingthemultidisciplinary adviceduring thetransitionprocess(e.g.,measurements ofthe FullScaleIntelligenceQuotienttoprovideadequatecareeradvice).

Other advicesincluded support by healthcare workers, e.g., in findinghousing,financialsupport,vocationaltrainingorpsychoso- cialsupport.Allpatientsvisitedthewhole‘carousel’atleastonce.

Somepatientswerefollowedbythetransitionclinic’sneurologist forashortperiod(forexamplebecauseofadiagnosticwork-upor afterarecentchangeinAEDprescription),othershadafewfollow- upvisitsforfurthersupportbythepsychologist,socialworkeror educationalist/vocationalcounselor.Transitionisagradualprocess, andtherewasnosetnumberofmaximumvisitstothetransition clinic. The total numberof visits atthe transition clinic varied, depending on the medical or psychosocial problems of the individual,butfinishedaftertwoorthreevisitspreferably.After completing the transition clinic, the medical transition, i.e. the transfer from paediatric to adult medical care was facilitated.

Patientswereeitherreferredtoanadultneurologistatthetertiary referralepilepsycenter,toanexternaladultneurologist,or,incaseof seizureremissionafterwithdrawalofAEDs,toageneralpractitioner.

Inourpreviousstudy[11],wescoredpatientswhovisitedthe transition clinic on three transitional domains, namely their medicalperformance,educational/vocationalperformanceandthe development of their own independence/separation/identity.

Scores are further defined in Table 1. As mentioned [11], no validated scoring systemto assess the level of functioning on transitionaldomainsinadolescentsoryoungadultswithepilepsy existed.Therefore,wedevelopedourownscoringsystem,basedon theSydneyPsychosocialReintegrationScaleVersion2(SPRS-2),a validatedscoringsystemforpatientswithtraumaticbraininjury.

In our scoring system, scores ranged from 0 (normal), 1 (suboptimal), to 2 (poor). To cope with the wide range of intellectualabilitiesandthemaximumlevelsoffunctioningofthe individual patients, and the comorbid conditions, scores were individually allocated by the transitionclinic’s neurologist and psychologistwithrespecttotheoptimalleveloffunctioningwhich canbeachievedbytheindividual.

Wealsodevelopedariskprofilescoringsystem[11].Thisrisk profile score too was individually allocated by the transition clinic’sneurologistandpsychologistandrepresentedthepatient’s riskforfutureadversepsychosocialoutcome.Ariskprofilescoreof 3indicatedthatthepatienthadpoorperspectivesforlong-term psychosocial outcome, a score of 2 indicated a substantial increased (‘moderate’) risk for adverse psychosocial outcome, andascoreof1indicatedalowrisk(‘noobviousrisk’)forlong- termpsychosocialoutcome[11].

Allscoreswereallocatedbythetransitionclinic’sneurologist and psychologist. If no agreement occurred, discussions were requireduntilconsensus.

Last, theinterventions that tookplace during thetransition clinicwererecordedandcategorizedinthreegroupsaccordingto theinterventions,namely:(a)transitioncliniconly;(b)transition clinicincombinationwithasingleintervention;eithermedical(by theneurologist/neuropsychologist),orsocial(bythesocialworker or educationalist/vocational counselor); (c) transition clinic in combinationwithamultidisciplinaryapproach(bothmedicaland thesocialworkeroreducationalist).

2.2. Studypopulationandstudyprocedure

Allpatients in this follow-upstudywererecruited fromthe study population of our previous study [11]. The minimum

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durationbetweentheinitialvisitatthetransitionclinicandthe invitationforthestudywassixmonths,themaximumduration was three years. Because patients were transitioned from paediatrictoadultmedicalcare, andwereno longerin follow- up at the transition clinic or at our tertiary referral center, a questionnairewassenttoeverypatientwhovisitedourEpilepsy Transition Clinic from March 2012 until September 2014. The questionnairescontainedquestionsregardingthepatient’smedi- cal,educational/vocationalstatus,andindependence.Tobemore specific:weaskedpatients toreporttheir seizurefrequencyby fillingoutanumberandadditionallychoosingtheoption daily/

weekly/monthy/yearly.Dataabouttheir currentandpast treat- mentoptions,theirneurologistandnumberofvisitsperyear,and thenumberofhospitaladmittancesduetoepilepsywerecollected withthe questionnaire. Furthermore, we asked to report their currenteducationoremploymentstatus, their financialincome and,ifapplicable,financial guardianship,relationships, housing, level of independence by completing several household tasks.

Finally,alistofmedicalandsocietalhealthcareworkers,e.g.,the number and frequency of different (health) care providers the patientwasincontactwithatthemoment,wasassessed.

Based on thepatientreported outcome,previously collected baseline statisticswerecompared to thecurrent outcome. The abovementionedperformance scoresand theriskprofile score were re-evaluated and re-allocated (Table 2) according to the definitionsinTable1.

Dataofallpatientswho gavewritteninformedconsent, and fullycompletedthequestionnaire,wereenteredinanIBMSPSS database.

2.3. Statisticalanalysis

Allstatistical analyseswereperformedby IBMSPSS Version 21.Weuseddescriptivestatisticstocomputefrequencies(n)and percentages(%)ofcategoricalvariablesandtogiveanoverviewof baselineandcurrentstatistics.Meansarepresentedwithstandard deviation(SD)andrange.

First,patientsweregroupedaccordingtotheirpastandcurrent riskprofilescores.Patientswithimprovementoftheirriskprofile score,or patients witha persistent lowrisk profile score,were groupedingroup1;patientswithamoderateriskprofilescorewere groupedingroup2;patients withadeteriorationofriskprofile scoreorpersistenthighriskprofilescoreweregroupedingroup 3.Furtherinformationaboutthegroupingofpatientsisprovidedin Table2.Aftergroupingofpatients,patient’sdemographic,medical andsocialcharacteristicswerecomparedusingtheaforementioned criteria for continuous or dichotomous variables. The involved medicaland societalhealth careworkerswereclassifiedintwo ways. In ourfirst analysis theywereclassifiedas dichotomous variables(yes/no)andcomparedbetweengroupsusingtheChi- SquareTest.Inoursecondanalysis,thefrequencyoftheinvolved healthcareworkerswasclassifiedasacontinuousvariableand comparedusingtheIndependent-SamplesTTest.Thethresholdfor significancewasp<0.05inbothanalyses.

Second,weperformeda two-tailedPaired-SamplesTTestto evaluatethedifferenceinpatientcharacteristics(thecontinuous variables),theperformancescores,andriskprofilescoreovertime between baseline and at follow-up. A p-value <0.05 was consideredstatisticallysignificant.

Third, we conducted descriptive discriminant analyses to determinethepredictivevaluesofpatient-relatedintrinsicfactors (definedastheriskfactorsfoundinourpreviousstudy[11])and transitioninterventionsforfinaloutcome.Weusedthedifference (delta) in the three transitional performance scores and the differenceinriskprofilescoresasdependentvariables.Weused twotypesofindependentvariables:first,thecharacteristicsofthe transitionintervention(interventionsduringtransition(ascate- gorizedinSection2above),durationoftimesincefirstvisitatthe transition clinic, and age at first visit at the transition clinic);

second, the risk factors we found in our previous study [11]

(intelligence level, seizure frequency, and an unstable and unsupportive family environment). However, to compare the impactoftransitioninterventionstotheimpactofpatient-related factors,andtoavoidanyconfusionintheanalyses,wechoseto namethepreviouslyfoundriskfactorsas‘patient-relatedintrinsic factors’throughoutthismanuscript.Thepatient-relatedintrinsic factors and the interventions were entered in two separate discriminantanalysesforeachdependentvariable.

Table1

Definitionsofpreviouslydefinedmedical,educational/vocationalandindependence/separation/identityperformancescores.

Normal(Score0) Suboptimal(Score1) Poor(Score2)

Medicalperformance score

Lowseizurefrequencyorseizurefreedom.

Nocomorbidconditions.

Mediumseizurefrequency(monthly).One mentalorphysicalcomorbidcondition.

Highseizurefrequency(daily,weekly).

Multiplementalorphysicalcomorbid conditions.

Educational/vocational performancescore

Maximumeducational/vocational opportunitieswithrespecttothepatient’s individualmentalabilitiesandmaximum leveloffunctioning.

Underemployment,academic underachievementSuboptimal

educational/vocationalopportunitieswith respecttothepatient’smentalabilitiesand maximumleveloffunctioning.

Nostudyorunemployment.Inabilityto keepajob.Pooreducational/vocational opportunitieswithrespecttothepatient’s individualmentalabilitiesandmaximum leveloffunctioning.

Independence/

separation/identity performancescore

Maximumlevelofindependenceand separationfromparents.Or:patientdoes notrequirehelpondailyactivities,making choices,andhouseholdchores,withrespect tothepatient’smentalabilitiesand maximumleveloffunctioning.

Suboptimallevelofindependenceand separationfromparents.Or:patientneeds anyhelpofparentsondailyactivities, choicesandhouseholdchores,withrespect tothepatient’smentalabilitiesand maximumleveloffunctioning.

Poorlevelofindependenceandseparation fromparents.Or:patientneedshelpof parentsonalmostanydailyactivities, choices,andhouseholdchores,withrespect tothepatient’smentalabilitiesand maximumleveloffunctioning.

ThistablewaspublishedbeforeinEpilepsy&Behavior[11].

Table2

Groupinganddistributionpatternofpatients’pastandcurrentriskprofilescores.

Group1:Improvementofriskprofilescoreor persistentlowriskprofilescore

Totalnumberof patients(n=66) Pastscore:3!currentscore:1(2) 2

Pastscore:3!currentscore:2(1) 7 Pastscore:2!currentscore:1(1) 14

1=1 11

Total 34(52%)

Group2:Stablemoderateriskprofilescore

2=2 6(9%)

Group3:Deteriorationofriskprofilescoreor persistenthighriskprofilescore

Pastscore:1!currentscore:2(+1) 1 Pastscore:1!currentscore:3(+2) 3 Pastscore:2!currentscore:3(+1) 7

3=3 15

Total 26(39%)

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2.4. Ethics

ThisstudywasapprovedbytheMedicalEthicsCommitteeof Kempenhaeghe. Patients could participate voluntarily. Written informedconsentwasobtainedfromallparticipants.

3. Results

3.1. Respondercharacteristics

3.1.1. Responderversusnon-responderanalysis

A totalof 114 patients were assessedat baseline and were invited to participate in this study. In total, completed ques- tionnaireswereobtained from66 patients (57.9%); 48 patients (42.1%)werenon-responding.Thepresenceofanunsupportive/

unstablefamilyenvironmentwassignificantlydifferentbetween respondersandnon-responders(21.2%vs.39.6%,p=0.04).Non- respondersalsohad asignificantly lower(=worse)performance score in the past for their level of independence/separation/

identity (1.12 vs. 1.48, p=0.02), and a significantly higher (=worse)riskprofilescore(2.14vs.2.44,p=0.04).Amongtheother characteristicsno statisticallysignificantdifferenceswerefound betweenrespondersandnon-responders.

3.1.2. Patientcharacteristics

Patientcharacteristicsatbaselineandatfollow-upareshownin Table 3. In total, 35 men (53.0%) and 31 women (47.0%) participated in this study. Their mean age was 18.9 years at baseline (median=18.6, SD=2.2) and 20.8 years at follow-up (median=20.7, SD=2.3). The mean Full Scale Intelligence Quotient was 83 (median=81, SD=16.9). The mean age at diagnosisofepilepsywas8.1years(median=8.1,SD=5.0),with ameandurationofepilepsyof10.6yearsatbaseline(median=9.5, SD=5.3) and 12.6 years at follow-up (median=11.9,SD=5.4).

Fifty-twopatients(78.8%)hadalocalization-relatedepilepsy,of which the cryptogenic type was most common (35 patients, 53.0%).Atbaseline,39outof66patients(59.1%)wereseizure-free foroneyear,comparedto38outof66patients(57.6%)atfollow- up.(Thedifferenceinseizure frequencyovertime hada p-vale of 0.81). Less patients were using polytherapy at follow-up (29patients(43.9%)vs.32previously(48.5%),p=0.27),andnine patients (13.6%) were not using AEDs anymore, compared to 7patients(10.6%)atbaseline.Theirself-reportedAEDadherence was higher at follow-up (80.3% vs. 57.6%). Compared to the baseline characteristics, more patients were independent from theirparentsatfollow-up(42vs.30,63.6%vs.45.5%respectively), andmorepatientsweresociallyparticipatingatfollow-up(50vs.

47,75.8%vs.71.2%respectively).Eightpatients(12.1%)wereliving eitherindependentlyorinasupportedaccommodationcompared to4patients(6.1%)atbaseline.Morepatientswereemployedat follow-up (31 vs. 42, 47.0% vs. 63.6%), and less patients were studying(13vs.6,19.7%vs.9.1%)orinaninternshipduringtheir study(13vs. 1,19.7% vs1.5%).Finally, morepatientswerenot studyinganymoreandhadnotfoundajobafterwardscompared tobaseline(unemployment7vs.17,10.6%vs.25.8%).Thirty-six patients(54.5%)hadasalaryoutofajoborinternship,whereas 29patients(43.9%)wereonsomekindofgovernmentalfinancial support.

TransitionclinicinterventionsareshowninTable4.Themean durationoffollow-upbetweenthetransitioninterventionsandthe currentstudywas23.7months(median=24.1,SD=10.4).After theirfirstvisitattheepilepsytransitionclinic,adiagnosticwork- upwasdonein56patients(84.8%),involving35(53.0%)clinical neuropsychologicalassessmentsandEEGrecording.Furthermore, 17MRIs(25.8%)and22laboratorytests(33.3%)wereperformed.

Twenty-two patients (33.3%) werebriefly admitted (<24h) to

complete the diagnostic work-up. Forty-two patients (63.6%) underwentachangeinAEDprescriptionbasedontheevaluations ofthemultidisciplinarytransitionclinic,ofwhich3patients(4.5%) werewomeninchild-bearingageusingvalproate.Aftervisiting the transition clinic, two patients (3.0%) were referred for the implantationofavagalnervestimulator.

Thesocialworkerwasconsultedin26patients(39.4%),e.g., to provide help with housing assistance (15 patients, 22.7%), for adviceabout financialguardianship(6patients,9.1%) orto assist in the separation from parents (4 patients, 6.1%). The educationalist/vocationalcounselorprovidededucationalassis- tancein 18patients (27.3%),vocationalassistance (6patients, 9.1%) or vocational training (4 patients, 6.1%). Psychosocial assistancewasindicatedin10patients(15.2%).

In43patients(65.2%)amedicaltransitionwasfacilitatedtoan adult neurologist at our tertiary referral epilepsy center, and sixteen patients (24.2%) were referred to an external adult neurologistforfurtherepilepsycare.Sevenpatients(10.6%)with seizure remission after AED withdrawal no longer needed specialized epilepsy care and were referred to their general practitioner.

Summarized, 12 patients (18.2%) had only one visit at the transition clinicwithout further consultation, diagnosticproce- dureorfollow-upinthetransitionoutpatientclinic;20patients (30.3%)visited thetransition clinicand had eithera diagnostic follow-uporafollow-upconsultationwiththepsychologistorthe socialworkerandeducationalist;34patients(51.5%)visitedthe transitionclinicandhadamultidisciplinaryfollow-upincludinga medicalintervention(eitherdiagnosticwork-uporchangeinAED prescription)incombinationwithaconsultationatthepsycholo- gist,socialworkeroreducationalist.

AsshowninTable5,theriskprofilescoreimproved,butnot statistically significant (2.14 at baseline vs. 1.97 at follow-up, p=0.12).Themedicalperformancescoreimprovedfrombaseline mean1.09,SD=0.87toamean0.60,SD=0.90(p<0.001),the educational/vocationalperformancescoreimprovedfrom1.06, SD=0.86tomean 0.82,SD0.89(p=0.01);theindependence/

separation/identity performance score improved from 1.12, SD=0.80to0.94,SD=0.88(p=0.04).

3.2. Medicalandsocietalhealthcareworkers

Abeneficialoutcomewasnotsignificantlycorrelatedwiththe involvementofmedicalandsocietalhealthcareworkersbothin termsoftypeofhealthcareworkerandquantityofsupport,except fortheinterventionbyapsychologist(23.5%comparedto3.1%, p=0.02).

3.3. Descriptivediscriminantanalyses

We used the interventions and the patient-related intrinsic factors[11]aspredictivevariablesinadiscriminantanalysesto evaluatetherelativeimpactofthevariablesonthedeltainrisk profile score and performance scores. As mentioned above in Section 3, all scores improved at follow-up. The results of the discriminantanalysisareshowninTable6.

3.3.1. Improvementinmedicaloutcome

The interventions combined showed a sensitivity of 56.5%

andaspecificityof52.6%onimprovementofmedicaloutcome.

Theinterventionscombinedcouldpredict56.1%oftheclassifica- tionofmedicalimprovement.

The patient-related intrinsic factors showeda sensitivity of 80.4%andaspecificityof52.6%.Intotal72.7%oftheimprovement inmedicaloutcomecanbepredictedwhencombiningthepatient- relatedintrinsicfactors.

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3.3.2. Improvementinindependence/separation/identity

Theinterventionscombinedshowedasensitivityof38.7%and a specificity of 54.2% on the improvement of independence/

separation/identityoutcome. Incombinationthe interventions

could predict 43.9% of the improvement of independence outcome.

The patient-related intrinsicfactors showed a sensitivity of 41.9%andaspecificityof29.2%.Intotal42.4%oftheimprovement Table3

Demographic,epilepsy-relatedandpsychosocialvariablesatbaselineandatfollow-up.

Baselinecharacteristics Atfollow-up p-value

Median SD Range Median SD Range

Gender Men Women

35(53.0%) 31(47.0%) Meanage

Youngerthan18yearsofage

18.9 27(40.9%)

18.6 2.2 15–25 20.8

6(9.1)

20.7 2.3 16–26

MeanFullscaleIntelligence(FSIQ) IQ100

IQ90–100 IQ70–90 Notassessed

83 31(47.0%) 19(28.8%) 15(22.7%) 1(1.5%)

81 16.9 51–113

Meandurationofepilepsy(years) 10.6 9.5 5.3 0.4–19.6 12.6 11.9 5.4 1.2–22.1

Meanageatdiagnosisofepilepsy(years) 8.1 8.1 5.0 (0.1–17.4) Typeofepilepsy

Localization-relatedepilepsy Idiopathic

Symptomatic Cryptogenic Generalizedepilepsy

Idiopathic Symptomatic Cryptogenic Notclassifiedyet

52(78.8%) 2(3.0%) 15(22.7%) 35(53.0%) 13(21.2%) 10(15.2%) 3(4.5%) 0(0%) 1(1.5%) Seizurefrequency

Daily Lastweek Lastmonth Lastyear

Seizurefree>1year Unknown

3(4.5%) 7(10.6%) 10(15.2%) 3(4.5%) 39(59.1%) 5(7.6%)

6(9.1%) 5(7.6%) 7(10.6%) 9(13.6%) 38(57.6%) 1(1.5%)

0.81

MeannumberofAEDs NocurrentAEDtreatment Monotherapy

Polytherapy(2–4AEDs)

1.56 7(10.6%) 27(40.9%) 32(48.5%)

1.0 0.95 0–4 1.45

9(13.6%) 27(40.9%) 29(43.9%)

1.0 0.98 0–4 0.27

Self-reportedAEDadherence Yes/mostlikelyyes No

NocurrentAEDtreatment Unknown

38(57.6%) 7(10.6%) 7(10.6%) 14(21.2%)

53(80.3%) 4(6.1%) 9(13.6%) 0(0%) Previoustherapies

Epilepticsurgery VagalNerveStimulator Ketogenicdiet

4(6.1%) 2(3.0%) 1(1.5%)

4(6.1%) 4(6.1%) 1(1.5%) Specialeducationprogram

Inthepast/ever Current

28(42.4%)

15(22.7%) 6(9.1%)

Livingarrangements Athomewithparents Independently

Supportedaccommodation Unknown

61(92.4%) 2(3.0%) 2(3.0%) 1(1.5%)

58(87.9%) 3(4.5%) 5(7.6%) 0(0%)

Socialparticipation 47(71.2%) 50(75.8%)

Independence 30(45.5%) 42(63.6%)

Unsupportive/unstablefamilyenvironment 15(22.7%)

Employment Yes No Internship

Studentwithoutajob Unknown

31(47.0%) 7(10.6%) 13(19.7%) 13(19.7%) 2(3.0%)

42(63.6%) 17(25.8%) 1(1.5%) 6(9.1%) 0(0%) Financialincome(somepatientshad>1income)

Job/salary/internship Governmentalsupport Parents

36(54.5%) 29(43.9%) 6(9.1%) Dataarepresentedasnumber(n,%).Meansarepresentedwithmedian,standarddeviation(SD)andrange.

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inindependence canbepredictedwhen combiningthepatient- relatedintrinsicfactors.

3.3.3. Improvementineducational/vocationaloutcome

Theinterventionscombinedshowedasensitivityof57.5%anda specificityof45.5%ontheimprovementofeducation/vocational outcome. In combination the interventionscouldpredict 53.0%

oftheimprovementofeducationaloutcome.

The patient-related intrinsic factors showeda sensitivity of 45.0%andaspecificityof59.1%.Intotal51.5%oftheimprovement ineducational/vocationoutcomecanbepredictedwhencombin- ingthepatient-relatedintrinsicfactors.

Table4

Transitionclinicinterventions.

Median SD Range

Durationoffollow-upatthetransitionclinic(months) 23.7 24.1 10.4 6.6–40.3

Diagnosticwork-upafterfirstvisitatthetransitionclinic (somepatientshad>1typeofdiagnosticintervention) Clinicalneuropsychologicalassessment

EEG MRI

Admittancefordiagnosticwork-up Laboratory

56(84.8%) 35(53.0%) 35(53.0%) 17(25.8%) 22(33.3%) 22(33.3%)

AEDchangeafterevaluationatthetransitionclinic ReasonAEDchange

Epilepsyremission Sideeffects SwitchAED AddingAED IncreasedoseAED DecreasedoseAED Womeninchildbearingage

42(63.6%)

12(18.2%) 10(15.2%) 5(7.6%) 5(7.6%) 4(6.1%) 3(4.5%) 3(4.5%) Consultationstransitionclinic(somepatientshad>1typeofintervention/consultation)

Socialworker Housingassistance

Reasonimprovingfamilysupport

Reasonimprovingseparation/individualization Reasonfinancialadvice

Reasonincreasingsocialinteractionandsupport Reasonplanningdailyactivities

Educationalist/vocationalcounselor Educationalassistance

Vocationalassistance Vocationaltraining Psychologicalassistance

26(39.4%) 15(22.7%) 5(7.6%) 4(6.1%) 6(9.1%) 1(1.5%) 1(1.5%) 25(37.9%) 18(27.3%) 6(9.1%) 4(6.1%) 10(15.2%) Referralto

Adultneurologistwithintheepilepsycenter Externalreferraltoadultneurologist Generalpractitioner

43(65.2%) 16(24.2%) 7(10.6%) Typeofinterventionssummarized

Transitioncliniconly

Transitionclinic+monodisciplinaryintervention Transitionclinic+multidisciplinaryintervention

12(18.2%) 20(30.3%) 34(51.5%) Dataarepresentedasnumber(n,%).Meansarepresentedwithmedianandrange.

Table5

Medical, educational/vocational and independence/separation/identity perfor- mancescoresandriskprofilescoreatbaselineandatfollow-up.

Baseline Follow-up p-value Medical

performancescore

1.09(0.87) 0.60(0.90) <0.001

Educational/vocational performancescore

1.06(0.86) 0.82(0.89) 0.01

Independence/separation/

identityperformancescore

1.12(0.80) 0.94(0.88) 0.04

Riskprofilescores 2.14(0.76) 1.97(0.89) 0.12 Dataarepresentedasmeanscoreswithstandarddeviation(SD).

Table6

Sensitivityandspecificityofinterventionsandpatient-relatedintrinsicfactorsinrelationtoimprovementofpsychosocialormedicaloutcome.

Deltascore Sensitivity Specificity Correctlyclassified

Medicalperformancescore Interventions 56.5% 52.6% 56.1%

Patient-relatedintrinsicfactors 80.4% 52.6% 72.7%

Independence/separation/identityperformancescore Interventions 38.7% 54.2% 43.9%

Patient-relatedintrinsicfactors 41.9% 29.2% 42.4%

Educational/vocationalperformancescore Interventions 57.5% 45.5% 53.0%

Patient-relatedintrinsicfactors 45.0% 59.1% 51.5%

Riskprofilescore Interventions 46.2% 58.8% 54.5%

Patient-relatedintrinsicfactors 23.1% 61.8% 45.5%

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3.3.4. Improvementinriskprofilescore

Theinterventionscombinedshoweda relativemodestsensi- tivityof46.2%andspecificityof58.8%inclassifyingthegroups.In totalthetypeofinterventionscouldpredictclassificationofrisk improvementin54.5%.

Thepatient-relatedintrinsicfactorscombinedhadasensitivity of 23.1% on an improvement on the risk profile score, with a specificityof61.8%.Thepatient-relatedintrinsicfactorscombined couldpredictcorrectclassificationofriskimprovementin45.5%of thepatients.

4. Discussion

This study compared changes in transition characteristics from baseline to a follow-up on average two years later in 66patientswithameanageof18.9yearsandameanFullScale Intelligence Quotient of 83. On average they had a mean durationofepilepsyof12.6yearsatfollow-up.Thisistherefore a groupwith chronic(mostlycryptogeniclocalization-related) epilepsy that transits with epilepsy from childhood to adult- hood.Norelevantchangeswerefoundforseizurefrequency,but withrespecttotreatment,lesspatientswereonpolytherapy,in more patients all AEDs were withdrawn and AED adherence hadimproved. Intermsof transition outcomes,more patients were living independently from their parents, more were socially participating, and more patients were employed at follow-up.

The results of the performance scores improved, which is in line with the aforementioned descriptive results: the risk profile score improved, but this was not statistically significant.

Themainobjectiveofthisstudywastoevaluatethelong-term effectsofamultidisciplinarytransitioninterventioncomparedto theimpactofpatient-relatedintrinsicfactorsontheimprovement inmedicaland psychosocialoutcome.Thus,thecontributionof transition interventions on the positive delta (difference) in performancescores.Thediscriminantanalysescombinedindicat- edthatthepatient-relatedintrinsicfactorscombinedareastrong predictor of improvement in medical outcome (72.7%). Our interpretation is that the patient-related intrinsic factors are inherent characteristics of the patients, e.g., the fact that the majority of the patients had a chronic epilepsy, and define a relativelystablesituation.

Thetransitioninterventionsontheotherhandareanequally strongpredictoras patient-related factorsfor improvement in educational/vocational outcome, independence, and the im- provement in the overall risk score. Here transition inter- ventions can have more influence than the patient-related intrinsicfactorsonimprovementof theeducational/vocational outcome.

Inpredictingafavorableoverallrisk,thesensitivityoftype ofinterventionismuchhigherthanthepatient-relatedintrinsic factors,withequalspecificity.Thisagainillustratestheimpact oftransitioninterventionsfor theoverallpositiveresult.Thus, the type of transition intervention contributed more to an improvementof theriskprofile scorethanthepatient-related intrinsic factors, which is understandable given the type of patient-relatedintrinsicfactors(i.e.,relativestablefactorsthat all will have a similar influence at follow-up compared to baseline).

Theimprovementinpsychosocialoutcomecannotbeexplained by the involvement of individual health care workers or the frequencyofappointmentswithhealthcareworkers.

Nocomparativestudiescanbefoundinliterature,sincethisis thefirststudyshowingtheeffectsofamultidisciplinarytransition interventionafterlong-termfollow-up.

4.1.1. Strengthsandlimitations

This study has some methodological limitations. The first limitationistheuseofquestionnairestoobtainmoreinformation atfollow-up.Sincewearefollowingpatientsthroughthetransition process,mostpatientswerenolongerinfollow-upatourtransition clinicoratourtertiaryreferralhospital.Inthepilot-phaseofour study,manypatientsindicatedthattheywouldnotparticipateinthe studywhentheyhadtocomeovertoourcenterforaninterview because of the long distance to our tertiary epilepsy center.

Therefore,wehavetorelyontheself-reporteddata.

Also, we were unable to re-evaluate the most significant variable in ourprevious study, namelyan unsupportive family environment.Thisvariablewassignificantlyworseamong non- responders. Further, non-responders had a significantly worse psychosocialoutcomeatbaseline,indicatingthattheresponders toourquestionnairesprobablyhaveabetterchanceforabeneficial long-term psychosocial outcome at baseline. Therefore, results of this studymight not begeneralizable totheadolescent and youngadultpopulationwithepilepsy.

Last,the‘patient-relatedintrinsicfactor’isbasedontheresults ofpreviouslyfoundriskfactors.Multiplevariableswerenottested.

5. Conclusion

Basedontheoverallimprovementofpsychosocialoutcomein mostpatients,and theinfluenceofa transitioninterventionon medical, educational/vocational outcome and the overall risk score, it is likely that adolescents with epilepsy benefit from visitingamultidisciplinaryepilepsytransitionclinic.

Conflictofintereststatement

Theauthorshavenoconflictsofinteresttodeclare.

Acknowledgments

Thisresearchwasfunded by theprovinceof Noord-Brabant (‘Leefbaarheid@Branbant’), The Netherlands, withgrand number C2134073.

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