www.jped.com.br
ORIGINAL
ARTICLE
Prolonged
toilet
training
in
children
with
Down
syndrome:
a
case---control
study
夽
Flávia
Cristina
de
Carvalho
Mrad
a,b,∗,
André
Avarese
de
Figueiredo
a,
José
de
Bessa
Jr.
c,
José
Murillo
Bastos
Netto
a,daUniversidadeFederaldeJuizdeFora(UFJF),DepartamentodeCirurgia,NúcleoInterdisciplinardePesquisaemUrologia(NIPU),
JuizdeFora,MG,Brazil
bUniversidadeFederaldeMinasGerais(UFMG),DepartamentodePediatria,BeloHorizonte,MG,Brazil cUniversidadeEstadualdeFeiradeSantana(UEFS),DepartamentodeCirurgia,FeiradeSantana,BA,Brazil
dFaculdadedeCiênciasMédicasedaSaúdedeJuizdeFora(SUPREMA),HospitaleMaternidadeTherezinhadeJesus,
DepartamentodeCirurgia,JuizdeFora,MG,Brazil
Received23December2016;accepted25April2017 Availableonline1September2017
KEYWORDS
Children; Downsyndrome; Functional constipation; Lowerurinarytract symptoms;
Toilettraining
Abstract
Objectives: ChildrenwithDownsyndromehavedelayedpsychomotordevelopment,whichis
afactor that influences thelevel ofdifficultyin toilettraining. The currentstudy aimsto estimatetheagetoilettrainingstartsandcompletesinchildrenwithDScomparedtochildren withnormalpsychomotordevelopmentandtoevaluatethemethodandtypeoftoilettraining mostfrequentlyused,aswellasitsassociationwithlowerurinarytractsymptomsandfunctional constipation.
Methods: Acase---controlstudy wascarried outfrom2010to2015.Allparentscompleteda questionnairedesignedtoassessthetoilettrainingprocess.Lowerurinarytractsymptomswere assessedthroughtheapplicationoftheDysfunctionalVoidingSymptomScore.Thepresenceof functionalconstipationwasassessedaccordingtotheRomeIIIcriteria.
Results: Thestudy included93children with Downsyndromeand204children withnormal
psychomotordevelopment(controlgroup[CG]).Themeanageoftoilettrainingonsetwas22.8 monthsinthosewithDSand17.5monthsintheCG(p=0.001).InchildrenwithDS,themeanage whencompletingtoilettrainingwas56.2monthsand27.1monthsintheCG(p=0.001).Among childrenwithDS,femalescompletedtoilettrainingearlier(p=0.02).Thetoilettrainingmethod usedmostoftenwaschild-orientedapproachinbothgroups.Noassociationwasobservedwith
夽
Pleasecitethisarticleas:MradFC,Figueiredo AA,BessaJ,NettoJM.ProlongedtoilettraininginchildrenwithDownsyndrome:a
case---controlstudy.JPediatr(RioJ).2018;94:286---92.
∗Correspondingauthor.
E-mail:flaviamrad@terra.com.br(F.C.Mrad).
https://doi.org/10.1016/j.jped.2017.06.011
thepresenceoflowerurinarytractsymptomsorfunctionalconstipationandtheageofbeginning andcompletingtoilettraininginbothgroups.
Conclusion: ChildrenwithDownsyndromeexperiencedprolongedtoilettrainingtime. Prospec-tivelongitudinalstudiesareessentialtogaininsightintothetoilettrainingofthesechildren. ©2017PublishedbyElsevierEditoraLtda.onbehalfofSociedadeBrasileiradePediatria.Thisis anopenaccessarticleundertheCCBY-NC-NDlicense(http://creativecommons.org/licenses/ by-nc-nd/4.0/).
PALAVRAS-CHAVE
Crianc¸as;
SíndromedeDown; Constipac¸ão funcional; Sintomasdotrato urinárioinferior; Treinamento esfincteriano
Treinamentoesfincterianoemcrianc¸ascomSíndromedeDown:estudocaso-controle
Resumo
Objetivos: Crianc¸ascomsíndromedeDownapresentamdesenvolvimentopsicomotoratrasado, fatorqueinfluenciaoníveldedificuldadedotreinamentoesfincteriano.Opresenteestudotem comoobjetivoestimaraidadeemqueotreinamentoesfincterianoéiniciadoeconcluídoem crianc¸ascomSDemcomparac¸ãoacrianc¸ascomdesenvolvimentopsicomotornormal,avaliaro métodoeotipodetreinamentoesfincterianoutilizadocommaiorfrequência,bemcomosua associac¸ãocomsintomasdotratourinárioinferioreconstipac¸ãofuncional.
Métodos: Umestudocaso-controlefoirealizadode2010a2015. Todosospaispreencheram um questionáriodestinadoaavaliaroprocessodetreinamentoesfincteriano.Osintomasdo tratourinárioinferiorforamavaliadospormeiodaaplicac¸ãodoDysfunctionalVoidingSymptom Score.Apresenc¸adeconstipac¸ãofuncionalfoiavaliadadeacordocomoscritériosRomaIII. Resultados: Oestudoincluiu93crianc¸ascomsíndromedeDowne204crianc¸ascom desenvolvi-mentopsicomotornormal(GrupodeControle(GC)).Aidademédiaemqueascrianc¸asiniciaram otreinamentoesfincterianofoide22,8mesesnaquelascomSDe17,5mesesnoGC(p=0,001). Emcrianc¸ascomSD,aidademédiaaoconcluirotreinamentoesfincterianofoide56,2meses e27,1mesesnoGC(p=0,001).Entreascrianc¸ascomSD,asdosexofemininoconcluíramo treinamento esfincterianomaiscedo(p=0,02).Ométododetreinamentoesfincterianomais utilizadofoiaabordagemvoltadaparaacrianc¸aem ambososgrupos.Nãohouveassociac¸ão comapresenc¸adesintomasdotratourinárioinferiorouconstipac¸ãofuncionaleaidadeno inícioenaconclusãodotreinamentoesfincterianoemambososgrupos.
Conclusão: Crianc¸ascomsíndromedeDownapresentaram tempodetreinamento esfincteri-ano prolongado. Estudos longitudinaisprospectivos são essenciais para obter uma visão do treinamentoesfincterianodessascrianc¸as.
©2017PublicadoporElsevierEditoraLtda.emnomedeSociedadeBrasileiradePediatria.Este ´
eumartigoOpenAccesssobumalicenc¸aCCBY-NC-ND(http://creativecommons.org/licenses/ by-nc-nd/4.0/).
Introduction
Down syndrome (DS) is the most common chromosomal malformation,1 witha medianlife expectancyof approxi-mately58years.2DSbirthsaccountforabout1---3per1000 livebirths.3Approximately64%ofindividualswithDShave atleastonemajorassociatedcongenitalanomaly.1Several studiesshowahighprevalenceofabnormalfindingsinthe lowerurinarysystem,whichcanleadtoprogressivekidney orurinarydisorders.1,4,5ChildrenwithDShavedelayed psy-chomotordevelopmentandproblemswithlearning,6being themostcommongeneticcauseofintellectualdisability.2
Toilet trainingfor a child withany disability, including DS,isonethemostchallengingdevelopmentalmilestones, asitis for any child.7,8 Toilettrainingmethods have fluc-tuatedoverthelast100years.9Child-orientedapproachis recommendedbytheAmericanAcademyofPediatrics(AAP) forchildrenwithnormalpsychomotordevelopment,which suggestsbeginningthetoilettrainingprocessonlywhenthe children shows readiness signs.9---11 Therefore, this model of toilet training comprises three variant forces in child
development:physiologicalmaturation,externalfeedback, and internal feedback.10,11 An alternative approach, is the intensive, structured, and parent-oriented method for toilet training in one day based on the principles of conditioningandimitationdevelopedbyAzrinandFoxx.9,12 Other methods that have emerged include the assisted infanttoilettraining,elimination communication,andthe daytimewettingalarm.9
45.7months,8,16 andthemeanageat completionoftoilet trainingwasreportedtobebetween48and66months.8,16---18 ABrazilianstudywithchildrenwithintellectualdisabilities ofvariousetiologies,including15withDS,showedthat99% weretoilettraineduntil60monthsage.19
Thecurrentstudyaimedtoestimatetheagetoilet train-ingstartsand completesin childrenwithDS comparedto childrenwith normalpsychomotor development,evaluate themethodandtypeoftoilettrainingmostfrequentlyused, andthereadinesssignsidentified,aswellastheassociation betweentoilet trainingand lowerurinarytractsymptoms (LUTS)andfunctionalconstipation.
Patients
and
methods
The study group comprised 127 children with DS, aged 3---14years,whoregularlyattendedaspecializedclinicfor DS from February 2010 through February 2015. A group comprisedof225childrenwithnormalpsychomotor devel-opment,already toilet trained,matched for gender, age, and socioeconomic status, who attended a pediatric and adolescentcliniccomprisedthecontrolgroup(CG).
Children with a current urogenital disorder, untreated hypothyroidism,currentuseofmedicationsorwhohad dis-easesknowntointerferewithbladderorsphincterfunction, thosewhostillworediapersuntiltheendofthestudy,and thoseagedlessthan3yearswerenotincluded.
A questionnaire based on the study by Stadtler et al. and Mota et al. was developed and applied to the par-ents toassess thehistory of toilet training,includingage at which a child started and completed toilet training, readinesssignspresented,useofpottychairortoilet,and toilet training methods,10,15 (Appendix A) urinary abnor-malities,presenceofchronicdiseases,useofmedications, andsocioeconomicstatus.Ageatwhichtoilettrainingwas completedwasdefined astheage at whicha child main-tainedfullbladder andbowelcontrol,withoutany failure inholding urine or stoolduring the dayand night.14 Chil-drenwithDSwhouseddiapersinthefirstevaluationwere re-evaluatedaftercompletingtoilettraining,whenthe par-entsansweredthequestionnaire.Thisquestionnaireaimed tocollect the child’s history andhas not been externally validated.TheprevalenceofLUTSwasestimatedusingthe DysfunctionalVoidingSymptomScore(DVSS),20adaptedand validatedforthispopulation.21Thecutoffvaluestoindicate thepresenceofLUTSweregreaterthan6forfemalesand greaterthan9 formales.The presenceof functional con-stipationwasassessedaccordingtotheRomeIIIcriteria.22 Childrenwereonlyevaluatedwhentoilettrainingwas com-pleted.Acompletephysicalexamandstandardassessment wasperformedonallsubjects(includingevaluationofthe lumbosacralregion,searchingforoccultspinaldysraphism). Parentswereaskedabouthistoryofurinarytractinfection, andaurineculturewascollected.
Data were expressed as means±SD, medians, and interquartile ranges, or asabsolute values and fractions. Student’st-testwasusedtocomparecontinuousvariables, while categorical variables were compared using Fisher’s exact test. Standardized mean difference (SMD) and the respectiveconfidenceintervalswereadditionallyexpressed asameasureoftheeffectssizes.Alltestsweretwo-sided;
p<0.05 and 95% confidence interval(CI) wereconsidered statisticallysignificantandwereperformedusingGraphPad Prism® (Prism, version 7.00 for Windows, GraphPad Soft-ware,CA,USA).
The current study (amendment protocol number 1960.019.2010) was approved by the University Ethics Committee, and all parents or those responsible for the patientsinbothgroupssignedaninformedconsentform.
Results
Ofthe127childrenwithDS,93wereincludedinthestudy. Ofthese,30childrenwhouseddiapersinthefirstevaluation werere-assessedaftercompletingtoilettraining,whenthe parentsansweredthequestionnaire.Ofthe34notincluded, threerefused toparticipate, 14were younger thanthree yearsofage, onegirlwasdiagnosedwithWestsyndrome, two boys had a posterior urethral valve, one girl had a decompensatedventricularseptaldefect,andfivechildren werediagnosedwithuntreatedhypothyroidism.Eight chil-dren had not completedtoilet trainingby the end of the study(threegirlswithameanageof3.9±2yearsandfive boyswithameanageof4.3±2.3years);ofthese,allhad untreatedfunctionalconstipation.TheCGconsistedof204 childrenwithnormalpsychomotordevelopment.
The mean age of patients when the questionnaire was answered was 7.7±2.0 years in children with DS and 7.3±3.1 years in the CG (p=0.24). Females repre-sented 47.3% (44/93) of the children with DS and 49.5% (101/204) of the CG (p=0.80). No statistical difference wasobservedregardingsocioeconomicstatusbetweenthe groups(p=0.36).
Children with DS initiated their toilet training at age of 22.8±5.3 months and in CG, at 17.5±4.4 months (SMD=5.52, 95% CI 4.26---6.78, p=0.0001). The time requiredtocompletetoilettrainingwas33.4±11.7months in children with DS and 9.6±6.2 months in the CG (SMD=24.04, 95% CI 21.94---26.14, p=0.0001). The mean ageatwhichtoilettrainingwascompletedwas56.2±12.6 monthsinthechildrenwithDSand27.1±8.6monthsinthe CG(SMD=29.12,95%CI27.08---31.47,p=0.0001;Table1).
In children with DS, the mean age to complete toi-let training in girls was53.1±12.7 months, and in boys, 58.9±12.1months(SMD=−5.89,95%CI−10.99to−0.80, p=0.02).IntheCG,nostatisticaldifferencewasobserved regarding gender:the meanagefor girls tocomplete toi-lettrainingwas 27.8±8.9monthsand inboys, 27.1±6.9 months(p=0.54).
Table1 MeanagefortoilettraininginchildrenwithDown syndromeandinthecontrolgroup.
TT Meanage (months)
DS CG p-Value
Start 22.8±5.3 17.5±4.4 0.001 Completion 56.2±12.6 27.1±8.6 0.001 Time 33.4±11.7 9.6±6.2 0.0001
TT, toilet training; DS, Down syndrome; CG, control group.
Regarding readiness signs, 90% of the parents of chil-drenwithDSrespondedthatabilitytowalkanddiscomfort withwetdiaperswasobserved,1%reportedthatthechild pointed or walked to the bathroom or potty chair, 1% reportedthatthechildindicatedtheneedtovoid,and8% showednoreadinesssigns.80%oftheparentsintheCGdid not remember the readinesssigns; therefore, comparison betweenthegroupswasnotpossible.
The child-oriented approachwasthemethodof choice fortoilettrainingin87.1%(81/93)ofchildrenwithDSand in81.9% (167/204)ofthoseof theCG(p=0.31).Allother familiesusednon-specifiedmethodsfortoilettraining.
Whenassessingthetypeofequipmentusedduringtoilet training,88.2% (82/93)of thechildren withDSand71.6% (146/204)oftheCGusedapottychair(p=0.30),and11.8% (11/93)ofthechildren withDSand28.5%(58/204)of the CGusedthetoiletwithseatreducer(p=0.10).
LUTSwasobservedin19.4%(18/93)ofthechildrenwith DSandin14.7%(30/204)ofthoseinCG(p=0.31).The pres-enceoftheLUTSwasnotassociatedwiththeaverageage of start and completionof toilet training in either group (p>0.05).
Functional constipation wasobserved in 32.3% (30/93) of thechildren withDSand in34.8% (71/204) ofthosein CG(p=0.69).Itwasfoundthat30%(9/30)ofchildrenwith DSand32.4%(23/71)ofthoseinCGwithfunctional consti-pationwerealreadyreceivingtreatmentwithpolyethylene glycol (p=0.84). The presence of functional constipation was notassociated with the mean age of start and com-pletionoftoilettraininginbothgroups(p>0.05).
Onlyasmallnumberofparents ofthechildren withDS (16%[15/93])andofthechildrenintheCG(14.2%[29/204]) soughtmedicaladviceontoilettraining(p=0.51).
Discussion
Few studies address the process of toilettraining in chil-dren with disabilities,9,23 includingthose withDS.8,16---18 It is essentialtoassess thebesttimetostart toilettraining processbyobservingthechild’sdevelopmentalreadiness.10 Contemporary literature has demonstrated that readiness signsinchildrenwithnormalpsychomotordevelopmentare welldevelopedbetween24and36monthsofage13,24 and, inthosewithDS,after36monthsofage.8Readinesssigns include mainly motor skills, appropriate language, desire to please based on a positive relationship with the par-ent,abilitytoimitatetheparents,andspecificskillsabout toilet training (staying dry for more than twohours, dis-comfortwithwet/dirtydiapers,andindicatingtheneedto void).8,10,11,13 In the present study, 90% of the parents of children withDS reported that they considered the abil-ity to walk and the discomfort by wet diapers sufficient tobegintoilettraininginthesechildren, whichmayhave contributedtoanearlytoilettraininginitiationinthese chil-dren(22.8monthsold).Theearlystartintoilettrainingin thispopulationwasconfirmedwhencomparingthepresent resultstothecurrentguidelinesfromtheNationalDown Syn-drome Society (NDSS),8 whichrecommend waiting for the thirdbirthdayforinitiatetoilettraining.Powersetal.also demonstratedalaterage(45.6months)ofinitialinterestin toilettrainingintheirpatients.16
TheinclusionofaCGallowedtheauthorstodemonstrate a29-monthdelayforcompletingtoilettraininginchildren withDS (56vs.27 months).In arecent studywitha sim-ilardesign, Powers etal. identified adelay of 39months andan age of 66 monthsfor toilet training completion.16 Althoughthemeanageattoilettrainingcompletioninthat studywas10monthshigher,ashorterperiodoftoilet train-ing(20.4months)wasobservedintheirstudycomparedto thepresentDSchildren(34months).Thisdifferencecanbe explainedbythefactthatDSchildrenincludedinthisstudy initiatedtoilettrainingatanearlierage(22.8months)than thoseinthe studyby Powersetal.16 (45.6months).It is, therefore,importanttoemphasizethatan earlyinitiation oftoilettraininginchildrenwithDSappearstohavethe dis-advantageofalongerperiodoftraining,buttheadvantage ofayoungerageofcontinenceacquisition.
Thebesttimetoinitiatetoilettraininginchildrenwith DSisnotyetknown.InBrazilianchildrenwithnormal psy-chomotordevelopment,itwasdemonstratedthatthosewho begantobetrainedearlier(before24months)hadalonger trainingperiod,whencomparedwiththosewhosetraining startedlater, nodifferences were observedregarding the meanageofcontinenceacquisition.15Itisworthnotingthat thedelayincontinenceacquisitionisoneofthefactorsthat canpostponeelementaryschoolentryforchildrenwithDS.25 Thisisrelevant,sincetheimprovementinsocialskills out-comesforthechildrenwhostartschoolattheusualagehas been demonstrated,being paramountfor theinclusion of thesechildren.26
GirlswithDSappeartohaveanearliermean developmen-talagewhencomparedwithboyswithDSofthesameage.27 Thisfindingisconsistentwiththepresentresults,inwhich toilettrainingwascompletedsoonerinfemaleswithDSthan intheirmalepeers.However,thesefindingsarein disagree-mentwiththeresultsreportedbyPowersetal.,16whodid notfinda significant difference between gendersin their groupofchildrenwithDS.RegardingtheCG,nodifference betweengenderswasfoundinthisstudy.Nonetheless,other studiesin childrenwithnormalpsychomotor development showedthatgirlsacquirednearlyalltheskillstocomplete toilettrainingmorequicklythanboysbyapproximately2---3 months.13,15
Therearenospecificguidelinesfortoilettrainingin chil-dren with DS.8 In the present study, 87.1% and 88.2% of parentsofchildrenwithDSusedachild-orientedapproach andapottychairfortoilettraining,respectively,similarto thoseintheCG.Inthepresentstudy,parentsreportedthe useof thechild-oriented approach, identifyingjusta few readinesssigns,whichmayhavedeterminedanearlystart ofthetoilettrainingprocess.Nofamilyreportedtheuseof theintensive andstructuredapproach(‘‘toilet trainingin aday’’method).Furthermore,noassociationwasobserved betweentheageoftoilettraininganduseofpottychair,as foundbyTarhanetal.whodescribedthatchildrenwhoused thistypeofequipmentcompletedtheirtoilettrainingata youngerage.14
age thechildren started or completed toilet trainingand thepresence of LUTS. Ina study of children withnormal psychomotor development,no relationship between com-pletingtoilettrainingbefore24monthsofageandvoiding dysfunction was found.28 In another recent study, void-ingdysfunctionwasassociatedwithtoilettrainingstarting either before24months or after36 monthsof age. How-ever,voidingdysfunctionwasobservedonlyinthepresence offunctionalconstipation.29Nostudiesthatdemonstratean associationbetweentheageofstartingorcompletingtoilet trainingandLUTSinchildrenwithDSwereretrieved.
Nodifferencewasfoundintheprevalenceoffunctional constipationamongthegroupsin thepresentstudy.Some studies showed that constipation could make defecation painful,andthismaybeoneofthereasonsachildmayresist toilettraining.23Functionalconstipationwasnotassociated withtheage oftoilet traininginitiationand thetimethe childtakestocompleteit.Itisimportanttoreportthatnone ofthechildrenwithDSincludedinthestudywerediagnosed withHirschsprung’sdisease.
It is alsoworth noting that, in thepresent study,only 16% and 14.2% of parents of children with DS and in the CG,respectively,sought medicaladviceontoilettraining. Nostatisticaldifferenceswereobserved,eventhough chil-drenwithDSshoweddelayeddevelopment.Thesefindings arecompatiblewithanotherstudy,inwhich15%ofparents soughtmedicalinstructions.15
This present studyhas severallimitations. The present resultsmaynotbegeneralizabletothepopulationatlarge, asthiswasaconveniencesample.Theinformationprovided reliedon thememory ofthe parents or caregivers. Cases tendtoattempttoidentifywhatmight have causedtheir disease; healthy controls have no such motivation. Thus, betterrecall amongcases is common.30 This mayexplain thefact that parents of children withdisabilitiestend to have better recollection of theimportant facts that hap-penedtotheirchildrenthanthoseofchildrenwithnormal psychomotordevelopment.
ThereadinesssignsfortoilettraininginDSchildrenstill need tobeclearlydescribed.The findingspresented here areimportanttodemonstratetoparentsor caregivers,as wellastotheprofessionalsinvolvedinmonitoringchildren withDS,theimportancetodeterminewhatthesesignsare inordertobeginthetoilettrainingprocess.
Itmaybeimportanttocarryoutinterventionstudiesin which parentsreceive professionalguidance,which would also allow comparing the child-oriented approach with a morestructuredapproach.Atthismoment,itisknownthat mostoftheparentsusetocenteredapproach,butitisstill notknownwhichmethodfortoilettraininginchildrenwith DSismosteffective.
Inconclusion,childrenwithDSstarttheirtoilettraining processlaterthanthosewithnormalpsychomotor develop-ment,takealongertimefor completingit,doingsoatan olderagethanthosewithoutanydisabilities.BoyswithDS finish their toilet training at an older age than girls with this syndrome. The mostly usedtoilet training method is the child-oriented approach. LUTS and constipation were notassociatedwithtoilettrainingneitherinDSchildrennor inthosewithnormalpsychomotordevelopment.
Conflicts
of
interest
Theauthorsdeclarenoconflictsofinterest.
Acknowledgments
Theauthorswouldliketothankalltheparticipantsfortheir dedicationandperseverance,andtothanktheAmbulatory InterdisciplinaryTeamofDownSyndromeoftheDepartment of Health of Children and Adolescents (Brazilian Unified HealthSystem).
Appendix
A.
Toilet training questionnaire for parents or caregivers of the children with Down syndrome and normal psychomotor development
At what age did you begin toilet training your child? ____________ months
Did your child show any signs that he/she wanted to stop using diapers?
Did not show ( )
You do not remember ( )
He/she stayed dry for more than 2 hours ( )
He/she pointed or walked to the potty ( )
He/she pointed or walked to the bathroom ( )
He/she was bothered by wet diapers ( )
He/she warned that he/she wanted to urinate or defecate ( )
What method did you use to toilet-train your child?
Child-oriented (he/she gave signs that it was time to withdraw) ( )
You thought it was the right time to toilet-train ( )
Removed the diapers in one day ( )
Your son/daughter was taught from birth and he/she has never worn diapers ( )
Used the alarm system ( )
Other. Which?_____________________________________________ ( )
What type of equipment did you use for toilet training?
Potty chair ( )
Toilet ( )
Toilet with footrest ( )
Toilet with seat reducer ( )
Toilet with seat reducer and footrest ( )
Other. Which?_____________________________________________ ( )
At what age did your child finish toilet training?
Age at which diapers were no longer worn during the day...months
Age at which diapers were no longer worn during the night...months
Did you ask for any medical advice to begin or during your child’s toilet training process?
Yes ( )
No ( )
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