www.rpped.com.br
REVISTA
PAULISTA
DE
PEDIATRIA
ORIGINAL
ARTICLE
Validation
of
questionnaires
to
assess
quality
of
life
related
to
fecal
incontinence
in
children
with
anorectal
malformations
and
Hirschsprung’s
disease
Arthur
Loguetti
Mathias
a,
Ana
Cristina
Aoun
Tannuri
a,
Mariana
Aparecida
Elisei
Ferreira
b,
Maria
Mercês
Santos
a,
Uenis
Tannuri
a,∗aInstitutodaCrianc¸adoHospitaldasClínicasdaFaculdadedeMedicinadaUniversidadedeSãoPaulo(USP),SãoPaulo,SP,Brazil bFaculdadedeMedicinadaUniversidadedeSãoPaulo(USP),SãoPaulo,SP,Brazil
Received15March2015;accepted16June2015 Availableonline28December2015
KEYWORDS
Digestivesystem abnormalities; Hirschsprung’s disease; Qualityoflife; Questionnaire; Children; Adolescents
Abstract
Objective: Surgicaltreatmentofanorectalmalformations(ARMs) andHirschsprung’sdisease
(HD)leadstoalterationsinbowelhabitsandfecalincontinence,withconsequentqualityoflife impairment.TheobjectivesweretocreateandvalidateaQuestionnairefortheFecal Inconti-nenceIndex(FII)basedontheHolschneiderscore,aswellasaQuestionnairefortheAssessment ofQualityofLifeRelatedtoFecalIncontinenceinChildrenandAdolescents(QQVCFCA),based ontheFecalIncontinenceQualityofLife.
Methods: The questionnaireswere applied to 71 children submitted tosurgical procedure,
intwo stages.Validity was tested bycomparing the QQVCFCAandageneric qualityoflife questionnaire(SF-36),andbetweenQQVCFCAandtheFII.Agroupof59normalchildrenwas usedascontrol.
Results: Attwostages,45.0%(32/71)and42.8%(21/49)ofthepatientshadfecalincontinence.
ItwasobservedthattheQQVCFCAshowedasignificantcorrelationwiththeSF-36andFII (Pear-son’scorrelation0.57),showingthatthequalityoflifeisdirectlyproportionaltoimprovementin fecalincontinence.Qualityoflifeinpatientswithfecalincontinenceisstillgloballyimpaired, when comparedwithcontrolsubjects (p<0.05,Student’st-test).Therewerealsosignificant differencesbetweentheresultsofchildrenwithARMsandchildrenwithHD.
Conclusions: QQVCFCAandFIIareusefultoolstoassessthequalityoflifeandfecalincontinence
inthesegroupsofchildren.ChildrenwithARMssubmittedtosurgicalprocedureandHDhave similarqualityoflifeimpairment.
©2015SociedadedePediatriadeS˜aoPaulo.PublishedbyElsevierEditoraLtda.Thisisanopen accessarticleundertheCCBYlicense(https://creativecommons.org/licenses/by/4.0/).
∗Correspondingauthor.
E-mail:[email protected](U.Tannuri).
http://dx.doi.org/10.1016/j.rppede.2015.06.022
PALAVRAS-CHAVE
Anomaliasdosistema digestivo;
Doenc¸ade Hirschsprung; Qualidadedevida; Questionário; Crianc¸as; Adolescentes
Validac¸ãodequestionáriosparaavaliac¸ãodaqualidadedevidarelacionadaà continênciafecalemcrianc¸ascommalformac¸õesanorretaisedoenc¸a deHirschsprung
Resumo
Objetivo: O tratamento cirúrgico das malformac¸ões anorretais (MAR) e da doenc¸a de
Hirschsprung(DH) levaaalterac¸õesdohábito intestinaleincontinênciafecal comprejuízo daqualidadedevida.OsobjetivosforamcriarevalidaroQuestionárioparaoÍndicede Con-tinênciaFecal(ICF),baseadonoHolschneiderCriteria,bemcomooQuestionárioparaAvaliara QualidadedeVidaRelativaàContinênciaFecalemCrianc¸aseAdolescentes(QQVCFCA),baseado noFecalIncontinenceQualityofLife.
Métodos: Osquestionáriosforamaplicadosem71crianc¸asoperadas,emduasetapas.Avalidade
foitestadapormeiodacomparac¸ãodoQQVCFCAeumquestionáriogenéricodequalidadede vida(SF-36) eentre oQQVCFCAe oICF. Umgrupo de59 crianc¸asnormais foiusado como controle.
Resultados: Nasduasetapas,45,0%(32/71)e42,8%(21/49)dospacientesapresentaram
incon-tinênciafecal.Verificou-sequeoQQVCFCAapresentoucorrelac¸ãosignificativacomoSF-36eo ICF(correlac¸ãodePearson0,57)emostrouqueaqualidadedevidaédiretamenteproporcional àmelhoriadacontinênciafecal.Aqualidadedevidanopacientecomincontinênciafecalestá aindacomprometidaglobalmente,emcomparac¸ãocomosindivíduoscontroles(p<0,05;teste
tdeStudent).Nãohouveaindadiferenc¸asignificativaentreosresultadosdecrianc¸ascomMAR ecrianc¸ascomDH.
Conclusões: OQQVCFCAeoICFsãoinstrumentosúteisparaaavaliac¸ãodaqualidadedevidae
daincontinênciafecalnessesgruposdecrianc¸as.Crianc¸asoperadasdeMAReDHapresentam comprometimentossemelhantesdaqualidadedevida.
©2015SociedadedePediatriadeS˜aoPaulo.PublicadoporElsevierEditoraLtda.Esteéumartigo OpenAccesssobalicençaCCBY(https://creativecommons.org/licenses/by/4.0/deed.pt).
Introduction
AnorectalmalformationsandHirschsprung’sdiseaseare con-genitaldisordersaffectingapproximately1:5000livebirths. Surgicalcorrectionshouldbedoneearlyandthemain objec-tive is the anatomical reconstruction of structures with normalbowel habits.1 However,constipationand/or fecal
incontinencearefrequent,withimportantconsequenceson
personal,social,andprofessionalspheresthatmayreflectin
adulthood.Thus,patientsmaysufferstrongnegativeimpact
onqualityoflife(QoL).QoLisdefinedbytheWorldHealth
Organizationas‘‘theindividual’sperception oftheir
posi-tioninlifeinthecontextofthecultureandvaluesystems
inwhich theyliveandin relationtotheir goals,
expecta-tions,standardsandconcerns’’.2Thus,QoLisasubjective
datacomprisingseveralareasandshouldbeevaluated
indi-viduallyandbasedontheexpectationsofpatientsandtheir
relatives.
InthePediatricSurgeryandLiverTransplantationService
oftheChildren’sInstituteofHospitaldasClínicasda
Facul-dadedeMedicinadaUSP----where15newcasesareattended
onaverageper year----fecalcontinence is assessed
subjec-tively on three levels (good, fair and poor) that do not
objectivelyreflectreality.Inthisservice,theissueofquality
oflifeisnotthorough.Onlycasesinwhichpsychosocial
prob-lemsareexternalizedoraremoreseriousarereferredtoa
psychologistorsocialworker.Thus,itbecamenecessaryto
applyquestionnairestoevaluatephysical(fecalcontinence)
andpsychosocial(qualityof life)performanceofpatients,
sothatpediatricianandsurgeoncanperformeffectiveand
positiveinterventionsduringthefollow-up.Wedidnotfind
instrumentsthatmettheneedsofourtargetpopulationin
theliterature. Theobjectiveof thisstudy wastodevelop
andvalidatenewquestionnairestoassessqualityoflifeand
fecalincontinence,fromapopulationofchildrenundergoing
surgeryforanorectalmalformationrepairorHirschsprung’s
diseaseinclinicalfollow-upinourclinic.
Method
The Questionnaire for the Fecal Incontinence Index (FII), based on the Clinical Evaluation of Fecal Conti-nence (Holschneider Criteria)3 and the Questionnaire for
the Assessment of Quality of Life Related toFecal
Conti-nenceinChildrenandAdolescents(QQVCFCA),basedonthe
FecalIncontinenceQualityofLife(FIQL)werecreated.4The
created questionnaireswere submitted to the translation
andculturaladaptationprocesses,thenthevalidationstep
wasinitiated.Forthis,theyweresentalongwiththe
Short-Form36(SF-36)5questionnaireviamailtovolunteers,and
withoutthepresenceofaninterviewer.
TheClinicalEvaluationofFecalContinence
(Holschnei-der Criteria) is an established and widely used index in
pediatricsurgery;however,ithasnotvalidatedtranslation
intoPortugueseanditsquestionsarefitforinterview
fromcorrespondingauthor)wascreatedbasedon
Holschnei-derCriteria,consistingof8questionseasilyunderstoodon
proceduresofeverydaylife(questions1---5)andondiarrhea,
constipation,anduseofauxiliarytreatment(questions6---8).
ItmeetstheHolschneider’scriteria.Thus,itwaspossibleto
createascoringsystemof0---6,withvalueof0---2pointsfor
eachquestion.Therangeof0---5indicatespoorcontinence,
6---10faircontinence,11---15goodcontinenceandmaximum
score,and16normalfecalcontinence.
Thequestionnairetoassessqualityofliferelatedtofecal
continenceinchildrenandadolescents(QQVCFCA;available
from corresponding author) was based on the translation
intoPortugueseandvalidationofFIQL.FIQLisa
question-naireof29questionsforuseinadults,whichhasquestions
thatareconsideredrepetitiveandsomethataddress
situa-tions of severe depressionand sex, considered unsuitable
for childrenandadolescents.Thus,16questionswerenot
used;thewordingof10questionswasmodifiedbutkeptthe
same meaning;3 questions were maintained;and 5 were
added,totaling18questions.Thefifthoption,‘‘noneofthe
answers’’,waschangedto‘‘othercause’’,whichprovedto
be more in line withthe explanatory text. This standard
questionnaire was submitted to analysis by a
multidisci-plinaryteamthatchangedit:theQQVCFCAwasexpanded
from18to24questions;andthefifthoption,‘‘othercause’’
hasbeendeleted.Atthattime,thesuggestionwastoleave
theanswerblank.Theoptions‘‘Istronglyagree’’,‘‘I
some-whatagree’’,‘‘Idisagreeabit’’,and‘‘Istronglydisagree’’
were considered difficult to understand and replaced by
‘‘almostalways’’,‘‘sometimes’’,‘‘rarely’’,and‘‘never’’.
The same domains of theoriginal were covered: lifestyle
(7 questions),behavior (7 questions),depression (7
ques-tions),andembarrassment(3issues).Questions 22and23
arerelatedtopatient’sopinionandsatisfactionwiththeir
ownhealthandbowelfunction.Eachquestionhasascoreof
1---4(1=worstsituation).Thefinalscoreisobtainedby
sum-mingthemeanscoreobtainedineachdomain,andranges
from4to16.
TheSF-36(MedicalOutcomesStudy36-ItemShort-Form
HealthSurvey)isagenerictoolforassessingthequalityof
life,whichiseasytoadministerandunderstand.Itisa
mul-tidimensionalquestionnaireconsistingof36items,grouped
in eight domains: physical functioning, bodily pain,
gen-eralhealth,vitality,socialfunctioning,emotionalaspects,
andmentalhealth.Thisquestionnairewasalsoappliedand
the results were compared with the results obtained in
QQVCFCA.
The questionnaires were administered in 85 patients,
between 4 and 19 years, undergoinganorectal repair for
malformationsorHirschsprung’sdisease,withfollow-upin
theclinic.Only10patientshad4---6years,valueconsidered
insufficient.Therefore,theywerenotusedinthisstudy.Of
the75remainingpatients,4respondedonlytotheFIIand
werealsoexcluded.Thus,71patients,from7to19years,
whohadanorectalmalformationsorHirschsprung’sdisease,
whosesurgical treatment hasbeencompletedfor atleast
sixmonths,and whoagreed toparticipatewereenrolled.
Patients with some degree of impaired
neuropsychomo-tor development and neurological and urinary disorders
involving the sphincter control were excluded. Of the 71
cases,31 were Hirschsprung’s disease:23 cases of classic
form (rectosigmoid aganglionosis); 7 cases of total colic
aganglionosis; one case of intestinal neuronal dysplasia.
Ofthe total, 22 were maleand 9 female. The remaining
cases, 40 were of anorectal malformations: 8 low form;
15 intermediate and/or high form; 4 persistent cloaca;
onevesicular-intestinal fissure;one rectalatresia; 11 not
reported(children operated in other services referred to
theInstitutionduetointestinalconstipation).
Control groupconsisted of 59healthy individuals
with-outbowelfunctioncomplaintsanddemographicallysimilar
to the assessed patients. They were selected in
outpa-tientclinicofpediatricsurgeryandsufferedfromcommon
surgical conditions, suchas inguinal hernia andphimosis.
Similartothestudy groups, the questionnaireswere sent
bymail.
Themeasurementpropertiesstudiedwere
reproducibil-ityandvalidity.Toevaluatereproducibility,FIIandQQVCFCA
weresenttwicetothevolunteersbymail.Betweenthefirst
andsecondsending, therewasanintervalof 6daysto14
months.Ofthe71initialpatients,22didnotrespondtothe
secondsent questionnaires.Ofthe49 remainingpatients,
24haveanorectalmalformationsand25haveHirschsprung’s
disease.
Convergent and discriminant validity was tested. To
assess the construct validity, incontinent patients also
responded to the generic questionnaire known as Short
Form-36,alreadyvalidated inourmidst.The resultswere
correlated with those of QQVCFCA and then the FII and
QQVCFCAresultswerecompared.Results ofpatients with
anorectal malformations and Hirschsprung’s disease were
alsocompared.Discriminantvaliditywasevaluatedwiththe
application of the questionnaires in 71 operated patients
and 59 healthy volunteers. The mean scores for each
group on FII, QQVCFCA, and each of its domains were
compared.Bray---Curtisindex,Pearson’scoefficient,and
Stu-dent’st-testwereused.Thesignificancelevelwas0.05.
Results
Based on the FII, it was found that among the initial 71 patients,continencewaspoorin16,fairin16,goodin26, andnormalin13patients.Among49patientswhoalso par-ticipatedinthesecondphase,itwasfoundthatofthe25 withHirschsprung’s disease,6 hadpoor, 6had fair, 9had good, and 4 had normal continence. Among the 24 with anorectalmalformations,continencewaspoorin4,fairin5, goodin12,andnormalin3patients.Inshort,45.07%(32/71) and42.85%(21/49)ofpatientshadfecalincontinenceinthe twophasesofquestionnaireapplication.
The reproducibility of the results obtained from both questionnairescouldbeseenovertimethroughtheir appli-cation in the same patients at two different times. The interval between these times ranged from 6 days to 14 months.ThefirstapplicationwastermedFII1andQQVCFCA1 andthesecondFII2andQQVCFCA2.
Table1 ComparisonofsimilaritybetweenFII1and2andQQVCFCA1and2anditsdomains. Timeintervalbetween
questionnaireapplications
FII1×FII2 QQVCFCA1× QQVCFCA2
LS1×LS2 BEH1×BEH2 DEP1×DEP2 EMB1×EMB2
6daysto1month 0.95 0.96 0.94 0.97 0.93 0.92
2to3months 0.92 0.93 0.89 0.96 0.92 0.90
4to6months 0.92 0.96 0.94 0.97 0.95 0.94
6to14months 0.96 0.94 0.95 0.93 0.94 0.85
All 0.94 0.95 0.95 0.96 0.93 0.90
FII,fecalincontinenceindex;QQVCFCA,questionnairefortheassessmentofqualityofliferelatedtofecalincontinenceinchildrenand adolescents;LS,lifestyle;BEH,behavior;DEP,depression;andEMB,embarrassment.Thenumber1afterthecriterionrelatestothefirst applicationofthequestionnaires;thenumber2referstothesecondapplicationofthesamequestionnaires.
Note:Bray---CurtisIndex:0---0.19---notsimilar;0.20---0.39---littlesimilarity;0.40---0.59---mediumsimilarity;0.60---0.79---highsimilarity; 0.80---1.0---maximumsimilarity.
Table2 ComparisonofFIIandQQVCFCAanditsdomainsbetweenhealthyvolunteers(group‘‘normal’’)andchildren with anorectalmalformationsorHirschsprung’sdisease(group‘‘patient’’).
Mean Standard deviation
25th percentile
75th percentile
Median
ICF
Normal 15.37 1.0 15.0 16.0 16.0
Patient 10.4 4.48 6.0 15.0 11.0
QQVCFCA
Normal 15.2 0.9 15.0 15.7 15.5
Patient 11.2 3.3 8.6 14.8 11.2
LS
Normal 3.9 0.3 4.0 4.0 4.0
Patient 3.1 0.9 2.3 4.0 3.4
BEH
Normal 3.9 0.2 3.8 4.0 4.0
Patient 3.0 0.8 2.5 3.8 3.1
DEP
Normal 3.6 0.31 3.4 3.8 3.6
Patient 2.7 0.83 2.1 3.5 2.7
EMB
Normal 3.9 0.2 4.0 4.0 4.0
Patient 2.3 1.11 1.3 3.6 2.0
FII,fecalincontinenceindex;QQVCFCA,questionnairefortheassessmentofqualityofliferelatedtofecalincontinenceinchildrenand adolescents;LS,lifestyle;BEH,behavior;DEP,depression;andEMB,embarrassment.PerformedwithStudent’st-test,withp<0.05and statisticallydifferentresults.
Toassessconstructvalidity,Pearsoncorrelationwasused and values from 0.50 to 0.74 were considered as rea-sonablecorrelation and values between 0.75 and 0.99 as strongcorrelation.Directcorrelationwasobservedwiththe value of 0.5 between the results of QQVCFCA and SF-36, appliedto71patients.Thus,theresultsfor qualityoflife aresimilarwiththeapplication oftheSF-36orQQVCFCA. Comparing FII and QQVCFCA, it is noted that there is a dependency between them (index of 0.82). There is also a strong correlation between the FII with each domain of QQVCFCA individually (lifestyle: 0.75; behavior: 0.78; absence of depression: 0.78; absence of embarrassment: 0.71).Thus,thebettertheresultsoffecalincontinence,the betterthequalityoflifeandallparametersevaluatedinthe domains.
Asfor discriminantvalidity,it wasfoundthatthe qual-ity of life of fecal incontinent patients is still hampered globallyin all domains andFII (meanscores of qualityof
life: 11.2; lifestyle: 3.1; behavior: 3.0; depression: 2.6; embarrassment:2.3,andFII:10.4)comparedwithhealthy volunteers(15.2;3.9;3.9;3.6;3.9,and15.4,respectively), and there is a significant statistical difference between groups when Student’s t-test was applied, with p<0.05 (Table 2). Moreover, there was no significant difference
between theresults of childrenwithanorectal
malforma-tionsandHirschsprung’sdisease(Table3).
Discussion
About 30---50% of patients with anorectal malformations or Hirschsprung’s disease may have some degree of fecal incontinence,6similarlytowhatwasobservedinthisstudy.
In general, itaffects the occupational, social,emotional,
sporting,andsexualareasandmayleadtopsychiatric
Table3 ComparisonofFIIandQQVCFCAanditsdomainsbetweenchildrenwithanorectalmalformationsandHirschsprung’s disease.
Mean Standard deviation
25th percentile
75th percentile
Median
ICF
HD 10.0 4.4 5.5 13.5 11.0
ARM 10.9 4.6 6.5 15.0 13.0
QQVCFCA
HD 11.2 3.4 8.1 15.0 11.0
ARM 10.8 3.6 7.6 14.9 10.5
LS
HD 3.1 0.8 2.3 4.0 3.5
ARM 3.1 0.8 2.3 4.0 3.0
BEH
HD 3.1 0.8 2.5 4.0 3.3
ARM 2.9 0.9 2.2 3.7 3.2
DEP
HD 2.7 0.8 1.8 3.4 2.5
ARM 2.5 1.0 1.6 3.8 2.3
EMB
HD 2.2 1.2 1.2 3.6 1.6
ARM 2.3 1.1 1.0 3.4 2.0
HD,Hirschsprung’sdisease;ARM,anorectalmalformations;FII,fecalincontinenceindex;QQVCFCA,questionnairefortheassessment ofqualityofliferelatedtofecalincontinenceinchildrenand adolescents;LS,lifestyle;BEH,behavior;DEP,depression; andEMB, embarrassment.
closelyrelatedtoQoL.7Inareviewarticleofseveral
stud-ies,itwasshownthatpatientswithimpairedbowelfunction
alsohaveimpairedQoL,withthiscorrelationrangingfrom
small, medium or strong.1 Despite advances in surgical
pediatric techniques, fecal incontinence remains a
com-mon problem for patients.8 Therefore, during outpatient
care, just asking about the number of bowel movements
and subjectivelyevaluating the patientstatus hasproven
insufficient and superficial. It is important to include the
concept of QoL; that is, the extent to which these
phys-ical/psychological/socialproblemsaffect thefunctionality
andexpectationsofthepatient.Therefore,theassessment
ofQoLandfecalincontinenceshouldbedoneusing
objec-tive, quantitative, and reproducible methods that allow
long-termfollow-upofpatients.
There are several tools for that purpose in the
liter-ature that are specific and validated, such as the Fecal
Incontinence Questionnaire,9 Fecal Incontinence
Sever-ity Index,10 Fecal Incontinence Quality of Life (FIQL),11
Hirschsprung’s Disease Anorectal Malformation QoL
Ques-tionnaire (HAQL),12 Medical Outcomes Study 36-Item
Short-FormHealthSurvey,13andClinicalEvaluationof
Conti-nence(HolschneiderCriteria).Inthepresentinvestigation,
thequestionnairesusedweretranslatedandadapted into
Portuguese.Allcases wereevaluatedbythemain
investi-gator(ALM), the participationof the originalauthors was
considered infeasible for practical reasons. Such conduct
wasalsosupportedbytheInstitutionalReviewBoardofthe
Institution.
The HAQL, for example, is specifically designed for
patientswithanorectalmalformationsorHirschsprung’s
dis-ease.Itis asurveywith39---42questionsansweredby the
patient or their parents/guardians. It consists of 10---11
subscales, with items related to laxative diet,
constipat-ingdiet,diarrhea,fecalincontinence,urinarycontinence,
social functioning, emotional functioning, body image,
physicalsymptoms,andsexualfunctioning.Theanswersare
providedbythepatient,bytheparents,orboth,depending
onage.Thus,thereisaformfor parentsofchildren aged
6and7years;aformforparentswithchildren8---11years
andadolescents12---16years;andanadultformforpatients
aged17yearsormore.13
The FIQL, in turn, validated in English in 2000, was
designed to evaluate the quality of life in any patients
withfecal incontinence. It consists of 29 items assessing
fourdomains: lifestyle, behavior, depression, and
embar-rassment.Thesamequestionnaire wasused,regardlessof
patientage.10
Althoughmanyinstruments arefound intheliterature,
noneisadequatetoaddress thetargetpopulationof
Insti-tutoda Crianc¸a,São Paulo, Brazil.Part of the population
attended at the Institute has low cultural level,
mak-ingitdifficult/impossibletoapplyquestionnairesin other
languages.Therearealsoquestionnaireswithoptions
con-sidered complex and difficult to understand, such as the
FIQL that has the following options: ‘‘strongly agree’’,
‘‘somewhatagree’’,‘‘somewhatdisagree’’,‘‘strongly
dis-agree’’,and‘‘notapplicable’’.Inaddition,somepatients
failtoregularlyattendtheclinicabout2---3yearsafterthe
surgicaltreatmentduetopooradherencetotreatment or
eventodifficultaccesstohealthcare.Still,manypatients
liveinremoteareasandhavefewresourcesfor
transporta-tion, which complicates the application and interviews.
Finally, we also found questions considered inappropriate
for children involving sexuality and situations of severe
translation,culturaladaptation,andvalidationoftwo
ques-tionnaires,FII and QQVCFCA, congruent withARMs or HD
patients.FIQLandHolschneiderCriteria,presentedabove,
wereusedasmodels.
Theprocessoftranslation,culturaladaptationand
vali-dationofquestionnaireshasbeenmadeinothercountries.
Minguezetal.validatedtheFIQLintoSpanish,keptthe29
questionsand applied it in patients withmean age of 60
yearswithfecalincontinenceofanyetiology.Incontinence
severitywasalsoratedanditsrelationtodomainsofFIQL.12
Rullieretal.validated theFIQLintoFrench,withastudy
similartothatbyMinguezetal.,and100patientswithmean
ageof57yearswereevaluated.7InSweden,Wiganderetal.
translatedandculturallyadaptedtheHAQL.The
question-naireshadtwoextraquestionsregardingtheoriginalHAQL:
thefirstaskedifthepatienthaddifficultyunderstandingthe
questionsoriftheyfounditodd;thesecond, iftherewas
somethingtheywould liketoadd.13 InBrazil,Yusufetal.
validatedtheFIQLintoPortuguese,butwithoutadaptingit
tochildrenandadolescents,themeanageofpatientswas
52.8years.14
Inall thesestudies,the questionnairesandassessment
tools of QoL and fecal continence underwent a detailed
processoftranslationandculturaladaptation, inorderto
suit the beliefs, customs, daily life, behavior, and
socio-economicconditionsofthetargetpopulation.Inthepresent
study, the importance of this step was perceived by the
significant number of fundamental changes in the
origi-nalquestionnairesused,suchassuppressionof questions,
changeinthewordingofquestions,creatingquestionsand
changingoptions.Inaddition,amultidisciplinaryteamwas
necessary to structure and, after an initial assessment,
modifyoncemorethequestionnairesagain.Thisthorough
processensuresanimprovementofthefinalquestionnaires.
Moreover, reproducibility and validity of instruments
were tested to evaluate the reliability. Reproducibility
proved stable, with indices always above 0.85. Thus, it
is concludedthat therewasa significant similarity in the
resultsofthequestionnairesintwoseparateapplicationsin
thesamepatients.Thetimeintervalbetweenapplications
ranged from6 days to14 months, withno change in the
resultsaswell.Theprovenreproducibilityinthetime
inter-valof6daysto1monthisespeciallyimportant.Inashort
period,suddenchangesintheevolutionofallpatientswould
beunlikely,whichcouldcompromisetheresults.Inalonger
period,thereproducibilitycouldnothavebeenprovendue
tochangesintheclinicalstatusofpatients,ifinterventions
withmorepronouncedeffectshadbeenmade.
Duringthevalidationphase,therewasareasonable
cor-relationbetweenSF-36andQQVCFCA,withacoefficientof
0.57consideredstatisticallysignificant.Itconfirmsthatthe
analyzed questionnaire has adequate validity when
com-paredwitha sensitivityinstrumentalreadyestablished.It
was also seen a direct relationship between the FII and
QQVCFCA. This showed that quality of life is affected in
patients withpoor control of fecalcontinence. This
rela-tionshipwasalsoseenbetweenFIIandQQVCFCA’sdomains;
so that, the better the fecal continence, the better the
lifestyleandbehaviorindicesandabsenceofdepressionand
embarrassment.Finally,inthediscriminativevalidation,it
was found that in patients with anorectal malformations
orHirschsprung’s diseasethescores arelowerthaninthe
controlpopulation,whichprovestheimpactoffecal
incon-tinenceonqualityoflifeofpatients.Itwasfoundthatthe
greatestimpactonqualityoflifeofpatientsoccursinthe
domainsofdepressionandembarrassment.
The loss to follow-up of 22 (30%) patients during the
reproducibilityprocessovertimemayberegardedasastudy
limitation.Thesepatientsmaynothaveunderstoodthatit
wasaresearchprocessinwhichthequestionnairewassent
twice intentionally and not accidentally. Thus, improving
theunderstandingof volunteersisneededduringresearch
activities.
Theassociation,inparticular,ofanorectalmalformations
withothercongenitalandgeneticdiseasesiscommonand
affectsasignificantpercentageofpatientsattendedatICr.
Thus,patientswithDownsyndrome,forexample,werenot
included in the inclusion criteria due to the presence of
other factors that could interfere with the evaluation of
qualityoflife,confoundingfactors.Thus,thereisagreat
needforthecreationandvalidationofspecificmethodsto
assesssuchchildren,intermsofbothfecalcontinenceand
qualityoflife.
Finally,itisalsoimportantthatyoungchildrenwhowere
notincludedinthisstudybecausethenumber(only10)was
consideredunrepresentativeofthetotalbeevaluatedinthe
future. Inthiscase, differentquestionnairesandforms of
recreationalevaluationcouldbeproposed,duetodifficulty
ofunderstandingandabstractionnecessaryfortheanswers.
Inconclusion,thecreationoftheFIIandQQVCFCA
fol-lowed the trend of several centers around the world to
create and validate their own questionnaires. The
vali-dation process wassatisfactory and provided instruments
inPortugueseforevaluationofchildrenwithanorectal
mal-formationsandHirschsprung’sdisease.Itcanbeusedboth
in the medicalpracticeand insurveys. It is alsoa model
thatstandardizes theevaluation,allowing itsuseinother
centers.
Funding
Programa de Iniciac¸ão Científica da Universidade de São Paulo(PIC-USP),modalitySantander.
Conflicts
of
interest
Theauthorsdeclarenoconflictsofinterest.
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