Rev. Bras. Reumatol. vol.55 número2

w w w . r e u m a t o l o g i a . c o m . b r

REVISTA

BRASILEIRA

DE

REUMATOLOGIA

Original

article

Assessment

of

fatigue

and

dryness

in

primary

Sjögren’s

syndrome:

Brazilian

version

of

“Profile

of

Fatigue

and

Discomfort

–

Sicca

Symptoms

Inventory

(short

form)

(PROFAD-SSI-SF)”

夽

Samira

Tatiyama

Miyamoto

,

Maurício

Aquino

Paganotti

,

Érica

Vieira

Serrano

,

Raquel

Altoé

Giovelli

,

Valéria

Valim

a_{EscolaPaulistadeMedicina,UniversidadeFederaldeSãoPaulo,SãoPaulo,SP,Brazil} b_{UniversidadeFederaldoEspíritoSanto,Vitória,ES,Brazil}

c_{UniversidadedeVilaVelha,Vitória,ES,Brazil}

d_{EscolaSuperiordeCiências,SantaCasadeMisericórdiadeVitória,Vitória,ES,Brazil}

a

r

t

i

c

l

e

i

n

f

o

Articlehistory:

Received27May2014 Accepted6October2014 Availableonline6January2015

Keywords:

Sjögren’ssyndrome Questionnaire Validationstudy Evaluation PROFAD-SSI-SF

a

b

s

t

r

a

c

t

Objective:Toperformacross-culturaladaptationandvalidationoftheProfileofFatigue andDiscomfort–SiccaSymptomsInventory(shortform)(PROFAD-SSI-SF)questionnaire assessingthesubjectiveaspectsofthesymptomsofprimarySjögrensyndrome(pSS),for theBrazilianPortugueselanguage.

Method:Conceptual,oftheitem,semanticandoperationalequivalenceswereevaluated. TheBrazilianversionofPROFAD-SSI-SFwasadministeredto62womenwithpSSaccording totheEuropean-Americanconsensus2002toassessmeasurementequivalence.␣-Cronbach

wasusedforinternalconsistency;intraclasscorrelationcoefficient(ICC)forintraobserver reproducibility;andSpearmancorrelationcoefficientforvaliditybycomparingwithPatient Global Assessment(PaGA), EULARSjögren’s SyndromePatientReportedIndex (ESSPRI), FunctionalAssessmentofChronicIllnessTherapyFatigueSubscale(FACIT-F)andEuroQOL (EQ-5D).

Results:TheinternalconsistencyofPROFAD,SSIandtotalscorewas0.80;0.78;and0.87, respectively.TheintraobserverreproducibilityoftotalPROFADwas0.89;oftotalSSIwas 0.86;andtotalscorewas0.89.Intermsofvalidity,PROFADcorrelatedsignificantlywithPaGA (r=0.50),FACIT-F(r=0.59),ESSPRI(r=0.58)andalldomainsofEQ-5D,withtheexceptionof Mobility.Ontheotherhand,SSIcorrelatedsignificantlywithPaGA(r=0.43),FACIT-F(r=0.57), ESSPRI(r=0.55)andmostdomainsofEQ-5D.ThetotalscoreofPROFAD-SSI-SFhada non-statisticallysignificantcorrelationonlywithMobilitydomainandwith1–100rangeofEQ-5D.

夽

Institution:RheumatologyOutpatientClinic,HospitalUniversitárioCassianoAntôniodeMoraes(HUCAM),UniversidadeFederaldo EspíritoSanto(UFES),Vitória,ES,Brazil.

∗ _{Correspondingauthor.}

E-mail:val.valim@gmail.com(V.Valim).

http://dx.doi.org/10.1016/j.rbre.2014.10.002

Conclusion: ThePortugueseversionofPROFAD-SSI-SFprovedtobeanadaptable, repro-ducibleandvalidtoolfortheBrazilianPortugueselanguage.

©2014ElsevierEditoraLtda.Allrightsreserved.

Avaliac¸ão

da

fadiga

e

da

secura

na

síndrome

de

Sjögren

primária:

versão

brasileira

do

“Profile

of

Fatigue

and

Discomfort

–

Sicca

Symptoms

Inventory

(short

form)

(Profad-SSI-SF)”

Palavras-chave:

SíndromedeSjögren Questionário Estudodevalidac¸ão Avaliac¸ão

Profad-SSI-SF

r

e

s

u

m

o

Objetivo: Realizaraadaptac¸ãotransculturaleavalidac¸ãodoProfileofFatigueand Dis-comfort–SiccaSymptomsInventory(shortform)(PROFAD-SSI-SF),questionárioqueavalia osaspectossubjetivosdossintomasdasíndromedeSjögrenprimária(SSp),paraalíngua portuguesabrasileira.

Método: Foiavaliadaaequivalênciaconceitual,deitem,semânticaeoperacional.Aversão brasileiradoProfad-SSI-SFfoiaplicadaa62mulherescomSSpconformeconsenso europeu-americanode2002paraavaliaraequivalênciademensurac¸ão.Foiusadoo␣-Cronbach

paraconsistênciainterna;coeficientedecorrelac¸ãointraclasse(ICC_{)parareprodutibilidade}

intraobservador;ecoeficientedecorrelac¸ãodeSpearmanparavalidadecorrelacionadocom oPatientGlobalAssessment(PaGA),EULARSjögren’sSyndromePatientReportedIndex(ESSPRI),

FunctionalAssessmentofChronicIllnessTherapyFatigueSubscale(FACIT-F)eEuroQOL(EQ-5D).

Resultados: AconsistênciainternadoPROFAD,doSSIedapontuac¸ãototalfoide0,80,0,78e 0,87,respectivamente.AreprodutibilidadeintraobservadordoPROFADtotalfoide0,89;do SSItotalde0,86edapontuac¸ãototalde0,89.Navalidade,oPROFADapresentoucorrelac¸ão significativacomoPaGA(r=0,50),FACIT-F(r=0,59),ESSPRI(r=0,58)etodososdomínios doEQ-5D,comexcec¸ãodamobilidade.JáoSSIapresentoucorrelac¸ãosignificativacom oPaGA(r=0,43),FACIT-F(r=0,57),ESSPRI(r=0,55)eamaioriadosdomíniosdoEQ-5D.A pontuac¸ãototaldoPROFAD-SSI-SFsónãoobtevecorrelac¸ãoestatisticamentesignificante comodomíniomobilidadeeescala1a100doEQ-D5.

Conclusão: AversãoemportuguêsdoPROFAD-SSI-SFmostrouseradaptável,reprodutívele válidaparaalínguaportuguesabrasileira.

©2014ElsevierEditoraLtda.Todososdireitosreservados.

Introduction

Primary Sjögren’s syndrome (pSS) is a systemic disorder

characterized by lymphocytic infiltration and progressive

destruction ofexocrine glands;however, the inflammatory

processcanaffectanyorgan.Theclinicalmanifestationscan besplitintotwoaspects:(1)benign,butdisabling,findings,as dryness,painandfatigue,affectingthemajorityofpatients and(2)systemicfindingsthatcanbepotentiallysevere, affect-ingabout20–40%ofpatients.1_{Duetothelackofuniversally}

acceptedclassificationcriteria,theevaluationoftheincidence andprevalenceofthediseasevariessignificantly,depending

onthecriteriaused.Theprevalencemay rangefrom0.03%

to2.7%ofthe population.2 _{Inthe Brazilianpopulation,the}

prevalence,accordingtotheAmerican-Europeancriteria, is 0.17%.3

Inrecentdecades,expertsestablishedaconsensusthatit iscriticaltoobtainobjectivemeasuresofsaliva4_andtear5

pro-duction,aswellastoassessdrynessandfatiguesymptoms, qualityoflifeandactivityanddiseasedamage,throughvalid andreliableassessmentinstruments,6–8 _{bothtoimprovethe}

accuracyofclinicalassessmentbeforeandaftertreatment,as toenableconductingclinicaltrials.

Therearefewself-assessmentinstrumentsfocusedonthe perceptionofthepatienttoassessthesubjectiveaspectsof his/hersymptomsinthisdisease,astheProfileofFatigueand Discomfort(PROFAD),9,10_{SiccaSymptomsInventory(SSI)}10,11

andthemostrecentlytoolcreatedbyEULARandalready val-idatedforBrazilianPortugueselanguage,theEULARSjögren’s SyndromePatientReportedIndex(ESSPRI),1,12_whichisa

ques-tionnaireconsistingofthreedomains: fatigue,drynessand pain,assessedbyascalenumberedfrom0to10,withequal weights,andwhosefinalscoreistheaverageofthescoresof thethreedomains.

TheProfileofFatigue andDiscomfort –SiccaSymptoms

Inventory(longform)(PROFAD-SSI-LF)consistsof64questions scoredfrom0to7foreightdomains:somaticfatigue, men-tal fatigue,arthralgia,vascular dysfunction,oculardryness, oraldryness,vaginaldrynessandcutaneousdryness.9,10

which consists of 19 questions distributed in the same 8 domainsofthelongversion,havingtwoscores,onefor PRO-FADandtheotherforSSI.PROFADhasnineitemssplitinto fourdomains: cutaneousfatigue, mentalfatigue,arthralgia andvascular,andSSIhas10itemssplitinto fourdomains: oculardryness,oraldryness,vaginaldryness,andcutaneous dryness.Inbothtoolsthefinalscoreisthesumofitsfour domains,andvariesfrom0to28.11_{Itsadvantageisthe}

dis-tinctionoffatigueinphysicalandmentaltypes,aswellasin sitesofdryness,whichdonotalwaysappearsimultaneously andatthesameintensity.

Recent studies in which PROFAD, SSI and ESSPRI were

correlated showedthat all thesetoolsexhibited significant

correlation amongthemselves and with the patient global

assessmentfordiseaseactivity,thusshowingconfirmationof constructvalidity.13,14 _{PROFAD wasalsovalidated forother}

rheumaticdiseases,suchassystemiclupuserythematosus

and rheumatoid arthritis,and it has been shownthat this

toolhavemoderatetohighsensitivity(65–85%)andspecificity (55–77%).9

Besidesthe needforvalidation ofthis tool,it iscritical thatithasitspsychometricpropertiestestedinseveral lan-guagesandindifferentpopulationsofpatientswithpSS.This wouldallowtherealizationofcross-culturalstudiesand of comparisonsbetweennationalandinternationalstudies,thus providingthescientificcommunicationbetweencountriesof differentlanguages.15–17_{Thisstudyaimstoconducta}

cross-culturaladaptationandvalidationofPROFAD-SSI-SFforthe BrazilianPortugueselanguage.Anattemptwasalsomadeto evaluatethepsychometricpropertiesofatotalscoreforthe instrument,althoughtheoriginalversiondidnotpresentit.

Method

Thisisacross-sectionalobservationalstudyapprovedbythe

Ethics Committeein Research of the Health Science

Cen-ter, UFES (Universidade Federal do Espírito Santo), under

Opinionnumber008/10anddevelopedintheSjögren’s

Syn-dromeAmbulatory,RheumatologyOutpatientClinic,Hospital UniversitárioCassianoAntoniodeMoraes(HUCAM),Vitória, EspíritoSanto.

Theproceduresfortheconductionofacross-cultural

adap-tationofPROFAD-SSI-SFfollowedthemethodologyproposed

byHerdman,Fox-RushbyandBadia(1998),18_{whichcoverssix}

steps:conceptual,ofitem,semantic, operational, measure-mentandfunctionalequivalence.

Conceptual, of item, semantic and operational

equiv-alences of PROFAD-SSI-SF were checked at the time of

translation and retranslation (back translation), bya

com-mittee formed by a rheumatologist and a physiotherapist

specializedinRheumatology,bothusedinthemanagement

ofpSS and fluentin English, and an English teacher. The

translationofPROFAD-SSI-SFwasperformedindependently

bytwoEnglishlanguageteachershavingPortugueseastheir

nativelanguageand awareofthepurposeofthe study.On

retranslation,thisversionwassubmittedtoatranslationinto EnglishbytwootherEnglishteachershavingtheEnglishas

their native language and who were unaware ofthe

origi-nalversionandofthepurposeofthestudy.Toevaluatethe

semanticequivalence,thePortugueseconsensusversionwas appliedto20consecutive patientsdiagnosedwithpSSbya

rheumatologistusedinmanagingpatientswithpSS.

Sixty-two patients were selected for the evaluation of

measurement equivalence across psychometric properties,

accordingtoStreinerandNorman(2008)19_{andKirshnerand}

Guyatt (1995) methodology20_{; the calculationto composing}

this sample wasbased on the use ofatleastfive patients perdomainoftheinstrument.21_{Theinclusioncriteriawere:}

diagnosisofpSSaccordingtotheAmerican-European classi-ficationcriteriaforSjögren’ssyndrome,22 _{18yearsofageor}

over,andsigningafreeandinformedconsentterm.Patients

withotherconcomitantautoimmunediseaseswereexcluded.

Diseaseactivitywassubjectivelyassessedasinactiveand active(low,moderateandhighactivity),accordingtotheonset orworseningofpotentiallyreversiblesignsandsymptomsof diseaseinthelastfourweeks,definedbyamedicalexpert,as wellasbyEULARSjögren’sSyndromeDiseaseActivityIndex (ESSDAI), whose score ranges from 0 to 123.17,23 _The

psy-chometricpropertiesevaluatedwere:reliability(intraobserver reliability) and construct validity. The 62 patients under-went clinical evaluationbyaphysicianusedinthecareof patientswithpSS,withcompletionofthefollowing

instru-ments: PROFAD-SSI-SF, Patient Global Assessment (PaGA),

ESSPRI, Functional Assessment of Chronic Illness Therapy

FatigueSubscale(FACIT-F)24,25_{andEuroQOL(EQ-5D)}26_ina

sin-glemoment(visit1)andaftertwodays(visit2).

We opted to replace the self-application of the

instru-mentforaface-to-faceinterviewintheevaluationstagesof

semanticequivalenceandofmeasurement,consideringthat

thischangedoesnotinvalidatetheuseofthequestionnaire byself-application27_{; thiscouldbeexplainedbythe lackof}

practiceofpatientsincompletingself-administered question-naires,orbytheirlowlevelofschooling,28_{andalsotoavoidthe}

exclusionofilliteratepatientsorthosewitheyeproblems.29

␣-Cronbachwasusedinthe analysisofinternal

consis-tency;forintra-observerreproducibility,theintraclass corre-lationcoefficient(ICC)wasused.Fortheconstructvalidityof thecomparisonofPROFAD-SSI-SFwithPaGA,ESSPRI,FACIT-F andEQ-5D,theSpearmancorrelationcoefficientwasused.In allanalyzes,thelevelofsignificancewasacceptedasp≤0.05.

Results

Inthephaseofconceptual,ofitem,semanticandoperational equivalence,changesinquestions14and19wereintroduced. Inquestion14,inthestudyofsemanticequivalence,theliteral translationof“blurredvision”wouldbevisãoturva;however, the wordturva wasreplacedbyembac¸ada,asthis isaterm considered morepopular. Likewise,the term “swollen sali-varyglands”inquestion19wouldmeaninchac¸odasglândulas salivares,butglândulassalivaresisatechnicaltermdifficultto understandand locateforthe Brazilianpopulation;thus,it wasreplacedbyregiãonorostoàfrentedasorelhaseabaixodo queixoinchados.Therewasnoquestionoritemmisunderstood bymorethan15%ofthe20patientsinthepre-test.Thus,the finalBrazilianlanguageconsensusversionwasobtained.

In the evaluation of measurement equivalence across

Table1–Demographicandclinicalcharacteristicsof62 patientswithprimarySjögren’ssyndrome.

Variables n(%)orMean± SD

Age(years) 49.4 ± 11.6

Femalegender 62(100)

Timeelapsedfromthefirstsymptoms(years) 7.2 ± 5.4

Timesincediagnosis(years) 3.0 ± 3.3

Intervalbetweensymptomsanddiagnosis (years)

4.3 ± 4.9

Systemicmanifestationsinthelastvisit 29(46.8)

Inactivedisease 29(46.8)

Activedisease

Lowactivity 24(38.7)

Moderateactivity 9(14.5)

ESSDAI 4.95 ± 6.73

Clinicalfeatures:

Objectivexerophthalmia(SchirmerI and/orRoseBengal)

48(77.4)

Objectivexerostomia(Salivary flow/Parotidscintigraphy)

53(85.5)

Lymphocyticsialadenitis≥1focus-score 56(90.3)

Anti-Ro 27(43.6)

Anti-La 12(19.4)

Previousparotidglandswelling 16(25.8)

SD,standarddeviation;ESSDAI,EULARSjögren’sSyndromeDisease ActivityIndex.

comingfromthe Sjögren’sSyndromeAmbulatory, Rheuma-tologyOutpatientClinicHUCAM,wereincluded.

Themeanagewas49.4±11.6years.Thedurationofillness was7.2±5.4years;46.8%ofthepatientshadsomesystemic manifestationand97.9%hadbeenmedicatedwith immuno-suppressive agents in some time (Table 1). Most patients (56.41%) hada lowlevel ofschooling (<8years),and 8.06% wereilliterate.Mostpatientsexhibiteddiseaseactivity(53.2%),

however,therewas apredominanceoflowactivity disease

(38.7%).Thislow activitywas confirmedbythe meantotal scoreof4.95withESSDAI(0–39),inwhich11.3%ofpatientshad ascore≥12.ThemeanofPROFADwas18.68±6.23;ofSSIwas 18.19±6.40;andofthetotalscoreofPROFAD-SSI-SF(obtained byaveragingPROFADandSSIresults)was18.34±5.69(Table2). TheinternalconsistencyofPROFAD,SSIandofthetotal scorewasconsideredhigh,anda␣-Cronbachcorresponding

to0.80;0.78;and0.87,respectively,wasobtained.

TheintraobserverreproducibilityofPROFADwas0.89;of SSI,0.86;andofthetotalscorewas0.89,showinghigh

repro-ducibility. The concordance ofthe questions and domains

betweenthetwovisitswasconsideredasgood toexcellent (Table3).

WithrespecttotheconstructvalidityofPROFAD, statis-ticallysignificant correlationsofdomains “Somaticfatigue” and“Arthralgia”withallothervariableswereobtained.The domain “Mental fatigue” had astatistically significant cor-relation with PaGA, FACIT-F, ESSPRI (Fatigue, Dryness, and total)andEQ-D5(Pain/Discomfortand Anxiety/Depression).

Ontheotherhand,thedomain“Vascular”showeda

statis-ticallysignificant correlationonly withFACIT-Fand ESSPRI (Pain,Drynessandtotal).AstoPROFAD,alackofstatistically significantcorrelationonlyoccurredwiththedomain “Mobil-ity”ofEQ-D5(Table4).Withrespecttotheconstructvalidity

ofSSI,thedomain“Cutaneousdryness”showedastatistically significantcorrelationonlywithFACIT-F;thedomain “Vagi-naldryness”withFACIT-F,ESSPRI(Fatigue,Dryness,andtotal) andEQ-D5(SelfcareandUsualactivities);thedomainOcular drynesswithPaGA,FACIT-FandESSPRI;andthedomain“Oral dryness”,withallvariables.ThetotalscoreofSSIshowed sta-tisticallysignificant correlationswithPaGA,FACIT-F,ESSPRI andEQ-D5(Mobility,SelfcareandUsualactivities)(Table5).On theotherhand,thetotalscoreofPROFAD-SSI-SFonlylackeda statisticallysignificantcorrelationwiththedomain“Mobility” andwith1–100scaleofEQ-D5(Table6).

Discussion

Theeditorialofthisjournalin200630_{highlightedthe}

impor-tanceofthetranslationandvalidationofassessmenttoolsin rheumatologyfortheBrazilianPortugueselanguage, remin-ding that, withagood-quality instrument availabletothat effect, the creationofa newinstrument requiresa greater commitmentoftimeandcost.However,itisnotenoughthat the instrument be simplytranslated; it is critical an accu-rate assessment ofits translation and cultural adaptation, aswellasanevaluationofitsmeasurement(psychometric) properties afterthecompletionofthis process,evenwhen

such properties have already been demonstrated with the

originalinstrument,asimportantculturaldifferencesmaybe present.30,31

PROFAD-SSI-SFwasoriginallywrittenintheEnglish lan-guage,withquestionsappropriatetoitsownculture.Hence, forapplicationofthisinstrumenttoourpopulation,we pro-ceededtoaculturalequivalence,sothatthetoolcouldactually beadequatelyinterpretedbybothpatientandevaluator.

Demographic data and clinical characteristics of 62

patientsinthestageofmeasurementequivalenceevaluation were similartothosefoundinother studiesofassessment

toolsforpSS(PROFAD,SSI,PROFAD-SSI-SFandESSPRI) and

theircohorts:prevalence>93%inwomen,meanagebetween 47and62years,longdiseasedurationaveraging7.21,9–12_years,

and mean interval between symptoms and diagnosis of5

years,whichdemonstratesalongdelaytimefordiagnosisof thisdisease,anaspectalreadyreportedbyothers.32

TheBrazilianversionofPROFAD-SSI-SFhasdemonstrated

high internal consistency(0.80 and 0.78), aswell asin the validation study ofthe originalversion,whose ␣-Cronbach

was 0.99.11 _{The intraobserver reproducibility (r=0.69–0.85)}

also was considered high, similar to the reproducibility of the original PROFAD-LF (r=0.67–0.79), tested 24h after the first application.9 _{Thereproducibilityofthedomains}

“Ocu-lardryness”(r=0.92)and“Oraldryness”(r=92)inthestudy ofPROFAD-SSI-SFdevelopmentwasalsosimilartothatinthe presentstudy:0.83and0.92,respectively.33

In the validation study of PROFAD-LF, the questions in

thedomain“Somaticfatigue”showedcorrelationonlywith

the domains “Vitality” of SF-36 (r=−0.53–0.70) and “Physi-calhealth”ofWHOQOL-BREF(r=−0.62andr=−0.69),9_unlike

Table2–DescriptivemeasuresofPROFAD-SSI-SF,ESSPRI,EQ-D5,FACIT-FandPaGA.

Variables Mean(±) Minimum Median Maximum SD

PROFAD

Somaticfatigue 5.37 0.50 6 7 1.72

Mentalfatigue 5.1 0 6 7 2.17

Arthralgia 5 0 5.5 7 1.98

Vascular 3.21 0 3.5 7 3.03

TotalPROFAD 18.68 1.5 18.63 28 6.23

SSI

Cutaneousdryness 4.79 0 6 7 2.57

Vaginaldryness 4 0 5 7 2.96

Oculardryness 5.38 0 60 7 1.85

Oraldryness 4.02 0 4.20 7 1.85

TotalSSI 18.19 3.53 18.33 28 6.40

PROFAD-SSI-SF,total 18.34 2.52 18.13 27.63 5.69

ESSPRI

Pain 7.9 0 8 10 2.53

Fatigue 6 0 7.5 10 3.8

Dryness 6.54 0 7 10 2.76

Total 6.81 1.33 7. 10 2.12

EQ-5D

Mobility 1.66 1 2 2 0.48

Selfcare 1.35 1 1 2 0.48

Usualactivities 1.68 1 2 3 0.57

Pain/Discomfort 2.18 1 2 3 0.53

Anxiety/Depression 2.05 1 2 3 0.66

Scale1–100 56.73 0 60 100 24.21

FACIT-F 30.73 1 31 52 11.35

PaGA 7.58 2 8 10 2.15

PROFAD,ProfileofFatigueandDiscomfort;SSI,SiccaSymptomsInventory;PROFAD-SSI-SF,ProfileofFatigueandDiscomfort–SiccaSymptoms Inventory(shortform);PaGA,Patient’sGlobalAssessment;FACIT-F,FunctionalAssessmentofChronicIllnessTherapyFatigueSubscale;ESSPRI, EULARSjögren’sSyndromePatientReportedIndex;EQ-5D,EuroQOL;SD,standarddeviation.

Scoresofinstruments:PROFAD,0–28;SSI,0–28;PROFAD-SSI-SFtotal,0–28;ESSPRI0–10;EQ-5D,domains–1–3,range–0–100;FACIT-F,0–52; PaGA,0–10.

Table3–Intraobserverreproducibilitybyquestions,domainsandtotalofPROFADandSSI.

PROFAD SSI

Variables CCI Variables CCI

Questions

Question1 0.71 Question10 0.74

Question2 0.69 Question11 0.91

Question3 0.76 Question12 0.72

Question4 0.75 Question13 0.69

Question5 0.78 Question14 0.91

Question6 0.86 Question15 0.82

Question7 0.78 Question16 0.79

Question8 0.80 Question17 0.87

Question9 0.78 Question18 0.80

Question19 0.83

Domains

Somaticfatigue 0.85 Cutaneousdryness 0.74

Mentalfatigue 0.89 Vaginaldryness 0.91

Arthralgia 0.80 Oculardryness 0.83

Vascular 0.78 Oraldryness 0.92

PROFAD,total 0.89 SSItotal 0.86

Table4–SpearmancorrelationofPROFADwithPaGA,FACIT-F,ESSPRIandEQ-5D.

Variables PROFAD

Somaticfatigue Mentalfatigue Arthralgia Vascular Total

PaGA 0.60a _0.26b _0.66a _{0.23 0.50}a

FACIT-F 0.72a _0.43a _0.51a _0.37b _0.59a

ESSPRI

Pain 0.43a _{0.13 0.66}a _0.33b _0.49a

Fatigue 0.45a _0.50a _0.31b _{0.21 0.46}a

Dryness 0.43a _0.32b _0.42a _0.25b _0.42a

Total 0.53a _0.46a _0.58a _0.32b _0.58b

EQ-5D

Mobility 0.28b _{−0.03 0.35}b _{0.06 0.18}

Selfcare 0.49a _{0.22 0.31}b _{0.01 0.27}b

Usualactivities 0.45a _{0.12 0.22 0.13 0.28}b

Pain/Discomfort 0.45a _0.29b _0.54a _{0.23 0.43}a

Anxiety/Depression 0.48a _0.33b _0.31b _{0.07 0.29}b

Scale1–100 −0.42a _{−0.06 −0.36}b _{−0.24 −0.31}b

PROFAD,ProfileofFatigueandDiscomfort;PaGA,Patient’sGlobalAssessment;FACIT-F,FunctionalAssessmentofChronicIllnessTherapy FatigueSubscale;ESSPRI,EULARSjögren’sSyndromePatientReportedIndex;EQ-5D,EuroQOL.

a _P<0.001.

b _P<0.05.

Surprisingly, the domain “Mental fatigue” showed a

significant but low correlation with the domain

“Anx-iety/Depression” of EQ-5D (r=0.33). However, this low

correlationwasalsoobservedbetweenthequestionsofthe domain“Mentalfatigue”oftheoriginalPROFAD-LFwiththe domains“Mentalhealth”ofSF-36(r=−0.27to−0.44),and “Psy-chologicalhealth”ofWHOQOL-BREF(r=−0.32to−0.47)and ofHospitalAnxietyandDepressionScale(HADS)(r=−0.34to 0.48).9

Thedomain“Arthralgia”ofPROFAD-SSI-SFinthis study

showedasignificantcorrelationwith“Mobility”(r=0.35)and “Pain/Discomfort”domains(r=0.54)ofEQ-5D.Thiswasalso

found in the correlation of the questions of “Discomfort”

domainoftheoriginalPROFAD-LFwith“Pain”domainof SF-36(r=0.65)and“Physicalhealth”domainofWHOQOL-BREF (r=0.62).9

In the validation study of ESSPRI, a good correlation

betweentotal scoreofPROFAD (r=0.66) andofSSI(r=0.56)

with PaGA1 _{was obtained – values similar to those in the}

presentstudy(0.50and0.43,respectively).

As important as the objective measures is the

assess-ment of pSS symptoms focused on patient’s perception,

both with the aim todetermine the clinical improvement

in clinical trials, as to observe the progressof patients in

Table5–SpearmancorrelationofSSIwithPaGA,FACIT-F,ESSPRIandEQ-5D.

Variables SSI

Cutaneousdryness Vaginaldryness Oculardryness Oraldryness Total

PaGA 0.20 0.16 0.59a _0.444a _0.43a

FACIT-F 0.31b _0.42a _0.43a _0.611a _0.57a

ESSPRI

Pain 0.08 0.23 0.29b _0.33b _0.30b

Fatigue 0.07 0.34b _0.38b _0.46b _0.41a

Dryness 0.22 0.62a _0.35b _0.55a _0.63a

Total 0.13 0.49a _0.41a _0.57a _0.55b

EQ-5D

Mobility 0.01 0.19 0.168 0.26b _0.27b

Selfcare 0.05 0.35b _{0.100 0.28}b _0.29b

Usualactivities 0.22 0.33a _{0.025 0.35}b _0.37b

Pain/Discomfort 0.07 0.13 0.232 0.30b _0.24

Anxiety/Depression 0.05 0.16 0.197 0.37b _0.22

Scale1–100 0.107 0.02 −0.231 −0.33b −0.11

SSI,SiccaSymptomsInventory;PaGA,Patient’sGlobalAssessment;FACIT-F,FunctionalAssessmentofChronicIllnessTherapyFatigueSubscale; ESSPRI,EULARSjögren’sSyndromePatientReportedIndex;EQ-5D,EuroQOL.

a _P<0.001.

Table6–SpearmancorrelationofPROFAD-SSI-SFtotal withPaGA,FACIT-F,ESSPRIandEQ-5D.

Variables PROFAD-SSI-SFtotal

PaGA 0.536b

FACIT-F 0.699b

ESSPRI

Pain 0.324a

Fatigue 0.423b

Dryness 0.441b

Total 0.545b

EQ-5D

Mobility 0.242

Selfcare 0.351a

Usualactivities 0.421b Pain/Discomfort 0.383a Anxiety/Depression 0.332a Scale1–100 −0.249

PROFAD-SSI-SF,ProfileofFatigueandDiscomfort–SiccaSymptoms Inventory(shortform);PaGA,Patient’sGlobalAssessment;FACIT-F, FunctionalAssessmentofChronicIllnessTherapyFatigueSubscale; ESSPRI,EULARSjögren’sSyndromePatientReportedIndex;EQ-5D, EuroQOL.

a _P<0.001.

b _P<0.05.

clinicalpractice.UnlikeESSPRI,whichhasonlythree ques-tions(domains),dilutingrelevantgroupsofsymptomsthat

are not always present together, PROFAD evaluates

differ-entaspectsoffatigue(somaticandmental)and discomfort

(arthralgia and vascular), and SSI evaluates dryness

(cuta-neous, vaginal dryness, ocular dryness, and oral dryness)

separately,andthisturnsPROFAD-SSI-SFintoamore conve-nienttooltouseinclinicalpracticeformonitoringpatients’ symptoms,andindetailingsymptomsinclinicalresearch.

Conclusion

TheBrazilianPortugueseversionofPROFAD-SSI-SF(Annex1) provedtobeanadaptable,reproducibleandvalidtoolforthe PortugueselanguageandcanbeusedintheBraziliancontext. Inaddition,thetotalscoreobtainedbyaveragingPROFAD andSSI,whichisnotcalculatedintheoriginalversion,has demonstratedinternalconsistency,reproducibilityand valid-ity.

Funding

FinancialsupportofConselhoNacionaldeDesenvolvimento

CientíficoeTecnológico(CNPq),Brazil.

Conflicts

of

interest

Theauthorsdeclarenoconflictsofinterest.

Annex

1.

Profile

of

Fatigue

and

Discomfort

–

Sicca

Symptoms

Inventory

(short

form)

(PROFAD-SSI-SF)

in

Brazilian

Portuguese

language

Porfavor,avalieoquantoosseussintomastêmestadoruinsnapiorfasenasúltimasduassemanas,assinalandoumnúmerode 0a7.

1.Amaiordificuldadequeeutivenasduasúltimassemanascomnecessidadededescanso,mesentindocansado,

exaustoouprecisandodormir:

nenhumanecessidadededescansar 0 1 2 3 4 5 6 7 tãoruimquantovocêpuder

imaginar 2.Amaiordificuldadequeeutivenasduasúltimassemanasparacomec¸arumaatividade,usandomuitoesforc¸o parafazerascoisasousentindocomoseestivesse“numabatalha”:

nenhumadificuldadeparacomec¸ar

umaatividade

0 1 2 3 4 5 6 7 tãoruimquantovocêpuder

imaginar

3.Amaiordificuldadequeeutivenasduasúltimassemanasparacontinuarumaatividade,mesentindofacilmente

exaustoousemenergia:

nenhumadificuldadeemcontinuar

umaatividade

0 1 2 3 4 5 6 7 tãoruimquantovocêpuder

imaginar

4.Amaiordificuldadequeeutivenasduasúltimassemanascomfaltadeforc¸anosmeusmúsculosoumesentindo fraco:

nenhumafaltadeforc¸a 0 1 2 3 4 5 6 7 tãoruimquantovocêpuder

imaginar 5.Amaiordificuldadequeeutivenasduasúltimassemanasemnãopensarclaramenteouachandodifícilme concentrar:

nenhumproblema 0 1 2 3 4 5 6 7 tãoruimquantovocêpuder

imaginar 6.Amaiordificuldadequeeutivenasduasúltimassemanasesquecendocoisasoucometendoerros:

nenhumproblema 0 1 2 3 4 5 6 7 tãoruimquantovocêpuder

7.Amaiordificuldadequeeutivenasduasúltimassemanascomdesconfortonosmeusmembros,como,por exemplo:desconfortooudornasgrandesjuntas(quadril,joelho,ombro)ounosmeusmúsculosoudorportodoo corpo:

nenhumproblema 0 1 2 3 4 5 6 7 tãoruimquantovocêpuder

imaginar 8.Amaiordificuldadequeeutivenasduasúltimassemanascomdorouinchac¸onosdedosoupunhos:

nenhumproblema 0 1 2 3 4 5 6 7 tãoruimquantovocêpuder

imaginar 9.Amaiordificuldadequeeutivenasduasúltimassemanascomasmãosdesconfortavelmentefrias:

nenhumproblema 0 1 2 3 4 5 6 7 tãoruimquantovocêpuder

imaginar 10.Amaiordificuldadequeeutivenasduasúltimassemanascompelesecaoucoceira:

nenhumproblema 0 1 2 3 4 5 6 7 tãoruimquantovocêpuder

imaginar

11.Amaiordificuldadequeeutivenasduasúltimassemanascomsecuravaginalcomo,porexemplo:momentosde

desconfortodurantearelac¸ãosexualdevidoàsecuravaginal:

nenhumproblema 0 1 2 3 4 5 6 7 tãoruimquantovocêpuder

imaginar

12.Amaiordificuldadequeeutivenasduasúltimassemanascomolhossensíveiscomo,porexemplo:sensac¸ãode areia,dor,queimac¸ão,coceiraouirritac¸ão.

nenhumproblema 0 1 2 3 4 5 6 7 tãoruimquantovocêpuder

imaginar 13.Amaiordificuldadequeeutivenasduasúltimassemanascomirritac¸ãonosolhoscomo,porexemplo:nos

ambientescomfumac¸a,sentindodesconfortoaovento,noarcondicionadooulugarescombaixaumidade:

nenhumproblema 0 1 2 3 4 5 6 7 tãoruimquantovocêpuder

imaginar

14.Amaiordificuldadequeeutivenasduasúltimassemanasnavisão(mesmousandoóculos)como,porexemplo, visãoembac¸ada,fraca,comlimitac¸ãoparaleitura,paraassistirtelevisãooudirigirànoite,verateladocomputador ouateladocaixaeletrônicodebancos:

nenhumproblema 0 1 2 3 4 5 6 7 tãoruimquantovocêpuder

imaginar

15.Amaiordificuldadequeeutivenasduasúltimassemanasparamealimentarcomo,porexemplo:sentiraboca seca,dificuldadeparaengoliralimentos,necessidadedeingerirlíquidosparaengolirosalimentos,alimentos entalados,necessidadederetirarrestosdealimentosdabocaouapreciarmenosacomida:

nenhumproblema 0 1 2 3 4 5 6 7 tãoruimquantovocêpuder

imaginar

16.Amaiordificuldadequeeutivenasduasúltimassemanascomgargantaounarizsecoscomo,porexemplo:boca secaquandorespirando,dificuldadeemfalarcombocaseca,necessidadedeingerirlíquidoparafalarfacilmente, sentindoonarizseco,gargantaseca,ouarcondicionadoressecandoaminhaboca:

nenhumproblema 0 1 2 3 4 5 6 7 tãoruimquantovocêpuder

imaginar 17.Amaiordificuldadequeeutivenasduasúltimassemanascommauhálitocomo,porexemplo:sentindoqueo hálitotinhacheiroruim,salivagrossa:

nenhumproblema 0 1 2 3 4 5 6 7 tãoruimquantovocêpuder

imaginar 18.Amaiordificuldadequeeutivenasduasúltimassemanascomnecessidadedelíquidoparamolharaminha bocacomo,porexemplo:levaralgoparabebernacama,precisarbeberalgoduranteanoite,acordarànoitepara urinar,ternecessidadeurgenteemurinar:

nenhumproblema 0 1 2 3 4 5 6 7 tãoruimquantovocêpuder

imaginar

nenhumproblema 0 1 2 3 4 5 6 7 tãoruimquantovocêpuder imaginar

SCORECALCULATION

PROFAD:meanof“somaticfatigue”(1+2+3+4/4)+“mentalfatigue”(5+6/2)+“arthralgia”(7+8/2)+“vascular”(9) domains

SSI:meansof“cutaneousdryness”(10)+“vaginaldryness”(11)+“oculardryness”(12+13+14/3)+“oculardryness” (15+16+17+18+19/5)

PROFAD-SSI-SFtotal:meanofPROFAD+SSI

r

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f

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