Quality of Life of Persons Quality of Life of Persons with Tourette Syndromewith Tourette SyndromeWebinarWebinar

July 19, 2007July 19, 2007Presented by AUCD and supported by Cooperative Presented by AUCD and supported by Cooperative

Agreement U59/CCU321285Agreement U59/CCU321285--01 from the National Center on 01 from the National Center on Birth Defects and Developmental Disabilities (NCBDDD) at Birth Defects and Developmental Disabilities (NCBDDD) at

Center for Disease Control and Prevention (CDC) Center for Disease Control and Prevention (CDC)

Webinar AgendaWebinar AgendaI. Welcome & Introduction I. Welcome & Introduction –– Sue Lin, MS Project Sue Lin, MS Project

Director, AUCDDirector, AUCD--NCBDDD Cooperative AgreementNCBDDD Cooperative AgreementII. Presentation II. Presentation

Quality of Life Among Youth with Tourette Quality of Life Among Youth with Tourette Syndrome Syndrome –– Sam Zinner, MD & Tari Topolski, PhD Sam Zinner, MD & Tari Topolski, PhD (WA UCEDD) (WA UCEDD)

III. DiscussantIII. DiscussantSue LeviSue Levi--Pearl, V.P. and Director of Medical and Pearl, V.P. and Director of Medical and Scientific Programs at Tourette Syndrome Scientific Programs at Tourette Syndrome Association Association

IV. Question and AnswerIV. Question and Answer

NCBDDDNCBDDD--AUCD Cooperative AgreementAUCD Cooperative Agreement

Strengthen the nation's capacity Strengthen the nation's capacity to carry out public health and to carry out public health and disability activities disability activities Foster collaborations among Foster collaborations among AUCD, its members, and AUCD, its members, and NCBDDD NCBDDD Facilitate a wide range of Facilitate a wide range of research, education, and research, education, and dissemination activities. dissemination activities.

Research Topics of Interests (RTOI)Research Topics of Interests (RTOI)

RTOI are specific research area of significance identified by scRTOI are specific research area of significance identified by scientists at ientists at NCBDDD, CDC. Past RTOI projects have focused on the following NCBDDD, CDC. Past RTOI projects have focused on the following areas: areas: Health Communication and Education Health Communication and Education Prevention of Secondary Conditions Prevention of Secondary Conditions Healthcare Cost Analysis Healthcare Cost Analysis Quality of Life Studies Quality of Life Studies Developmental Factors and Outcomes Developmental Factors and Outcomes Health Promotion Interventions Health Promotion Interventions CoCo--Morbidity Prevalence Studies Morbidity Prevalence Studies

Specific disabilities areas include: autism, Down syndrome, DuchSpecific disabilities areas include: autism, Down syndrome, Duchenne enne muscular dystrophy, hearing loss, fetal alcohol syndrome, spina muscular dystrophy, hearing loss, fetal alcohol syndrome, spina bifida, bifida, and Tourette syndrome. and Tourette syndrome.

Presenters and Discussant Presenters and Discussant

Sam Zinner, MD

University of Washington

Tari Topolski, PhD

University of Washington

Sue Levi-Pearl, V.P. and Director of

Medical and Scientific Programs

at TSA

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Quality of Life among Youth Quality of Life among Youth with Tourette Syndromewith Tourette Syndrome

Samuel H. Zinner, MDSamuel H. Zinner, MDTari D. Topolski, PhDTari D. Topolski, PhD

University of WashingtonUniversity of Washington

Supported bySupported byNational Center on Birth Defects and National Center on Birth Defects and

Developmental DisabilitiesDevelopmental Disabilities(NCBDDD) (NCBDDD)

in cooperation within cooperation with

Assn of University Centers on Disabilities Assn of University Centers on Disabilities (AUCD)(AUCD)

Learning ObjectivesLearning Objectives

QoL definitionsQoL definitionsQoL in Tourette syndromeQoL in Tourette syndromeOur projectOur project

Project AimsProject Aims

Identify factors that contribute to Identify factors that contribute to QoL in QoL in youth (TS and TS+)youth (TS and TS+)

Is a TSIs a TS--QoL specific measure needed?QoL specific measure needed?Impact of TS on the FamilyImpact of TS on the Family

LongLong--range Goalsrange Goals

Outcomes measurement Outcomes measurement devdev’’tt phasesphases1.1. Create assessment modulesCreate assessment modules2.2. Validate among different populationsValidate among different populations3.3. Confirm in clinical practiceConfirm in clinical practice

Why Measure Quality of Life?Why Measure Quality of Life?

Goal of intervention Goal of intervention –– QoL indistinguishable from unaffected QoL indistinguishable from unaffected

peerspeers

WallanderWallander, Schmidt and , Schmidt and KootKoot, 2001, 2001

Concepts in Quality of LifeConcepts in Quality of Life

Perceived Perceived

vs.vs.

HealthHealth--relatedrelated

Determinants of Health and Determinants of Health and Perceived Quality of LifePerceived Quality of Life

Social & Cultural

Economic & Political

Physical & Geographic

Health, Education & Social Care

External to Individual

Symptoms Functional Status

Health Perceptions Opportunity

Biology & Life Course

Lifestyle & Health Behavior

Personality & Motivation

Values & Preferences

General

Quality

of Life

Health Status and Health-Related Quality of Life

Internal to Individual

Illness Behavior

SurvivalPatrick, 1993

Definition of Quality of LifeDefinition of Quality of Life

Perceptions of onePerceptions of one’’s position in lifes position in life–– in context of their culture & value systemsin context of their culture & value systems–– goals, expectations, standards & concernsgoals, expectations, standards & concerns

World Health Org. QOL Group 1994, 1997World Health Org. QOL Group 1994, 1997

Quality of Life as a LensQuality of Life as a Lens

Integrates internal & external influences Integrates internal & external influences Incorporates individualIncorporates individual’’s perspectives perspectiveRelates individualRelates individual’’s perceptions to s perceptions to othersothers’’Pinpoints what is important to individualsPinpoints what is important to individuals

Generic Generic vsvs Specific QoL MeasuresSpecific QoL Measures

GenericGeneric–– compare different populationscompare different populationsSpecificSpecific–– capture concerns of specific populationscapture concerns of specific populations–– evaluate interventions evaluate interventions –– identify subtle influencesidentify subtle influences

YQOL Conceptual ModelYQOL Conceptual Model"My Evaluation Of…"

Social Relationships Sense of Self Environment

• adult support • belief in self • engagement, activities• caring for others • being oneself • good education• family relations • mental health • liking neighborhood• freedom • physical health • monetary resources• friendships • spirituality • personal safety• participation • view of future• peer relations

General Quality of Life

• enjoying life• feeling life is worthwhile• satisfied with one’s life

"…my relationshipswith others"

"…my life"

"…my feelings aboutmyself"

"…my opportunities &obstacles"

Tourette syndrome and QoLTourette syndrome and QoL

Studies so farStudies so far––ElstnerElstner et al et al (2001)(2001)––Bernard et alBernard et al (2006)(2006)––StorchStorch et al et al (2007)(2007)––FaqihFaqih (2007)(2007)

Cultural influencesCultural influences

TOURETTE

OCD

LD Depression ADHD

GAD

“PLUS”

TOURETTE

Genericquantitative

QoL instrument

Tics

Psycho-social

Comorbidconditions

Other

Quantitative process

Qualitative process Interviews

TS-specificquantitative

QoL instrument

Genericquantitative

QoL instrument

Quantitative process

Qualitative process

TS-specificquantitative

QoL instrument

Assessment InstrumentsAssessment Instruments

QuantitativeQuantitative–– SelfSelf--administered reportsadministered reports–– Other psychometric instruments Other psychometric instruments –– Structured interviewStructured interview

QualitativeQualitative–– SemiSemi--structured interviewstructured interview

Tourette syndrome and QoLTourette syndrome and QoL

SitesSites––U. Washington U. Washington (USA)(USA)––U. British Columbia U. British Columbia (Canada)(Canada)RecruitmentRecruitment

Assessment Step: QuantitativeAssessment Step: Quantitative

ParticipantsParticipants

DiagnosisDiagnosisTSTS--onlyonly TS+TS+

UWUW 3333UBCUBC 1818 2525 4343

TotalTotal 3838 5858 9696

2020TotalTotal5353SiteSite

Assessment Step: QuantitativeAssessment Step: Quantitative

Participant characteristicsParticipant characteristics

Age (n = 96)Age (n = 96) Mean (SD)Mean (SD)

At tic onset At tic onset 5.6 (2.3) 5.6 (2.3)

At diagnosis At diagnosis 9.2 (2.7) 9.2 (2.7)

At time of participationAt time of participation 13.5 (2.4) 13.5 (2.4)

Participant features Participant features -- GenderGender

Female (n = 9)

Male (n=87)

Participant features Participant features -- EthnicityEthnicity

Caucasian

Hispanic

African American

Amer. Indian/Alaskan Asian/Pacific

Comorbid frequenciesComorbid frequencies

FeatureFeature Med Interview Med Interview n (%)n (%)

Current Symptoms

ADHDADHD 44 (46%) 44 (46%) 9 (09%)OCDOCD 44 (46 %) 44 (46 %) 53 (55%)AnxietyAnxiety 22 (23 %)22 (23 %) 11 (12%)11 (12%)DepressionDepression 14 (15%)14 (15%) 8 (08%)LD 25 (26 %)25 (26 %) 54 (56%)

Additional frequenciesAdditional frequencies

FeatureFeature nn (%)(%)

CoproCopro--phenomena phenomena hxhx 18 (19 %) 18 (19 %)

Rage Attacks (now)Rage Attacks (now) 17 (18 %) 17 (18 %) Integrative health Integrative health hxhx 22 (23 %) 22 (23 %) Fine motor difficultyFine motor difficulty 50 (52 %) 50 (52 %)

Family HistoryFamily HistoryUBCUBCn=43n=43

%%

UWUWn=53n=53

%%

TotalTotaln=96n=96

%%TicsTics 61.961.9 56.656.6 58.958.9

ADHDADHD 52.852.8 43.943.9 48.948.9

OCDOCD 41.541.5 56.656.6 50.050.0

Youth QoL Compared with OthersYouth QoL Compared with Others

76.676.6RelationshipsRelationships75.575.5SelfSelf

80.480.377.489.489.4GeneralGeneral Quality Quality

of Lifeof Life

79.878.4Environment/CultureEnvironment/Culture

MLMLn=52n=52

ADHDADHDn=55n=55

TSTSn=96n=96

ControlControln=107n=107YQOLYQOL--R DomainR Domain

80.9 69.2 73.4

Scores are on a scale of 0-100;

82.7 72.2 72.7

89.4 81.4

QoL Comparisons QoL Comparisons Between Youth PopulationsBetween Youth Populations

All condition groups reported lower QoL All condition groups reported lower QoL Scores than youth with no chronic Scores than youth with no chronic conditionsconditionsSense of Self Sense of Self TS lower than ML TS lower than ML RelationshipsRelationshipsEnvironmentEnvironment = ADHD= ADHDGeneral Quality of LifeGeneral Quality of Life

During the past 4 weeks, how often has your child a. . .

TS onlyTS onlyn=38n=38

TS+TS+n=54n=54

ADHDADHDn=19n=19

ControlControln=8n=8

behavior caused problems in your family

1.34bc

(0.99)2.19 (1.01)

2.16 (0.77)

1.20bc

(0.42)

spent time with a friend outside of school

2.972.97bc

(0.92)(0.92)2.262.26(0.99)(0.99)

2.162.16(1.07)(1.07)

3.40bc

(0.52)had serious emotional/ mental problems

0.610.61(0.89)(0.89)

1.501.50(1.13)(1.13)

1.211.21(0.79)(0.79)

0.100.10(0.32)(0.32)

missed an activity because of physical/emotional problems

0.370.37b

(.068)(.068)1.001.00(1.12)(1.12)

0.740.74(0.87)(0.87)

0.100.10b

(0.32)(0.32)

Mothers Reports on Contextual QoLMothers Reports on Contextual QoL

a YQOL contextual items are scored on a scale of 0 to 5 with 0 =Never and 4 = Very Often bsig from TS+ p<.05 c Differs from ADHD group at the alpha <.05 level

MothersMothers’’ Reports of Impact on the Reports of Impact on the FamilyFamily

TS Only Mean (sd)

TS Plus Mean (sd)

ADHD Mean (sd)

Control Mean (sd)

Negative Parenting 6.47 (4.69) 12.61 (5.50) 13.28 (6.31) 4.50 (2.97)

Positive Parenting 11.79 (4.84) 8.89 (4.42) 6.88 (4.36) 12.63 (4.11)

Social Impact 2.50 (4.11) 6.22 (7.00) 6.60 (6.54) 0.25 (0.577)

Financial Impact 2.87 (3.57) 5.31 (4.55) 6.44 (3.28) 1.63 (3.36)

Marital Impact 3.21 (3.20) 7.22 (3.90) 7.89 (5.27) 2.67 (2.46)

Sibling Impact 3.24 (2.68) 7.30 (4.99) 7.05 (3.92) 2.40 (1.06)

Generic v Specific QoL MeasuresGeneric v Specific QoL Measures

GenericGeneric–– compare different populationscompare different populationsSpecificSpecific–– capture concerns of specific populationscapture concerns of specific populations–– evaluate interventions evaluate interventions –– identify subtle influencesidentify subtle influences

Determinants of Health and Determinants of Health and Perceived Quality ofPerceived Quality of LifeLife

Social & Cultural

Economic & Political

Physical & Geographic

Health, Education & Social Care

External to Individual

Symptoms Functional Status

Health Perceptions Opportunity

Biology & Life Course

Lifestyle & Health Behavior

Personality & Motivation

Values & Preferences

General

Quality

of Life

Health Status and Health-Related Quality of Life

Internal to Individual

Illness Behavior

SurvivalPatrick, 1993

Assessment Step: QualitativeAssessment Step: Qualitative

InterviewsInterviews

SemiSemi--structured itemstructured item--generationgenerationSiteSite # Subjects# Subjects

UWUW 18181515

TOTALTOTAL 3333UBCUBC

Module Development ProcessModule Development ProcessSemi-structured interviews (33 youth with/TS)

Item pool developmentExpertopinion

Items selected based on theoretical approach

Focus groups: young adults w/TS

SemiSemi--structured Interviews:structured Interviews:Question samplesQuestion samples

How would you describe a person with How would you describe a person with a a ““good/poorgood/poor”” QoL?QoL?If you could change one thing about If you could change one thing about your life, what would it be?your life, what would it be?Would you say people treat you Would you say people treat you differently because of your TS, either differently because of your TS, either positively or negatively?positively or negatively?

Item Pool Development:Item Pool Development:Text selection by 2 membersText selection by 2 members

Sociology

Social psychology

Developmentalpsychology

Ecologicalpsychology

Anthropology

Interview transcripts

Developmentalpediatrics Relevant text

Item Pool Development:Item Pool Development:Code generation by 2 membersCode generation by 2 members

Relevant text

Sorted into Basic DomainsEnv Rel

SelfGen QoL

Item Pool Development:Item Pool Development:Draft items writtenDraft items written

Relevant textBasic domains

Draft items

My peers judge me because of my tics

Because of my tics it is difficult to make friends

“it makes it difficult to make friends, especially in the teenager years because people

are just so judgmental on

what you look like and what you do.”

Env Rel

SelfGen QoL

Item CriteriaItem Criteria

Item must be:Item must be:–– specific to TSspecific to TS–– relevant to all youth with TSrelevant to all youth with TS–– amenable to changeamenable to change–– frequently mentioned by youth with TSfrequently mentioned by youth with TS–– likely to discriminate w/symptom severitylikely to discriminate w/symptom severity

Item CriteriaItem Criteria

Item should:Item should:–– evaluate a feeling, sensation or perceptionevaluate a feeling, sensation or perception–– have semantic equivalence in other have semantic equivalence in other

languageslanguages–– be in the parlance of the youthbe in the parlance of the youth

Results from InterviewsResults from Interviews

Generic QOL measure doesnGeneric QOL measure doesn’’t address:t address:

teasing & bullying issues identified in teasing & bullying issues identified in the interviewsthe interviews

environmental issues such as feeling environmental issues such as feeling like they cannot go to some public like they cannot go to some public placesplaces

YQOLYQOL--TS in developmentTS in development

ConclusionsConclusions

Preliminary results show:Preliminary results show:––youth w/ TS experience a poorer QOL youth w/ TS experience a poorer QOL

than youth w/o chronic conditionsthan youth w/o chronic conditions––there is a significant impact upon the there is a significant impact upon the

familyfamily––there is a need for a TSthere is a need for a TS--specific QOL specific QOL

measuremeasure

CollaboratorsCollaboratorsUniversity of WashingtonUniversity of Washington

Samuel H. Zinner, MDSamuel H. Zinner, MDTari D. Topolski, PhDTari D. Topolski, PhDTodd C. Edwards, PhDTodd C. Edwards, PhDDonald L. Patrick, PhD, MSPHDonald L. Patrick, PhD, MSPH

University of British ColumbiaUniversity of British ColumbiaRoger D. Freeman, MDRoger D. Freeman, MD

AssistantsAssistantsUniversity of WashingtonUniversity of Washington

Ren Taylor, BSRen Taylor, BSKatie White, BAKatie White, BAVanessa White, BAVanessa White, BA

University of British ColumbiaUniversity of British ColumbiaStefan CaneyStefan CaneyLauren GilesLauren Giles

Contact InformationContact Information

Tari D. Topolski, PhDTari D. Topolski, PhDU. Washington U. Washington -- Seattle QoL GroupSeattle QoL Grouptopolski@u.washington.edutopolski@u.washington.edu

Samuel H. Zinner, MDSamuel H. Zinner, MDU. Washington U. Washington –– DepDep’’t of Pediatricst of Pediatricsszinner@u.washington.eduszinner@u.washington.edu