Challenges for economic evaluation when doing research with people with learning disabilities

Claire Hulme, John O’Dwyer, Louise Bryant, Amy Russell, Allan House on behalf of the OK Diabetes Research Team

Academic Unit of Health Economics, University of Leedsc.t.hulme@leeds.ac.uk0113 343 0875

Funded by National Institute of Health Research, Health Technology Assessment Research Programme 10/102/03: Managing with Learning Disability and Diabetes.OK Diabetes. The views expressed are those of the authors and not necessarily those of the NHS, the NIHR or the Department of Health

Introduction

• Research with hard to reach vulnerablegroups presents myriad challenges

• This is particularly pertinent for peoplewith learning difficulties

• People with learning difficulties werenot involved in research that was aboutthem even as interviewees until the1980s

• Methodological difficulties include atendency to acquiesce because somuch of their lives are controlled byothers

• How do we know if the person reallyunderstands what we are asking them?

• Economic evaluations rely on healthcare or health insurance records orforms such as the CSRI completed bythe service provider rather than bypeople with learning disabilitiesthemselves

• This presentation reports on thedevelopment and testing of datacollection methods for use in aneconomic evaluation within a RCT for amanualised supported diabetes self-management programme for peoplewith mild/moderate learningdisabilities: the OK Diabetes study

• Background: Learning difficulties and diabetes

• A little about the research and overall study ‘challenges’

• The Challenges for economic evaluation so far

• Discussion

Background: Diabetes

• Prevalence of diabetes rising nationally

• Leeds (Pop ~725k) there are >27000 onthe QOF diabetes register and inBradford (Pop ~520k) there are 26500on the QOF diabetes register

• This gives population prevalence thatare typical of published figures fromelsewhere of 4% and 5%

• There are 3.2 million people diagnosedwith diabetes in the UK

• An estimated 630,000 people have thecondition, but don’t know it

• Diabetes develops when glucose can’tenter the body’s cells to be used asfuel. This happens when either:

• There is no insulin to unlock the cells(Type 1)

• There is not enough insulin or theinsulin is there but not workingproperly (Type 2)

Background: Learning disabilities

• People with a learning disability havepoorer health

• More likely to have additional healthproblems e.g. weight, mental healthand respiration

• Have poorer health outcomes

• CIPOLD (Confidential Inquiry intoPremature deaths Of people withLearning Disabilities)

• Men with learning disabilities die onaverage 13 years earlier than thosewithout ; women on average 20 yearsearlier; 22% under age of 50 when theydied

• Most common underlying problem washeart /circulatory disorders and cancer

• Most common immediate problem wasa respiratory

• More likely to have multiple long termconditions

• Delays in diagnosis and treatment

• Lack of communication betweenprofessionals; co-ordination of careacross different disease pathways andservice providers

Learning disabilities and diabetes

• Type 2 diabetes disproportionately affects people with a learning disability

• Studies of GP data (NHS MiQuestsystem) in the UK shown higher prevalence of diabetes in people with LD (Glover et al, 2012)

• This higher rate has also been found in USA, Netherlands and Canada

• People with LD are more likely to be admitted to hospital as an emergency with complications of diabetes

• Admission to hospital for AmbulatoryCare Sensitive Conditions (ACSCs)indicates potential weaknesses inprimary care that need addressing

• ACSCs are chronic conditions for whichit is possible to prevent acuteexacerbations and reduce the need forhospital admission through activemanagement. Examples includecongestive heart failure, diabetes,asthma, angina, epilepsy andhypertension.

Supported self-management

• Self-management is a standard part of the NHS response to long-term conditions

• It is unclear whether supported self-management, which is widely promoted in other areas, would be of benefit in improving the health of people with mild/moderate learning disability and type 2 diabetes

• Supported self-management programmes have an educational component

• Problem-solving strategies; • Goal setting and planning behaviour

change• Self-monitoring• Effective use of resources – including

healthcare• Work with supporters• Managing emotions

Research question

• Is it possible to develop a practicable programme of supported self-management for people with mild/moderate learning disability and type 2 diabetes?

• And evaluate it in an RCT?

• Type 2 as it has lifestyle management aspects

• Insulin use needs very specific advice to calculate doses

Phase 1:

• identify people with learning disability and type 2 diabetes and characterize their diabetes management and control

• Develop a supported self-management programme

Phase 2:

• Undertake a feasibility RCT

Learning disabilities or difficulties?

• Phase 1: Identifying people with a learning disability and type 2 diabetes

• We have asked:

– GPs

– Charities like Tenfold and People in Action

– Secondary Care e.g. LGI diabetes clinic

– Community LD Team

– LD Housing providers

• Learning difficulty is difficult to define and identify, especially at the milder end of the spectrum

• It can be defined statistically based on test scores, which typically show a negatively-skewed distribution, and in those terms, it is often said that 2% of the general population will have some degree of learning difficulty

• However, the picture becomes more complex when functional impairment in real-world activities is built into the definition

Learning disabilities or difficulties?

• Functional deficit may not be entirely attributable to intellectual impairment but to (for example) emotional or social problems or missed schooling

• Conversely, an adult with intellectual impairment may not come to the attention of statutory or non-statutory agencies if he or she is functioning independently or is well supported by family or some other informal carer

• The functional approach to definition is now widespread

• Learning disability (referring to an intellectual impairment) and learning difficulty (referring to a functional state)

• Learning disability often refers to specific deficits such as dyslexia, even when it is not associated with more general intellectual impairment or functional deficit

• We use the term learning difficulties to encompass all types of intellectual and educational deficit that lead to problems with self-management,

Case definition

• Not a diagnostic checklist but a guide tohelp identify possible participants

• Activities

• Can/do they:

• Read, write, manage money, look aftertheir personal care, tell the time, cook,have difficulty in communicating withother people?

• Remembering

• Can they remember:

• Significant things about themselves (e.g. birthday), significant things about their environment (e.g. where they live), when to do things (get up, what time dinner is), what you have said?

• Life experience

• Have/do they:

• Attended a special school, or statement of special educational need; attend a day centre; live outside a hospital or a LD residential service; have people who support them e.g. care manager, advocate, or informal supporter?

Learning disability in Leeds

• Estimated 14,000 people in Leeds with a learning disability

• Only 2128 on LD register

• 3300 in receipt of paid support

• QOF Diabetes register in Leeds has 27,000 people

• Only 98 of these are also on LD register

• Assuming 2% of adults have LD, should be 540 (approx.)

Recruitment from primary care:

• Read-code based searches

• Advice to GPs re: alternative approaches

• Regular mailings & newsletters

• Face to face meetings (GPs & Practice Managers)

• Attendance at events

• Engagement with and support from WSY&B CSU

• WYCLRN support

• But... recruitment slow

Recruitment from primary care:

• Non-response from GPs

• Uncomfortable referring vulnerable population

• Reluctance to discuss with those without formal diagnosis of LD

• Primary Care LD registers limited

• Lack of support from PCRN

• Higher proportions using insulin -reduced ‘pool’

• Difficulty contacting potential participants

• More cases than expected come from outside healthcare

Recruitment Beyond Healthcare

Local

Authority

LD

CharitiesFurther

EducationAdvocacy Employment

Citizens

Advice

Bureau

Disability

Employment

Advisors

(Job Centre)

MENCAP

employability

service

Foundation

Skills

Advisors FE

colleges

Fulfilling

Lives Day

Centres

Advocacy

Charities in

all 3 areas

Carers

Community

Volunteering

e.g. The

Conservation

Volunteers

Supported

housing

providers

Carers orgs. In

all 3 areas:

events and

newsletters

Health

Charities

HALE Bradford,

Zest Health for

Life, Feelgood

Factor

MENCAP,

Thru the

Maze, People

1st Bradford/

Keighley,

Tenfold

1:1 Support

Services

e.g. St

Anne’s

Where we are now.....

• We have produced all the necessary materials for a trial:

– information; consent;

– Protocol; CRFs; outcome measures

– Intervention + adherence measure

• We have learned a great deal about recruitment from the target population

• We on target to recruit 200 participants of whom at least 75% will be eligible to participate and have already expressed an interest in being re-contacted

What about the economic evaluation?

• Resource use

• HES – but need for community resources

• GP records – completed by the GP or GP surgeries

• TPP Systmone

• Client questionnaires – interviewer administered

• Outcomes

• EQ-5D interview administered

• Client questionnaires

• 4 week recall except diet, previous day

• Interview administered by researcher

• Often there will be a supporter present at the interview

• Form designed to ask about health and social care, employment, travel, and diet (change in diet important part of the self-management intervention)

• Each question on the EQ-5D printed on an A4 sheet as interview aid

Obtaining data from GPs

• Forms were simplified and contained only 10 questions for brevity

• Tick box plus number of visits/appointments

• Researcher regularly sent reminders and visited to talk through completion Challenges:

• Skip sections

• Refuse to complete

• Incomplete forms

• Use tick box but not number

• This was even from GPs who are very research ‘savvy’

• Why?

• Many of the contracts are recorded as free text in the records – e.g. phone contact with the GP; referral letters from psychiatrist – not coded

• Time intensive to go through

• Dependent on the knowledge of the practice manager or whoever is tasked with completing the form

• Some admin staff reluctant to hand over data – don’t feel they have the authority

Possible solutions…

• Find out who the client sees most often in the GP surgery – send the form to them?

• But… if this is the GP this will be likely delegated to the practice manager

• Often practice nurses do health checks?

• Lack of coded data – free text is a worry in data returns to Systmone

Client questionnaire

• Supporter can be formal or informal

• Informal supporters often also have LD

• Formal supporters have incomplete knowledge

• Concept of time: poor recall of GP visits etc

• Recall big events such as hospital, A&E – but often these are actually years ago

• Confusion over hospital and clinic visits

• Little understanding of medication/prescriptions as these tend to be sent by the pharmacy or collected by a supporter

• No recall of some GP consultations as they are not involved e.g. their supporter may phone the GP on their behalf

• In terms of domestic help needed (social care) most in our cohort live in shared homes so the paid supporter does these things – although there are different levels of help

• Means of travel to health care appointment is always remembered; given bus numbers and for some there is a staff driver

Client questionnaire

• Employment status is an emotive question: Found the question upsetting

• Defensive about never working

• Almost all at home on disability benefit

• Felt employers wouldn’t give them a job

• Recall of food was also challenging

• Most recalled evening meal but there was confusion over earlier meals

• Reluctance to admit to snacks or drinks during the day

• Asked if what they ate the previous day was usual or unusual – no difficulties answering this

• Found ‘more or less’ very difficult as the food eaten might be different

EQ-5D

• This was perceived by the researcher to be the most difficult part of the questionnaire

• Respondents had a lot of difficulty with the terminology

• In particular the change in terminology within the domains:

• From no problems and some problems to confined (to bed) or unable (to wash or dress)

• The change from self care (no problems) to some problems washing and dressing

• In self care often the supporter helps washing and dressing and as such no problems

• Didn’t understand what ‘performing’ meant

• In the pain/discomfort and anxiety/depression domains found the terms moderate and extreme baffling

• In the anxiety/depression domain didn’t understand the term anxious

Discussion

• Four primary impressions from this preliminary data: acquiescence, time, terminology, control

• Previous research that highlights acquiescence – telling the person the person what they want to hear might be at play here - particularly within the food section

• There appears to be a real challenge in the conception of time apparent in the 4 week and previous day recall

• The terminology – especially in the EQ-5D was a clear problem. Input in the phrasing of the resource use questions was received from the advisory group and more widely from the third sector but the EQ-5D is validated and can’t be changed

• The level of control over activities of daily living is also clear in everything from transport, to household chores, to cooking (and therefore to an extent food intake?)

• If we include only those with supporters do we exclude those with mild/very mild LDs?

What now?

• These are preliminary results we are still collecting data

• Immediately after each interview the researcher complete a questionnaire that records level of difficulty answering each question, whether the supporter helped, and free text with their perceptions – this should provide a rich dataset

• Overall…

• Unlikely GPs will provide robust data on community health use but we will explore better targeting who completes the form

• The amount of free text rather than coded items. Need to explore how Systmone address this

• Challenges in acquiescence, time, terminology, control

• More input from supporter? – but only includes those with supporters? And assumes the supporter has that knowledge (formal/informal/advocate)

• How to measure HRQoL?

Thanks!