Challenges for economic evaluation when doing research with people with learning disabilities - Claire Hulme, Professor of Health Economics, University of Leeds

1. 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 Leeds
c.t.hulme@leeds.ac.uk
0113 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

2. Introduction
• Research with hard to reach vulnerable
groups presents myriad challenges
• This is particularly pertinent for people
with learning difficulties
• People with learning difficulties were
not involved in research that was about
them even as interviewees until the
1980s
• Methodological difficulties include a
tendency to acquiesce because so
much of their lives are controlled by
others
• How do we know if the person really
understands what we are asking them?
• Economic evaluations rely on health
care or health insurance records or
forms such as the CSRI completed by
the service provider rather than by
people with learning disabilities
themselves
• This presentation reports on the
development and testing of data
collection methods for use in an
economic evaluation within a RCT for a
manualised supported diabetes self-
management programme for people
with mild/moderate learning
disabilities: the OK Diabetes study

3. • Background: Learning difficulties and diabetes
• A little about the research and overall study ‘challenges’
• The Challenges for economic evaluation so far
• Discussion

4. Background: Diabetes
• Prevalence of diabetes rising nationally
• Leeds (Pop ~725k) there are >27000 on
the QOF diabetes register and in
Bradford (Pop ~520k) there are 26500
on the QOF diabetes register
• This gives population prevalence that
are typical of published figures from
elsewhere of 4% and 5%
• There are 3.2 million people diagnosed
with diabetes in the UK
• An estimated 630,000 people have the
condition, but don’t know it
• Diabetes develops when glucose can’t
enter the body’s cells to be used as
fuel. This happens when either:
• There is no insulin to unlock the cells
(Type 1)
• There is not enough insulin or the
insulin is there but not working
properly (Type 2)

5. Background: Learning disabilities
• People with a learning disability have
poorer health
• More likely to have additional health
problems e.g. weight, mental health
and respiration
• Have poorer health outcomes
• CIPOLD (Confidential Inquiry into
Premature deaths Of people with
Learning Disabilities)
• Men with learning disabilities die on
average 13 years earlier than those
without ; women on average 20 years
earlier; 22% under age of 50 when they
died
• Most common underlying problem was
heart /circulatory disorders and cancer
• Most common immediate problem was
a respiratory
• More likely to have multiple long term
conditions
• Delays in diagnosis and treatment
• Lack of communication between
professionals; co-ordination of care
across different disease pathways and
service providers

6. Learning disabilities and diabetes
• Type 2 diabetes disproportionately
affects people with a learning disability
• Studies of GP data (NHS MiQuest
system) 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 Ambulatory
Care Sensitive Conditions (ACSCs)
indicates potential weaknesses in
primary care that need addressing
• ACSCs are chronic conditions for which
it is possible to prevent acute
exacerbations and reduce the need for
hospital admission through active
management. Examples include
congestive heart failure, diabetes,
asthma, angina, epilepsy and
hypertension.

7. 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

8. 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

9. 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

10. 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,

11. Case definition
• Not a diagnostic checklist but a guide to
help identify possible participants
• Activities
• Can/do they:
• Read, write, manage money, look after
their personal care, tell the time, cook,
have difficulty in communicating with
other 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?

12. 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.)

13. 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

14. Recruitment Beyond Healthcare
Local
Authority
LD
Charities
Further
Education
Advocacy 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

15. 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

16. 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

17. 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

18. 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

19. 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

20. 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

21. 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

22. 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?

23. 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?

24. Thanks!