1. The role of feasibility and scoping studies in the development of the programme grant: Does specialist teenage and young adult cancer care improve outcome? Susie Pearce Health Services Researcher for Young People with Cancer Faith Gibson, Rachel Taylor, Lorna Fern, Catherine O’ Hara, Jeremy Whelan

2. Overview Towards a programme of research: Background Methodological underpinnings Piloting and feasibility studies The NIHR programme grant Concluding thoughts

3. Background Very little known about experiences of TYA with cancer 2000 new cases in the UK each year (13-24 year olds) Cancer accounts for 11% of deaths in this age group Poorer outcomes compared to other age groups Place of care, poor involvement in clinical trials, delays in diagnosis, unfavourable tumour biology, all thought to contribute to these outcomes.

4. Range of malignancies and treatment Cancer superimposed the normal developmental and psychosocial needs of TYA Importance of disease and age appropriate settings of care Improving Outcomes Guidance (IOG) for Children and Young people’s Cancer (NICE 2005) 13 Principal treatment centres in England. However, as yet limited evidence for the development of specialist services National and international need for a substantial body of work to identify the benefit

5. Aims To develop a programme of research to determine whether specialist care in England, for young people aged 13-24 years is associated with improved outcomes during and after treatment.

6. Challenges How? What study design? –Ideally randomised controlled trial, BUT Services are already in place Unethical to randomise to specialist care vs. not Variation in services across country What sample? –Too much variability and small numbers to be single centre –National cohort, BUT How do you identify ALL young people? How do you recruit ALL young people? What should the outcomes be? What exactly is TYA cancer care?

7. Methodological framework Medical research council’s framework for developing and evaluating complex interventions and theories of complex health care evaluation Specialist TYA cancer care - a complex health care intervention Evaluation requires a non standard, multifaceted approach

8. Complex intervention Several interacting components Flexibility in the tailoring of delivery is permitted There are a number of outcomes There are a number of groups and organisational levels targeted by the complex intervention

9. MRC framework for evaluating complex interventions

10. Essence of Care: Phase I

11. Key Questions for this first phase of work What is the current service provision for TYA with cancer in England and what are the anticipated changes in the next 5 years? What is the nature of specialist cancer care? What young people’s experiences of cancer care? What are the key characteristics of a specialist unit? What patient reported outcome measures are available and validated for this age range? How can we rapidly identify all new diagnoses of cancer in those aged 13-24? Can clinical data be obtained accurately and timely from existing NHS databases/ registries? What are young people’s preferences for a longitudinal survey? Is there a framework available to address the methodological challenges of conducting a nationwide survey

12. Study 1: Current & future policy underpinning TYA cancer Semi-structured interviews with four commissioners of TYA cancer services Documentary analysis of implementation plans from all SHA’s –Progress reports to the National cancet Action team from Sept ‘08 to Nov ‘09

13. What this informed… Aspects of study design: –Multi-centre, national –What TYA cancer care will look like in 5 years time. The reality versus the policy. –Emphasized importance of a work stream in main programme of research to more fully explore the varied contexts of TYA specialist care

14. Study 2: Nature of TYA cancer care Non-participant observation –Single principle treatment centre and shared care units –Observation over 2 weeks of all key events –All areas providing TYA cancer care Semi-structured interviews –Three health care professionals –Three young people

15. What this informed… An in depth understanding of the complex nature of TYA cancer care and processes of multidisciplinary working within one Principal Treatment Centre The methodology, including the development of an observation schedule for a multiple case study of half of the principal treatment centres and networks of care in England. These case studies will use non-participant observation and semi structured interviews and document analysis Guided the sampling strategy for a multiple case study

16. Studies 3 - 5 Study 3: Towards a definition of TYA cancer care unit Study 4: Identifying the professional competencies for TYA cancer care Study 5: Young people’s experiences of cancer care  Workshops with young people ( n=11) and health professionals ( n=22)

17. What studies 3-5 informed… Importance of young people as co-researchers –Involve young people in study design –Involve young people in study conduct and analysis Highlighted the pattern of communication in TYA healthcare Initial survey for an international Delphi survey in professional competency in TYA cancer care Quality of life as important as survival –Implication for primary outcome

18. Study 6: Self-reported outcome after treatment for cancer Literature review –Search terms related to outcome, i.e., quality of life –Cochrane + 4 databases –Young people aged 13 – 45 years with cancer (US definition of ‘young adult’) –Any study design –Study quality assessed with Cesario/CONSORT/STROBE guidelines

19. What this informed… Identify significant non-medical outcomes Understand young people’s experiences of cancer [care] –Underpin content of the Essence of Care, ‘Brightlight’ Survey

20. Study 7: Outcome measures validated for young people aged 13 – 24 years Literature review –Search terms related to outcome, i.e., quality of life –Cochrane + 4 databases –Participants <16 years and ≥19 years –Only quantitative designs –Instrument quality assessed with McMaster Outcome Measure Rating Form

21. What this informed… Identify outcome measures that can be used in the cohort study. Develop a toolkit of outcome measures used in TYA, with a validity & reliability profile, for use in future research

22. Study 8: Young people’s preference for participation in a longitudinal study Recruitment in two principal treatment centres Six young people aged 17 – 21 years Complete outcome measures –PedsQL, EORTC, INPATSAT, HUI2, EQ5D –Feasibility, acceptability Semi-structured interviews

23. What this informed… Primary and secondary outcome measures Study design issues particularly around acceptable methods of data collection in a cohort study Importance of involving young people in study design

24. Study 8: Evaluation of NHS datasets Aims –Identify all young people within 90 days of diagnosis –To provide clinical data Review national datasets Criteria: –Minimum data: NHS number, name, gender, age, address, diagnosis & place of care Feasibility study of processes

25. Results: identification of a dataset

26. What this informed… Cancer Waiting Time dataset had overall completeness of 99% –Young people can be identified within 90 days Only suspected cancer therefore needs confirmation Additional data in other datasets –Clinical data available without relying on medical notes Quality of data is not confirmed Missing data Lack of reliable death data Further study in programme grant to validate the national data sets through the cohort study

27. Study 10: Framework for the Essence of Care study Challenges for Phase II –Can we identify all newly diagnosed young people in England in 2012? –How can they be recruited?????? –How can data be collected?????? Yes

28. Solutions NIHR funded project = access to NCRN nurses –Will they recruit young people? Search existing national cohort studies (e.g. Millennium Cohort Study) How do they data collect? –Commercial research companies –Is this economically feasible? Yes

29. Developing the programme grant Identification of co-applicant team Feeding pilot studies into programme grant development Regular co applicant team meetings through the feasibility phase Ongoing refinement and stages of submission Successful NIHR Applied health programme grant £1999,957. ‘Do Specialist Cancer Services for TYA add value’ Cohort to begin recruitment 1/7/12.

30. The 2012 TYA Cancer Cohort Study: developing team working UCLH: Dr Jeremy Whelan (CI) Susie Pearce Martin Lerner UCL: Dr Julie Barber Professor Steve Morris Professor Rosalind Raine Dr Rachel Taylor University of Leeds: Dr Richard Feltbower St James’ University Hospital Dr Dan Stark Cancer Research UK Dr Lorna Fern GOSH/LSBU Professor Faith Gibson NCAT Louise Hooker NWCIS Dr Tony Moran Dr Catherine O’Hara NCRI CSG TYA CCG: Hannah Millington

31. Workstream 1: the description of specialist TYA cancer care Explore the culture of care through non- participant observation, interviews and documents analysis Identify the specialist competencies and added value of specialist health professionals through a Delphi survey Validate a bespoke scale to categorise 3 levels of TYA care (TYA Cancer Specialism Scale)

32. Workstream 2: cohort study of all TYA diagnosed during an 18 month period Accrual within 3 months of diagnosis and follow-up will continue for 3 years Relate the level of specialist care received to: –Patient reported outcome. QL, satisfaction with care. –Clinical processes and outcomes –Experience of cancer care. –Achieving social and educational milestones –Geographic and socio-demographic inequalities

33. Workstream 3: health economics Compare costs to the NHS and personal social services between TYA receiving different levels of specialist care Estimate the cost to TYA and families of specialist care Calculate the cost effectiveness of different levels of TYA care

34. Workstream 4: intervention development & service evaluation Workshops with clinicians and policy makers Aiming to throughout the programme of research to feed findings into practice, service development, policy making and the development of new areas of research

35. Summary Complex evaluation of health care need complex study designs MRC framework for evaluating complex interventions –Importance of feasibility & pilot studies –Importance of understanding processes and context Importance of these stages for successful programme development grant

36. Conclusions Challenge of the development of feasibility studies in synchrony with the development of the programme submission Feasibility and development work ongoing Importance of the development of team working (and the most appropriate team) through this process. Role of nurses in understanding the complexities of health care; leading practice and service delivery. Nurses skills as researchers are thus crucial to the facilitation, development, management and leadership in programmes of research evaluating service delivery and patient experience and outcome.