Health

Children’s Palliative Outcome Scale

Person-centred care is a core health value of modern health care. The overarching aim of the Children's Palliative Care Outcome Scale (C-POS) is to develop and validate a person-centred outcome measure (PCOM) for children, young people (CYP) and their families affected by life-limiting and life-threatening conditions (LLLTC). A recent systematic review has highlighted that no measures suitable for use in this population currently exist. In addition, the development of such a measure has been highlighted as an international research priority. This novel study will draw together a unique multidisciplinary collaboration and seek to engage children within the research process, rather than relying on proxy data. Currently, children and young people are often neglected in the research process and this study hopes to change this.

The C-POS study addresses a current gap in both methods and evidence: repeated reviews and policies have called for scientific advancement to develop, validate and implement PCOMs for CYP and their families facing LLLTC. Currently, no valid tool exists, due to the complexities of self-report among children who often have profound communication difficulties, as well as sensitivities around the subject matter, and lack of evidence on their symptoms and concerns. Therefore, this population has so far been neglected in terms of research activity and evidence.

Aims

C-POS aims to develop, validate, and implement a person- and family-centred outcome measure that can be used by CYP and their families affected by LLLTC. The study will identify CYP, family, professional and commissioner priority concerns and outcomes in paediatric palliative care, which will be used to inform item generation and initial measure development. Through cognitive interviewing initial C-POS versions will be revised. The validation process further aims to test measures for comprehensibility, acceptability, feasibility, reliability, validity, and responsiveness. Lastly, the project aims to implement the C-POS in paediatric palliative care practice, and develop guidance for both wider implementation and adapt existing POS translation protocol to be suitable for the C-POS.

Methods

C-POS uses a mixed-methods approach, including methods such as semi-structured interviews, cognitive interviewing, a Delphi survey, and self-report surveys. C-POS is an ambitious study that, for the first time, will enable the measurement of person-centred outcomes of paediatric palliative care. This will be a turning point in the scientific study of a hitherto neglected group.

Our steering group is vital in directing our progress and consists of an expert group of researchers, clinicians, medical ethicists and patient and public involvement members. In addition to the two core patient and public members, we have a vital partnership with ‘Generation R’, the Great Ormond Street Young People’s Advisory Group, who have supported us in all phases of C-POS to date.

Impact

The C-POS will be the first validated children and family centred outcome measure for use in paediatric palliative care. The study brings together families, researchers, health and social care professionals and champions children’s voices in the research, rather than relying on proxy data. The tool will be implemented into routine clinical practice which if used regularly to measure and document change in patient’s status, can inform and improve care for children and young people with life-limiting and life-threatening illnesses.

Once implemented in the UK, the existing online Palliative Care Outcome Scale (POS) translation protocol and implementation guidance will be adapted for wider European implementation of the novel C-POS.

Project team

Myra Bluebond-Langner

Research Project Investigator

Debbie Braybrook

Research Associate

Katherine Bristowe

Herbert Dunhill Lecturer

Lucy Coombes

PhD Candidate

Clare Ellis-Smith

Lecturer in Palliative Care

Lorna Fraser

Research Project Investigator

Daney Harðardóttir

Research Assistant

Richard Harding

Vice Dean (International) and Director and Global Lead, Cicely Saunders Institute of Palliative Care, Policy & Rehabilitation

Irene Higginson

Executive Dean, Florence Nightingale Faculty of Nursing, Midwifery & Palliative Care

Fliss Murtagh

Visiting Professor

Anna Roach

Research Project Investigator

Hannah Scott

Research Assistant

Effective start/end date: 1/09/2018 - 28/02/2023

Phase 1 – Development

Identify CYP, their families, professional and commissioner priorities for outcomes of care and measure completion methods (qualitative interviews).
A systematic review on optimal recall period, response format and administration mode in CYP measure completion.
Delphi survey to establish priorities for items to be included in C-POS.
Generate initial C-POS version(s)

The clinical sites and local Principal Investigators who supported with Phase 1 qualitative interviews are Royal Marsden Hospital (Dr AK Anderson), King’s College Hospital (Dr Rachel Burman and Dr Steve Marshall), Great Ormond Street Hospital for Children (Dr Finella Craig), Martin House Hospice (Dr Michelle Hills), Leeds NHS Trust (Dr Sue Picton), Evelina London Children's Hospital (Dr Jo Laddie), Northern Ireland Children’s Hospice (Dr Deborah Burns and Julie Hall-Carmichael), East Anglia's Children's Hospices (Dr Linda Maynard), Addenbrookes Hospital (Dr Pat Sartori).

Phase 2 – Validation

Cognitive interviews to determine the extent to which CYP can participate, and their interpretation and completion of the proposed items.
Refine C-POS and finalise ready for subsequent reliability and validity testing.
Determine test-retest reliability, internal consistency, construct validity and responsiveness.

The clinical sites and local Principal Investigators who are supporting with Phase 2 cognitive interviews are Royal Marsden Hospital (Dr AK Anderson), Martin House Hospice (Dr Michelle Hills and Dr Debbie Box), Leeds NHS Trust (Dr Sue Picton), Evelina London Children's Hospital (Dr Jo Laddie), Northern Ireland Children’s Hospice (Julie Hall-Carmichael and Andrina McCormack), East Anglia's Children's Hospices (Dr Linda Maynard).

Phase 3 – Implementation

Develop implementation guidance for routine practice & hold launch workshops for children, families and clinicians
Adapt existing online POS translation protocol and implementation guidance for wider European implementation of the novel C-POS.

Publications

Coombes L, Wiseman T, Lucas G, Sangha A and Murtagh F (2016). Health-related quality-of-life outcome measures in paediatric palliative care: A systematic review of psychometric properties and feasibility of use. Palliative Medicine, 30 (10); 935-949. https://journals.sagepub.com/doi/full/10.1177/0269216316649155

Harding R, Chambers L and Bluebond-Langner (2019) Advancing the science of outcome measurement in paediatric palliative care. International Journal of Palliative Nursing, 25 (2); 72-79. https://doi.org/10.12968/ijpn.2019.25.2.72

Namisango, E., Bristowe, K., Allsop, M. J., Murtagh, F. E., Abas, M., Higginson, I. J., ... & Harding, R. (2019). Symptoms and concerns among children and young people with life-limiting and life-threatening conditions: A systematic review highlighting meaningful health outcomes. The Patient-Patient-Centered Outcomes Research, 1-41 https://link.springer.com/article/10.1007/s40271-018-0333-5

Namisango, E., Bristowe. K., Murtagh, F., Downing, J., Powell, R., Abas, M., ... & Harding, R. (2020). Towards person-centred quality care for children with life-limiting and life-threatening illness: Self-reported symptoms, concerns and priority outcomes from a multi-country qualitative study. Palliative Medicine, 34 (3); 319–335. https://journals.sagepub.com/doi/abs/10.1177/0269216319900137

Coombes, L., Roach, A., Bristowe, K., Ellis-Smith, C., Bluebond-Langner, M., Downing, J., Braybrook, D., and Harding, R. (2020). Priority outcomes for children and young people with life-limiting conditions: an in-depth qualitative interview study. Abstracts from the 11th EAPC World Research Congress Online, 7th – 9th October 2020. Palliative Medicine, 34(1_suppl), pp.165. https://doi.org/10.1177/0269216320958098

Coombes, L., Bristowe, K., Ellis-Smith, C., Downing, J., Bluebond-Langner, M., Fraser, L., Chambers, L., and Harding, R. (2020). Maximising participation in patient reported outcome measures (PROMs) for children and young people: a systematic review of optimal recall period, response format and administration mode. Abstracts from the 11th EAPC World Research Congress Online, 7th – 9th October 2020. Palliative Medicine, 34(1_suppl), pp.172 https://doi.org/10.1177/0269216320958098

Roach, A., Braybrook, D. and Marshall, S. (2021) ‘Reflective insights from developing a palliative care children and young people’s advisory group’, Palliative Medicine, 35(3), pp. 621–624. https://doi.org/10.1177/0269216320976035

Coombes, L., Bristowe, K., Ellis-Smith, C., Aworinde, J., Fraser, L. K., … & Harding, R. (2021). Enhancing validity, reliability and participation in self-reported health outcome measurement for children and young people: a systematic review of recall period, response scale format, and administration modality. Quality of Life Research. https://doi.org/10.1007/s11136-021-02814-4

Conferences

10th APPM Paediatric Palliative Care Study Day and Research Day. London, November 2019:

Lucy Coombes, ‘The Children’s Palliative Outcome Scale’

Palliative care Outcome Scale (POS) Workshop, February 2020 ‘Outcome Measurement in Palliative and End of Life Care’. Cicely Saunders Institute, King’s College London:

Debbie Braybrook, ‘Development of the Children’s Palliative Care Outcome Scale (C-POS): What matters to families facing serious illness?’

11th EAPC World Research Congress Online, October 2020:

Lucy Coombes, ‘The Children’s Palliative Outcome Scale (C-POS) Study - EU research update’
Lucy Coombes, ‘Maximising participation in patient-reported outcome measures (PROMs) for children and young people: a systematic review of optimal recall period, response format and administration mode’ (Poster)
Lucy Coombes, ‘Priority outcomes for children and young people with life-limiting conditions: An in-depth qualitative interview study’ (Poster)

Palliative care Outcome Scale (POS) Workshop, March 2021:

Lucy Coombes and Debbie Braybrook, ‘The Children’s Palliative Outcome Scale (C-POS) Study’

ECHO Northern Ireland Paediatric Palliative Care Network, March 2021:

Lucy Coombes, ‘Patient-Centred Outcomes and the C-POS Study’

5th UK National Patient Reported Outcome Measures (PROMs) Research Conference, June 2021:

Debbie Braybrook ‘"if they knew what I thought was important … that would help a lot more”: Methods for involving children and young people with life-limiting and life-threatening conditions in the development of a paediatric palliative care outcome measure’
Lucy Coombes ‘Enhancing validity, reliability and participation in self-reported health outcome measurement for children and young people: a systematic review and recommendations for recall period, response scale format, and administration modality’