Paediatric Hip Conditions: Psychosocial and economic dimensions of impact on quality of life

Lead Research Organisation: Cardiff University
Department Name: Healthcare Studies

Abstract

The understanding, diagnosis, and treatment of PHC is rapidly evolving. A variety of novel hip preserving surgical techniques are now utilised worldwide. These surgeries are now viewed as desirable alternatives compared with persistent hip pain, joint degeneration and hip replacement in a young population. However this is still an underserved patient group where the full social, economic and QoL impact of living with these conditions over the life course has not been explored. We know treatment and care are not always optimal, referral to specialised care can be slow and there is limited research available comparing outcomes for different treatments (Nunley et al 2011).
This is an emerging research area and a focus of current work of the applicants (TG, AFL, JH, RK). Our core group now includes ST and EE. We aim to develop a research agenda and research network that continues beyond the seminar series, bringing together researchers from diverse academic and clinical backgrounds and countries. We have begun to generate a wider group of multi-disciplinary academics, patients and clinicians to create an international research network. JH has international links with the invited speakers, Michael Millis and Prof Ganz, TG has links with the Nicola Rumsey specialising in body image , we will also draw on the expertise of others working in similar fields David Flynn (QoL scoliosis), and AFL with Profs Sugden and Christensen (education). In addition we build on our existing strong patient link for speakers.
The seminar series aims to facilitate engagement between academic researchers, clinicians and persons with a PHC to discuss and debate issues concerning diagnosis, treatment decision-making and post-surgical care and to develop a research agenda to promote evidence-based practice and measurement of what matters to persons with a PHC and their family. It has six specific objectives;
1.Develop a fuller understanding of the QoL impact of PHCs across the life-course, centred on the experiences and perspectives of persons with these conditions and their significant others.
2.Enable clinicians and academics currently engaged in the care or research surrounding PHC to share perspectives and ideas with persons with these conditions and learn from research in other paediatric conditions with long-lasting impact,
3.Develop ways to raise awareness of the patient and family perspective across the treatment and caring professions
4.Share and exchange multi-disciplinary knowledge around the treatment and care of PHCs both within and between parts of the UK, European and the USA
5.Explore economic-related, educational and body image dimensions of living with a PHC condition over time
6.Develop supportive strategies to embed successfully the assessment of patient concerns and impact of the conditions into routine clinical practice across the life-course.
Six, one-day interactive seminars, with informative pre-circulated presentations, will be undertaken, concluding with action points:
i.Uncovering the Expert Patient/Lay and Familial Perspective
ii.Measuring the Concerns, Consequences and QoL Impact of PHC:
iii.International Healthcare Diagnosis and Treatment Approaches: Clinical Perspectives: Monitoring and Assessing Outcomes of PHC within Routine Practice
iv.Childhood and Young Persons Impact on Schooling and Social Participation
v.Communicating, Awareness and Support for Persons with PHC

A key benefit will be development of an inter-disciplinary research agenda in this emerging field which moves beyond the surgical and clinical perspective to embrace the person with a PHC and their family. A dialogue during and after the seminar series will be supported by a website providing seminar papers, discussion forums and links to proposed research. We will link this to existing hip, charitable and patient forums. The creation of a strong research network and action plans ensures its sustainability and future collaboration.

Planned Impact

The seminar series brings together academics (health sciences, health services research, health economics and education), persons with a PHC and informal carers and clinicians (GP, specialist hip surgeons, rehabilitation team members) to develop a collaborative inter-disciplinary research agenda. The explicit intention is to enable a shift from a multi- to an inter-disciplinary approach, with a strong patient dimension. A core set of participants will be invited to attend all the seminars; additional persons will be invited to individual seminars (medical students for Seminars 3 & 4; persons from education and disability to Seminar 5).

The first two seminars (1, 2) focus on sharing expert patient/lay and familial perspectives (1) of health and well-being concerns, consequences and QoL impact surrounding living with a PHC across the life-course, and economic-related dimensions and ways these can be explored (2). (3) explores clinical practice in different countries with a view to share best practice. (4) builds on this with a focus on outcomes monitoring and its implementation in practice and the potential for information sharing across healthcare agencies. (5) focuses on the impact of PHCs on childhood and young persons. (6) addresses the key area of communicating, awareness-raising and support for persons with a PHC.

Potential beneficiaries include:
- Patients and family members/informal carers
- Practitioners: NHS and private, across levels (GP, general and specialist hip surgeons, hip rehabilitation team) and teachers
- Academic researchers: clinical effectiveness researchers; trialists; health and social science, economics and educational researchers in the UK and beyond
- Policy makers: NHICE, clinical commissioning groups from future research studies

The work will contribute to the nation's health, wealth and culture by:
- Addressing an ESRC strategic challenge, health and well-being, for PHCs across the life-course, the consequences of ill-health on social and economic participation and developing models of good practice, providing a foundation for enhanced evidence-based practice and clinical outcomes studies
- Development of an inter-disciplinary research agenda promoting early diagnosis, prevention of early disability and patient-centred assessment tools of the health and well-being and QoL impact of PHC.
- Developing a cross-Universities, sectors and patient collaborative, inter-disciplinary research thematic network to be involved in research bids and advising on emerging research and practice implications.
- Providing a platform for collaborative international EU Horizon 2020 research bids, addressed at the core societal challenge of developing and preserving good health, centred on issues of importance to patients
- Enhancing insight into health and well-being, economic-related and QoL impact areas from the patient perspective to enable development of patient-focused assessment tools
- Promoting ongoing monitoring and assessment in hip clinical practice of impact on patients across service levels and in evaluation studies, contributing to the development of models of good treatment and care practice.
- Identifying potential supportive curative and preventive self-care strategies and support for treatment decision making, tailored to patient concerns. Disseminating to: i clinicians - via GP and specialist interest groups, European Hip Society and International Dysplasia Institute websites and professional journals; ii medical, physio- and occupational therapy post-graduate/registration educationists for CPD courses; iii academic community, via peer-reviewed publications in health, social science and medical journals; iv patients - via project website, links on web-based patient forums and patient information leaflets (via GPs, charities and clinical teams); v social media utilising existing social network sites. All supported by development and maintenance of a project web-site.
 
Description 1. Established Significant New Knowledge, Identification of Research Needs:
• Established perspectives, experiences and impact on young persons of living with a Paediatric Hip Condition (PHC), importance of the child speaking for him/herself and need to:
(i) Use child-centred data collection approaches
(ii) Involve young patients in research studies.
• Using other young persons' chronic conditions to expand thinking into the impact of PHCs on their lives e.g. scoliosis.
• Responding to the current lack of appropriate patient-centred outcome measures (PROMS) by developing fit-for-purpose child-centred, validated PROMS for clinical research.
• Establishing the need for national neo-natal hip ultrasound screening, including robust training, as a major preventative strategy, notably for DDH, enhancing international research and practice (e.g. Coventry,UK; Austria) and building the RCT evidence base required by NHS (E), NHICE and CCGs
• Exploring cost implications of failure to screen, including litigation following misdiagnosis.
• .Recognising varied access to specialist hip centres, potentially restricting early recognition, treatment and management of patients with PHC.
• Need/value of step-by-step 'programme planning' research approach to build evidence, linked to translation of research to healthcare improvement.
• Establishing shared set of core methodological values..

2. Novel Research Methods Established
• Engaging with children and young people who have directed the purpose and methods of our research.
• Establishing new methodological approaches to assess health economic impact of PHCs for patient, family, health service as well as the financial and societal costs of misdiagnosis, using cost-utility studies.
• New PROM designed and being validated; methods developed for embedding PROM in routine clinical practice ensuring patient-centred and meaningful reported outcome measures.
• Methods of diagnostic optimisation by engaging with GPs and physiotherapists.

3. Important New Research Resources Identified
• Criticality of prioritising voices of child/young person and parents with PHCs and their contribution to identifying research questions and throughout the research process
• PHCs charities, particularly STEPS, DHH UK and the Perthes Association, involving them in our research, utilising their websites/patient stories and PHC information leaflets. Building on the Cardiff Hip-Pain website, Facebook page (https://www.facebook.com/HipPainCardiff/?ref=aymt_homepage_panele) which facilitates linking with patients and raising wider awareness of paediatric hip issues.
• RCT research on Perthes at Alder Hey Children's Hospital, their urgent need for child-centred measure development on the child's and families experience and impact of Perthes on their lives.
• Emerging research links with Harvard University, Boston, USA (http://www.childrenshospital.org/doctors/michael-millis)
• Development of project-specific websites (Universities of Cardiff and Leeds) with Cardiff Hip Pain website being information resource site for PHCs linking it to relevant charities and work with Alder-Hey.

4. Expansion of Research Networks and Collaborations
• Build our network to include European and other UK clinical specialists (surgeons, physiotherapists, health economists plus academics with research track record on children and young person's conditions)
• Linkage of research to Cardiff and Vale University Health Board (Wales)
• Linkage with USA research team (Mr Millis) MD Professor of Orthopaedic surgery. http://www.childrenshospital.org/doctors/michael-millis) and Prof Tschauner and his extensive research base (Austria)
• Develop current work with Alder-Hey and Perthes RCT, network members' involvement in developing child-friendly PROMs for their Children's Hip Disease (ORCHiD) study.
Exploitation Route The findings maybe used to educate health practitioners about paediatric hip conditions.
There is a need to raise general awareness of the conditions.
Exploring ways to embed outcome mesaures in routine practice
Understanding more about rehabilitation and how to take forward intervientions into non specialist areas.
Sectors Communities and Social Services/Policy,Education,Healthcare,Leisure Activities, including Sports, Recreation and Tourism

URL http://medhealth.leeds.ac.uk/info/638/seminars/1793/paediatric_hip_conditions_seminar_series
 
Description The finding and outcome of the seminar series have been distributed to patient user groups and health professionals working in paediatric hip conditions. Mention facebook site and links with charities etc.To enhance wider communication we have also established a presence on Twitter and an accessible Blog We have been using social media to publicise our work and findings. We have developed an outcome measure- the measure is currently being validated internationally through the IHDI. The work has helped to raise awareness of the socio-economic impact of DDH many users contact our group though social media, We have also raised awareness at conferences and Royal college of surgeons meetings
First Year Of Impact 2015
Sector Healthcare
Impact Types Societal,Policy & public services

 
Description raising awareness of peadiatric hip conditions to physios
Geographic Reach National 
Policy Influence Type Influenced training of practitioners or researchers
 
Description Saudi Ministry PhD studentships
Amount ر.س.‏ 450,000 (SAR)
Organisation Ministry of Health 
Sector Public
Country Rwanda
Start 10/2017 
End 10/2020
 
Description Saudi Ministry PhD studentships 2 studentships
Amount ر.س.‏ 450,000 (SAR)
Organisation Saudi Arabian Cultural Bureau 
Sector Public
Country Canada
Start 10/2017 
End 10/2020
 
Title Development of a quality of life measure 
Description Development of a tool that will specifically measure the quality of life of adults with hip dysplasia 
Type Of Material Improvements to research infrastructure 
Year Produced 2016 
Provided To Others? Yes  
Impact It is being validated in Birmingham they are using it in practice as part of the validation process . It is influencing decision making in clinical consultations. It is an indirect result of teh seminar series. The seminar has helped to publicise the work and engage clinicians in using the tool in the future. 
 
Title Outcome measure Psycho-social impact of hip dysplasia 
Description Measurement tool that can be used to measure the psychosocial impact of Paediatric hip conditions, in can be used in decision making and measuring the progression of the disease and the impact of any surgery or treatments. 
Type Of Material Physiological assessment or outcome measure 
Year Produced 2018 
Provided To Others? No  
Impact It will be implemented into practice as over the next year following final validation. 
 
Description Alder Hey Hospital 
Organisation Alder Hey Children's Hospital
Country United Kingdom 
Sector Hospitals 
PI Contribution Develop a child friendly outcome measure
Collaborator Contribution Collecting data at Alder from parents and children to develop the measure
Impact the development of a child friendly outcome measure for paediatric hip conditions
Start Year 2017
 
Description Alder Hey Hospital 
Organisation Alder Hey Children's Hospital
Country United Kingdom 
Sector Hospitals 
PI Contribution Develop a child friendly outcome measure
Collaborator Contribution Collecting data at Alder from parents and children to develop the measure
Impact the development of a child friendly outcome measure for paediatric hip conditions
Start Year 2017
 
Description Biomechanics partnership 
Organisation Cardiff University
Country United Kingdom 
Sector Academic/University 
PI Contribution We are developing projects around the Biomechanics of Hip Dysplasia and Perthes Disease. We are currently co-supervising 2 PhD students who are working to consider the basic science around movement deviations in Biomechanics.
Collaborator Contribution Joint PhD supervision of 2 students. Steps and DDHUK will support us in recruiting patients.
Impact This is very early in the collaboration but we anticipate one paper will be written this year a systematic review of the gait deviations in Perthes Disease.
Start Year 2017
 
Description Collaborating with charities DDH UK and Steps International 
Organisation STEPS UK
Country United Kingdom 
Sector Charity/Non Profit 
PI Contribution We are working together by engaging patients and carers through the charities to engage in the development of research proposals but also to recruit participants into future studies
Collaborator Contribution The partners are supporting our work by providing professional and clinical expertise, providing access to patients and carers who can influence and comment on research applications and also help us to develop future grant applications
Impact we have received an extension to out ESRC grant to develop research applications and to raise awareness of DDH both charities will play a pivotal role in the next 2 seminars.
Start Year 2016
 
Description Collecting data for PhD study looking at diagnostic indicators for DDG 
Form Of Engagement Activity A formal working group, expert panel or dialogue
Part Of Official Scheme? No
Geographic Reach National
Primary Audience Study participants or study members
Results and Impact Much interest in the research and the sample group are going to participate n further development of the tool in the future.
Year(s) Of Engagement Activity 2017
 
Description ESRC Seminar 
Form Of Engagement Activity Participation in an activity, workshop or similar
Part Of Official Scheme? No
Geographic Reach National
Primary Audience Professional Practitioners
Results and Impact Paediatric Hip Conditions:
Psychosocial and Economic Dimensions of Impact on Quality of Life
A Series of Six Seminars
Funded by the ESRC (Nov 2014-Oct 2016)

SEMINAR 1: Uncovering the Expert Patient and Familial Perspective
Introduction
The seminar, held at Cardiff University on 13th February 2015 (10am to 4pm), explored patient and familial perspectives on having and living with a paediatric hip condition. Its aim was: 'to develop a fuller understanding of the quality of life (QoL) impact of paediatric hip conditions across the life-course centred on the experiences and perspectives of persons with these conditions and their significant others.' The 40-50 participants ranged from academics with an interest in the topic area and/or highly relevant methodological expertise in the area, clinicians (in particular, physiotherapists, nurses and orthopaedic surgeons) and invited patients and members of their family. This short document draws attention to key points raised, implications and further research and practice-related issues arising.

The Morning (Chair: Professor Andrew Long, University of Leeds)

Following a welcome to participants from Professor Patricia Rice, Pro-Vice Chancellor, Cardiff University, and, from the Chair, a brief overview of the seminar series and programme for the day, the first presentation was given by Mr Phillip Thomas (Consultant Orthopaedic, Hip, Surgeon, Cardiff and Vale NHS Trust). His talk centred on the Aetiology, Diagnosis, Treatment and Patient Experiences of the three conditions (Developmental Dysplasia of the Hip, DDH; Perthes disease; and Slipped Upper Femoral Epiphyses, SUFE) and highlighted their impact on patients and their families. Four key themes emerged: (i) their difficulties in dealing with the diagnosis; (ii) common questions they asked; (iii) effects on the wider social context, especially schooling (absenteeism, social networking); and (iv) difficulties faced by parents, the child and young person in managing the condition.

The first keynote presentation was given by Dr Tina Gambling (Senior Lecturer & Health Psychologist, Healthcare Sciences, Cardiff University; and expert patient) and entitled Living with a Paediatric Hip Condition. Her talk, divided into three parts, focused mainly on hip dysplasia providing some insight into research findings on this condition, drawing on earlier work, led by herself, conducted with Mr John O'Hara, Professor Andrew Long and Liz Evans. Three key issues were highlighted: (i) issues surrounding long delays in obtaining appropriate specialist referral and definitive diagnosis; (ii) coping with decision-making over treatment options; (iii) coping with very invasive surgery at a young age and extensive and long process of rehabilitation; and (iv) effects on their education, work and social life. The second part of the keynote provided rich insight into patient perspectives with three young people, Verity, Melissa and Luke, telling their experiences and the impact of their PHC on their life. This was followed by three sets of parents with their child / young person focusing on the impact on the family and the child/teenager (Leanne and Ella, Deborah and Rickie, and Kyle and Lacey). Key emerging factors included: late diagnosis, being taken seriously by the GP and other clinicians; effects on the whole family and the stress incurred; lengthy times out of school/work; and psychological impact and damage.
The second keynote presentation was given by Mr John O'Hara (Consultant Orthopaedic, Hip, Surgeon, Royal Orthopaedic Hospital Birmingham). He explored the Pathology and History of Treatment of the three hip conditions and, in particular, highlighted the difficulties specialist centres face and the need for prompt diagnosis and treatment to deliver the best outcomes. The two keynotes were followed by a short 'questions and discussion' session between the speakers, the patients and parents, and the audience.
The Afternoon (Chair: Professor Monica Busse-Morris, Cardiff University)

The third keynote presentation was given by Dr Heidi Williamson, Senior Research Fellow & Health Psychologist, Centre for Appearance Research, University of the West of England, Bristol) and entitled Supporting Patients with Appearance-Related Issues and Concerns. Her talk centred on body image issues facing young adults with appearance-altering conditions. Focus lay on important psychological factors which predispose someone to body image issues and challenges faced by young people at school and in their social life (for example, staring, teasing, unsolicited questioning, feeling judged and feelings of being avoided) and effects on their psychological health and social confidence. She also provided insight into interventions that could be delivered by healthcare professionals to address these concerns, including supportive websites (for example, for Perthes), screening for psychological distress and offering supportive advice.

The seminar participants then divided into small discussion groups and were asked to share their perspectives on three issues: (i) young persons and family experiences and QoL, social and psychological impact of living with a hip problem and others' responses to the person with hip difficulties; (ii) clinical implications for patient-clinician interactions and clinical practice; and (iii) further research implications.


Key patient and family concerns related to:
• QoL, social and psychological effects (frustrations in the family; anxiety; fear; embarrassment; anger: how to explain/verbalise how much pain they were in; frustration at not being listened; having to stay at home, not going out or to school; others' and teachers' not understanding the condition; and a need for ongoing support;
• Difficulties with 'peers' with little or no understanding of pain or the conditions);
• Their need to be informed about treatment options;
• Psychological and social problems arising from misdiagnosis and late diagnosis;
• The need for greater primary and non-hip specialist awareness of PHCs and their impact on the patient;
• The need for better healthcare practitioner communication to reduce/avoid the patient feeling neglected by the need for repetition of their problems;
• Importance of clinicians' really 'listening' to the patient and family.

Potential clinical implication identified related to:
• The issue of communication, including X-rays and results of tests (clinician to clinician including onto rehabilitation);
• Education and training of healthcare staff in relation to PHCs; the criticality of raising awareness in primary care and non-hip specialist consultants about the conditions and need for earlier specialist diagnosis;
• Implications of late diagnosis in relation to treatment options and recovery times;
• How best to support patients in patient-treatment decision-making and to develop their own coping strategies.

Research implications included:
• The potential of taking a longitudinal perspective (following patients and their families from diagnosis through their healthcare journey);
• Mapping and understanding the patients' family support network, leading onto to how to support this social network; research over the ill-health trajectory in order to gain a multi-dimensional understanding of the QoL psychological and social impact of the conditions;
• Exploration of cost-effectiveness of diagnosis and treatment options and outcomes.

All of these implications will be re-addressed in subsequent seminars in the series.
Year(s) Of Engagement Activity 2015
 
Description ESRC Seminar 2 
Form Of Engagement Activity Participation in an activity, workshop or similar
Part Of Official Scheme? No
Geographic Reach National
Primary Audience Professional Practitioners
Results and Impact Paediatric Hip Conditions:
Psychosocial and Economic Dimensions of Impact on Quality of Life
A Series of Six Seminars
Funded by the ESRC (Nov 2014-Oct 2016)
SEMINAR 2: Measuring the Concerns, Consequences and QoL Impact of PHC: How are Patient Concerns assessed in Research on PHCs and Other Paediatric Conditions?
Introduction
This seminar, held at Birmingham NHS Foundation Trust on 3rd July 2015 (Learning and Development Suite, 10am to 3:30pm), followed on from seminar 1 which explored patient and familial perspectives on having and living with a Paediatric Hip Condition (PHC). Seminar 2 focused on the measuring of patients' concerns and the consequences and quality of life (QoL) impact of PHCs and similar paediatric conditions.
The 12 seminar participants ranged from academics with an interest or relevant methodological expertise in the topic area, clinicians (particularly physiotherapists and orthopaedic surgeons) and a patient representative. This short document draws attention to key points raised, implications and further research and practice-related issues arising.

The Morning (Chair: Mr John O'Hara, Royal Orthopaedic Hospital Birmingham)
Dr Tina Gambling and Mr John O'Hara welcomed the attendees and highlighted the importance of taking into account patients' and their families' perspectives when considering how to design tools to measure the impacts of living with a PHCs condition. Dr Gambling provided a brief introduction to the ESRC seminar series and an overview of the first seminar which collected rich information on how these conditions affected patients and their families. Mr O'Hara welcomed participants to Birmingham, outlining the long connection of the hospital to orthopaedics, providing an overview of hip resurfacing techniques and sharing a good practice illustration (a 21 year old patient with only a mild delay in diagnosis; after three years of surgery and recovery, now training for a half-marathon.)
The first keynote presentation was given by Dr Sandy Tubeuf (Health Economist, University of Leeds). She began by providing a short introduction to what health economics is (the application of economics to the topic of health and healthcare, with a view to guide choices in health care in context of scarce resources and to maximise the health benefit). She then moved on to consider how different methods could be used to assess costs and benefits (cost-benefit analysis, cost effectiveness and cost-utility analysis). She provided an overview of some examples relating to PHC, pointing out that at present there are no studies evaluating the cost utility of a health care intervention for PHC, despite this method being the preferred method for NHS decision making. Dr Tubeuf reflected on the difficulties behind developing these studies and posed two main questions: (i) can all the costs saved to the NHS be measured? (ii) can the impact of quality of life and ultimately QALYs be measured in the case of PHC? Her keynote in Seminar Three (22nd October, 2015) will explore these issues directly.
The second keynote presentation was given by Mr John O'Hara (Consultant Orthopaedic, Hip, Surgeon, Royal Orthopaedic Hospital Birmingham). He focused on the current status of treatment of young adult hip disease and the importance of early diagnosis for health and QoL outcomes. He estimated that, for every one young person with a PHC, there are up to ten people with less severe cases who are undiagnosed. Mr O'Hara then drew on research led by Dr Gambling, in which participants had waited an average of 8.4 years to receive the right diagnosis, with likely severe impacts on the patient's and family lives. Other research conducted by Mr O'Hara's and his team on a 30-year follow-up of a patient case series indicated that, if the operation is done below the age of 20, 89% of patients are happy with their quality of life 20 years later; if patients are over 25 years of age, only 76% are so and, if over 30 years of age only 67%. In conclusion, he pointed to the need to raise awareness of the condition in primary and secondary healthcare. In the subsequent short discussion, it was noted that a potential output from the seminar series, and linked research, would be the development of tools to promote awareness and assist early diagnosis and treatment.
The Afternoon Chair (Dr Tina Gambling)
The third keynote was given by Dr Josette Bettany-Saltikov, Chartered Physiotherapist and Senior Lecturer in Research Methods at the Institute of Health and Social Care in Teesside University. Her presentation, entitled Measuring the consequences, concerns and QOL impact on young people with idiopathic scoliosis (IS), demonstrated a number of common concerns of IS patients with those of young people with a PHC. She began with an introduction to IS (a condition characterised physical by curvature of the spine), which affects around 0.47-5.2% of the population but is 7 times more common for girls. IS is normally first detected by family and friends; like PHCs, there is no screening programme for IS in the UK and in consequence late diagnosis is common (in the UK, the condition is often not detected until children have a curvature of 40 or 50 degrees). Without treatment, the condition worsens and leads to the need for surgery, often having significant physical and psychological impact on the person's and family's life. Dr Bettany-Saltikov then presented findings from a number of studies she has conducted to evaluate the IS patient group and suggest common issues of concern between IS & PHCs in young people. A 2010 study highlighted that patients and their families need timely, pertinent and specific information. Her talk continued by considering aspects of monitoring progression and treatment effectiveness, including X-ray and associated problems with repeated X-rays at 3-6 monthly intervals increasing cancer risk. She pointed to alternative methods of spinal assessment and imagining and went on to explore Physiotherapy Scoliosis Specific Exercises (PSSE). These are individually adapted to the curve of a patient and personalised to a particular case.
The fourth and final keynote of the day was given by Dr Tina Gambling and Mrs Liz Evans (Cardiff University) on a paper jointly authored with Prof Andrew Long (University of Leeds), entitled Improving the patient experience and measuring outcomes. Dr Gambling began by re-emphasising the need to measure the quality of life (QoL) impact on patients of their hip condition and treatment, as well as addressing the diagnostic challenges of hip dysplasia. She provided an overview of current measures being used in clinical practice, none of which explored QoL. She then discussed a patient-centred questionnaire being developed by the research team, which is grounded in patient views and aims to explore the 'QoL Concerns, Impacts and Consequences' of PHCs. The measure is designed to be both useful to the patient and clinician and to aid patient-clinician treatment decision making and outcomes monitoring. The second part of the talk was delivered by Mrs Liz Evans and focused on the diagnosis of Adult Hip Dysplasia. There was a need for better understanding of what happens at the time of diagnosis. Her secondary analysis of 81 life stories from Dr Gambling and Prof Long's study identified a range of issues which may point to common signs that could indicate a hip problem. She then outlined her future research plan centred on developing an assessment tool for diagnosis and monitoring of changes for physiotherapists and other healthcare professionals (for example, radiologists).
Group Discussion
The group discussion explored some key issues which had arisen throughout the day's presentations and discussed implications for research and practice. The main issues identified were:
• A main problem is late diagnosis of PHCs.
o Educating physiotherapists might be more effective than trying to work with GPs. Physiotherapists have more time and see the patient more than once. Commonly, GPs refer the person to the physiotherapist, and then onto a consultant.
o Patient education: books and forums are excellent resources and provide links to the other people/specialists in the field which may help patients with their condition's trajectory.
o Physiotherapy studies/training: it was highlighted that undergraduate education of physiotherapists does not cover dysplasia; in contrast to podiatrists who receive much more training on these conditions.
• Potential to raise diagnosis rate and shorten time of diagnosis:
o For hip dysplasia, symptoms are common; this should help with developing an assessment tool/score. Final diagnosis is normally undertaken following an X-ray but this has to be done by an expert, otherwise it may not be diagnosed correctly.
o But many health professionals are not taught about the condition, so the real incidence rate is not being captured. It was mentioned that Birmingham has an average of 3 or 4 new patients per week, but many cases go undetected.
o There is a need to develop networks across different hospitals in the UK (e.g. core, periphery etc.), so that patients are referred early on to the right experts.
• Patient-centred measure and the empowering of patients with HIP conditions:
o Many of the professional participants thought that the way forward is to empower patients.
o Patients need a forum to which to send all their cases, experiences, etc., so a stronger evidence base is built. It was suggested that the seminar series website could be used to create such forum.
• Enhancing clinical practice:
o An important issue arises with patients of 30-40 years of age who have a hip problem, such as mild dysplasia, for whom surgery does not work. Based on the discussions about IS, it seemed important to explore whether physiotherapy could help. Research is needed here.
• Developing a pool of experts/networks:
o There was extensive discussion on the geographical coverage of specialist centres in the UK and the need to map them. Specialist centres included: Birmingham, London (UCL and Guys), Manchester, Newcastle, Southampton (only for children) and not Sunderland and Scotland, with none in Cambridge, Nottingham or Sheffield/Leeds and little cover in Wales.
o Young patients are commonly sent to centres in London and East Midlands.
Year(s) Of Engagement Activity 2015
 
Description ESRC Seminar 3 
Form Of Engagement Activity Participation in an activity, workshop or similar
Part Of Official Scheme? No
Geographic Reach National
Primary Audience Professional Practitioners
Results and Impact Paediatric Hip Conditions:
Psychosocial and Economic Dimensions of Impact on Quality of Life
A Series of Six Seminars
Funded by the ESRC (Nov 2014-Oct 2016)
SEMINAR 3: 'Monitoring and Assessing Outcomes' and 'Measuring the Health Economic Impact' of PHCs within Routine Clinical Practice.

Introduction
The seminar, held at Leeds University on 22rd October 2015 (Marks and Spencer Building Suite, 10am to 4.00pm), followed on from Seminar 2 (July 2015) which explored issues surrounding economic evaluation in general and for persons with a PHC and the concerns, consequences and QoL impact of the conditions on young people. Seminar 3 addressed two issues: monitoring and assessing outcomes within routine practice and measuring the health economic impact of PHCs.

The 10 seminar participants ranged from academics with an interest or relevant methodological expertise in the topic area and a patient representative. This short document draws attention to key points raised, implications and further research and practice-related issues arising. The morning was ably chaired by Jenny Hewison (Professor of HSR, Institute of Health Sciences, University of Leeds) and the afternoon by Dr Tina Gambling and Liz Evans (School of Health Sciences, Cardiff University).

Dr Tina Gambling welcomed the attendees and highlighted the importance of monitoring and assessing outcomes and measuring the health economic impact of PHCs within routine clinical practice, as opposed to research. Dr Gambling also provided a short overview of the ESRC seminar series and issues explored in the first and second seminars. These in particular had explored rich information on how PHCs affect young people and their families and begun to consider better ways to explore their consequences, patient concerns and impact of PHC.

The first keynote presentation was given jointly by Professor Andrew Long (School of Healthcare, University of Leeds) and Dr Joanne Greenhalgh (School of Sociology and Social Policy, University of Leeds). Their talk explored issues surrounding embedding outcome measures within routine practice. It focused on two areas: (i) what is the evidence on whether or not feedback to clinicians on patient-reported outcome measures (PROMs) improves patient care and, if so, in what ways; and (ii) how can a PROM be successfully introduced, adopted and used within patient-clinician interactions in decision-making?

Dr Greenhalgh considered briefly the history of PROMs, their complexity, the exponential increase in the number of outcome measures (OMs) being developed and the emerging interest and body of literature focused around their use in clinical practice at two levels: patient care; and as an aggregate performance measure of care quality. In essence, the current body of evidence suggests some impact on patient care and varied evidence on the impact on patient healthcare outcomes. Evidence from Cochrane reviews also highlighted a general lack of clarity in the field, especially about the appropriate goals of PROMs and the mechanisms by which these might be achieved. Joanne pointed to the long implementation chain and the need to explore this at each stage, and evidence on potential facilitating and inhibiting factors to take-up into practice. She then presented an informative logic model depicting the potential modes of influence of a PROM pre- during and post-clinical consultations/intervention. A potentially highly valuable approach to build on this would be to use realist evaluation methods, including a realist reviews; this approach focuses on explaining how and why an intervention does or does not work.

Professor Andrew Long then explored some theoretical literature, drawn from implementation science and performance review and the potential of particular research approaches to enable successful embedding of an innovation into routine clinical practice, He began by drawing on Lewis (2015) who argues that, rather than just asking 'do clinicians use OMs?' and 'how might they use them more effectively?', we need to revisit the core questions of 'why are we doing this?' and 'what is outcome measurement and performance management (PM) for?'. Doing so leads to a realisation of the significance of the location of power in organisations and leads to the search for answers to questions such as: 'why and when are PM measures and OMs developed and promoted?', 'why may their introduction be resisted?' and 'how might the measures become purposely distorted by persons in the organisation?' Attempts to embed an innovation, such as a new PROM, in routine practice need to pay heed to such questions.

Andrew then returned explicitly to explore possible approaches that may enhance the successful introduction, and sustained use, of a PROM. One particular valuable theory to draw on was normalisation process theory. This argues that practices become embedded in social contexts as a result of people working, individually and collectively, to implement them, paying heed to four generative mechanisms: coherence, cognitive participation, collective action and reflexive monitoring. Finally, he explored the potential research approaches to employ to enhance successful embedding. A core issue was the need to engage with all participants in the clinical setting, ensuring their ownership of the innovation, and then working with them, for example, using the approaches of collaborative action research and/or soft systems methodology. Successful embedding of a new outcome measure into routine practice would thus be enhanced if the clinicians and patients identify coherent arguments for adopting the measure, judge it to be of value and useful, and own and engage in the adoption process. This keynote was then followed by three linked 'case study' presentations.

The first from Dr Penny Wright (Leeds Institute of Cancer and Pathology, University of Leeds) explored practical issues and lessons learned from implemented a computer-based assessment scheme, eCAS, to collect co-morbidity data. She began by pointing out that routine measurement of PROMs linked with clinical data across the patient pathway is increasingly important for informing future care planning. She then explored issues surrounding thesurrounding the development and implementation of an innovative electronic 'PROMs from Cancer Survivors' (ePOCS) system. This was developed to collect PROMs data, all completed by patients online at specified post-diagnostic time-points, and integrate these with clinical and treatment data in cancer registries. Penny then drew out key lessons learned from the three clinic implementation of eCAS.

A number of key messages for successful implementation were noted. These included: space (the need to negotiate exclusive use of a room close to the major clinic activities with network access); engagement with all staff from the beginning and finding out how the system could fit in/be adapted to suit this clinic (all staff groups involved must 'buy in' to it, and one clinical staff member have overall responsibility for the implementation process); training & support (try to get the training organised to 'piggy-back' onto other meetings and ask established users to mentor colleagues); the need for the activity to be recognised in annual reviews. Finally, Penny recommended that implementation should first take place in a 'not too busy/complex clinic; identify and work with a potential early adopter; and, finally, use the up-and-running implementation site as an exemplar with champions from the clinic as advocates.

The second case study was given by Liz Evans (School of Health Sciences, Cardiff University) was entitled 'my opposition to patient-centred care'. She highlighted the diagnostic value of paying close attention to the symptoms and ill-health features reported by patients during clinical assessment of their painful hip. Extracts from patients demonstrated the wide-range and significant consequences that have occurred when clinicians failed to act on patient reports. In essence, patients were expressing their feelings that their comments were not taken seriously and, in consequence, their lives were 'on hold' due to delayed or mis-diagnosis. There is thus a real opportunity to use patient reported information to expand our understanding of PHCs enabling us to develop a clearer clinical picture of the conditions. She emphasised that physiotherapists are well placed and have the opportunity to gather such information. Liz then moved on to consider the conflicting aims of, and tension between, 'patient-centred care' and 'clinical pathways'. There was a risk that a focus on patient-centred care may lead to patients being somewhat detached from the activities of the multiple clinicians who seemingly are orbiting around them. Patient reports of having diagnostic assessments by a range of clinicians where the same physical tests were applied repeatedly but the diagnosis remained allusive went some way to confirming this. A case for the patient being part of the team, rather than its centre/focus, was recommended.

The third case study was given by Dr Tina Gambling (School of Health Sciences, Cardiff University) explored some issues surrounding the current validation and future implementation of a PHC-specific outcome measure in routine clinical care. She began by outlining reasons for developing a new measure and, in particular, the need to capture the broader impact of a PHC on the young person's QoL. Building on earlier qualitative research, the developed measure is both grounded in patient views, with the aim of exploring 'QoL Concerns, Impacts and Consequences' of PHCs, and designed to be both useful to the patient and clinician, in particular, in aiding patient-clinician treatment decision making and outcomes monitoring. The validation study is currently being conducted at the Royal Orthopaedic Hospital Birmingham, with the first step focusing on exploring patient and clinical utility of the measure, and ways that this can be enhanced. Initial findings highlighted the importance of both the collation and then discussion of patient-based views on the wider QoL aspects and impact of the PHC. However this needs to be balanced with what can be discussed in a busy clinic environment. On the one hand, patients would like their voices to be heard; on the other hand, the orthopaedic surgeons are primarily concerned with physical function and physiological features of the hip.

The second keynote was presented by Dr Sandy Tubeuf (Health Economist, University of Leeds), entitled 'assessing costs, effectiveness and cost-effectiveness of paediatric hip conditions: why we should and how we can: Part II.' She began by briefly recapping on her earlier keynote (Seminar 2) by drawing attention to the focus of health economics and the nature of economic evaluation. Her keynote then centred on exploring available evidence from the research literature on each of the three approaches to economic evaluation within PHCs: cost-benefit analysis (CBA); cost-effectiveness analysis (CEA); and cost-utility analysis (CUA). Illustrating CBA was the example of Ultrasound Neonatal Screening for DDH, where the costs included medical equipment, fees for ultrasound examinations, personnel and training, etc., and the benefits were measured in terms of treatment costs (surgical and non-surgical) for children with/without the ultrasound (late case). However, Sandy pointed out, CBA was not favoured in the NHS, as it is not easy always to measure benefits in monetary terms. Illustrating CEA was the example of a study on the proportion of infants and children with DDH who had surgical interventions. In CEA, benefits are measured in 'natural' units (e.g. here, number of late diagnosed DDH per annum, splintage rate, chances of having a non-arthritic hip at the age of 60). Again, such an approach is not favoured in the NHS as the approach only compares interventions technically but the outcome measure is context-specific and not standardised; in contrast, the NHS needs to make decisions and choices across a range of health care interventions and needs to measure benefits with a standardised outcome such as quality-adjusted life years (QALY).

Sandy's final set of illustrations centred on a range of studies using CUA, the approach favoured in the NHS; in particular, NICE will recommend a health care intervention to the NHS worth =£20,000/£30,000 per QALY. The National Institute for Health and Care Excellence (NICE) explicitly use a range between £20,000 and £30,000 per QALY to recommend health care interventions to the NHS. In other words, treatments costing less than £20,000 per QALY gained are considered cost-effective while treatments costing more than £30,000 per QALY are not cost-effective; between £20,000 and £30,000 per QALY gained, NICE judges various dimensions such as uncertainty, innovation and whether there are non-health outcomes or end of life considerations before its decision. CUA is useful for decision-makers to make health care decisions across a broad set of treatments for various health conditions and their ability to measure health gains with the same unique output, here QALY, ensure a fair and comparable decision-making.

Finally, Sandy then explored different perspectives on costs, and then of benefits, in the context of PHCs, illustrating these by exploring the multiple perspectives that had been used in economic evaluations of PHCs. She concluded by emphasising the lack of cost-utility analyses and high-quality economic evaluations in PHC. Moreover, few studies have collected data on costs to the family and none have done so comprehensively, raising the question of whether these costs could be weighed against the direct and indirect cost of health care in short/medium term. More broadly, there was a need to adopt a 'decision analysis modelling' approach, extrapolating the costs and the consequences using probabilities and assumptions based on health record as PHC has a long term impact on quality of life gain and costs to the health care system.

Group Discussion
The group discussions explored a range of key issues which had arisen throughout the day's presentations and discussed implications for research and practice. The main issues identified were:
• A main problem is the lack of integration of outcome measurement in the area of PHCs.
• Measures specific to PHC do not exist at present; measurement is primarily functional, using measures designed for a different population (e.g. older people with osteo-arthritis).
• Patients are treated in busy specialist clinics; this could make it potentially very difficult to incorporate and integrate PROMs into routine clinical practice. There was, however, great potential, building on Dr Wright's presentation, of on-line/electronic capture; this might be better suited to a busy orthopaedic clinic setting.
• There is a definitive need to understand more about the condition and patient concerns and its long-term impact of the clinical course and its impact on patients' everyday lives.
• Decision-making is difficult for late diagnosed patients; supporting material on QoL impacts on the patients or guides on 'patient-reported features to look for' would be helpful.
• Understanding more about the conditions may help diagnosis; a clearer clinical picture would be potentially helpful for primary care, with healthcare professionals thus being alerted to possible nuances of the conditions.
• Further information and insight is needed about the healthcare interactions that patients may experience prior to receiving a definitive diagnosis: for example, who do they visit, and for how many times? How many tests/investigations do they experience (e.g. X-rays, scans)? And are they routinely requesting medication? Collection of such routine data may provide some predictive information that we could then use to alert practitioners; moreover, it can provide valuable information for a better understanding of the patient and health provider costs incurred and be used in economic decision analysis modelling.
• The true incidence rate/diagnosis rate of PHCs is not being captured. For example, it was pointed out that, while the Birmingham orthopaedic clinic has an average of 3 to 4 new patients per week, many cases go undetected in primary care and are thus not be referred on to specialist centres for investigation, treatment and care.
• It is important to shorten the time to diagnosis, and thus reduce delayed diagnosis.
• There is a need to develop clinical networks across different hospitals in the UK, in part to ensure that patients are referred early on to the right experts/specialist centres.

The next seminar explores International Healthcare Diagnosis and Treatment Approaches: Clinical Perspectives: Sharing Perspectives on Clinical Practice and Developing Evidence Base. The seminar will be held at Cardiff University at a date to be arranged in March 2016.
Year(s) Of Engagement Activity 2015
 
Description ESRC Seminar 5 
Form Of Engagement Activity A formal working group, expert panel or dialogue
Part Of Official Scheme? No
Geographic Reach National
Primary Audience Professional Practitioners
Results and Impact v. Childhood and Young Persons Impact: exploring effects on schooling, personal development, sports and wider social participation and fears over early disability. The keynote speaker for this session would included an educationalist who could inform the group about interruptions to schooling due to ill health and the impact this has on children and young adults. We would also included co-applicant Mr John O'Hara and his Paediatric Hip colleagues from The Birmingham Children's Hospital. Also Linda Milnes who has reached extensively young people with chronic conditions
Year(s) Of Engagement Activity 2016
 
Description ESRC Seminar 6 
Form Of Engagement Activity A formal working group, expert panel or dialogue
Part Of Official Scheme? No
Geographic Reach National
Primary Audience Professional Practitioners
Results and Impact vi. Communicating, Awareness and Support for Persons with one of the PHC: exploring issue surrounding explaining about a PHC to others, clinical awareness raising, self-care options and treatment decision making, including web-based discussion forums and charities. For this seminar we would invited speakers from the charities STEPS and the Perthes Association. This was really useful as they have many forums for support we were able to gain insight into the issues patient and carers face. We linked patients charties and professionals the output of this will be to work together to develop information for patients on the issues that matter to then
Year(s) Of Engagement Activity 2016
 
Description Seminar 4 
Form Of Engagement Activity A formal working group, expert panel or dialogue
Part Of Official Scheme? No
Geographic Reach International
Primary Audience Professional Practitioners
Results and Impact Seminar 4 International Healthcare Diagnosis and Treatment Approaches: Clinical Perspectives: sharing perspectives on clinical practice and developing evidence base. For the keynote talks in this session we would invited our European, International and UK Specialists Hip surgeons. We raised awareness of the differing approaches to screening Universal versus risk based and explored different UK regional protocols.
Year(s) Of Engagement Activity 2016
 
Description Skype meeting with INternational Hip dysplasia Institute 
Form Of Engagement Activity A talk or presentation
Part Of Official Scheme? No
Geographic Reach International
Primary Audience Supporters
Results and Impact We discussed future international projects and the use and development of the outcome measure in other centres
Year(s) Of Engagement Activity 2017
 
Description Symbios HipPLAN 2018 meeting 
Form Of Engagement Activity A formal working group, expert panel or dialogue
Part Of Official Scheme? No
Geographic Reach International
Primary Audience Professional Practitioners
Results and Impact Meeting co-convened by : Symbios UK Ltd and The Cardiff and Vale Orthopaedic Centre (CAVOC) A specialist company that develops individualised hip implants. This was a workshop which civered many theme including: the most up to date surgical techniques, Childhood deformities of the hip and the challenges that lay ahead for the Young Adult Arthrioplasty Surgeon

We were able to contribute to discussions around how to measure outcomes and patient experience, this will lead to further collaborations with the company.
Year(s) Of Engagement Activity 2018