Background
Patients severely affected by progressive long-term neurological conditions such as Multiple Sclerosis (MS), Parkinson’s Disease (PD), Multiple-System Atrophy (MSA), Progressive Supranuclear Palsy (PSP) (grouped together as Parkinsonism), and Motor Neuron Disease (MND) have unmet physical and psychological needs [
1,
2], similar to cancer patients [
3] and experience problems in coordination and continuity of care [
4]. For example, Parkinsonism patients experience on average more than 10 symptoms [
5], which also include many non-motor symptoms such as fatigue or psychological problems [
6]. These diseases also pose significant demands on caregivers [
7] who experience unmet needs [
8].
Such complex, person-focused needs highlight the potential for specialist palliative care involvement. This has been recommended by the National Service Framework for long term conditions for patients with long-term neurological conditions early in the disease trajectory [
9]. Palliative care aims to deliver physical, psychological, emotional and spiritual care for patients with progressive and serious illness, and their caregivers. Despite this recommendation, patients severely affected by neurological conditions often have limited access to specialist palliative care services [
10].
A main factor limiting palliative care involvement in long term neurological conditions (LTNC) is the lack of evidence-based knowledge on how this should be done. So far, only a few models of integration between neurology and palliative care have been described. One Canadian palliative care service for patients with PD [
11] seems to improve patients’ symptoms over 3 months [
12]. However, exactly how and when patients are referred and the exact intervention format is unclear. In the UK, an integrated model in advanced MS showed improvements in patients’ symptoms, caregiver burden and costs [
13,
14] after a 6-week intervention in which palliative care and neurology worked closely together. However, whether this model would also be possible and successful in other neurological conditions and settings is unclear.
To develop and test better integrated models of care between neurology and specialist palliative care for patients with several progressive long-term neurological conditions (such as MS, PD, MSA, PSP, and MND) across different care settings and services, it is needed to explore the existing type and level of integration. Such insight is still largely missing. Therefore, this study aimed to map the integration between neurology and specialist palliative care services in different centres within the UK. This is an important first step to develop and test new models of care, which could ultimately lead to better care and improved patients’ outcomes.
Discussion
This mapping exercise showed wide variation across centers in the UK regarding the neurology and specialist palliative care services available and their level of integration. The eight sites ranged in geographical location and catchment area, which was (non-linearly) reflected in the number of patients seen within each site. Exploration of the selected sites and the service provision informs understanding on the current integration between neurology and specialist palliative care, and the need and opportunities for better integrated models of care.
Overall, our results indicate that in the UK neurology patients do not often access specialist palliative care, although differences between disease groups are present. This might be problematic, as it has been estimated that between 63 and 82 % of all deaths would have needed palliative care involvement [
15]. Moreover, neurology patients experience palliative care symptoms such as pain, breathlessness, worry and fatigue [
3], are at the end of life regularly hospitalised [
15] and it is not uncommon that they die here (quality issues with death registration data should be taken into account) [
16]. Of course, neurological diseases are heterogeneous and in our sample, MS patients were seen least by specialist palliative care, followed by Parkinsonism (with more involvement for MSA and PSP then PD) and MND. This was reflected in organizational differences; the integration between specialist palliative care and neurology was least developed in MS but joint clinics or MDT meetings took place in most sites for MND patients.
Our data suggest that variations in specialist palliative care involvement are related to disease trajectory rather than disease prevalence. MND generally has a poor prognosis of a few years. This is followed by MSA and PSP, although there are subgroups in MSA such as the parkinsonian variant which may have a prognosis of 15 years. Despite the fact that PD and MS might not be fatal in themselves, they do lead to deterioration and progressive symptoms, which for MS consists of fluctuations of periods of long stability punctuated by crises, while for PD this is marked by a mixture of motor and non motor symptoms with functional consequences underpinning the advanced PD state. There are at any time 5,000 people with MND, 120,000 with PD, 3,000 with MSA, 4,000-10,000 with PSP, and 100,000 with MS in the UK [
17].
There are several potential barriers, next to the unpredictable prognosis without a clear referral cut-off point, to integration between specialist palliative care and neurology. On the one hand, specialist palliative care experts have been found to be reluctant to take on care for non-cancer patients, due to a lack of disease-specific knowledge [
17]. On the other hand, neurologists might not refer patients as they might not always see a role for palliative care in their (complex) patients, associate palliative care with death and dying [
18], or are afraid to diminish patients’ hope by introducing palliative care [
19]. Last, it could be that patients themselves might be reluctant to accept a referral, for reasons such as the association of palliative care with end-of-life care or longstanding relations with neurological services and service providers. These barriers are important to take into account and to address when developing potential models of integrated care.
Various models could explore how neurology and palliative care can complement each other so that patients may best benefit. For example, specialist palliative care could be used as an ‘add-on’ approach used in time of need. Palliative care would then be provided in addition to neurology care, without taking over. It would also imply that palliative care is provided based on need rather than on prognosis or disease stage. This reflects a shifting trend and is supported by research showing that Parkinsonism patients’ symptoms over time are better predicted by their initial level of symptoms than by their disease stage [
5]. Such models fit also within the trend of a generalist and specialist palliative care approach [
20], where neurology providers are taught and expected to provide primary palliative care in less complex situations. The America Stroke Association has recently endorsed such an approach for stroke patients [
21]. While all providers caring for stroke patients should be able to provide primary palliative care (e.g., develop appropriate goals of care), referral to specialist palliative care should be done if necessary. While such models seem plausible, we believe they should also be rigorously tested before widely implemented.
We are currently in the process of testing such a new model within some of the sites from this mapping exercise. This exercise provided important contextual data to develop this model in line with the preparation phase of the MRC framework for developing and evaluating complex interventions [
22]. Incorporating this information, our model builds further upon an earlier study showing that an integrated approach improved MS patients’ and caregivers’ outcomes, while decreasing costs [
13,
14]. However, whether similar effects can be found across other diseases, managed in different settings, with varied service provisions is unclear and might become apparent from our own and future trials.
Potential factors to take into account when developing and evaluating integrated models are disease groups, co-terminus between neurology and specialist palliative care catchment areas and patients’ background characteristics. Disease groups differ, as discussed, in progression and survival. Next, the co-terminus between neurology and palliative care services could influence collaborations. If, as was the case for most sites in our sample, neurology catchment areas exceed palliative care catchment areas, neurology services need to build relationships with different palliative care teams, who might operate according to different models and which might influence integration approaches. Last, patient characteristics could influence needed care. For example, patients’ from ethnic minority groups have been found to suffer from more aggressive MS trajectories compared to white British patients [
23] and are more inclined to attribute the source of their illness to supernatural powers (e.g., fate) and less to biomedical factors (e.g., genetics) [
24]. They are also known to access palliative care at disproportionally lower rates [
25]. These and other factors might vary between sites and might influence the most appropriate provided services or integration.
This mapping exercise had limitations. Most importantly, as we asked clinicians to provide the detailed data about not only their service provisions, but also the number of patients they see and catchment area, issues with the quality of the data emerged. Not all sites were providing all information. One of the areas that often lacked data was the population and square mileage figure. In these situations, the researchers tried to manually find them. In some cases we had to look up additional information from websites or year reports. This also served as a check for the quality of information provided, although it was not feasible to check all provided information. We were, however, impressed by the willingness from both palliative care and neurology providers to provide us with the needed information. A final limitation of this exercise was that we focused on neurology and palliative care provisions, but not on rehabilitation care (both in specialist rehabilitation centres and in the community; however in one site a rehabilitation service was involved in ongoing support of patients). This should be examined in the future, as rehabilitation can play an important role in caring for neurology patients, for example, in symptom management but also in the provision of equipment and coordination of services [
26]. Other settings, such as specialist care homes, and disciplines, such as elderly care, might also play a role and be relevant to explore.
Acknowledgements
We would like to thank all clinicians and other staff who helped to complete the mapping exercise.
OPTCARE Neuro is a multicentre clinical trial funded by the National Institute for Health Research, Health Services and Delivery Research Programme (NIHR HS&DR, 12/130/47), led by King’s College London, Cicely Saunders Institute, Department of Palliative Care, Policy & Rehabilitation, UK. The views expressed in this publication are those of the authors and not necessarily those of the NHS, the National Institute for Health Research or the Department of Health.
Co-Chief Investigators: Higginson, Gao.
Co-investigators: Al-Chalabi, Benz, Burman, Byrne, Chaudhuri, Crosby, Evans, Hotopf, Jackson, Leigh, McCrone, Murtagh, Pickles, Silber, Wilcock, Young.
Project Manager: Hepgul, van Vliet (1st Apr 14 to 1st Apr 15).
Site PIs: Brighton – Lindsay; Cardiff – Byrne; Liverpool – Young; London – Burman, Higginson, Murtagh; Nottingham – Crosby.
Trial statisticians: Gao, Pickles.
Health Economists: McCrone, Yi.
Collaborators: Turner-Stokes, C Murphy.
Intervention Leads: Nottingham – Crosby; London - Bajwah, Dawkins; Liverpool – Groves; Cardiff – Byrne; Brighton - Lindsay.
OPTCARE Neuro team members: Prof Ammar Al-Chalabi, Dr Sarah Awan, Dr Sabrina Bajwah, Dr Cynthia Benz, Dr Rachel Burman, Dr Anthony Byrne, Prof K Ray Chaudhuri, Dr Vincent Crosby, Ms Joanna Davies, Ms Marsha Dawkins, Dr Catherine Evans, Ms Mim Evans, Ms Sarah Farnan, Dr Wei Gao, Dr Karen Groves, Dr Nilay Hepgul, Prof Irene Higginson, Prof Matthew Hotopf, Dr Diana Jackson, Mrs Paramjote Kaler, Dr Nigel Leigh, Dr Fiona Lindsay, Ms Cathann Manderson, Prof Paul McCrone, Ms Caroline Murphy, Dr Fliss E M Murtagh, Mrs Jenifer Newton, Ms Caty Pannell, Ms Louise Pate, Prof Andrew Pickles, Dr Eli Silber, Miss Debbie Tonkin, Prof Lynne Turner-Stokes, Dr Liesbeth van Vliet, Dr Andrew Wilcock, Dr Deokhee Yi, Prof Carolyn Young.
Study Steering Committee members: Prof Marie Fallon (Chair), Dr Cynthia Benz, Prof Mogens Groenvold, Prof William Hollingworth, Ms Denise Howel,, Prof Huw Morris, Mr Foster Murphy, Dr Diane Playford, Prof Julia Riley, Prof Jane Seymour, Dr Andrew Wilcock.
Data Monitoring and Ethics Committee members: Prof Mike Bennett (Chair), Prof Gunn Grande, Dr David Oliver, Prof Raymond Voltz, Prof Stephen Walters.
Patient and Public Involvement Committee members: Dr Cynthia Benz, Mr Sanjay Chadha, Mr David Charlton, Mr Colin Fellows, Ms Helen Findlay, Mrs Savita Jain.
The UKCRC-registered King’s Clinical Trials Unit at King’s Health Partners is part funded by the National Institute for Health Research (NIHR) Biomedical Research Centre for Mental Health at South London and Maudsley NHS Foundation Trust and King’s College London and the NIHR Evaluation, Trials and Studies Coordinating Centre (NETSCC). We would like to thank Ms Joanna Kelly for the management of the eCRF database, and Mrs Beverley White-Alao for providing advice on the Trial Management process.
The Collaboration for Leadership in Applied Health Research and Care (CLAHRC) South London is part of the National Institute for Health Research (NIHR), and is a partnership between King’s Health Partners, St. George’s, University London, and St George’s Healthcare NHS Trust. This publication is independent research supported by the National Institute for Health Research Collaboration for Leadership in Applied Health Research & Care Funding scheme. The views expressed in this publication are those of the author(s) and not necessarily those of the NHS, the National Institute for Health Research or the Department of Health.
Prof Irene J Higginson is an NIHR Senior Investigator.
Prof Ammar Al-Chalabi’s work is part of an EU Joint Programme - Neurodegenerative Disease Research (JPND) project, which is supported through the following funding organisations under the aegis of JPND -
www.jpnd.eu (United Kingdom, Medical Research Council and Economic and Social Research Council), and the EuroMotor project, funded by the European Community’s Health Seventh Framework Programme (FP7/2007–2013; grant agreement number 259867). AAC receives salary support from the National Institute for Health Research (NIHR) Dementia Biomedical Research Unit and Biomedical Research Centre in Mental Health at South London and Maudsley NHS Foundation Trust and King’s College London.
Authors’ contributions
IH, WG, VC, AW, AB, AA, KRC, CE, ES, CY developed the design of the study. LV, WG, IH and DF carried out the mapping exercise and interpreted data. LV prepared the manuscript with input from WG and IJH. All authors helped retrieve data, and commented the draft versions and agreed with the final version.