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The economic burden of amyotrophic lateral sclerosis: a systematic review

DOI:10.1007/s10198-021-01328-7
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K. Achtert
K. Achtert
K. Achtert
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Linda Kerkemeyer at LiKe Healthcare Research GmbH
Linda Kerkemeyer
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Objectives This systematic review aimed to comprehensively collect and summarise the current body of knowledge regarding the cost-of-illness of amyotrophic lateral sclerosis, to identify cost-driving factors of the disease and to consider the development of costs over the course of disease. Further, the review sought to assess the methodological quality of the selected studies.MethodsA systematic review was performed using the databases MEDLINE, Embase, Cochrane Library and PsycINFO. Studies examining the economic burden of amyotrophic lateral sclerosis on a patient or national level written in English or German published from the year 2001 onwards were included. Additional searches were conducted. Study characteristics and results were extracted and compared.ResultsIn summary, 20 studies were included in this review. Most studies investigated costs per patient, amounting to total costs between €9741€ to €114,605. Six studies confirmed a rise in costs with disease progression, peaking close to the death of a patient. National costs for amyotrophic lateral sclerosis varied between €149 million and €1329 million.Conclusion Most of these studies suggest the economic burden of amyotrophic lateral sclerosis to be considerable. However, further research is needed to establish a cost-effective health policy in consideration of disease severities.
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Vol.:(0123456789)
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The European Journal of Health Economics (2021) 22:1151–1166
https://doi.org/10.1007/s10198-021-01328-7
ORIGINAL PAPER
The economic burden ofamyotrophic lateral sclerosis: asystematic
review
K.Achtert1· L.Kerkemeyer1
Received: 14 July 2020 / Accepted: 27 May 2021 / Published online: 18 June 2021
© The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature 2021
Abstract
Objectives This systematic review aimed to comprehensively collect and summarise the current body of knowledge regard-
ing the cost-of-illness of amyotrophic lateral sclerosis, to identify cost-driving factors of the disease and to consider the
development of costs over the course of disease. Further, the review sought to assess the methodological quality of the
selected studies.
Methods A systematic review was performed using the databases MEDLINE, Embase, Cochrane Library and PsycINFO.
Studies examining the economic burden of amyotrophic lateral sclerosis on a patient or national level written in English or
German published from the year 2001 onwards were included. Additional searches were conducted. Study characteristics
and results were extracted and compared.
Results In summary, 20 studies were included in this review. Most studies investigated costs per patient, amounting to total
costs between €9741€ to €114,605. Six studies confirmed a rise in costs with disease progression, peaking close to the death
of a patient. National costs for amyotrophic lateral sclerosis varied between €149 million and €1329 million.
Conclusion Most of these studies suggest the economic burden of amyotrophic lateral sclerosis to be considerable. However,
further research is needed to establish a cost-effective health policy in consideration of disease severities.
Keywords Amyotrophic lateral sclerosis· Motor neuron disease· Cost-of-illness· Economic burden· Systematic review·
Healthcare costs
Introduction
Some years ago, a popular social media campaign raised
the awareness of amyotrophic lateral sclerosis (ALS) sig-
nificantly. Millions of people got inspired to take part in
this social media phenomenon called ‘Ice Bucket Challenge’
by pouring a bucket of ice water on their heads. Besides
increasing awareness for this progressive neurodegenerative
disease another primary goal of the Ice Bucket Challenge
was to raise money for its research [1]. To date, the disease is
incurable and will eventually lead to a patient’s death [2–4].
ALS is a rare disease with global incidence rates ranging
from 0.5 to 3.6 cases per 100,000 persons [5]. The incidence
is increasing with advancing age, peaking at about 60 to
75years. The disease is very rare under 30years and almost
not prevalent in under 20years olds [5–7]. The mean age is
at 56 to 58years [2, 8, 9]. Due to a short survival period of
about 2 to 4years after diagnosis, the global prevalence of
the disease is quite low amounting from 1.1 to 8.2 cases per
100,000 persons [10]. With a proportion of 1.5:1, the condi-
tion slightly affects men more often than women though at a
declining rate [5, 6]. ALS is characterized by the simultane-
ous appearance of signs of lower and upper motor neuron
degeneration. With disease progression the patients typically
experience atrophies and paralyses which can either begin at
the upper or the lower extremities [2, 3, 11]. Furthermore,
in the remaining cases patients suffer from a bulbar course,
where they first experience articulation problems and dys-
phagia [12]. Regardless of where the symptoms first occur,
in the terminal phase most patients suffer from a generalized
paralysis as well as a respiratory insufficiency [2, 9]. The
treatments commonly aim to attenuate the symptoms and
halt the deterioration of the disease. For many years, Riluzol
treatment has been the only widely approved drug therapy
with positive effects to prolong the survival with ALS [13].
* K. Achtert
achtert@inav-berlin.de
1 Institute forApplied Health Services Research (inav GmbH),
Berlin, Germany
Content courtesy of Springer Nature, terms of use apply. Rights reserved.
... 6 Institute of Cognitive Sciences and Technologies, C.N.R, Rome, Italy. 7 Azienda Ospedaliero Universitaria Citta' della Salute e della Scienza, Turin, Italy. 8 Department of Neurology, Johns Hopkins University Medical Center, Baltimore, MD, USA. ...
... 6). The high fatality rate and the severe physical burden associated with ALS have further enhanced worldwide awareness of the disease, especially after the public attention attracted by the Ice Bucket Challenge [7][8][9][10] . ...
Amyotrophic lateral sclerosis: translating genetic discoveries into therapies
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  • Apr 2023
Recent advances in sequencing technologies and collaborative efforts have led to substantial progress in identifying the genetic causes of amyotrophic lateral sclerosis (ALS). This momentum has, in turn, fostered the development of putative molecular therapies. In this Review, we outline the current genetic knowledge, emphasizing recent discoveries and emerging concepts such as the implication of distinct types of mutation, variability in mutated genes in diverse genetic ancestries and gene-environment interactions. We also propose a high-level model to synthesize the interdependent effects of genetics, environmental and lifestyle factors, and ageing into a unified theory of ALS. Furthermore, we summarize the current status of therapies developed on the basis of genetic knowledge established for ALS over the past 30 years, and we discuss how developing treatments for ALS will advance our understanding of targeting other neurological diseases.
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... The last Lancet Global Burden of Disease report indicated that the worldwide years of life lost in 2019 ranged between 11 and 14 years in people with PD (GBD 2016Parkinson's Disease Collaborators, 2018Golbe and Leyton, 2018) and AD (GBD 2016Dementia Collaborators, 2019Zhao et al., 2021a) and up to 25 years in people with motor neuron diseases. The estimated annual cost for AD, PD and ALS in our era of symptomatic treatments range from US $225 billion in China (Yang and Chen, 2017;Jia et al., 2018;Li et al., 2019;Song et al., 2020;Xu et al., 2020) to US $411 billion in Europe (Tinelli et al., 2016;Cimler et al., 2019;Achtert and Kerkemeyer, 2021) and US $632.5 billion in the United States Achtert and Kerkemeyer, 2021;Thorpe et al., 2021;Alzheimer's Association, 2022). ...
... The last Lancet Global Burden of Disease report indicated that the worldwide years of life lost in 2019 ranged between 11 and 14 years in people with PD (GBD 2016Parkinson's Disease Collaborators, 2018Golbe and Leyton, 2018) and AD (GBD 2016Dementia Collaborators, 2019Zhao et al., 2021a) and up to 25 years in people with motor neuron diseases. The estimated annual cost for AD, PD and ALS in our era of symptomatic treatments range from US $225 billion in China (Yang and Chen, 2017;Jia et al., 2018;Li et al., 2019;Song et al., 2020;Xu et al., 2020) to US $411 billion in Europe (Tinelli et al., 2016;Cimler et al., 2019;Achtert and Kerkemeyer, 2021) and US $632.5 billion in the United States Achtert and Kerkemeyer, 2021;Thorpe et al., 2021;Alzheimer's Association, 2022). ...
Disease-modifying vs symptomatic treatments: Splitting over lumping
Chapter
  • Jan 2023
Clinical trials of putative disease-modifying therapies in neurodegeneration have obeyed the century-old principle of convergence, or lumping, whereby any feature of a clinicopathologic disease entity is considered relevant to most of those affected. While this convergent approach has resulted in important successes in trials of symptomatic therapies, largely aimed at correcting common neurotransmitter deficiencies (e.g., cholinergic deficiency in Alzheimer's disease or dopaminergic deficiency in Parkinson's disease), it has been consistently futile in trials of neuroprotective or disease-modifying interventions. As individuals affected by the same neurodegenerative disorder do not share the same biological drivers, splitting such disease into small molecular/biological subtypes, to match people to therapies most likely to benefit them, is vital in the pursuit of disease modification. We here discuss three paths toward the splitting needed for future successes in precision medicine: (1) encourage the development of aging cohorts agnostic to phenotype in order to enact a biology-to-phenotype direction of biomarker development and validate divergence biomarkers (present in some, absent in most); (2) demand bioassay-based recruitment of subjects into disease-modifying trials of putative neuroprotective interventions in order to match the right therapies to the right recipients; and (3) evaluate promising epidemiologic leads of presumed pathogenetic potential using Mendelian randomization studies before designing the corresponding clinical trials. The reconfiguration of disease-modifying efforts for patients with neurodegenerative disorders will require a paradigm shift from lumping to splitting and from proteinopathy to proteinopenia.
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... Patients become increasingly dependent on caregiver support, including the administration of treatment [7][8][9], which signi cantly affects the quality of life (QoL) and creates a great socioeconomic burden. [10,11] A cure for ALS it is not available yet and riluzole in Europe is the only approved Disease Modifying Treatment [13,14]. To date, riluzole is available in two formulations: tablets (50 mg) and oral suspension (5 mg/mL in 300 mL bottles). ...
Amyotrophic Lateral Sclerosis Patients’ Preferences on Riluzole Treatment in Europe
Preprint
Full-text available
  • Mar 2023
The Patient Preference Study aims to understand unmet needs related to riluzole management in patients with Amyotrophic Lateral Sclerosis (ALS) and which characteristics of a newly formulated product could better match their preferences. The survey involved 117 patients treated with riluzole, in four European countries. The dysphagic patients were least satisfied with both riluzole tablet and oral suspension and with ease in self-administration; up to 61% of respondents postponed or missed the treatment, due to swallowing difficulties and need for assistance. Overall, 56% of tablet and 53% of oral suspension users regularly crushed or mix riluzole with beverages, respectively; patients that always manipulate riluzole showed low patient satisfaction with the formulation and considered the risk of choking and pneumonia the most worrisome events. The study evaluated the driver factors in choosing/switching the therapy: 97% of ALS patients declared a low risk of choking. The study finally evaluated the level of preference of attributes of a new product: the most relevant were ease in use (4.3/5), convenient/portable packaging (4.0/5) and oral-dissolving properties without tongue motility (3.9/5). The Patient Preference Study suggests that patients have several unmet needs and preferences that could be addressed by new products, e.g. oral film technologies.
View
... Caring for patients with ALS is medically and financially resource intensive. Furthermore, there is a considerable burden on caregivers [11,12]. Most patients with ALS are conscious and have normal sensory function. ...
Factors affecting do-not-resuscitate decisions among patients with amyotrophic lateral sclerosis in Taiwan
Article
Full-text available
Amyotrophic lateral sclerosis (ALS) is a neurodegenerative disease. Usually, patients survive for approximately 2-4 years after the onset of the disease, and they often die of respiratory failure. This study examined the factors associated with signing a "do not resuscitate" (DNR) form in patients with ALS. This cross-sectional study included patients diagnosed with ALS between January 2015 and December 2019 in a Taipei City hospital. We recorded patients' age at disease onset; sex; presence of diabetes mellitus, hypertension, cancer, or depression; use of invasive positive pressure ventilator (IPPV) or non-IPPV (NIPPV); use of nasogastric tube (NG) or percutaneous endoscopic gastrostomy (PEG) tube; follow-up years; and number of hospitalizations. Data from 162 patients were recorded (99 men). Fifty-six (34.6%) signed a DNR. Multivariate logistic regression analyses revealed that the factors associated with DNR included NIPPV (OR = 6.95, 95% CI = 2.21-21.84), PEG tube feeding (OR = 2.86, 95% CI = 1.13-7.24), NG tube feeding (OR = 5.75, 95% CI = 1.77-18.65), follow-up years (OR = 1.13, 95% CI = 1.02-1.26), and number of hospital admissions (OR = 1.26, 95% CI = 1.02-1.57). The findings suggest that end-of-life decision making among patients with ALS may often be delayed. DNR decisions should be discussed with patients and their families during the early stages of disease progression. Physicians are advised to discuss DNR with patients when they can speak and to offer palliative care options.
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Epidemiology and economic burden of amyotrophic lateral sclerosis in the United States: a literature review
Article
  • Feb 2023
Objective: This review sought to gain a comprehensive, up-to-date understanding of the epidemiology and cost and healthcare resource use (HCRU) burden of amyotrophic lateral sclerosis (ALS) in the US, at a patient and national level. Methods: A targeted literature review (TLR) to identify epidemiological evidence (prevalence, incidence, mortality, survival), and systematic literature review (SLR) to identify cost and HCRU data published since January 2016, were performed. MEDLINE databases and Embase searches were conducted in January 2021. Key congresses (2019-2020) and bibliographies of relevant SLRs were hand-searched. Two high-quality SLRs were reviewed for additional cost data published between January 2001-2015. Registry and database studies were prioritized for epidemiological evidence. To allow comparison between studies in this publication, only evidence from the US was considered, with costs inflated to the 2020/2021 cost-year and converted to US dollars. Results: Eight studies from the epidemiology TLR, and eighteen from the cost and HCRU SLR, were extracted. Reported ALS incidence in the US was ∼1.5 per 100,000 person-years, and point prevalence ranged from 3.84-5.56 per 100,000 population. Total US national costs spanned ∼$212 million-∼$1.4 billion USD/year, and variably consisted of direct costs associated with HCRU and indirect costs. Conclusions: The national cost of ∼$1.02 billion USD/year (estimated using a prevalence of 16,055 cases) best aligns with prevalence estimates found in the TLR (equating to ∼13,000-18,000 cases). However, large-scale, population-based studies are necessary to precisely assess US epidemiology of ALS and capture all costs needed to inform cost-effectiveness models and resource planning.
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Costs of illness in amyotrophic lateral sclerosis (ALS): A cross-sectional survey in Germany
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Background: Amyotrophic lateral sclerosis (ALS) is a fatal neurodegenerative disorder. Consequently, patients undergo a multidisciplinary treatment that often requires intensive use of medical resources. This study provides an estimate on the cost of illness depending on the clinical severity while also analysing the patients' health-related quality of life. Methods: Primary data from patients and caregivers was collected through a standardised questionnaire. Direct medical, direct non-medical and indirect costs were calculated using the latest German health economic guidelines. Patients were divided into five groups according to the King's staging system. Health-related quality of life was assessed using EuroQoL Group EQ-5D-5L™ questionnaire. Influencing factors on both total cost and quality of life were examined. Results: The mean annual total cost of illness was 78,256€ per patient while the lifetime cost per patient was estimated at 246,184€. The prevalence based total burden yearly therefore was 519,776,352€ in Germany. Nearly half of the costs were attributable to informal care. With increase of the clinical severity stage, costs rose and quality of life decreased. The score of the revised Amyotrophic Laterals Sclerosis Functional Rating Scale was identified as one major influencing factor on total costs, while subjective impairment in daily activities and classification into a care level as opposed to having no care level influenced patients' quality of life. Conclusion: It is essential to understand the socioeconomic burden of a disease. These data can be used to improve patient care standards and quality of life while also serving as a basis for cost-benefit analyses during the approval process of new treatments.
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Current status and direct medical cost of amyotrophic lateral sclerosis in the region of Catalonia: A population-based analysis
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Introduction Amyotrophic lateral sclerosis is a neurodegenerative disease that leads to motor weakness. There is no cure, and treatment focuses on slowing down progression, which is achieved by a multidisciplinary approach. Hence, it is vital to understand the population needs for an optimal management of the disease. Objectives To evaluate the current status of amyotrophic lateral sclerosis in the region of Catalonia, how the disease is managed and its direct medical costs. Methods Records corresponding to 841 patients diagnosed between the year 2007 and 2017 were analysed in a retrospective population-based study, including data from primary care centres, hospitals (inpatient and outpatient care), extended care facilities and mental health centres. Results Mean diagnosis age was 66.11 years (SD = 12.61) and 52.79% of admitted patients were males. On average, 14.91 months elapsed between diagnosis and death, and the mean age of death was 72.64 years (SD = 12.00). Patients were admitted 10.70 times per year, mostly into primary care (86.50%), although most expenses were concentrated in hospital inpatient care. The mean cost per patient per year was €1,168. The 83.24% of patients had more than 4 systems affected by chronic conditions. Conclusions Primary care is of utmost importance in ALS attention in Catalonia, which may have a direct impact in reducing hospitalisation costs. Nonetheless, the expenses linked to inpatient care represent the biggest portion of total costs. Patients’ healthcare usage patterns and the high proportion of patients with multiple chronic conditions should be taken into account in order to adapt and improve guidelines and healthcare systems.
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Viral Ice Buckets: A Memetic Perspective on the ALS Ice Bucket Challenge’s Diffusion
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This paper presents an exploratory memetic perspective on the diffusion pattern of the ALS Ice Bucket Challenge. More precisely, the paper contributes to research on social learning, cultural evolution, and social contagion by shedding light on endogenous (meme-related) as well as exogenous (structural) properties that may have influenced the Ice Bucket Challenge’s diffusion. In a first pillar, we present a descriptive memetic analysis of the diffusion pattern, including an evaluation of the Ice Bucket Challenge according to memetic criteria for successful replication. In the second pillar, we present an agent-based simulation model designed to illuminate the influence of particular social network characteristics on the Ice Bucket Challenge’s diffusion. By combining these two pillars, we contribute to the advancement of memetic theory, narrowing the gap between a solely meme-centered perspective and social network analysis.
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The Economic Burden of Cardiovascular Disease in Type 2 Diabetes: A Systematic Review
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  • Feb 2018
Background Cardiovascular diseases (CVDs) constitute major comorbidities in type 2 diabetes mellitus (T2DM), contributing substantially to treatment costs for T2DM. An updated overview of the economic burden of CVD in T2DM has not been presented to date. Objective To systematically review published articles describing the costs associated with treating CVD in people with T2DM. Methods Two reviewers searched MEDLINE, Embase, and abstracts from scientific meetings to identify original research published between 2007 and 2017, with no restrictions on language. Studies reporting direct costs at either a macro level (e.g., burden of illness for a country) or a micro level (e.g., cost incurred by one patient) were included. Extracted costs were inflated to 2016 values using local consumer price indexes, converted into US dollars, and presented as cost per patient per year. Results Of 81 identified articles, 24 were accepted for analysis, of which 14 were full articles and 10 abstracts. Cardiovascular comorbidities in patients with T2DM incurred a significant burden at both the population and patient levels. From a population level, CVD costs contributed between 20% and 49% of the total direct costs of treating T2DM. The median annual costs per patient for CVD, coronary artery disease, heart failure, and stroke were, respectively, 112%, 107%, 59%, and 322% higher compared with those for T2DM patients without CVD. On average, treating patients with CVD and T2DM resulted in a cost increase ranging from $3418 to $9705 compared with treating patients with T2DM alone. Conclusions Globally, CVD has a substantial impact on direct medical costs of T2DM at both the patient and population levels.
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July 2017 ENCALS statement on edaravone
Article
Full-text available
  • Oct 2017
Neurologists of the ENCALS centers throughout Europe have discussed the potential of edaravone as a new therapy for amyotrophic lateral sclerosis (ALS, Motor Neuron Disease, MND) at the ENCALS meeting, 18–20 May 2017, in Ljubljana, Slovenia. In May 2017, the US Food and Drug Administration (FDA) granted a license for the drug known as edaravone (licensed in Japan in 2015 as Radicut®) for the treatment of ALS in the United States (to be marketed as Radicava®). We are not aware of any official request from Mitsubishi Tanabe Pharma, the manufacturer of edaravone, to the European Medicines Agency (EMA) to register the drug for use in ALS in Europe. However, edaravone can be imported to Europe from Japan or the United States. The FDA approval of edaravone is based on a single positive clinical trial. The ENCALS neurologists were of the view that the outcome of this trial requires a balanced and considered interpretation when considering how best to advise those with ALS and their families. This study showed that edaravone may slow disease progression in ALS, but the disease-modifying effect was limited to a subgroup of ALS patients with distinct clinical characteristics. For ALS patients without those characteristics there is currently no evidence for a therapeutic benefit of edaravone.
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Dying of amyotrophic lateral sclerosis: Health care use and cost in the last year of life
Article
  • Oct 2019
Objective: To describe health care service utilization and cost for decedents with and without amyotrophic lateral sclerosis (ALS) in the last year of life. Methods: Using linked health administrative data, we conducted a retrospective, population-based cohort study of Ontario, Canada, decedents from 2013 to 2015. We examined demographic data, rate of utilization, and cost of health care services in the last year of life. Results: We identified 283,096 decedents in Ontario, of whom 1,212 (0.42%) had ALS. Decedents with ALS spent 3 times as many days in an intensive care unit (ICU) (mean 6.3 vs 2.1, p < 0.001), and twice as many days using complex continuing care (mean 12.7 vs 6.0, p < 0.001) and home care (mean 99.1 vs 41.3, p < 0.001). A greater percentage of decedents with ALS received palliative home care (44% vs 20%, p < 0.001) and palliative physician home visits (40% vs 18%, p < 0.001) than decedents without ALS. Among decedents with ALS, a palliative physician home visit in the last year of life was associated with reduced adjusted odds of dying in hospital (odds ratio 0.65, 95% confidence interval 0.48–0.89) and fewer days spent in the ICU. Mean cost of care in the last year of life was greater for those with ALS ($68,311.98 vs $55,773.48, p < 0.001). Conclusions: In this large population-based cohort of decedents, individuals with ALS spent more days in the ICU, received more community-based services, and incurred higher costs of care in the last year of life. A palliative care physician home visit was associated with improved end of life outcomes; however, the majority of patients with ALS did not access such services.
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Cochrane Handbook for Systematic Reviews of Interventions
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  • Sep 2019
The revised edition of the Handbook offers the only guide on how to conduct, report and maintain a Cochrane Review ? The second edition of The Cochrane Handbook for Systematic Reviews of Interventions contains essential guidance for preparing and maintaining Cochrane Reviews of the effects of health interventions. Designed to be an accessible resource, the Handbook will also be of interest to anyone undertaking systematic reviews of interventions outside Cochrane, and many of the principles and methods presented are appropriate for systematic reviews addressing research questions other than effects of interventions. This fully updated edition contains extensive new material on systematic review methods addressing a wide-range of topics including network meta-analysis, equity, complex interventions, narrative synthesis, and automation. Also new to this edition, integrated throughout the Handbook, is the set of standards Cochrane expects its reviews to meet. Written for review authors, editors, trainers and others with an interest in Cochrane Reviews, the second edition of The Cochrane Handbook for Systematic Reviews of Interventions continues to offer an invaluable resource for understanding the role of systematic reviews, critically appraising health research studies and conducting reviews.
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Health Utilities and Costs for Motor Neurone Disease
Article
  • Aug 2019
Background: Motor neurone disease (MND) places a significant burden on patients, their carers, and healthcare systems. Objectives: To estimate health utilities and costs of MND within the UK setting. Methods: Patients with MND, recruited via 22 regional clinics, completed a postal questionnaire of a cost and quality-of-life survey. Health outcome assessment included the EuroQoL (EQ)-5D-5L, EQ-5D-visual analogue scale, Amyotrophic Lateral Sclerosis Utility Index, and the Amyotrophic Lateral Sclerosis Functional Rating Scale-Revised. Clinical staging was based on the Kings and Milano-Torino (MiToS) systems. The questionnaire asked about patients' use of primary, secondary, and community care services in the previous 3 months. Variability in total costs was examined using regression models. Results: 595 patients were included in the health utility analysis, of whom 584 patients also completed a resource use questionnaire. Mean health utility decreased and costs increased between consecutive Kings stages, from 0.76 (95% CI 0.71-0.80) and £1096 (£757-£1240) in Kings stage 1, to 0.50 (0.45-0.54) and £3311 (£2666-£4151) in stage 4, respectively. The changes by MiToS stages were from 0.71 (0.69-0.73) and £1115 (£937-£1130) in MiToS stage 0, to 0.25 (0.07-0.42) and £2899 (£2190-£3840) in stage 2. Kings stages 3 and 4 and MiToS stages 1 and 2, respectively, were significant in explaining variability in total costs. Conclusions: The impact of MND on health utilities and costs differs by disease severity. The data provided here can be used in cost-effectiveness analyses and to inform decision-making regarding healthcare provision for people with MND.
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