Healthcare Utilisation and Satisfaction with Care in Patients with Amyotrophic Lateral Sclerosis - An Observational Study

INTRODUCTION

Amyotrophic lateral sclerosis (ALS) is a neurodegenerative disease primarily affecting upper and lower motor neurons and characterized by progressive muscle weakness and wasting. Besides muscular symptoms, cognitive and behaviour changes can occur, being more frequent in severe stages of the disease [1]. Other commonly reported impairments include fatigue, anxiety, depression, and pain [2]. The European incidence rate is approximately 3 per 100 000 people-years, with a slightly higher rate among men [3, 4]. Depending on the site of onset, ALS can be described as spinal, i.e., starting with muscular signs in the limbs, or bulbar which starts with speech and swallowing difficulties. Bulbar-onset ALS is present in about 25%of patients and is associated with worse prognosis [5, 6]. Although most patients with ALS die from respiratory failure within 2–4 years of diagnosis, the inter-individual variety is substantial and some survive for decades [7, 8].

There is no cure for ALS and clinical management focus on treatment and care to alleviate symptoms, and to improve survival and health-related quality of life (HRQL). Published guidelines recommend, inter alia, regular support from a multidisciplinary care team, symptomatic and disease-modifying medical treatment, percutaneous endoscopic gastrostomy feeding to improve nutrition and mechanical ventilation to improve respiratory function [9, 10]. Since ALS has a vast impact on functioning with activity limitations and participation restrictions in social and lifestyle activities [11, 12], patients will need a large amount of healthcare services. Thus, besides the need for medical care, patients with ALS have a need for other healthcare and social services, such as rehabilitation, advanced healthcare care in the home, special transport services, home care services and housing with special services due to disability.

The organisation of healthcare and social services differs internationally. Thus, the study results from different countries might not be extrapolated to other countries due to differences in healthcare systems and policies. Swedish citizens have, based on their needs, access to healthcare and social services to a low cost for the individual. However, the organisation and delivery of services vary between regions and municipalities. In the Stockholm county (Region Stockholm), the outpatient ALS clinic at the department of neurology at the Karolinska University Hospital has the main responsibility for the ALS specialist care. A cohort of patients, and their next of kin, from this ALS clinic has been followed every six-months during a three-year period [2, 13, 14]. The results have shown that patients, regardless of disease severity, experienced fatigue, anxiety, depression, and pain, and that about one-third experienced two or more of these impairments concurrently, i.e., at the same time point [2]. Further, activity limitations and participation restrictions were common, and the use of aids and services increased over time [2]. In addition, despite the provided social services, next of kin served as informal caregivers and devoted a significant amount of time to care duties [14].

However, for the assessment of healthcare needs and the planning and organisation of good quality care for patients with ALS, it is important to gain knowledge not only on functioning but also on which healthcare services they currently use, and their stated need and satisfaction with care. The literature on healthcare utilisation in patients with ALS is scarce and mainly focus on costs [15–17], especially at the end of life [18, 19]. The need for including the patient perspective, i.e., patient satisfaction with healthcare services has been highlighted to yield a fuller picture [20], but studies are scarce [21]. There is, further, a dearth of studies combining these aspects, i.e., use of and satisfaction with healthcare. Thus, the aims of the present study were to: a) describe the use of healthcare in this cohort of patients with ALS; b) explore differences in use of healthcare based on personal and disease-related factors c) describe satisfaction with care from the patients’ perspective, and d) explore differences in use of healthcare based on satisfaction with healthcare.

MATERIAL AND METHODS

RESULTS

Twenty-five of the 60 patients included at baseline died between data collections and 23 declined participation at some time-point during the three-year study. Thus, 39 patients participated in data collections at T2, 29 at T3, 17 at T4, 14 at T5 and T6, and 12 patients participated in data collections at T7. Information on patient characteristics is presented in Table 1. The median age was around 60 years, the majority lived with a family member, around a third had a university education, aids and services were frequently used, and this use appeared to increase over time.

Approximately half of the patients (52%) had the year before baseline been an inpatient with a median stay of 12 days (Table 2). Between 24%and 44%received inpatient care during each six-months period, i.e., from baseline until the last data-collection (Table 2). The total amount of inpatient days (i.e., from one year before baseline to the last data collection) was 1316 days where care at the neurology and rehabilitation departments composed two thirds of all inpatient care (Fig. 1). Altogether 37 individual patients received inpatient care at the neurology department and 10 at a rehabilitation department.

Fig. 1

Hospital inpatient care from one year before study start (T0) until the last participated data collection (T1–T7), proportion of inpatient days at different departments. Other inpatient care includes surgery, orthopaedics, and palliative care.

There were no significant between-group differences (age, sex, education level, time since diagnosis, and disease severity) in use of inpatient care at the departments of neurology, medicine and geriatrics or the summarised others which included surgery, orthopaedics, and palliative care. There was a significant difference in use of inpatient care at rehabilitation departments depending on time since diagnosis, p = 0.008. The median (minimum-maximum) stay for those with < 24 months since diagnosis was 18 (11–19) days compared to 37 (28–84) days for those with≥24 months since diagnosis.

All patients had registered outpatient contacts during the year before baseline (T0-T1), and all received outpatient care during the study period (Table 2). The median number of contacts per user varied over time, from 52 the year before baseline to 115 at T7 (Table 2). A total of 14036 outpatient contacts (i.e., from one year before baseline to the last data collection) were registered of which half were registered by providers of advanced healthcare in the home and a fifth by allied health professionals (Fig. 2). Distributions of outpatient contacts by healthcare personnel and sort of contacts are presented in Table 3. Over half of all outpatient contacts consisted of home visits, mainly performed by nurses. Outpatient contacts with physicians were mainly clinic visits followed by administrative contacts, i.e., without patient. Over half of the outpatient contacts with occupational therapists were home visits while half of the contacts for physiotherapists were visits at the clinic. Over time, all patients had registered outpatient contacts with physicians, 58 with nurses, 55 with physiotherapists, 50 with occupational therapists, 42 with dieticians, 36 with speech therapists, 33 with welfare officers and 5 patients had outpatient contacts with psychologists.

Fig. 2

Hospital outpatient care from one year before study start (T0) until the last participated data collection (T1–T7), proportion of contacts with different outpatient healthcare providers. Other outpatient care included contacts with medicine, surgery, orthopaedic, psychiatry, geriatrics, respiratory, ear/nose/throat and radiology clinics.

There were no significant between-group (age, sex, education level, time since diagnosis, and disease severity) differences in use of outpatient care from providers of advanced healthcare in the home, allied health professionals, and primary care. There were significant differences in use of outpatient ALS clinic care between groups based on time since diagnosis (p = 0.046) and disease severity (p = 0.006). The median (minimum-maximum) contacts for those with < 24 months since diagnosis was 15 (2–95) compared to 11 (2–37) for those with≥24 months since diagnosis. The median (minimum-maximum) contacts for those with an ALSFRS-R score between 0–24 (severe disease) was 10 (2–25) compared to 15 (2–95) for those with ALSFRS-R score between 25–48 (less severe). There was a significant difference in use of other outpatient care (i.e., including contacts with medicine, surgery, orthopaedic, psychiatry, geriatrics, respiratory, ear/nose/throat and radiology clinics) between groups based on age (p = 0.044). The median (minimum-maximum) contacts for those < 65 years was 8 (1–111) compared to 5 (2–33) for those≥65 years.

Satisfaction with the various dimensions of health care (i.e., art of care, availability, accessibility, continuity, participation in care planning, efficacy/outcome of care and finances) for those with a manifested need or being applicable from the perspective of patients with ALS was relatively stable over time (Table 4). In general, two thirds or more of the patients were satisfied with the art of care (sympathy or engagement from staff and kind treatment) and the continuity, i.e., meeting the same staff. As for availability, most patients were satisfied although less were satisfied with the availability of physicians. The proportion of patients being satisfied with access to welfare officers and psychologists was in general lower than for other healthcare personnel, even though a majority expressed that they had had contact with all expertise needed. Almost all patients wanted to participate in care planning, but few perceived that they had taken part. Regarding efficacy or outcome of care, over time around two-thirds to all patients expressing a need were satisfied.

There were no significant differences in satisfaction with care in those patients reporting a need at both baseline (T1) and at the first follow-up (T2). There were no statistically significant differences in use of healthcare between groups satisfied or not satisfied with healthcare except for outpatient contacts at T3. At that timepoint, the median (min-max) number of contacts were 22 (3–107) for those satisfied (n = 18) and 40 (6–278) for those not satisfied (n = 7), p = 0.017.

DISCUSSION

We have explored utilisation of healthcare and satisfaction with care in a cohort of patients with ALS who participated in our three-year observational study [2]. Over time, between a quarter and half of the patients used inpatient care, and two-thirds of the total number of inpatient days were spent at neurology and rehabilitation departments. All patients used outpatient care, and over half of the total number of contacts were with providers of advanced healthcare in the home and one-fifth with allied health professionals. Nurses performing home visits composed the largest proportion of outpatient contacts. Analyses revealed that patients with less severe disease and shorter time since diagnosis had significantly more contacts with the outpatient ALS clinic compared to those with severe disease and longer time since diagnosis. Satisfaction with the various dimensions of care was in general stable over time with around two-thirds or more of patients being satisfied. However, few patients perceived that they had taken part in care planning. Overall, there seemed to be no significant difference in use of healthcare between patients being satisfied with healthcare versus those not satisfied.

Healthcare utilisation data imply that patients with ALS mainly had outpatient contacts and that half, or less of the participating patients, utilised inpatient care. This is in line with findings from other neurological conditions in Sweden such multiple sclerosis [26] and stroke [28], but differ from the reported large amount of inpatient care in patients with Guillain-Barré syndrome [24]. Another striking difference is the amount of inpatient rehabilitation, where only 10 patients with ALS (16%) had any inpatient rehabilitation days registered in comparison to 31%of patients with multiple sclerosis [26] and 83%of patients with Guillain-Barré syndrome [24]. These differences can be due to the diverse courses of these diseases, or just mirror changes in the organisation of healthcare where the trend during the last decades is to reduce inpatient care in favour of outpatient care.

Most of the outpatient care consisted of contacts with personnel providing advanced healthcare in the home. In fact, almost two-thirds of all registered outpatient contacts were home visits. In addition to these visits, the patients also had home care provided by personnel from community-based social services, or community and government salaried personal assistants. Although all these services make it possible for patients to remain in their home even at the end of life, it may also induce a significant amount of strain for both patients and their families. The wish and struggle to maintain roles within the family and a sense of normality, as well as to retain control of personal and domestic space are factors highlighted by patients with ALS and their families [29–32]. However, the burden of care experienced by family caregivers might not depend on utilisation of healthcare and social services but on the subjective experience and psychological composition of the family caregivers [33].

Current guidelines on clinical management of ALS recommend that patients have contact with an experienced neurologist and receive regular support from a multidisciplinary care team [9, 34]. The ALS clinic at the department of neurology Karolinska University Hospital was developed over 30 years ago with the aim to improve the management and care for patients with ALS and their families in the Region Stockholm. Besides neurologists and nurses, team members from other hospital departments include occupational therapists, physiotherapists, welfare officers, speech therapists and dieticians. However, it is clear from our results that healthcare emerging from the ALS clinic only composed a small amount of the utilised outpatient care. Further, that contacts with the ALS clinic was reduced as patients became worse and needed advanced healthcare in the home. Studies have shown that it is important to individualise and involve patients and their caregivers, both through information and joint decision-making, to avoid experienced difficulties during care transitions [35, 36]. It would therefore be interesting to study these care transitions from one healthcare provider to another, for example from the ALS clinic to advanced healthcare in the home, from both the patients’ and the healthcare system perspective. Further, to adhere to the guidelines on clinical management of ALS, here is a need to develop a structured multidisciplinary care organisation among healthcare providers in outpatient care, beyond the multidisciplinary care provided by the ALS-clinic.

The management and care of ALS incorporate many different healthcare providers and there is a need for care coordination, a topic which has been reported to receive little attention in the literature [37]. Perhaps the found discrepancy between wanting and having participated in care planning illustrates the problem with care coordination and formulation of coherent care plans. Even though the Swedish Patient Act (2014:821) state the patient’s right to participate in all decisions about the care he/she will be receiving, many patients with ALS stated that they had not participated. Our findings of extensive use of outpatient care beside the rather small amount of care from the ALS clinic, clearly indicate the need of coordination between these entities and with the individual patient participating. An issue previously raised by patients with motor neuron diseases [38, 39]. Thus, there is a need for strategies to enhance the coordination of care and to incorporate the patients with ALS as full partners to their care-providers with a role in healthcare decisions [40], i.e., to establish a person-centered care [41] where care is based on the patient’s preferences, needs, and values, taking patient resources into account [42]. This may be facilitated by interdisciplinary or transdisciplinary care models which have shown to improve health outcome, resource utilisation and satisfaction in patients with ALS [43].

Satisfaction with care in our cohort of patients with ALS resembled previous findings in patients with multiple sclerosis [25, 26], where lower satisfaction with availability of physicians and psychologists, and accessibility to psychosocial support /counselling was found. Studies on satisfaction with care in patients with ALS and their family caregivers report concerns about inadequate emotional support [38], lack of continuity and coordination of services [38, 39], unmet needs [44] and difficulties to accessing services [45]. Even though most of our patients expressed being satisfied with the various dimensions of healthcare, patients being dissatisfied varied from around a third to a fifth depending on timepoint and dimensions. This indicate that there is room for improvement. Patients who receive tailored services seem more satisfied [46]. Thus, person-centered care should be adopted to increase both satisfaction with and the quality of healthcare services.

The major strength with this study is that data on healthcare utilisation came from the computerised register where all in- and outpatient care can be identified and linked to the individual patient and, thus, is not dependent on the patients’ memory. In addition, the prospective design with repeated data-collections by home visits to the patients, and the possibility to link patient characteristics and their perceived satisfaction with healthcare to all register-based data make this study unique. However, studying patients with ALS over time has its challenges due to the progressive nature of the disease. Even though recruitment was performed at an ALS clinic with probably the largest patient cohort in Sweden, the study sample was reduced already after one year which is a limitation of the study. Also, studying satisfaction with healthcare by use of a questionnaire does not give the full picture and should preferably be complemented by qualitative studies.

In conclusion, patients with ALS use hospital-based specialist care and other outpatient care in parallel with many healthcare providers involved. Our findings highlight the need for implementation of person-centred care to improve both coordination of care, care transitions and satisfaction with healthcare services.