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Optimizing quality of care for patients with ALS and their family caregivers

Creemers, H.W.J.

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Citation for published version (APA):Creemers, H. W. J. (2016). Optimizing quality of care for patients with ALS and their family caregivers

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4Cluster RCT of case management on pa� ents’ quality

of life and caregiver strain in ALS

Huub CreemersJan H. VeldinkHepke GrupstraFrans NolletAnita BeelenLeonard H. van den Berg.

Neurology 2014. Jan;82(1):23-31.

© Reprinted with permission from Wolters Kluwer Health

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ABSTRACT

Objec� vesTo study the eff ect of case management on quality of life (QOL), caregivers’ strain, and perceived quality of care (QOC) in pa� ents with amyotrophic lateral sclerosis (ALS) and their caregivers.

MethodsWe conducted a mul� center cluster randomized controlled trial, with the mul� disciplinary ALS care team as the unit of randomiza� on. During 12 months, pa� ents with ALS and their caregivers received case management plus usual care, or usual care alone. Outcome measures were the 40-item ALS Assessment Ques� onnaire (ALSAQ-40), Emo� onal Func� oning domain (EF); the Caregiver Strain Index (CSI); and the QOC score. These measures were assessed at baseline and at 4, 8, and 12 months.

ResultsCase management resulted in no changes in ALSAQ-40 EF, CSI, or QOC from baseline to 12 months. ALSAQ-40 EF scores in both groups were similar at baseline and did not change over � me (p = 0.331). CSI scores in both groups increased signifi cantly (p < 0.0001). Pa� ents with ALS from both groups rated their perceived QOC at baseline with a median score of 8, which did not change signifi cantly during follow-up.

ConclusionWithin the context of mul� disciplinary ALS care teams, case management appears to confer no benefi t for pa� ents with ALS or their caregivers.

Classifi ca� on of EvidenceThis study provides Class III evidence that case management in addi� on to mul� disciplinary ALS care does not signifi cantly improve health-related quality of life of pa� ents with ALS.

Eff ec� veness of case management in ALS

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INTRODUCTION

There is no cure for amyotrophic lateral sclerosis (ALS); treatment is limited to op� mal neuropallia� ve care, an� cipa� ng the relentless progression of muscle weakness and its consequences to daily ac� vi� es and par� cipa� on 1,2. In the Netherlands, in accordance with interna� onal guidelines,3-5 most pa� ents with ALS are supported by one of the 43 ALS teams that provide proac� ve mul� disciplinary ALS care. Neuropallia� ve care is diverse and complex. We know from clinical prac� ce and from QOC evalua� ons from pa� ents and caregivers that ALS care does not always adequately address the needs of pa� ents and their caregivers 6,7.

Case management has been suggested as an innova� ve strategy to op� mize complex care, thereby improving pa� ent sa� sfac� on, caregiver sa� sfac� on, and quality of life (QOL) 8. In case management, an individual supports the pa� ent and caregiver through the complex naviga� on of available resources to manage the consequences of ALS, with the aim of stabilizing QOL 9. Case management programs are o{ en directed at pa� ents with serious and life-threatening illness and increasingly incorporate pallia� ve care assessment and interven� ons 10.

In this study we inves� gated the added value of case management to mul� disciplinary ALS care. We hypothesized that case management in addi� on to usual care would improve QOL of pa� ents with ALS, caregivers’ burden, and perceived QOC of pa� ents with ALS and their informal caregivers.

METHODS

Study designWe performed a cluster randomized controlled trial (RCT) with the mul� disciplinary ALS team as the unit of randomiza� on (fi gure 1). Teams as a whole were randomized, instead of individual pa� ents, to avoid readjustment of each team’s treatment procedures during the study period owing to their experiences with case management. The full details of our trial protocol are available upon request from the correspondence address.

The primary research ques� on for this study was to determine whether case management in addi� on to usual mul� disciplinary ALS care would improve the QOL of pa� ents. The secondary research ques� ons were if supplemental case management would aff ect caregivers’ strain and the QOC of pa� ents and their primary informal caregivers compared with mul� disciplinary ALS care alone.

This study design provided Class III evidence for case management in addi� on to mul� disciplinary care on health related QOL for pa� ents with ALS, on caregiver burden and on QOC for pa� ents and their caregivers.

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Standard Protocol Approvals, Registra� ons, and Pa� ent ConsentsThe Medical Ethics Commi� ee of the Academic Medical Center in Amsterdam waived the need for ethical approval. Our RCT was registered at the Netherlands Trial Register, number NTR1270. Informed consent was obtained from all par� cipa� ng pa� ents and all caregivers.

ClustersWe invited all 43 Dutch mul� disciplinary ALS teams (clusters) to par� cipate. Pairs of teams were formed that were matched on three factors that may aff ect the QOC: the number of pa� ents with ALS the team supports during one year (≤10; 11−20; >20); the type of center the team is working in (outpa� ent department of a hospital or rehabilita� on center); and whether or not the team is cer� fi ed by the Dutch Associa� on for Pa� ents with Neuromuscular Disease as a “Neuromuscular Rehabilita� on Center” 11. Finally, a researcher (HC) performed computer generated randomiza� on of the teams. Alloca� on concealment was at the cluster level.

Par� cipantsThe rehabilita� on medicine consultant of each par� cipa� ng ALS team introduced the study to the eligible pa� ents and their most important informal caregiver. Pa� ents were not eligible for trial entry if they met one or more of the exclusion criteria: cogni� ve dysfunc� on (Mini Mental State Examina� on <20, 12 insuffi cient mastery of the Dutch language, and ins� tu� onaliza� on. The exclusion criterion for caregivers was insuffi cient mastery of the Dutch language.

BlindingIn the interven� on and control groups, the case manager administered the outcome assessment ques� onnaires during baseline home visits; assessments were unblinded for group assignment. Two independent researchers who were blinded to group assignments performed outcome assessments during follow-up at 4, 8, and 12 months.

Interven� onsUsual careUsual care in the Netherlands is neuropallia� ve care by mul� disciplinary, secondary care teams. Such teams consist of a rehabilita� on medicine consultant, an occupa� onal therapist, physical therapist, speech pathologist, die� cian, social worker, psychologist and consultant physicians (in neurology, respiratory and gastroenterology). Community and social services also play an important role in care for pa� ents with ALS and their caregivers. General prac� � oners, district nurses, home care services, paramedics, social workers, and voluntary workers par� cipate in these services.


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Case managementWe performed the interven� on “case management” at the individual par� cipant level. The predominant focus of the pa� ent advocacy case management model is more comprehensive coordina� on of services across the con� nuum of care, viewed from the pa� ent perspec� ve 8,13-16.

During the 12-month interven� on period, case management was provided by 2 experienced occupa� onal therapists, specialized in ALS care and trained in client-centered prac� ce, who used a client-centered approach to guide the par� cipants 17. The case manager had an independent posi� on outside, but in close contact with, the ALS team. The case manager’s role was to be a� en� ve to the needs of the par� cipants. The case manager provided par� cipants all of the informa� on needed to allow individual choices about how their needs would be met.

The case manager started the interven� on by visi� ng par� cipants at home at study entry and subsequently every three months. Between visits, contact was possible by telephone, e-mail, or in wri� ng. At the fi rst visit, the case manager provided par� cipants with addi� onal oral and wri� en informa� on about the procedures and objec� ves of the case management interven� on. The posi� on, tasks, and responsibili� es of the case manager in rela� on to the par� cipants’ usual care system were also elucidated.

The star� ng point for the case management interven� on was any soma� c, psychosocial, environmental, or care issue raised by the par� cipants. On the basis of the issue, the case manager discussed the aims to achieve and the steps necessary for achievement (client-centered approach). In response to the issues raised, the case manager undertook one or more of the following steps:

• Inform par� cipants’ mul� disciplinary ALS team and other professional caregivers involved.

• Provide par� cipants with wri� en and oral informa� on and advice.• Give emo� onal support to par� cipants.• Refer par� cipants to health care providers and agencies.• Mediate if problems arose between par� cipants and health care providers or

agencies.• Support par� cipants during their contact with agencies and suppliers.• Evaluate the handling of the issues raised by par� cipants.

During the quarterly home visits, the case manager monitored the par� cipants’ disease process with the aim to iden� fy symptoms or problems related to ALS progression or caregiver burden. For that purpose, a symptom checklist was used to monitor disease progression and to register changes in disease symptoms. When the case manager no� ced QOC problems or observed an increase in symptom burden, psychosocial problems, or caregiver burden, the issue was discussed with the par� cipants. Subsequently, steps toward solu� ons of the relevant issues were discussed and put into ac� on, if desired by the par� cipants.

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A{ er randomiza� on, the case manager and the rehabilita� on medicine consultant (as coordinator of the team) agreed upon lines of communica� on with the ALS team during the study. The case manager visited the ALS team before ini� a� on of the study to meet the team upon request. The case manager reported any ques� ons of the par� cipants, and ac� ons or issues no� ced by the case manager to the rehabilita� on medicine consultant.

Outcome measuresThe primary outcome was each pa� ent’s health-related QOL assessed with the 40-item ALS Assessment Ques� onnaire (ALSAQ-40), Emo� onal Func� oning domain (EF). The ALSAQ-40 contains 40 items incorporated in fi ve dis� nct areas of health: Physical Mobility, Ac� vi� es of Daily Living/Independence, Ea� ng and Drinking, Communica� on, and Emo� onal Func� oning. Each of the fi ve scales is transformed into a scale of 0 to 100 (100 indica� ng worst health) 18-21.

Secondary outcomes were caregiver strain assessed with the Caregiver Strain Index (CSI) 22 and Quality of Care (QOC). To score QOC, we asked pa� ents rate the total care provided on a 10-point scale, with 1 as very poor QOC and 10 as excellent QOC. Caregivers were asked to give two ra� ngs, one for the care provided to the pa� ent, and one for the care provided to the caregiver.

We monitored disease progression with the ALS Func� onal Ra� ng Scale-Revised (ALSFRS-R). The ALSFRS-R is a 48-point validated disease-specifi c measure to assess func� on 23; a lower score indicates more disability.

Data analysisA sample size of 65 pa� ents with ALS and their caregiver in each treatment group was planned. This group size has 80% power to detect a treatment diff erence of 11.9 points (SD: 26.25) in QOL scores on the ALSAQ-40 EF between the groups 20. This calcula� on is based on 4 repeated measurements with a within-person correla� on coeffi cient (� me interval 4 months) of 0.75 at a signifi cance level of 0.05 (one-sided) 24. We calculated the sample size without taking into account the intracluster correla� on coeffi cient, as the number of par� cipants per mul� disciplinary ALS team is rela� vely small.

We compared sociodemographic characteris� cs of the pa� ents and their caregivers, as well as the clinical characteris� cs of the pa� ents in both groups (par� cipa� ng vs non-par� cipa� ng and interven� on vs control) with 2-tailed independent t tests (for con� nuous variables) and chi-squared tests (for dichotomous and categorical variables).

We used linear mixed models with an unstructured covariance type (mul� level analysis) to analyse diff erences in outcomes between the interven� on and control group over � me and to adjust for the clustering of the data 25. In case of signifi cant diff erences in baseline characteris� cs of pa� ents and caregivers between the interven� on and control group, we adjusted for these variables.


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A linear mixed model does not require complete follow-up data from all par� cipants. We expected incomplete follow-up owing to decease or withdrawal. Sta� s� cal analysis was carried out on an inten� on to treat basis and data of all included pa� ents and caregivers, including those with incomplete sets of data were analysed. We calculated regression coeffi cients for the eff ects of case management compared with usual care as β values and standard errors (SE). Because outcomes are measured at the individual pa� ent level, the analysis must be adjusted for poten� al clustering in the data. The analysis yields maximum likelihood es� mates for the eff ects of � me, treatment group, and the � me � treatment group interac� on. All hypotheses were tested 2-sided, with a cri� cal value of <0.05.

RESULTS

Cluster randomiza� onThirty-one mul� disciplinary ALS teams (72%) agreed to par� cipate in the study (fi gure 1). We evaluated 16 interven� on teams and 15 control teams. No teams dropped out during the interven� on period. Par� cipa� ng teams were representa� ve of all Dutch ALS teams, and their service areas cover all parts of the country. The median cluster size was 4 pa� ents for the interven� on teams (range 0−10 pa� ents) and 2 pa� ents for the control team (range 0−17 pa� ents).

Par� cipantsFrom March 2009 to July 2011, 132 pa� ents and 126 of their caregivers par� cipated: 71 in the interven� on group and 61 in the control group (fi gure 1). Baseline sociodemographic and clinical characteris� cs of pa� ents and caregivers did not diff er between the case management and control group (table 1) or between par� cipants and non-par� cipants (supplementary table e-1). Most caregivers were partners (n=112).

During follow-up, the propor� on of missing assessments did not diff er signifi cantly between the interven� on group (pa� ents 111/284, caregivers 100/264) and the control group (pa� ents 92/244, caregivers 98/240). Reasons for missing outcome data were pa� ent death, pa� ent withdrawal, and pa� ent request to skip an assessment. The required number of pa� ents according to the study protocol (n=130) was reached.

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Figure 1 Flow of clusters and individual par� cipants.

ALS teams

invited to participate the study

n=44

Randomization of ALS teams

n=31

No consent

n=13

Completed the trial:

Patients n=43 Caregivers n=41

Death: n=23 Patient’s death: n=22

Stop: n=5 Stop: n=3

Number of evaluations of:

Patients n=173

Caregivers n=164

Intervention:

case management + usual care

Enrolment:

Patients n=71

Caregivers n=66

Enrollment of ALS teams

Control:

usual care

Enrolment:

Patients n=61

Caregivers n=60

Completed the trial:

Patients n=36 Caregivers n=35

Death: n=15 Patient’s death: n=15

Stop: n=10 Stop: n=10

Number of evaluations of:

Patients n=152

Caregivers n=142

Analysis

Intervention teams n=16

Patients n=155

ALS patients

No consent

n=166

Baseline

Control teams n=15

Patients n=143

Allocation of ALS teams

ALS = amyotrophic lateral sclerosis.


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Table 1 Baseline characteris� cs of study par� cipants.

Pa� ent variables Interven� on (n=71)

Control (n=61)

p Value

Mean age (SD), y 63 (11) 62 (11) 0.680Male 57 64 0.786Married 83 84 0.789Primary caregiver is partner 82 84 0.474Caucasian ethnicity 100 97 0.307College or university degree 42 31 0.190Employed 7 15 0.168Limb onset 73 79 0.466Median � me since onset (Q1-Q3), y 1.8 (1.2 – 3.5) 2.2 (1.2 – 3.9) 0.600Median � me since diagnosis (Q1-Q3), y 0.9 (0.3 – 1.7) 0.9 (0.4 – 2.3) 0.277Mean predicted FVC % (SD) 86 (23) 83 (24) 0.627PEG 14 21 0.275NIV/IV 17 18 0.864Riluzole 91 95 0.398Mean ALSFRS-R (SD) 32 (8) 32 (9) 0.685Mean ALSAQ-40 Emo� onal Func� oning (SD) 21 (18) 19 (17) 0.524Mean ALSAQ-40 sum score (SD) 37 (17) 37 (18) 0.919Mean quality of care ra� ng score (SD) 8.2 (0.8) 8.3 (0.8) 0.803Caregiver variables Interven� on

(n=66)Control (n=60)

p Value

Mean age (SD), y 59 (14) 57 (15) 0.922Female 70 62 0.342Married 94 90 0.463Caucasian ethnicity 97 95 0.463College or university degree 37 27 0.197Employed 23 32 0.305Mean CSI (SD) 6 (3) 5 (3) 0.184Mean quality of care score “care for the pa� ent” (SD) 8.0 (0.9) 8.2 (1.1) 0.298Mean quality of care score “care for the caregiver” (SD) 7.6 (1.4) 7.8 (1.6) 0.436

Abbrevia� ons: ALSAQ-40 = 40-item Amyotrophic Lateral Sclerosis Assessment Ques� onnaire; ALSFRS-R = Amyotrophic Lateral Sclerosis Func� onal Ra� ng Scale-Revised; CSI = Caregiver Strain Index; FVC% = percentage of the predicted value of forced vital capacity; IV = invasive ven� la� on; NIV = noninvasive ven� la� on; PEG = percutaneous endoscopic gastrostomy; Q1-Q3 = fi rst quar� le to third quar� le; SD = standard devia� on; y = years.Values are percentages unless otherwise indicated.

Case managementThe dura� on of home visits ranged from 60 to 180 minutes. The extent to which pa� ents and their caregivers relied on case management varied widely. Par� cipants diff ered in their need for informa� on, support, treatment, resources, and care (Supplementary Figure e-1). Seventeen pa� ents (24%) and their caregivers did not raise any ques� ons for the case

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manager. Twenty-nine par� cipants contacted the case manager at least once during the interval between visits. Thirty-one caregivers and 48 pa� ents raised at least one issue during the interven� on period. Ac� ons by the case manager were mostly related to emo� onal well-being (coping with loss, emo� ons, dependence and changes, caregiver strain, and private life) and consisted of providing a listening ear for par� cipants, discussing the impact of having ALS and the impact of symptoms on daily func� oning. Prac� cal ac� ons included providing oral and wri� en informa� on, reference to health care providers, and contac� ng/informing health care providers.

Eff ec� veness of case managementDespite signifi cant disease progression, emo� onal well-being as refl ected by ALSAQ-40 EF did not change over � me. No signifi cant diff erence in ALSAQ-40 EF score was detected between the interven� on group and the usual care group (table 2 and fi gure 2).

Caregiver strain (CSI) increased signifi cantly during the study period in both groups, without diff erences between the groups.

At baseline, pa� ents as well as caregivers rated the QOC as high (table 2). The QOC scores did not change over � me, and the interven� on did not signifi cantly aff ect the QOC ra� ng score.


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Table 2 Eff ects of case management and usual care at baseline and 4, 8, and 12 months.

Outcome measures

Case managementa

Mean (SD), nUsual carea

Mean (SD), nLinear Mixed Modelβ (SE); p-value

1. ALSAQ-40 Emo� onal Func� oning (0 – 100)b

Baseline4 months8 months12 months

21.3 (18.2), 7019.8 (14.6), 4321.5 (13.5), 2822.8 (16.4), 30

19.3 (17.0), 6119.4 (16.6), 3920.0 (16.1), 2219.1 (14.7), 27

Time: 0.54 (0.58)p = 0.35Time x Group: 0.33 (0.79)p = 0.68

2. CSI (0-13)b


5.8 (3.4), 667.1 (3.4), 427.0 (3.6), 257.9 (2.9), 29

5.0 (2.9), 596.4 (3.1), 377.4 (3.5), 207.3 (3.2), 24

Time: 0.60 (0.11)p < 0.0001c

Time x Group: -0.08 (0.15)p = 0.59

3. QOC report mark pa� ents with ALS (1 – 10)d


8.2 (0.8), 658.2 (0.9), 438.1 (1.0), 287.9 (0.9), 30

8.3 (0.8), 558.0 (0.7), 378.0 (1.1), 238.0 (0.7), 25

Time: -0.06 (0.04)p = 0.13Time x Group: -0.02 (0.05)p = 0.69

4. QOC report mark caregivers: pa� ent care (1 – 10)d


8.0 (0.9), 638.3 (0.8), 408.0 (0.9), 267.9 (0.7), 27

8.2 (1.1), 598.2 (0.8), 378.1 (0.8), 187.9 (0.9), 24

Time: -0.05 (0.04)p = 0.21Time x Group: 0.02 (0.06)p = 0.21

5. QOC report mark caregivers: caregiver care (1 – 10)d


7.6 (1.4), 567.7 (1.0), 387.3 (1.6), 257.3 (1.3), 25

7.8 (1.6), 497.7 (1.2), 337.6 (1.0), 167.1 (1.0), 22

Time: -0.10 (0.06)p = 0.12Time x Group: 0.004 (0.08)p = 0.96

Disease progression: ALSFRS-R (0 – 48)d


32.1 (7.9), 7129.9 (7.0), 4327.4 (7.1), 2724.0 (9.3), 28

31.6 (8.8), 6127.6 (10.2), 3727.8 (11.0), 2025.1 (11.5), 25

Time: -2.64 (0.40)p < 0.0001c

Time x Group: -0.26 (0.54)p = 0.63

Abbrevia� ons: ALSFRS-R = Amyotrophic Lateral Sclerosis Func� onal Ra� ng Scale Revised; ALSAQ-40 = ALS Assessment Ques� onnaire-40 items; CSI = Caregiver Strain Index; QOC = Quality Of Care; SD = standard devia� on; SE = standard error.β = mul� level regression coeffi cient for the eff ect of case management compared with usual care, and is an es� ma� on of the longitudinal rela� onship between the parameter and the outcome.Time = change over � me (4 months) in outcome for both groups; Time x treatment group interac� on term = change over � me (4 months) in outcome diff erent for both groups.a Intracluster correla� on coeffi cient (ICC): All ICC values were less than zero.b Score range; lower scores indicate a be� er situa� on.c p < 0.05.d Score range; higher scores indicate a be� er situa� on.

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Figure 2 Trajectories of ALSAQ EF score for pa� ents in the usual care group and interven� on group.

The lighter lines denote individual trajectories (i.e., individual pa� ents); the thick line represents the es� mated course during 12 months. ALS-AQ EF = 40-item Amyotrophic Lateral Sclerosis Assessment Ques� onnaire, Emo� onal Func� oning domain.

DISCUSSION

This cluster randomized clinical trial on case management in mul� disciplinary ALS care demonstrated no signifi cant benefi t of case management to the emo� onal func� oning of pa� ents, the level of strain experienced by the caregiver, or the level of sa� sfac� on of pa� ents and caregivers when added to standard mul� disciplinary care compared with


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standard care alone. The design and results of our study must be viewed in the context of the Dutch ALS care se ̀ ng. Par� cipants were recruited from ALS teams, and all received standard mul� disciplinary ALS care. Previous studies demonstrated that mul� disciplinary ALS care results in a be� er QOL and sa� sfac� on with QOC than non-specialized general care 1,4,5,26. We es� mate that at least 80% of pa� ents with ALS are presently being referred to a mul� disciplinary ALS team in the Netherlands 27. To further explore the eff ect of case management models in ALS, future studies must be conducted in other health care systems.

In other diseases, eff ec� veness of case management on pa� ents’ QOL has been demonstrated 28,29, although the evidence is limited due to inconsistent fi ndings 30.

The present study aimed to improve any shortcomings in the needs of pa� ents with ALS and their caregivers. Judging from the frequency of ques� ons and issues raised during the interven� on period, the interven� on did not result in an intensive use of case management by the par� cipants. The high scores on mental QOL and sa� sfac� on with care and the modest number of issues raised by pa� ents and their caregivers suggest that problems in Dutch mul� disciplinary ALS care, as iden� fi ed in daily prac� ce, are incidental, with rela� vely low impact on mental QOL and overall sa� sfac� on with ALS care.

In our study design, we deliberately chose a case manager who was not a member of the mul� disciplinary ALS team. Pa� ents and caregivers in this way had the opportunity to consult the independent case manager for a second opinion or when the ALS team did not adequately address the needs of pa� ents/caregivers. Regular contacts between the case manager and the team were assured to avoid overlap. The case managers’ professional background in our opinion did not infl uence our study results: their broad overview of the Dutch ALS care and their extensive experience in ALS care were appropriate for their signaling, suppor� ve and advisory func� on.

We chose an ALS-specifi c health-related QOL measure to evaluate the eff ect of case management. SMiLE 31 and SEIQOL 32 measure non-health-related factors of QOL and are more focused on psychological, existen� al and support factors. We cannot exclude an eff ect on those measures. However, the lack of eff ect does not seem to be due to the scales used as we did not fi nd an eff ect on any other outcome measure.

In addi� on to the lack of eff ec� veness for pa� ents with ALS, we were unable to demonstrate any improvement in caregivers’ burden. The increased caregiver burden over � me has been described earlier 33,34. This fi nding may result from the fact that most issues raised during the interven� on period were directly aimed at pa� ents and were less o{ en issues concerning caregiver strain. This possibility is illustrated by the content of the issues raised, which was mostly related to the provision of assis� ve products and technology, restric� ons in ac� vi� es and par� cipa� on, symptom treatment, psychosocial problems, and provision of pallia� ve care, illustra� ng that the focus was more on the pa� ent than the caregiver. Our fi nding of an increasing burden for caregivers indicates the need for a more prominent role of interven� ons targe� ng caregivers in future studies.

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The strengths of this study are its randomized controlled design and the large sample size of pa� ents with ALS, which is representa� ve for the Dutch ALS popula� on as we compare pa� ent characteris� cs (with respect to age at disease onset, sex, � me to diagnosis, and site of onset) with those of a recently described popula� on-based epidemiology study 27. In addi� on, we applied mul� level analysis to account for various poten� al confounding factors.

The number of missing data could be considered a limita� on of the study, but is inherent to longitudinal studies of pa� ents with ALS 35,36. Loss to follow-up is not expected to have a substan� al eff ect on the power of the study considering the size of the mul� level regression interac� on coeffi cient for the eff ect of case management. Irrespec� ve of the number of observa� ons per par� cipant, every par� cipant was included in the mul� level analyses, and these analyses deal appropriately with the varying numbers of observa� ons 25,37. Another possible limita� on of this study is the limited dura� on of the interven� on period (12 months). Wynia et al. 9 (2010) stated that although there is no evidence, it seems reasonable to assume that an “investment eff ect” is applicable for outcome variables such as QOL. However, 12 months is rela� vely long considering that ALS care is intensive during a rela� vely short disease period compared with other progressive diseases (e.g., mul� ple sclerosis). It is credible that the quarterly home visits, combined with the possibility of contact by telephone and e-mail between visits, allowed for our interven� on period to be long enough to build trust and stability in the case manager/par� cipant rela� onship 38,39.

Our data do not support the implementa� on of case management as part of ALS care. Because health care systems vary considerably between countries and regions, as does the rate of implementa� on of mul� disciplinary ALS care, our research design facilitates future case management research projects in other countries, as well as outside the context of specialized ALS teams. Such studies will decisively determine the eff ect of diff erent case management models in ALS care.

ACKNOWLEDGEMENT

The authors thank the pa� ents; their caregivers; the referring rehabilita� on medicine consultants; Marije Bolt (Academic Medical Center Amsterdam), who was one of the case managers; Maud Maessen, PhD (University Medical Center Utrecht), who contributed to conceptualiza� on of the trial; Gerda Valk, research nurse (University Medical Center Utrecht) and Kim Bibbe, psychology student (University Medical Center Utrecht), who conducted the outcome assessments; and Tim Zandbelt (University Medical Center Utrecht) and Hermieneke Vergunst (University Medical Center Utrecht), who conducted data entry.


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STUDY FUNDING

Funded by ZonMw, the Netherlands Organiza� on for Health Research and Development, within the Programme Pallia� ve Care grant agreement no. 1150.0008, and the Netherlands ALS Founda� on.

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Supplementary Table e-1 Baseline characteris� cs of study par� cipants versus non-par� cipants.

Variables Par� cipants (n=132)

Non-par� cipants(n=166)

p Value

Mean age (SD), years 62.9 (11.2) 63.4 (11.7) 0.702Male 65 57 0.151Married 83 77 0.375Primary caregiver is partner 83 67 0.312Caucasian ethnicity 99 96 0.326College or university degree 42 30 0.190Limb onset 76 70 0.233Median � me since onset (Q1-Q3), years 2.1 (0.3 - 12.6) 2.3 (0.1 - 22.7) 0.372Median � me since diagnosis (Q1-Q3), years 0.9 (0.05 - 12.1) 1.1 (0.01 - 22.2) 0.446Mean predicted FVC % (SD) 85 (22) 81 (21) 0.325PEG use 17 19 0.680NIV/IV use 17 13 0.273Riluzole 93 90 0.326Median ALSFRS-R (Q1-Q3) 33 (26 – 39) 32 (26 – 40) 0.868

Abbrevia� ons: ALSFRS-R = Amyotrophic Lateral Sclerosis Func� onal Ra� ng Scale-Revised; FVC% = percentage of the predicted value of forced vital capacity; IV = invasive ven� la� on; NIV = noninvasive ven� la� on; PEG = percutaneous endoscopic gastrostomy; Q1-Q3 = fi rst quar� le to third quar� le; SD = standard devia� on.Values are percentages unless otherwise indicated.

Supplementary Figure e-1 Themes of issues about which par� cipants raised ques� ons.

2

3

5

7

7

9

10

10

12

13

19

19

31

35

37

38

39

Social services / voluntary work

Supporting children

Respite care

Durable power of attorney

Work

Respiratory care / NIV

Advance directives / end of life decisions

Transportation services

Domestic life services

Nutrition / PEG

Finances

Personal care services

Palliative care provision

Psychosocial

Symptoms

Activities and participation

Provision of assistive products and technology

Number of ALS patients and/or caregivers who raised the issue

Abbrevia� ons: PEG, percutaneous endoscopic gastrostomy; NIV, non-invasive ven� la� on.

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