Cluster RCT of case management on patients' quality of life and caregiver strain in ALS
Abstract
OBJECTIVES: To study the effect of case management on quality of life, caregiver strain, and perceived quality of care (QOC) in patients with amyotrophic lateral sclerosis (ALS) and their caregivers. METHODS: We conducted a multicenter cluster randomized controlled trial with the multidisciplinary ALS care team as the unit of randomization. During 12 months, patients with ALS and their caregivers received case management plus usual care or usual care alone. Outcome measures were the 40-item ALS Assessment Questionnaire (ALSAQ-40), Emotional Functioning domain (EF); the Caregiver Strain Index (CSI); and the QOC score. These measures were assessed at baseline and at 4, 8, and 12 months. RESULTS: Case 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 time (p = 0.331). CSI scores in both groups increased significantly (p < 0.0001). Patients with ALS from both groups rated their perceived QOC at baseline with a median score of 8, which did not change significantly during follow-up. CONCLUSION: Within the context of multidisciplinary ALS care teams, case management appears to confer no benefit for patients with ALS or their caregivers. CLASSIFICATION OF EVIDENCE: This study provides Class III evidence that case management in addition to multidisciplinary ALS care does not significantly improve health-related quality of life of patients with ALS.
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As the disease progresses, ALS caregivers experience heightened feelings of psychological distress and burden.
“as the disease progresses, ALS caregivers experience heightened feelings of psychological distress and burden [6–8]”
The sample size calculation was based on HADS data of informal caregivers in a previously completed study.
“The sample size calculation was based on Hospital Anxiety Depression Scale (HADS) data of informal caregivers in a previously completed study [6].”
There is a need for psychosocial interventions for caregivers, but such interventions are still lacking.
“Previous studies have indicated that there is a need for psychosocial interventions for caregivers, but such interventions are still lacking [6, 20, 21].”
Cites
Folstein, M. F., Folstein, S. E., & McHugh, P. R. (1975) · Journal of Psychiatric Research · doi:10.1016/0022-3956(75)90026-6 · Cited by 88,282 · see citing works
Cedarbaum, J. M., Stambler, N., Malta, E., Fuller, C., Hilt, D., Thurmond, B., & Nakanishi, A. (1999) · Journal of the Neurological Sciences · doi:10.1016/s0022-510x(99)00210-5 · Cited by 3,307 · see citing works
Validation of a Caregiver Strain Index
Robinson, B. C. (1983) · Journal of Gerontology · doi:10.1093/geronj/38.3.344 · Cited by 1,426 · see citing works
The EFNS Task Force on Diagnosis and Management of Amyotrophic Lateral Sclerosis:, Andersen, P. M., Abrahams, S., Borasio, G. D., de Carvalho, M., Chio, A., Van Damme, P., Hardiman, O., Kollewe, K., Morrison, K. E., et al. (2012) · European Journal of Neurology · doi:10.1111/j.1468-1331.2011.03501.x · Cited by 1,117 · see citing works
Miller, R. G., Jackson, C. E., Kasarskis, E. J., England, J. D., Forshew, D. A., Johnston, W. S. W., Kalra, S., Katz, J., Mitsumoto, H., Rosenfeld, J., et al. (2009) · Neurology · doi:10.1212/wnl.0b013e3181bc0141 · Cited by 808 · see citing works
Twisk, J. W. R. (2006) · Cambridge University Press eBooks · doi:10.1017/cbo9780511610806 · Cited by 692 · see citing works
Miller, R. G., Jackson, C. E., Kasarskis, E. J., England, J. D., Forshew, D. A., Johnston, W. S. W., Kalra, S., Katz, J., Mitsumoto, H., Rosenfeld, J., et al. (2009) · Neurology · doi:10.1212/wnl.0b013e3181bc01a4 · Cited by 677 · see citing works
Client-Centred Practice: What does it Mean and Does it Make a Difference?
Law, M., Baptiste, S., & Mills, J. (1995) · Canadian journal of occupational therapy. Revue canadienne d'ergotherapie · doi:10.1177/000841749506200504 · Cited by 441 · see citing works
Applied Multilevel Analysis: A Practical Guide for Medical Researchers
Twisk, J. W. R. (2006) · Cited by 370 · see citing works
Multidisciplinary ALS care improves quality of life in patients with ALS
den Berg, J. P. V., Kalmijn, S., Lindeman, E., Veldink, J. H., de Visser, M., van der Graaff, M. M., Wokke, J. H. J., & van den Berg, L. H. (2005) · Neurology · doi:10.1212/01.wnl.0000180717.29273.12 · Cited by 344 · see citing works
Self-citationMoore, S., Corner, J., Haviland, J., Wells, M., Salmon, E., Normand, C., Brada, M., & Smith, I. (2002) · BMJ · doi:10.1136/bmj.325.7373.1145 · Cited by 304 · see citing works
Population based epidemiology of amyotrophic lateral sclerosis using capture-recapture methodology
Huisman, M. H. B., de Jong, S. W., van Doormaal, P. T. C., Weinreich, S. S., Schelhaas, H. J., van der Kooi, A. J., de Visser, M., Veldink, J. H., & van den Berg, L. H. (2011) · Journal of Neurology Neurosurgery & Psychiatry · doi:10.1136/jnnp.2011.244939 · Cited by 266 · see citing works
Self-citationA longitudinal study on quality of life and depression in ALS patient–caregiver couples
Gauthier, A., Vignola, A., Calvo, A., Cavallo, E., Moglia, C., Sellitti, L., Mutani, R., & Chiò, A. (2007) · Neurology · doi:10.1212/01.wnl.0000257093.53430.a8 · Cited by 256 · see citing works
Howgego, I. M., Yellowlees, P., Owen, C. A., Meldrum, L., & Dark, F. L. (2003) · Australian & New Zealand Journal of Psychiatry · doi:10.1046/j.1440-1614.2003.01131.x · Cited by 251 · see citing works
Aiken, L. S., Butner, J., Lockhart, C. A., Volk-Craft, B. E., Hamilton, G., & Williams, F. G. (2006) · Journal of Palliative Medicine · doi:10.1089/jpm.2006.9.111 · Cited by 187 · see citing works
Jenkinson, C., Brennan, C. A., Fitzpatrick, R. M., Bromberg, M. B., & Swash, M. (1999) · Journal of Neurology · doi:10.1007/bf03161085 · Cited by 134 · see citing works
The effect of noninvasive ventilation on ALS patients and their caregivers
Mustfa, N., Walsh, E., Bryant, V., Lyall, R. A., Addington-Hall, J. M., Goldstein, L. H., Donaldson, N., Polkey, M. I., Moxham, J., & Leigh, P. N. (2006) · Neurology · doi:10.1212/01.wnl.0000208957.88534.11 · Cited by 130 · see citing works
Effects of Case Management for Frail Older People or Those With Chronic Illness
Oeseburg, B., Wynia, K., Middel, B. J., & Reijneveld, S. A. (2009) · Nursing Research · doi:10.1097/nnr.0b013e3181a30941 · Cited by 123 · see citing works
Assessing individual quality of life in amyotrophic lateral sclerosis
Clarke, S., Hickey, A., O’Boyle, C. A., & Hardiman, O. M. (2001) · Quality of Life Research · doi:10.1023/a:1016704906100 · Cited by 115 · see citing works
Psychological health in patients with amyotrophic lateral sclerosis
Averill, A. J., Kasarskis, E. J., & Segerstrom, S. C. (2007) · Amyotrophic Lateral Sclerosis · doi:10.1080/17482960701374643 · Cited by 110 · see citing works
Turner‐Stokes, L. F., Sykes, N. P., & Silber, E. (2008) · Clinical Medicine · doi:10.7861/clinmedicine.8-2-186 · Cited by 93 · see citing works
van den Berg, J., de Groot, I. J., Joha, B., van Haelst, J., van Gorcom, P., & Kalmijn, S. (2004) · Amyotrophic Lateral Sclerosis and Other Motor Neuron Disorders · doi:10.1080/14660820410021258 · Cited by 83 · see citing works
Case management used to optimize cancer care pathways: A systematic review
Wulff, C. N., Thygesen, M. K., Søndergaard, J., & Vedsted, P. (2008) · BMC Health Services Research · doi:10.1186/1472-6963-8-227 · Cited by 66 · see citing works
Meaning in life in patients with amyotrophic lateral sclerosis
Fegg, M. J., Kögler, M., Brandstätter, M., Jox, R. J., Anneser, J. M. H., Haarmann-Doetkotte, S., Wasner, M., & Borasio, G. D. (2010) · Amyotrophic Lateral Sclerosis · doi:10.3109/17482961003692604 · Cited by 59 · see citing works
Renders, C., Valk, G. D., de Sonnaville, J. J. J., Twisk, J., Kriegsman, D., Heine, R., van Eijk, J. V., & van der Wal, G. (2003) · Diabetic Medicine · doi:10.1046/j.1464-5491.2003.01009.x · Cited by 58 · see citing works
Integrating Case Management and Palliative Care
Meier, D. E., Thar, W. E., Jordan, A. M., Goldhirsch, S. L., Siu, A. L., & Morrison, R. S. (2004) · Journal of Palliative Medicine · doi:10.1089/109662104322737395 · Cited by 54 · see citing works
Perceptions of quality of life in people with ALS: Effects of coping and health care
Foley, G., O'Mahony, P. D., & Hardiman, O. M. (2007) · Amyotrophic Lateral Sclerosis · doi:10.1080/17482960601164532 · Cited by 48 · see citing works
Quality of life in amyotrophic lateral sclerosis/motor neuron disease: A structured review
Epton, J., Harris, R., & Jenkinson, C. (2008) · Amyotrophic Lateral Sclerosis · doi:10.1080/17482960802163721 · Cited by 47 · see citing works
Case Management Model or Case Manager Type? That is the Question
Long, M. (2002) · The Health Care Manager · doi:10.1097/00126450-200206000-00009 · Cited by 38 · see citing works
Ng, L., Talman, P. S., & Khan, F. (2011) · International Journal of Rehabilitation Research · doi:10.1097/mrr.0b013e328344ae1f · Cited by 34 · see citing works
Wynia, K., Annema, C., Nissen, H., Keyser, J. D., & Middel, B. (2010) · BMC Health Services Research · doi:10.1186/1472-6963-10-142 · Cited by 26 · see citing works
Maessen, M., Post, M. W. M., Maillé, R., Lindeman, E., Mooij, R., Veldink, J. H., & van den Berg, L. H. (2007) · Amyotrophic Lateral Sclerosis · doi:10.1080/17482960601012541 · Cited by 25 · see citing works
Self-citationCase Management and Nursing Practice
Rhéaume, A., Frisch, S. R., Smith, A., & Kennedy, C. (1994) · JONA The Journal of Nursing Administration · doi:10.1097/00005110-199403000-00009 · Cited by 20 · see citing works
Jenkinson, C., Fitzpatrick, R. M., Swash, M., & Jones, G. L. (2007) · Clinical Rehabilitation · doi:10.1177/0269215506071123 · Cited by 20 · see citing works
Nurse Case Management: Negotiating Care Together Within a Developing Relationship
Yamashita, M., Forchuk, C., & Mound, B. (2005) · Perspectives In Psychiatric Care · doi:10.1111/j.1744-6163.2005.00012.x · Cited by 18 · see citing works
Rehabilitation care for patients with ALS in the Netherlands
van den Berg, J., Kalmijn, S., Lindeman, E., Wokke, J. H. J., & van den Berg, L. H. (2003) · Amyotrophic Lateral Sclerosis and Other Motor Neuron Disorders · doi:10.1080/aml.4.3.186.190 · Cited by 14 · see citing works
Mills, M. E., & Staggers, N. (1994) · JONA The Journal of Nursing Administration · doi:10.1097/00005110-199411000-00008 · Cited by 14 · see citing works
Modeling drop-outs in amyotrophic lateral sclerosis
Messina, P., & Beghi, E. (2011) · Contemporary Clinical Trials · doi:10.1016/j.cct.2011.09.013 · Cited by 14 · see citing works
Jenkinson, C., Harris, R., & Fitzpatrick, R. (2007) · Amyotrophic Lateral Sclerosis · doi:10.1080/17482960600989343 · Cited by 11 · see citing works
Physician-assisted death: dying with dignity?
Reagan, P., Hurst, R., Cook, L., Żylicz, Z., Otlowski, M. F. A., Veldink, J. H., van den Berg, L. H., & Wokke, J. H. J. (2003) · The Lancet Neurology · doi:10.1016/s1474-4422(03)00536-2 · Cited by 11 · see citing works
Self-citation
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Rooney, J. P. K., Byrne, S., Heverin, M., Tobin, K., Dick, A., Donaghy, C. G., & Hardiman, O. M. (2014) · Journal of Neurology Neurosurgery & Psychiatry · doi:10.1136/jnnp-2014-309601 · Cited by 223 · see citing works
Amyotrophic lateral sclerosis: improving care with a multidisciplinary approach
Hogden, A., Foley, G., Henderson, R. D., James, N., & Aoun, S. M. (2017) · Journal of Multidisciplinary Healthcare · doi:10.2147/jmdh.s134992 · Cited by 187 · see citing works
Sun, Y., Ji, M., Leng, M., Li, X., Zhang, X., & Wang, Z. (2022) · International Journal of Nursing Studies · doi:10.1016/j.ijnurstu.2022.104204 · Cited by 176 · see citing works
van Groenestijn, A. C., Schröder, C. D., Visser-Meily, J. M. A., Reenen, E. T. K.-V., Veldink, J. H., & van den Berg, L. H. (2015) · Amyotrophic Lateral Sclerosis and Frontotemporal Degeneration · doi:10.3109/21678421.2015.1038276 · Cited by 54 · see citing works
Self-citationGeng, D., Ou, R., Miao, X., Zhao, L., Wei, Q., Chen, X., Liang, Y., Shang, H., & Yang, R. (2016) · Journal of Clinical Nursing · doi:10.1111/jocn.13667 · Cited by 49 · see citing works
Factors related to caregiver strain in ALS: a longitudinal study
Creemers, H., de Morée, S., Veldink, J. H., Nollet, F., van den Berg, L. H., & Beelen, A. (2016) · Journal of Neurology, Neurosurgery & Psychiatry · doi:10.1136/jnnp-2015-311651 · Cited by 52 · see citing works
Self-citationA two-year longitudinal study on strain and needs in caregivers of advanced ALS patients
Bruletti, G., Comini, L., Scalvini, S., Morini, R., Luisa, A., Paneroni, M., & Vitacca, M. (2014) · Amyotrophic Lateral Sclerosis and Frontotemporal Degeneration · doi:10.3109/21678421.2014.974616 · Cited by 44 · see citing works
Aerobic Exercise Therapy in Ambulatory Patients With ALS: A Randomized Controlled Trial
van Groenestijn, A. C., Schröder, C. D., van Eijk, R. P. A., Veldink, J. H., Reenen, E. T. K., Groothuis, J. T., Grupstra, H. F., Tepper, M., van Vliet, R. O., Visser‐Meily, J. M. A., et al. (2019) · Neurorehabilitation and neural repair · doi:10.1177/1545968319826051 · Cited by 38 · see citing works
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Hobson, E., Baird, W. O., Bradburn, M. J., Cooper, C. L., Mawson, S., Quinn, A., Shaw, P. J., Walsh, T., & McDermott, C. J. (2019) · BMJ Open · doi:10.1136/bmjopen-2018-028526 · Cited by 31 · see citing works
Bakker, M., Creemers, H., Schipper, K., Beelen, A., Grupstra, H. F., Nollet, F., & Abma, T. (2015) · Amyotrophic Lateral Sclerosis and Frontotemporal Degeneration · doi:10.3109/21678421.2014.971811 · Cited by 30 · see citing works
Self-citationCordesse, V., Sidorok, F., Schimmel, P., Holstein, J., & Meininger, V. (2015) · BMC Health Services Research · doi:10.1186/s12913-015-0810-7 · Cited by 30 · see citing works
Janssen, T., Lodder, P., de Vries, J., Lepper, C. C. H. A. V. H.-D., Gobardhan, P., Ho, G., & van der Laan, L. (2020) · BMC Geriatrics · doi:10.1186/s12877-020-01579-8 · Cited by 13 · see citing works
Recommendations to support informal carers of people living with motor neurone disease
Bergin, S., & Mockford, C. (2016) · British Journal of Community Nursing · doi:10.12968/bjcn.2016.21.10.518 · Cited by 18 · see citing works
Xu, J., Bao, H., Qi, X., Wang, J., Yin, H., Shang, C., Tan, R. L.-Y., Wu, Q., & Huang, W. (2021) · Molecular Genetics & Genomic Medicine · doi:10.1002/mgg3.1760 · Cited by 17 · see citing works
de Wit, J., Beelen, A., Drossaert, C. H. C., Kolijn, R., van den Berg, L. H., Visser-Meily, J. M. A., & Schröder, C. D. (2018) · BMC Psychology · doi:10.1186/s40359-018-0232-5 · Cited by 12 · see citing works
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Johnson, S. W., Alonso, B., Faulkner, K., Roberts, H., Monroe, B., Lehman, L. A., & Kearney, P. J. (2017) · American Journal of Occupational Therapy · doi:10.5014/ajot.2017.024828 · Cited by 12 · see citing works
Cafarella, P. A., Effing, T., & Chur-Hansen, A. (2022) · Palliative & Supportive Care · doi:10.1017/s1478951522000311 · Cited by 6 · see citing works
Rose, L., Thaventhiran, T., Hobson, E., Rogers, R. L., James, K., Chu, P., Carter, B., Faull, C., Saha, S., Lee, J. S., et al. (2023) · Trials · doi:10.1186/s13063-023-07124-3 · Cited by 6 · see citing works
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Groß, M., & Summ, O. (2019) · Nervenheilkunde · doi:10.1055/a-0876-2651 · Cited by 5 · see citing works
Rush, C. L., Lester, E. G., Berry, J. D., Brizzi, K. T., Lindenberger, E. C., Curtis, J. R., & Vranceanu, A. (2023) · Translational Behavioral Medicine · doi:10.1093/tbm/ibad024 · Cited by 5 · see citing works
Lian, L., Zheng, M., He, R., Lin, J., Chen, W., Pei, Z., & Yao, X. (2022) · BMJ Open · doi:10.1136/bmjopen-2022-066402 · Cited by 4 · see citing works
(2015) · Amyotrophic Lateral Sclerosis and Frontotemporal Degeneration · doi:10.3109/21678421.2015.1089039 · Cited by 3 · see citing works
Rush, C. L., Lester, E. G., Manglani, H. R., Woodworth, E. C., Vitolo, O. V., D, M. F. M., Berry, J. D., Brizzi, K. T., Babu, S., Lindenberger, E. C., et al. (2023) · Amyotrophic Lateral Sclerosis and Frontotemporal Degeneration · doi:10.1080/21678421.2023.2224400 · Cited by 3 · see citing works
Rush, C. L., Lyons, C., Gittle, J., Seward, M., Scalia, J., Ho, D., Babu, S., Garret, M. A., Brizzi, K. T., Berry, J. D., et al. (2024) · Journal of Pain and Symptom Management · doi:10.1016/j.jpainsymman.2024.12.010 · Cited by 2 · see citing works
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Cafarella, P., Effing, T., & Chur‐Hansen, A. (2024) · Palliative & Supportive Care · doi:10.1017/s1478951524000877 · Cited by 1 · see citing works
Okoh, C., Mayall, L. A., Makin, S. M., Chen, C., & Zarotti, N. (2025) · Brain Sciences · doi:10.3390/brainsci15020112 · Cited by 1 · see citing works
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Soofi, A. Y., Kho, M. E., Dal, B.-H. V., & Lori, L. (2017) · International Journal of Neurodegenerative Disorders · doi:10.23937/ijnd-2017/1710001
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Assalone, F., Ortega, J., Samaniego, V. C., & Caro, I. A. (2026) · Journal of Health Psychology · doi:10.1177/13591053261449653
- Authors
- Creemers, H., Veldink, J. H., Grupstra, H., Nollet, F., Beelen, A., & van den Berg, L. H.
- Affiliations
- Amsterdam UMC Location University of Amsterdam (NL)
- From the Department of Rehabilitation (H.C., H.G., F.N., A.B.), Academic Medical Center Amsterdam, Netherlands ALS Center; and Department of Neurology (J.H.V., L.H.v.d.B.), Rudolf Magnus Institute of Neuroscience, Netherlands ALS Center, Utrecht.
- Venue
- Neurology
- Published
- 2014-01-07
- Volume
- 82
- Issue
- 1
- Pages
- 23-31
- ISSN
- 0028-3878 (ISSN-L)
- 1526-632X
- OpenAlex
- W2020948737
- Cited by
- 32
- References
- 40
- Citations by year
- 2026: 1 · 2025: 1 · 2024: 2 · 2023: 3 · 2022: 4 · 2021: 1 · 2020: 2 · 2019: 3 · 2018: 1 · 2017: 4 · 2016: 2 · 2015: 6 · 2014: 2
- FWCI
- 2.12
- Type
- journal article
- Language
- en
- Open access
- closed
- Locations
- Neurology (publishedVersion)
- PubMed (publishedVersion)
- Data Archiving and Networked Services (DANS) (submittedVersion)
- Pure Amsterdam UMC (submittedVersion)
- Topics
- Health Sciences › Medicine › Neurology › Amyotrophic Lateral Sclerosis Research
- Health Sciences › Health Professions › Pharmacy › Nursing care and research
- Health Sciences › Medicine › Psychiatry and Mental health › Dementia and Cognitive Impairment Research
- Keywords
- amyotrophic lateral sclerosis, case management, caregiver burden, health-related quality of life, perceived quality of care, multidisciplinary team, Caregiver Strain Index, emotional functioning
- MeSH terms
- Aged, Amyotrophic Lateral Sclerosis, Female, Follow-Up Studies, Humans, Male, Middle Aged, Quality of Life, Surveys and Questionnaires, Cluster Analysis, Caregivers, Case Management
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