A 16-year follow-up of walking function, fatigue, and pain in adults aged 34–65 years with spastic cerebral palsy
DOI:
https://doi.org/10.2340/jrm.v57.43295Keywords:
adults, Cerebral Palsy, Fatigue, follow-up study, Pain, walking., 6-minute walk test, expanded Timed Up and Go test, Gait Deviation Index., Gait AnalysisAbstract
Objective: This study aimed to describe the long-term changes in walking function, fatigue, and pain in adults with cerebral palsy.
Design: A 16-year follow-up study with paired comparisons.
Subjects: Adults with spastic cerebral palsy (n = 29) from a baseline study in 2008.
Methods: The mean age at follow-up was 50 (standard deviation 10) years.
Primary outcomes: Gait Deviation Index, 6 Minute Walk Test, Timed-Up-and-Go test, walking speed, Fatigue Severity Scale, and bodily pain (visual analogue scale, 0–100). Paired samples t-test (Wilcoxon signed rank test for non-parametric data) was used to assess differences between baseline and follow-up. Between-group differences were analysed using an independent samples t-test (Mann–Whitney U test for non-parametric data).
Results: Mean gait pattern deviations significantly (p-value = < 0.001) increased at follow-up compared with baseline for the full cohort. Walking speed decreased for the full cohort (–0.08 m/sec, p-value = 0.022), due to the bilateral group (–0.13 m/sec, p-value = 0.006). Walking capacity was maintained for the full cohort but decreased (mean diff: –84m, p-value = 0.035) for the bilateral group. Fatigue remained stable (p-value = 0.888). Pain decreased (p-value = 0.025) for the whole group, primarily due to the unilateral group (mean diff: 14 points on visual analogue scale, p-value = 0.031).
Conclusions: Gait pattern deviations increased for adults with cerebral palsy during this 16-year follow-up. Walking speed and capacity decreased for the bilateral group but were maintained for the unilateral group. Fatigue symptoms were high at baseline but did not change across this follow-up. Pain decreased, similar to the general population.
Downloads
References
Rosenbaum P, Paneth N, Leviton A, Goldstein M, Bax M, Damiano D, et al. A report: the definition and classification of cerebral palsy April 2006. Dev Med Child Neurol Suppl 2007; 109: 8–14.
Morgan P, McGinley JL. Cerebral palsy. Handbook of Clinical Neurology 2018; 159: 323–336.
https://doi.org/10.1016/B978-0-444-63916-5.00020-3 DOI: https://doi.org/10.1016/B978-0-444-63916-5.00020-3
Jonsson U, Eek MN, Sunnerhagen KS, Himmelmann K. Changes in walking ability, intellectual disability, and epilepsy in adults with cerebral palsy over 50 years: a population-based follow-up study. Dev Med Child Neurol 2021; 63: 839–845.
https://doi.org/10.1111/dmcn.14871 DOI: https://doi.org/10.1111/dmcn.14871
Benner JL, Noten S, Limsakul C, Van Der Slot WMA, Stam HJ, Selb M, et al. Outcomes in adults with cerebral palsy: systematic review using the International Classification of Functioning, Disability and Health. Dev Med Child Neurol 2019; 61: 1153–1161.
https://doi.org/10.1111/dmcn.14247 DOI: https://doi.org/10.1111/dmcn.14247
Boyer KA, Hayes KL, Umberger BR, Adamczyk PG, Bean JF, Brach JS, et al. Age-related changes in gait biomechanics and their impact on the metabolic cost of walking: Report from a National Institute on Aging workshop. Exp Gerontol 2023; 173: 112102.
https://doi.org/10.1016/j.exger.2023.112102 DOI: https://doi.org/10.1016/j.exger.2023.112102
Opheim A, Jahnsen R, Olsson E, Stanghelle JK. Walking function, pain, and fatigue in adults with cerebral palsy: a 7-year follow-up study. Dev Med Child Neurol 2009; 51: 381–388.
https://doi.org/10.1111/j.1469-8749.2008.03250.x DOI: https://doi.org/10.1111/j.1469-8749.2008.03250.x
Benner JL, Hilberink SR, Veenis T, Stam HJ, van der Slot WM, Roebroeck ME. Long-term deterioration of perceived health and functioning in adults with cerebral palsy. Arch Phys Med Rehabil 2017; 98: 2196–2205.
https://doi.org/10.1016/j.apmr.2017.03.013 DOI: https://doi.org/10.1016/j.apmr.2017.03.013
Ostensjø S, Carlberg EB, Vøllestad NK. Motor impairments in young children with cerebral palsy: relationship to gross motor function and everyday activities. Dev Med Child Neurol 2004; 46: 580–589.
https://doi.org/10.1111/j.1469-8749.2004.tb01021.x DOI: https://doi.org/10.1111/j.1469-8749.2004.tb01021.x
van der Slot WMA, Benner JL, Brunton L, Engel JM, Gallien P, Hilberink SR, et al. Pain in adults with cerebral palsy: a systematic review and meta-analysis of individual participant data. Ann Phys Rehabil Med 2021; 64: 101359.
https://doi.org/10.1016/j.rehab.2019.12.011 DOI: https://doi.org/10.1016/j.rehab.2019.12.011
Puce L, Pallecchi I, Chamari K, Marinelli L, Innocenti T, Pedrini R, et al. Systematic review of fatigue in individuals with cerebral palsy. Front Hum Neurosci 2021; 15: 598800.
https://doi.org/10.3389/fnhum.2021.598800 DOI: https://doi.org/10.3389/fnhum.2021.598800
Lennon N, Church C, Shrader MW, Robinson W, Henley J, Salazar-Torres JJ, et al. Mobility and gait in adults with cerebral palsy: evaluating change from adolescence. Gait Posture 2021; 90: 374–379.
https://doi.org/10.1016/j.gaitpost.2021.09.177 DOI: https://doi.org/10.1016/j.gaitpost.2021.09.177
Bonnefoy-Mazure A, De Coulon G, Lascombes P, Bregou A, Armand S. A 10.5-year follow-up of walking with unilateral spastic cerebral palsy. J Child Orthop 2023; 17: 173–183.
https://doi.org/10.1177/18632521231154975 DOI: https://doi.org/10.1177/18632521231154975
MacCarthy M, Heyn P, Tagawa A, Carollo J. Walking speed and patient-reported outcomes in young adults with cerebral palsy. Dev Med Child Neurol 2022; 64: 1281–1288.
https://doi.org/10.1111/dmcn.15225 DOI: https://doi.org/10.1111/dmcn.15225
Gravholt A, Fernandez B, Bessaguet H, Millet GY, Buizer AI, Lapole T. Motor function and gait decline in individuals with cerebral palsy during adulthood: a narrative review of potential physiological determinants. Eur J Appl Physiol 2024; 124: 2867–2879.
https://doi.org/10.1007/s00421-024-05550-y DOI: https://doi.org/10.1007/s00421-024-05550-y
Blair E, Langdon K, McIntyre S, Lawrence D, Watson L. Survival and mortality in cerebral palsy: observations to the sixth decade from a data linkage study of a total population register and National Death Index. BMC Neurol 2019; 19: 111.
https://doi.org/10.1186/s12883-019-1343-1 DOI: https://doi.org/10.1186/s12883-019-1343-1
Himmelmann K, Sundh V. Survival with cerebral palsy over five decades in western Sweden. Dev Med Child Neurol 2015; 57: 762–767.
https://doi.org/10.1111/dmcn.12718 DOI: https://doi.org/10.1111/dmcn.12718
Maanum G, Jahnsen R, Stanghelle JK, Sandvik L, Keller A. Effects of botulinum toxin A in ambulant adults with spastic cerebral palsy: a randomized double-blind placebo controlled-trial. J Rehabil Med 2011; 43: 338–347.
https://doi.org/10.2340/16501977-0672 DOI: https://doi.org/10.2340/16501977-0672
Schrack JA, Zipunnikov V, Simonsick EM, Studenski S, Ferrucci L. Rising energetic cost of walking predicts gait speed decline with aging. J Gerontol A Biol Sci Med Sci 2016; 71: 947–953.
https://doi.org/10.1093/gerona/glw002 DOI: https://doi.org/10.1093/gerona/glw002
McCormick A, Brien M, Plourde J, Wood E, Rosenbaum P, McLean J. Stability of the Gross Motor Function Classification System in adults with cerebral palsy. Dev Med Child Neurol 2007; 49: 265–269.
https://doi.org/10.1111/j.1469-8749.2007.00265.x DOI: https://doi.org/10.1111/j.1469-8749.2007.00265.x
Guyatt GH, Sullivan MJ, Thompson PJ, Fallen EL, Pugsley SO, Taylor DW, et al. The 6-minute walk: a new measure of exercise capacity in patients with chronic heart failure. Can Med Assoc J 1985; 132: 919–923.
ATS statement: guidelines for the six-minute walk test. Am J Resp Crit Care Med 2002; 166: 111–117.
https://doi.org/10.1164/ajrccm.166.1.at1102 DOI: https://doi.org/10.1164/ajrccm.166.1.at1102
Schwartz MH, Rozumalski A. The Gait Deviation Index: a new comprehensive index of gait pathology. Gait Posture 2008; 28: 351–357.
https://doi.org/10.1016/j.gaitpost.2008.05.001 DOI: https://doi.org/10.1016/j.gaitpost.2008.05.001
Maanum G, Jahnsen R, Stanghelle JK, Sandvik L, Larsen KL, Keller A. Face and construct validity of the Gait Deviation Index in adults with spastic cerebral palsy. J Rehabil Med 2012; 44: 272–275.
https://doi.org/10.2340/16501977-0930 DOI: https://doi.org/10.2340/16501977-0930
Rasmussen HM, Nielsen DB, Pedersen NW, Overgaard S, Holsgaard-Larsen A. Gait Deviation Index, Gait Profile Score and Gait Variable Score in children with spastic cerebral palsy: intra-rater reliability and agreement across two repeated sessions. Gait Posture 2015; 42: 133–137.
https://doi.org/10.1016/j.gaitpost.2015.04.019 DOI: https://doi.org/10.1016/j.gaitpost.2015.04.019
McMulkin ML, MacWilliams BA. Application of the Gillette Gait Index, Gait Deviation Index and Gait Profile Score to multiple clinical pediatric populations. Gait Posture 2015; 41: 608–612.
https://doi.org/10.1016/j.gaitpost.2015.01.005 DOI: https://doi.org/10.1016/j.gaitpost.2015.01.005
Mathias S, Nayak US, Isaacs B. Balance in elderly patients: the “get-up and go” test. Arch Phys Med Rehabil, 1986; 67: 387–389.
Podsiadlo D, Richardson S. The timed “Up & Go”: a test of basic functional mobility for frail elderly persons. J Am Geriatr Soc 1991; 39: 142–148.
https://doi.org/10.1111/j.1532-5415.1991.tb01616.x DOI: https://doi.org/10.1111/j.1532-5415.1991.tb01616.x
Krupp LB, LaRocca NG, Muir-Nash J, Steinberg AD. The Fatigue Severity Scale: application to patients with multiple sclerosis and systemic lupus erythematosus. Arch Neurol 1989; 46: 1121–1123.
https://doi.org/10.1001/archneur.1989.00520460115022 DOI: https://doi.org/10.1001/archneur.1989.00520460115022
Lerdal A, Wahl A, Rustoen T, Hanestad BR, Moum T. Fatigue in the general population: a translation and test of the psychometric properties of the Norwegian version of the fatigue severity scale. Scand J Public Health 2005; 33: 123–130.
https://doi.org/10.1080/14034940410028406 DOI: https://doi.org/10.1080/14034940410028406
Ware JE Jr, Sherbourne CD. The MOS 36-item short-form health survey (SF-36). I. Conceptual framework and item selection. Med Care 1992; 30: 473–483.
https://doi.org/10.1097/00005650-199206000-00002 DOI: https://doi.org/10.1097/00005650-199206000-00002
Hays RD, Sherbourne CD, Mazel RM. The RAND 36-Item Health Survey 1.0. Health Econ 1993; 2: 217–227.
https://doi.org/10.1002/hec.4730020305 DOI: https://doi.org/10.1002/hec.4730020305
Garratt AM, Stavem K. Measurement properties and normative data for the Norwegian SF-36: results from a general population survey. Health Qual Life Outcomes 2017; 15: 51.
https://doi.org/10.1186/s12955-017-0625-9 DOI: https://doi.org/10.1186/s12955-017-0625-9
Bohannon RW, Glenney SS. Minimal clinically important difference for change in comfortable gait speed of adults with pathology: a systematic review. J Eval Clin Pract 2014; 20: 295–300.
https://doi.org/10.1111/jep.12158 DOI: https://doi.org/10.1111/jep.12158
Correa KP, Devetak GF, Martello SK, de Almeida JC, Pauleto AC, Manffra EF. Reliability and Minimum Detectable Change of the Gait Deviation Index (GDI) in post-stroke patients. Gait Posture 2017; 53: 29–34.
https://doi.org/10.1016/j.gaitpost.2016.12.012 DOI: https://doi.org/10.1016/j.gaitpost.2016.12.012
Lundh S, Nasic S, Riad J. Fatigue, quality of life and walking ability in adults with cerebral palsy. Gait Posture 2018; 61: 1–6.
https://doi.org/10.1016/j.gaitpost.2017.12.017 DOI: https://doi.org/10.1016/j.gaitpost.2017.12.017
Svane C, Forman CR, Rasul A, Nielsen JB, Lorentzen J. Muscle contractures in adults with cerebral palsy characterized by combined ultrasound-derived echo intensity and handheld dynamometry measures. Ultrasound Med Biol 2022; 48: 694–701.
https://doi.org/10.1016/j.ultrasmedbio.2021.12.012 DOI: https://doi.org/10.1016/j.ultrasmedbio.2021.12.012
Geertsen SS, Kirk H, Lorentzen J, Jorsal M, Johansson CB, Nielsen JB. Impaired gait function in adults with cerebral palsy is associated with reduced rapid force generation and increased passive stiffness. Clin Neurophysiol 2015; 126: 2320–2329.
https://doi.org/10.1016/j.clinph.2015.02.005 DOI: https://doi.org/10.1016/j.clinph.2015.02.005
Baer HR, Thomas SP, Pan Z, Tagawa A, Carollo JJ, Heyn PC. Self-reported physical function is associated with walking speed in adults with cerebral palsy. J Pediatr Rehabil Med 2019; 12: 181–188.
https://doi.org/10.3233/PRM-180585 DOI: https://doi.org/10.3233/PRM-180585
Middleton A, Fritz SL, Lusardi M. Walking speed: the functional vital sign. J Aging Phys Act 2015; 23: 314–322.
https://doi.org/10.1123/japa.2013-0236 DOI: https://doi.org/10.1123/japa.2013-0236
Bohannon RW, Crouch R. Minimal clinically important difference for change in 6-minute walk test distance of adults with pathology: a systematic review. J Eval Clin Pract 2017; 23: 377–381.
https://doi.org/10.1111/jep.12629 DOI: https://doi.org/10.1111/jep.12629
Igarashi T, Miyata K, Tamura S, Otani T, Iizuka T, Usuda S. Minimal clinically important difference in 6-minute walk distance estimated by multiple methods in inpatients with subacute cardiovascular disease. Physiother Theory Pract 2024; 40: 1981–1989.
https://doi.org/10.1080/09593985.2023.2232014 DOI: https://doi.org/10.1080/09593985.2023.2232014
Maanum G, Jahnsen R, Frøslie KF, Larsen KL, Keller A. Walking ability and predictors of performance on the 6-minute walk test in adults with spastic cerebral palsy. Dev Med Child Neurol 2010; 52: e126–132.
https://doi.org/10.1111/j.1469-8749.2010.03614.x DOI: https://doi.org/10.1111/j.1469-8749.2010.03614.x
Gillett JG, Lichtwark GA, Boyd RN, Barber LA. Functional capacity in adults with cerebral palsy: lower limb muscle strength matters. Arch Phys Med Rehabil 2018; 99: 900–6.e1.
https://doi.org/10.1016/j.apmr.2018.01.020 DOI: https://doi.org/10.1016/j.apmr.2018.01.020
Jahnsen R, Villien L, Egeland T, Stanghelle JK, Holm I. Locomotion skills in adults with cerebral palsy. Clin Rehabil 2004; 18: 309–316.
https://doi.org/10.1191/0269215504cr735oa DOI: https://doi.org/10.1191/0269215504cr735oa
Wang B, Huang H. Effects of various exercise interventions on motor function in cerebral palsy patients: a systematic review and network meta-analysis. Neurol Sci 2024; 45: 5915–5927.
https://doi.org/10.1007/s10072-024-07741-z DOI: https://doi.org/10.1007/s10072-024-07741-z
Kear BM, Guck TP, McGaha AL. Timed Up and Go (TUG) Test: normative reference values for ages 20 to 59 years and relationships with physical and mental health risk factors. J Prim Care Community Health 2017; 8: 9–13.
https://doi.org/10.1177/2150131916659282 DOI: https://doi.org/10.1177/2150131916659282
Bohannon RW. Reference values for the timed up and go test: a descriptive meta-analysis. J Geriatr Phys Ther 2006; 29: 64–68.
https://doi.org/10.1519/00139143-200608000-00004 DOI: https://doi.org/10.1519/00139143-200608000-00004
Oude Lansink ILB, McPhee PG, Brunton LK, Gorter JW. Fatigue in adults with cerebral palsy: a three-year follow-up study. J Rehabil Med 2018; 50: 886–891.
https://doi.org/10.2340/16501977-2493 DOI: https://doi.org/10.2340/16501977-2493
Jahnsen R, Villien L, Stanghelle JK, Holm I. Fatigue in adults with cerebral palsy in Norway compared with the general population. Dev Med Child Neurol 2003; 45: 296–303.
https://doi.org/10.1111/j.1469-8749.2003.tb00399.x DOI: https://doi.org/10.1111/j.1469-8749.2003.tb00399.x
Rodby-Bousquet E, Alriksson-Schmidt A, Jarl J. Prevalence of pain and interference with daily activities and sleep in adults with cerebral palsy. Dev Med Child Neurol 2021; 63: 60–67.
https://doi.org/10.1111/dmcn.14678 DOI: https://doi.org/10.1111/dmcn.14678
Engel JM, Kartin D, Jensen MP. Pain treatment in persons with cerebral palsy: frequency and helpfulness. Am J Phys Med Rehabil/Assoc Academic Physiatrists 2002; 81: 291–296.
https://doi.org/10.1097/00002060-200204000-00009 DOI: https://doi.org/10.1097/00002060-200204000-00009
Hirsh AT, Kratz AL, Engel JM, Jensen MP. Survey results of pain treatments in adults with cerebral palsy. Am J Phys Med Rehabil/Assoc Academic Physiatrists 2011; 90: 207–216.
https://doi.org/10.1097/PHM.0b013e3182063bc9 DOI: https://doi.org/10.1097/PHM.0b013e3182063bc9
Jahnsen R, Villien L, Aamodt G, Stanghelle JK, Holm I. Musculoskeletal pain in adults with cerebral palsy compared with the general population. J Rehabil Med 2004; 36: 78–84.
https://doi.org/10.1080/16501970310018305 DOI: https://doi.org/10.1080/16501970310018305
Published
How to Cite
License
Copyright (c) 2025 Sandra L. Klund-Hansen, Arve Opheim, Terje Gjøvaag, Eivind Lundgaard, Grethe Månum, Linda Rennie
This work is licensed under a Creative Commons Attribution 4.0 International License.
All digitalized JRM contents is available freely online. The Foundation for Rehabilitation Medicine owns the copyright for all material published until volume 40 (2008), as from volume 41 (2009) authors retain copyright to their work and as from volume 49 (2017) the journal has been published Open Access, under CC-BY-NC licences (unless otherwise specified). The CC-BY-NC licenses allow third parties to copy and redistribute the material in any medium or format and to remix, transform, and build upon the material for non-commercial purposes, provided proper attribution to the original work.
From 2024, articles are published under the CC-BY licence. This license permits sharing, adapting, and using the material for any purpose, including commercial use, with the condition of providing full attribution to the original publication.
