Study protocol of a randomized controlled trial of home-based computerized executive function training for children with cerebral palsy

María García-Galant, Montse Blasco, Lee Reid, Kerstin Pannek, David Leiva, Olga Laporta-Hoyos, Júlia Ballester-Plané, Júlia Miralbell, Xavi Caldú, Xènia Alonso, Esther Toro-Tamargo, Mar Meléndez-Plumed, Francisca Gimeno, Marc Coronas, Emili Soro-Camats, Roslyn Boyd, Roser Pueyo, María García-Galant, Montse Blasco, Lee Reid, Kerstin Pannek, David Leiva, Olga Laporta-Hoyos, Júlia Ballester-Plané, Júlia Miralbell, Xavi Caldú, Xènia Alonso, Esther Toro-Tamargo, Mar Meléndez-Plumed, Francisca Gimeno, Marc Coronas, Emili Soro-Camats, Roslyn Boyd, Roser Pueyo

Abstract

Background: Cerebral palsy (CP) is frequently associated with specific cognitive impairments, such as executive dysfunction which are related to participation and quality of life (QOL). The proposed study will examine whether a computerized executive function (EF) training programme could provide superior benefits for executive functioning, participation, QOL and brain plasticity, as compared to usual care.

Methods: A single-blind randomized controlled trial (RCT) design will be performed. Thirty children with CP aged 8 to 12 years will participate in a home-based computerized multi-modal executive training programme (12 weeks, 5 days a week, 30 min a day training, total dose = 30 h). Thirty children with CP matched by age, sex, motor and intelligence quotient (IQ) will compose the waitlist group. Cognitive, behavioural, emotional, participation and QOL measures will be obtained at three time points: before, immediately after and 9 months after completing the training. Additionally, structural and functional (resting state) magnetic resonance images (MRI) will be obtained in a subsample of 15 children from each group. Outcomes between groups will be compared following standard principles for RCTs.

Discussion: The study will test whether the cognitive training programme exerts a positive effect not only on neuropsychological and daily functioning of children with CP but also on other measures such as participation and QOL. We will also use brain MRI to test brain functional and structural changes after the intervention. If this on-line and home-based training programme proves effective, it could be a cost-effective intervention with short- and long-term effects on EF, participation or QOL in CP.

Trial registration: ClinicalTrials.gov: NCT04025749. Registered 19 July 2019. Retrospectively registered.

Keywords: Cerebral palsy; Cognitive training; Computerized therapy; Executive functions; Neuroimaging; Participation; Quality of life.

Conflict of interest statement

The authors declare that they have no competing interests.

Figures

Fig. 1
Fig. 1
D-KEFS: Delis-Kaplan Executive Function System; MRI: Magnetic Resonance Imaging; NEPSY-II: A Developmental NEuroPSYchological Assessment-II; TOMAL: Test of Memory and Learning; WISC-V: Wechsler Intelligence Scale for Children, Fifth Edition; WNV: Wechsler Nonverbal Scale of Ability

References

    1. 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. 2007;49(s109):8–14.
    1. Oskoui M, Coutinho F, Dykeman J, Jetté N, Pringsheim T. An update on the prevalence of cerebral palsy: a systematic review and meta-analysis. Dev Med Child Neurol. 2013;55(6):509–519. doi: 10.1111/dmcn.12080.
    1. Gosling AS. Recent advances in the neuroimaging and neuropsychology of cerebral palsy. Appl Neuropsychol Child. 2017;6(1):55–63. doi: 10.1080/21622965.2015.1074914.
    1. Colver A, Rapp M, Eisemann N, Ehlinger V, Thyen U, Dickinson HO, et al. Self-reported quality of life of adolescents with cerebral palsy: a cross-sectional and longitudinal analysis. Lancet. 2015;385(9969):705–716. doi: 10.1016/S0140-6736(14)61229-0.
    1. Straub K, Obrzut JE. Effects of cerebral palsy on neuropsychological function. J Dev Phys Disabil. 2009;21:153. doi: 10.1007/s10882-009-9130-3.
    1. Stadskleiv K, Jahnsen R, Andersen GL, von Tetzchner S. Neuropsychological profiles of children with cerebral palsy. Dev Neurorehabil. 2018;21(2):108–120. doi: 10.1080/17518423.2017.1282054.
    1. Ego A, Lidzba K, Brovedani P, Belmonti V, Gonzalez-Monge S, Boudia B, et al. Visual- perceptual impairment in children with cerebral palsy: a systematic review. Dev Med Child Neurol. 2015;57(s2):46–51. doi: 10.1111/dmcn.12687.
    1. Pirila Silja, van der Meere Jaap J., Rantanen Kati, Jokiluoma Maria, Eriksson Kai. Executive Functions in Youth With Spastic Cerebral Palsy. Journal of Child Neurology. 2011;26(7):817–821. doi: 10.1177/0883073810392584.
    1. Bottcher L. Children with spastic cerebral palsy, their cognitive functioning, and social participation: a review. Child Neuropsychol. 2010;16(3):209–228. doi: 10.1080/09297040903559630.
    1. Laporta-Hoyos O, Ballester-Plané J, Póo P, Macaya A, Meléndez-Plumed M, Vázquez E, et al. Proxy-reported quality of life in adolescents and adults with dyskinetic cerebral palsy is associated with executive functions and cortical thickness. Qual Life Res. 2017;26(5):1209–1222. doi: 10.1007/s11136-016-1433-0.
    1. Novak I, Mcintyre S, Morgan C, Campbell L, Dark L, Morton N, et al. A systematic review of interventions for children with cerebral palsy: state of the evidence. Dev Med Child Neurol. 2013;55(10):885–910. doi: 10.1111/dmcn.12246.
    1. Simons DJ, Boot WR, Charness N, Gathercole SE, Chabris CF, Hambrick DZ, et al. Do “brain-training” programs work? Psychol Sci Public Interest. 2016;17(3):103–186. doi: 10.1177/1529100616661983.
    1. Conklin HM, Ogg RJ, Ashford JM, Scoggins MA, Zou P, Clark KN, et al. Computerized cognitive training for amelioration of cognitive late effects among childhood cancer survivors: a randomized controlled trial. J Clin Oncol. 2015;33(33):3894–3902. doi: 10.1200/JCO.2015.61.6672.
    1. Daley D, Van Der Oord S, Ferrin M, Danckaerts M, Doepfner M, Cortese S, et al. Behavioral interventions in attention-deficit/hyperactivity disorder: a meta-analysis of randomized controlled trials across multiple outcome domains. J Am Acad Child Adolesc Psychiatry. 2014;53(8):835–847. doi: 10.1016/j.jaac.2014.05.013.
    1. Davis JC, Marra CA, Najafzadeh M, Liu-Ambrose T. The independent contribution of executive functions to health related quality of life in older women. BMC Geriatr. 2010;10:16. doi: 10.1186/1471-2318-10-16.
    1. Eve M, O’Keeffe F, Jhuty S, Ganesan V, Brown G, Murphy T. Computerized working- memory training for children following arterial ischemic stroke: a pilot study with long-term follow-up. Appl Neuropsychol Child. 2016;5(4):273–282. doi: 10.1080/21622965.2015.1055563.
    1. Hardy KK, Willard VW, Allen TM, Bonner MJ. Working memory training in survivors of pediatric cancer: a randomized pilot study. Psychooncology. 2013;22(8):1856–1865. doi: 10.1002/pon.3222.
    1. McBride RL, Horsfield S, Sandler CX, Cassar J, Casson S, Cvejic E, et al. Cognitive remediation training improves performance in patients with chronic fatigue syndrome. Psychiatry Res. 2017;257:400–405. doi: 10.1016/j.psychres.2017.08.035.
    1. Best MW, Gale D, Tran T, Haque MK, Bowie CR. Brief executive function training for individuals with severe mental illness: effects on EEG synchronization and executive functioning. Schizophr Res. 2019;203:32–40. doi: 10.1016/j.schres.2017.08.052.
    1. Damholdt MF, Mehlsen M, O'Toole MS, Andreasen RK, Pedersen AD, Zachariae R. Web-based cognitive training for breast cancer survivors with cognitive complaints-a randomized controlled trial. Psycho-Oncology. 2016;25(11):1293–1300. doi: 10.1002/pon.4058.
    1. De Giglio L, De Luca F, Prosperini L, Borriello G, Bianchi V, Pantano P, et al. A low-cost cognitive rehabilitation with a commercial video game improves sustained attention and executive functions in multiple sclerosis: a pilot study. Neurorehabil Neural Repair. 2015;29(5):453–461. doi: 10.1177/1545968314554623.
    1. Eggenberger P, Wolf M, Schumann M, de Bruin ED. Exergame and balance training modulate prefrontal brain activity during walking and enhance executive function in older adults. Front Aging Neurosci. 2016;8:66. doi: 10.3389/fnagi.2016.00066.
    1. Diamond A. Executive Functions. Annu Rev Psychol. 2013;64:135–168. doi: 10.1146/annurev-psych-113011-143750.
    1. Løhaugen GC, Beneventi H, Andersen GL, Sundberg C, Østgård HF, Bakkan E, et al. Do children with cerebral palsy benefit from computerized working memory training? Study protocol for a randomized controlled trial. Trials. 2014;15:269. doi: 10.1186/1745-6215-15-269.
    1. Mak C, Whittingham K, Ross C, Boyd RN. Effect of mindfulness yoga programme MiYoga on attention, behaviour, and physical outcomes in cerebral palsy: a randomized controlled trial. Dev Med Child Neurol. 2018;60(9):922–932. doi: 10.1111/dmcn.13923.
    1. Cortese S, Ferrin M, Brandeis D, Buitelaar J, Daley D, Dittmann RW, et al. Cognitive training for attention-deficit/hyperactivity disorder: meta-analysis of clinical and neuropsychological outcomes from randomized controlled trials. J Am Acad Child Adolesc Psychiatry. 2015;54(3):164–174. doi: 10.1016/j.jaac.2014.12.010.
    1. Hardy JL, Nelson RA, Thomason ME, Sternberg DA, Katovich K, Farzin F, et al. Enhancing Cognitive Abilities with Comprehensive Training: A Large, Online, Randomized, Active-Controlled Trial. PLoS One. 2015;10(9):e0134467. doi: 10.1371/journal.pone.0134467.
    1. Piovesana AM, Ross S, Whittingham K, Ware RS, Boyd RN. Stability of executive functioning measures in 8-17-year-old children with unilateral cerebral palsy. Clin Neuropsychol. 2015;29(1):133–149. doi: 10.1080/13854046.2014.999125.
    1. Boyd Roslyn N, Mitchell Louise E, James Sarah T, Ziviani Jenny, Sakzewski Leanne, Smith Anthony, Rose Stephen, Cunnington Ross, Whittingham Koa, Ware Robert S, Comans Tracey A, Scuffham Paul A. Move it to improve it (Mitii): study protocol of a randomised controlled trial of a novel web-based multimodal training program for children and adolescents with cerebral palsy. BMJ Open. 2013;3(4):e002853. doi: 10.1136/bmjopen-2013-002853.
    1. Bilde PE, Kliim-Due M, Rasmussen B, Petersen LZ, Petersen TH, Nielsen JB. Individualized, home-based interactive training of cerebral palsy children delivered through the internet. BMC Neurol. 2011;11:32. doi: 10.1186/1471-2377-11-32.
    1. Reid LB, Rose SE, Boyd RN. Rehabilitation and neuroplasticity in children with unilateral cerebral palsy. Nat Rev Neurol. 2015;11(7):390–400. doi: 10.1038/nrneurol.2015.97.
    1. Manning KY, Fehlings D, Mesterman R, Gorter JW, Switzer L, Campbell C, et al. Resting state and diffusion neuroimaging predictors of clinical improvements following constraint-induced movement therapy in children with hemiplegic cerebral palsy. J Child Neurol. 2015;30(11):1507–1514. doi: 10.1177/0883073815572686.
    1. Inguaggiato E, Sgandurra G, Perazza S, Guzzetta A, Cioni G. Brain reorganization following intervention in children with congenital hemiplegia: a systematic review. Neural Plast. 2013;2013:356275. doi: 10.1155/2013/356275.
    1. Horowitz-Kraus T, Toro-Serey C, Difrancesco M. Increased resting-state functional connectivity in the cingulo-opercular cognitive-control network after intervention in children with reading difficulties. PLoS One. 2015;10(7):e0133762. doi: 10.1371/journal.pone.0133762.
    1. Astle DE, Barnes JJ, Baker K, Colclough GL, Woolrich MW. Cognitive training enhances intrinsic brain connectivity in childhood. J Neurosci. 2015;35(16):6277–6283. doi: 10.1523/JNEUROSCI.4517-14.2015.
    1. Chen N, Cai P, Zhou T, Thompson B, Fang F. Perceptual learning modifies the functional specializations of visual cortical areas. Proc Natl Acad Sci U S A. 2016;113(20):5724–5729. doi: 10.1073/pnas.1524160113.
    1. Suzuki T, Takeuchi S, Yoshinaga H. Dislocation dynamics and plasticity. Springer series in materials science v. 12. Berlin: Springer; 1991.
    1. Frantzidis C, Moraitou D, Ladas AK, Anderson-Hanley C, Barcelos NM, Zimmerman EA, et al. The Aerobic and Cognitive Exercise Study (ACES) for Community-Dwelling Older Adults With or At-Risk for Mild Cognitive Impairment (MCI): Neuropsychological, Neurobiological and Neuroimaging Outcomes of a Randomized Clinical Trial. 2019;1:76.
    1. Eliasson A-C, Krumlinde-Sundholm L, Rösblad B, Beckung E, Arner M, Öhrvall A-M, et al. The manual ability classification system (MACS) for children with cerebral palsy: scale development and evidence of validity and reliability. Dev Med Child Neurol. 2006;48(7):549–554. doi: 10.1017/S0012162206001162.
    1. Toronto AS. Screening test of Spanish grammar. Illinois: Northwestern University Press; 1973.
    1. Sabbadini M, Bonanni R, Carlesimo GA, Caltagirone C. Neuropsychological assessment of patients with severe neuromotor and verbal disabilities. J Intellect Disabil Res. 2001;45(2):169–179. doi: 10.1046/j.1365-2788.2001.00301.x.
    1. Ballester-Plané J, Laporta-Hoyos O, Macaya A, Póo P, Meléndez-Plumed M, Vázquez É, et al. Measuring intellectual ability in cerebral palsy: the comparison of three tests and their neuroimaging correlates. Res Dev Disabil. 2016;56:83–98. doi: 10.1016/j.ridd.2016.04.009.
    1. Mclean B, Blakeman M, Carey L, Ward R, Novak I, Valentine J, et al. Discovering the sense of touch: protocol for a randomised controlled trial examining the efficacy of a somatosensory discrimination intervention for children with hemiplegic cerebral palsy. BMC Pediatr. 2018;18(1):252. doi: 10.1186/s12887-018-1217-5.
    1. PREBO-6: Prediction of childhood Brain Outcomes in infants born preterm using neonatal MRI and concurrent clinical biomarkers [Internet]. ANZCTR. [cited 18 Sep 2019]. Available from:
    1. Boyd RN, Davies PS, Ziviani J, Trost S, Barber L, Ware R, et al. PREDICT-CP: study protocol of implementation of comprehensive surveillance to predict outcomes for school-aged children with cerebral palsy. BMJ Open. 2017;7(7):e014950. doi: 10.1136/bmjopen-2016-014950.
    1. Sakzewski L, Bleyenheuft Y, Boyd RN, Novak I, Elliott C, Reedman S, et al. Protocol for a multisite randomised trial of hand–arm bimanual intensive training including lower extremity training for children with bilateral cerebral palsy: HABIT-ILE Australia. BMJ Open. 2019;9(9):e032194. doi: 10.1136/bmjopen-2019-032194.
    1. Reid LB, Pagnozzi AM, Fiori S, Boyd RN, Dowson N, Rose SE. Measuring neuroplasticity associated with cerebral palsy rehabilitation: an MRI based power analysis. Int J Dev Neurosci. 2017;58:17–25. doi: 10.1016/j.ijdevneu.2017.01.010.
    1. García-Galant M, Cañas A, Blasco M, Laporta-Hoyos O, Ballester-Plané J, Caldú X, Miralbell J, Leiva D, Toro-Tamargo E, Alonso X, Meléndez-Plumed M, Gimeno F, Pannek K, Boyd NR, Pueyo R. Study protocol of a randomised controlled trial of online executive function training for children with cerebral palsy. Dev Med Child Neurol. 2018;60(s2):38.

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