Top

Published in:

Open Access 01-12-2011 | Study protocol

Upper limb children action-observation training (UP-CAT): a randomised controlled trial in Hemiplegic Cerebral Palsy

Authors: Giuseppina Sgandurra, Adriano Ferrari, Giuseppe Cossu, Andrea Guzzetta, Laura Biagi, Michela Tosetti, Leonardo Fogassi, Giovanni Cioni

Published in: BMC Neurology | Issue 1/2011

Abstract

Background

Rehabilitation for children with hemiplegic cerebral palsy (HCP) aimed to improve function of the impaired upper limb (UL) uses a wide range of intervention programs. A new rehabilitative approach, called Action-Observation Therapy, based on the recent discovery of mirror neurons, has been used in adult stroke but not in children. The purpose of the present study is to design a randomised controlled trial (RCT) for evaluating the efficacy of Action-Observation Therapy in improving UL activity in children with HCP.

Methods/Design

The trial is designed according to CONSORT Statement. It is a randomised, evaluator-blinded, match-pair group trial. Children with HCP will be randomised within pairs to either experimental or control group. The experimental group will perform an Action-Observation Therapy, called UP-CAT (Upper Limb-Children Action-Observation Training) in which they will watch video sequences showing goal-directed actions, chosen according to children UL functional level, combined with motor training with their hemiplegic UL. The control group will perform the same tailored actions after watching computer games. A careful revision of psychometric properties of UL outcome measures for children with hemiplegia was performed. Assisting Hand Assessment was chosen as primary measure and, based on its calculation power, a sample size of 12 matched pairs was established. Moreover, Melbourne and ABILHAND-Kids were included as secondary measures. The time line of assessments will be T0 (in the week preceding the onset of the treatment), T1 and T2 (in the week after the end of the treatment and 8 weeks later, respectively). A further assessment will be performed at T3 (24 weeks after T1), to evaluate the retention of effects. In a subgroup of children enrolled in both groups functional Magnetic Resonance Imaging, exploring the mirror system and sensory-motor function, will be performed at T0, T1 and T2.

Discussion

The paper aims to describe the methodology of a RCT for evaluating the efficacy of Action-Observation Therapy in improving UL activity in children with hemiplegia. This study will be the first to test this new type of treatment in childhood. The paper presents the theoretical background, study hypotheses, outcome measures and trial methodology.

Trial Registration

NCT01016496

Available only for authorised users

Bax M, Goldstein M, Rosenbaum P, Leviton A, Paneth N, Dan B, Jacobsson B, Damiano D, Executive Committee for the Definition of Cerebral Palsy: Proposed definition and classification of cerebral palsy. Dev Med Child Neurol. 2005, 47: 571-576. 10.1017/S001216220500112X.CrossRefPubMed

Uvebrant P: Hemiplegic cerebral palsy: aetiology and outcome. Acta Paediatr Scand. 1988, 345: 1-100.CrossRef

Hagberg B, Hagberg G, Olow I, von Wendt : The changing panorama of cerebral palsy in Sweden. VII. Prevalence and origin in the birth year period 1987-90. Acta Paediatr. 1996, 85 (8): 954-960. 10.1111/j.1651-2227.1996.tb14193.x.CrossRefPubMed

Himmelmann K, Hagberg G, Beckung E, Hagberg B, Uvebrant P: The changing panorama of cerebral palsy in Sweden. IX. Prevalence and origin in the birth-year period 1995-1998. Acta Paediatr. 2005, 94 (3): 287-294.CrossRefPubMed

Cioni G, Sgandurra G, Muzzini S, Paolicelli PB, Ferrari A: Forms of hemiplegia. The spastic forms of cerebral palsy A guide to the assessment of adaptive functions. Edited by: Ferrari A Cioni G. 2009, Milan: Springer-Verlag, 331-353.

Sakzewski L, Ziviani J, Boyd R: Systematic Review and Meta-analysis of Therapeutic Management of Upper-Limb Dysfunction in Children with congenital hemiplegia. Pediatrics. 2009, 123: e1111-1122. 10.1542/peds.2008-3335.CrossRefPubMed

di Pellegrino G, Fadiga L, Fogassi L, Gallese V, Rizzolatti G: Understanding motor events: a neurophysiological study. Exp Brain Res. 1992, 91: 176-180.CrossRefPubMed

Gallese V, Fadiga L, Fogassi L, Rizzolatti G: Action recognition in the premotor cortex. Brain. 1996, 119: 593-609. 10.1093/brain/119.2.593.CrossRefPubMed

Rizzolatti G, Fadiga L, Gallese V, Fogassi L: Premotor cortex and the recognition of motor actions. Brain Res Cogn Brain Res. 1996, 3: 131-141. 10.1016/0926-6410(95)00038-0.CrossRefPubMed

10.

Rizzolatti G, Fogassi L, Gallese V: The Mirror Neuron System: A Motor-Based Mechanism for Action and Intention Understanding. The Cognitive Neuroscience IV. Edited by: Gazzaniga M. 2009, The MIT Press, Cambridge USA, 625-640.

11.

Romani M, Cesari P, Urgesi C, Facchini S, Aglioti SM: Motor facilitation of the human cortico-spinal system during observation of bio-mechanically impossible movements. Neuroimage. 2005, 26: 755-763. 10.1016/j.neuroimage.2005.02.027.CrossRefPubMed

12.

Fadiga L, Craighero L, Olivier E: Human motor cortex excitability during the perception of others' action. Curr Opin Neurobiol. 2005, 15: 213-218. 10.1016/j.conb.2005.03.013.CrossRefPubMed

13.

Rizzolatti G, Fogassi L, Gallese V: Neurophysiological mechanisms underlying the understanding and imitation of action. Nat Rev Neurosci. 2001, 2: 661-670. 10.1038/35090060.CrossRefPubMed

14.

Buccino G, Riggio L: The role of the mirror neuron system in motor learning. Kinesiology. 2006, 38: 5-15.

15.

Fabbri-Destro M, Rizzolatti G: Mirror Neurons and Mirror Systems in Monkey and Humans. Physiology. 2008, 23: 171-179. 10.1152/physiol.00004.2008.CrossRefPubMed

16.

Alexander GE, Crutcher MD: Neural representations of the target (goal) of visually guided arm movements in three motor areas of the monkey. J Neurophysiol. 1990, 64: 164-178.PubMed

17.

Alexander GE, Crutcher MD: Preparation for movement: neural representations of intended direction in three motor areas of the monkey. J Neurophysiol. 1990, 64: 133-150.PubMed

18.

Kakei S, Hoffman DS, Strick PL: Muscle and movement representations in the primary motor cortex. Science. 1999, 285: 2136-2139. 10.1126/science.285.5436.2136.CrossRefPubMed

19.

Kakei S, Hoffman DS, Strick PL: Direction of action is represented in the ventral premotor cortex. Nat Neurosci. 2001, 4: 1020-1025. 10.1038/nn726.CrossRefPubMed

20.

Umiltà MA, Escola L, Intskirveli I, Grammont F, Rochat M, Caruana F, Jezzini A, Gallese V, Rizzolatti G: When pliers become fingers in the monkey motor system. Proc Natl Acad Sci USA. 2008, 105: 2209-2213. 10.1073/pnas.0705985105.CrossRefPubMedPubMedCentral

21.

Rochat MJ, Caruana F, Jezzini A, Escola L, Intskirveli I, Grammont F, Gallese V, Rizzolatti G, Umiltà MA: Responses of mirror neurons in area F5 to hand and tool grasping observation. Exp Brain Res. 2010, 204: 605-616. 10.1007/s00221-010-2329-9.CrossRefPubMedPubMedCentral

22.

Bonini L, Rozzi S, Serventi F, Simone L, Ferrari PF, Fogassi L: Ventral premotor and inferior parietal cortices make distinct contribution to action organization and intention understanding. Cereb Cortex. 2010, 20: 1372-1385. 10.1093/cercor/bhp200.CrossRefPubMed

23.

Fogassi L, Ferrari PF, Gesierich B, Rozzi S, Chersi F, Rizzolatti G: Parietal lobe: from action organization to intention understanding. Science. 2005, 308: 662-667. 10.1126/science.1106138.CrossRefPubMed

24.

Calvo-Merino B, Glaser DE, Grèzes J, Passingham RE, Haggard P: Action observation and acquired motor skills: an fMRI study with expert dancers. Cereb Cortex. 2005, 15: 1243-1249.CrossRefPubMed

25.

Calvo-Merino B, Grèzes J, Glaser DE, Passingham RE, Haggard P: Seeing or doing? Influence of visual and motor familiarity in action observation. Curr Biol. 2006, 16: 1905-1910. 10.1016/j.cub.2006.07.065.CrossRefPubMed

26.

Cross ES, Hamilton AF, Grafton ST: Building a motor simulation de novo: observation of dance by dancers. Neuroimage. 2006, 31: 1257-1267. 10.1016/j.neuroimage.2006.01.033.CrossRefPubMedPubMedCentral

27.

Buccino G, Vogt S, Ritzi A, Fink GR, Zilles K, Freund HJ, Rizzolatti G: Neural circuits underlying imitation learning of hand actions: an event-related fMRI study. Neuron. 2004, 42: 323-334. 10.1016/S0896-6273(04)00181-3.CrossRefPubMed

28.

Vogt S, Buccino G, Wohlshläger AM, Canessa N, Shah NJ, Zilles K, Eickoff SB, Freund HJ, Rizzolatti G, Fink GR: Prefrontal involvement in imitation learning of hand actions: Effects of practice and expertise. Neuroimage. 2007, 37: 1371-1383. 10.1016/j.neuroimage.2007.07.005.CrossRefPubMed

29.

Ertelt D, Small S, Solodkin A, Dettmers C, McNamara A, Binkofski F, Buccino G: Action observation has a positive impact on rehabilitation of motor deficits after stroke. Neuroimage. 2007, 36 (Suppl 2): T164-T173.CrossRefPubMed

30.

Franceschini M, Agosti M, Cantagallo A, Sale P, Mancuso M, Buccino G: Mirror neurons:action observation treatment as a tool in stroke rehabilitation. Eur J Phys Rehabil Med. 2010, 46: 1-7.

31.

Weller C, McNeil J: CONSORT 2010 statement: updated guidelines can improve wound care. J Wound Care. 2010, 19: 347-353.CrossRefPubMed

32.

Schulz KF, Altman DG, Moher D, for the CONSORT Group: CONSORT 2010 Statement: updated guidelines for reporting parallel group randomised trials. Ann Int Med. 2010, 152 (11): 726-732.CrossRefPubMed

33.

Bax M, Goldstein M, Rosenbaum P, Leviton A, Paneth N, Dan B, Jacobsson B, Damiano D, Executive Committee for the Definition of Cerebral Palsy: Proposed definition and classification of cerebral palsy, April 2005. Dev Med Child Neurol. 2005, 47: 571-576. 10.1017/S001216220500112X.CrossRefPubMed

34.

Sanger TD, Delgado MR, Gaebler-Spira D, Hallett M, Mink JW, Task Force on Childhood Motor Disorders: Classification and definition of disorders causing hypertonia in childhood. Pediatrics. 2003, 111: e89-97. 10.1542/peds.111.1.e89.CrossRefPubMed

35.

Bohannon RW, Smith MB: Interrater reliability of a modified Ashworth scale of muscle spasticity. Phys Ther. 1987, 67: 206-207.PubMed

36.

House JH, Gwathmey FW, Fidler MO: A dynamic approach to the thumb-in-palm deformity in cerebral palsy: evaluation and results in fifty-six patients. J Bone Joint Surg. 1981, 63: 216-225.PubMed

37.

Koman LA, Williams RMM, Evans PJ, Richardson R, Naughton MJ, Passmore L, Smith BP: Quantification of upper extremity function and range of motion in children with cerebral palsy. Dev Med Child Neurol. 2008, 50: 910-917. 10.1111/j.1469-8749.2008.03098.x.CrossRefPubMed

38.

Eliasson AC, Krumlinde-Sundholm L, Shaw K, Wang C: Effects of constraint-induced movement therapy in young children with hemiplegic cerebral palsy: an adapted model. Dev Med Child Neurol. 2005, 47: 266-275. 10.1017/S0012162205000502.CrossRefPubMed

39.

Boyd R, Sakzewski L, Ziviani J, Abbott DF, Badawy R, Gilmore R, Provan K, Tournier JD, Macdonell RA, Jackson GD: INCITE: A randomised trial comparing constraint induced movement therapy and bimanual training in children with congenital hemiplegia. BMC Neurol. 2010, 10: 4-10.1186/1471-2377-10-4.CrossRefPubMedPubMedCentral

40.

Hoare BJ, Wallen MA, Imms C, Villanueva E, Rawicki HB, Carey L: Botulinum toxin A as an adjunct to treatment in the management of the upper limb in children with spastic cerebral palsy (UPDATE) [review]. Cochrane Database Syst. 2010, 1: CD003469-

41.

Arner M, Eliasson AC, Nicklasson S, Sommerstein K, Hägglund G: Hand function in cerebral palsy: report of 367 children in a population-based longitudinal health care program. J Hand Surg Am. 2008, 33: 1337-1347. 10.1016/j.jhsa.2008.02.032.CrossRefPubMed

42.

Jebsen RH, Taylor N, Trieschmann RB, Trotter MJ, Howard LA: An objective and standardised test of hand function. Arch Phys Med Rehabil. 1969, 50: 311-319.PubMed

43.

Krumlinde-Sundholm L, Eliasson AC: Development of the Assisting Hand Assessment: a Rasch-built measure intended for children with unilateral upper limb impairments. Scand J Occup Ther. 2003, 10: 16-26. 10.1080/11038120310004529.CrossRef

44.

Krumlinde-Sundholm L, Holmefur M, Kottorp A, Eliasson AC: The Assisting Hand Assessment: current evidence of validity, reliability, and responsiveness to change. Dev Med Child Neurol. 2007, 49: 259-264. 10.1111/j.1469-8749.2007.00259.x.CrossRefPubMed

45.

Holmefur M, Krumlinde-Sundholm L, Eliasson AC: Interrater and intrarater reliability of the Assisting Hand Assessment. Am J Occup Ther. 2007, 61: 79-84.CrossRefPubMed

46.

Klingels K, Jaspers E, Van de Winckel A, De Cock P, Molenaers G, Feys H: A systematic review of arm activity measures for children with hemiplegic cerebral palsy. Clin Rehabil. 2010, 24: 887-900. 10.1177/0269215510367994.CrossRefPubMed

47.

Willis JK, Morello A, Davie A, Rice JC, Bennett JT: Forced use treatment of childhood hemiparesis. Pediatrics. 2002, 110: 94-96. 10.1542/peds.110.1.94.CrossRefPubMed

48.

Charles J, Wolf S, Schneider J, Gordon A: Efficacy of a child-friendly form of constraint-induced movement therapy in hemiplegic cerebral palsy: a randomized control trial. Dev Med Child Neurol. 2006, 48: 635-642. 10.1017/S0012162206001356.CrossRefPubMed

49.

Wallen M, Ziviani J, Evans R, Novak I: Modified constraint-induced therapy for children with hemiplegic cerebral palsy: a feasibility study. Dev Neurorehabil. 2008, 11: 124-133. 10.1080/17518420701640897.CrossRefPubMed

50.

Gordon AM, Schneider JA, Chinnan A, Charles JR: Efficacy of a hand-arm bimanual intensive therapy (HABIT) in children with hemiplegic cerebral palsy: a randomized control trial. Dev Med Child Neurol. 2007, 49: 830-838. 10.1111/j.1469-8749.2007.00830.x.CrossRefPubMed

51.

Aarts PB, Jongerius PH, Geerdink YA, van Limbeek J, Geurts AC: Effectiveness of modified constraint-induced movement therapy in children with unilateral spastic cerebral palsy: a randomized controlled trial. Neurorehabil Neural Repair. 2010, 24: 509-518. 10.1177/1545968309359767.CrossRefPubMed

52.

Gilmore R, Sakzewski L, Boyd R: Upper limb activity measures for 5- to 16-year-old children with congenital hemiplegia: a systematic review. Dev Med Child Neurol. 2009, 52: 14-21.CrossRefPubMed

53.

Randall M, Johnson L, Reddihough D: The Melbourne Assessment of Unilateral Upper Limb Function: Test administration manual. 1999, Melbourne: Royal Children's Hospital

54.

Randall M, Carlin JB, Chondros P, Reddihough D: Reliability of the Melbourne Assessment of Unilateral Upper Limb Function. Dev Med Child Neurol. 2001, 43: 761-767. 10.1017/S0012162201001396.CrossRefPubMed

55.

Randall M, Imms C, Carey L: Establishing validity of a modified Melbourne Assessment for children ages 2 to 4 years. Am J Occup Ther. 2008, 62: 373-383.CrossRefPubMed

56.

Klingels K, De Cock P, Desloovere K, Huenaerts C, Molenaers G, Van Nuland I, Huysmans A, Feys H: Comparison of the Melbourne Assessment of Unilateral Upper Limb Function and the Quality of Upper Extremity Skills Test in hemiplegic CP. Dev Med Child Neurol. 2008, 50: 904-909. 10.1111/j.1469-8749.2008.03123.x.CrossRefPubMed

57.

Speth LA, Leffers P, Janssen-Potten YJ, Vles JS: Botulinum toxin A and upper limb functional skills in hemiparetic cerebral palsy: a randomised trial in children receiving intensive therapy. Dev Med Child Neurol. 2005, 47: 468-473. 10.1017/S0012162205000903.CrossRefPubMed

58.

Wallen MA, O'Flaherty SJ, Waugh MC: Functional outcomes of intramuscular botulinum toxin type A in the upper limbs of children with cerebral palsy: a phase II trial. Arch Phys Med Rehabil. 2004, 85: 192-200. 10.1016/j.apmr.2003.05.008.CrossRefPubMed

59.

Wallen MA, O'Flaherty SJ, Waugh MC: Functional outcomes of intra-muscular boutlinum toxin type A and occupational therapy in the upper limbs of children with cerebral palsy: a randomised controlled trial. Arch Phys Med Rehabil. 2007, 88: 1-10. 10.1016/j.apmr.2006.10.017.CrossRefPubMed

60.

Arnould C, Penta M, Renders A, Thonnard JL: ABILHAND-Kids: a measure of manual ability in children with cerebral palsy. Neurology. 2004, 63: 1045-1052.CrossRefPubMed

61.

Biagi L, Cioni G, Fogassi L, Guzzetta A, Tosetti M: Anterior intraparietal cortex codes complexity of observed hand movements. Brain Res Bull. 2010, 81: 434-40. 10.1016/j.brainresbull.2009.12.002.CrossRefPubMed

62.

Cioni G, Montanaro D, Tosetti M, Canapicchi R, Ghelarducci B: Reorganisation of the sensorimotor cortex after early focal brain lesion: a functional MRI study in monozygotic twins. Neuroreport. 2001, 12 (7): 1335-1340. 10.1097/00001756-200105250-00009.CrossRefPubMed

63.

Guzzetta A, Staudt M, Petacchi E, Ehlers J, Erb M, Wilke M, Krägeloh-Mann I, Cioni G: Brain representation of active and passive hand movements in children. Pediatr Res. 2007, 61 (4): 485-490. 10.1203/pdr.0b013e3180332c2e.CrossRefPubMed

64.

Guzzetta A, Bonanni P, Biagi L, Tosetti M, Montanaro D, Guerrini R, Cioni G: Reorganisation of the somatosensory system after early brain damage. Clin Neurophysiol. 2007, 118 (5): 1110-1121. 10.1016/j.clinph.2007.02.014.CrossRefPubMed

65.

Morrone MC, Guzzetta A, Tinelli F, Tosetti M, Del Viva M, Montanaro D, Burr D, Cioni G: Inversion of perceived direction of motion caused by spatial undersampling in two children with periventricular leukomalacia. J Cogn Neurosci. 2008, 20 (6): 1094-1106. 10.1162/jocn.2008.20061.CrossRefPubMed

66.

Chilosi AM, Cipriani P, Pecini C, Brizzolara D, Biagi L, Montanaro D, Tosetti M, Cioni G: Acquired focal brain lesions in childhood: effects on development and reorganization of language. Brain Lang. 2008, 106 (3): 211-225. 10.1016/j.bandl.2007.12.010.CrossRefPubMed

67.

Guzzetta A, Pecini C, Biagi L, Tosetti M, Brizzolara D, Chilosi A, Cipriani P, Petacchi E, Cioni G: Language organisation in left perinatal stroke. Neuropediatrics. 2008, 39 (3): 157-163. 10.1055/s-0028-1085465.CrossRefPubMed

68.

Talairach J, Tournoux P: Co-Planar Stereotaxic Atlas of the Human Brain. 1988, Thieme Medical, New York

69.

Boynton GM, Engel SA, Glover GH, Heeger DJ: Linear systems analysis of functional magnetic resonance imaging in human V1. J Neurosci. 1996, 16: 4207-4221.PubMed

70.

Cohen J: Statistical power analysis for the behavioral sciences. Edited by: Hillsdate, NJ. 1988, Lawrence Erlbaum, 2

The pre-publication history for this paper can be accessed here:http://www.biomedcentral.com/1471-2377/11/80/prepub

Title: Upper limb children action-observation training (UP-CAT): a randomised controlled trial in Hemiplegic Cerebral Palsy
Authors: Giuseppina Sgandurra
Adriano Ferrari
Giuseppe Cossu
Andrea Guzzetta
Laura Biagi
Michela Tosetti
Leonardo Fogassi
Giovanni Cioni
Publication date: 01-12-2011
Publisher: BioMed Central
Published in: BMC Neurology / Issue 1/2011
Electronic ISSN: 1471-2377
DOI: https://doi.org/10.1186/1471-2377-11-80

Keynote webinar | Spotlight on medication adherence

Springer Medicine

Upper limb children action-observation training (UP-CAT): a randomised controlled trial in Hemiplegic Cerebral Palsy

Abstract

Background

Methods/Design

Discussion

Trial Registration

Keynote webinar | Spotlight on medication adherence

Springer Medicine

Abstract

Background

Methods/Design

Discussion

Trial Registration

Please log in to get access to this content

Other articles of this Issue 1/2011

Anti-N-methyl-D-aspartate receptor encephalitis with serum anti-thyroid antibodies and IgM antibodies against Epstein-Barr virus viral capsid antigen: a case report and one year follow-up

SMARCB1/INI1 germline mutations contribute to 10% of sporadic schwannomatosis

Chronic widespread musculoskeletal pain, fatigue, depression and disordered sleep in chronic post-SARS syndrome; a case-controlled study

Selective frontal neurodegeneration of the inferior fronto-occipital fasciculus in progressive supranuclear palsy (PSP) demonstrated by diffusion tensor tractography

Risk factors and prognosis of young stroke. The FUTURE study: A prospective cohort study. Study rationale and protocol

Long-term prognosis of symptomatic isolated middle cerebral artery disease in Korean stroke patients