Hyperkinetic movement disorders (HMD) are a heterogeneous group of neurological conditions among which dystonia is the predominant disorder and dyskinetic cerebral palsy the largest secondary dystonia group. Currently, there are no evidence-based, non-medical management options for childhood HMD. The Cognitive Orientation to daily Occupational Performance (CO-OP) Approach is a task-oriented, performance-based intervention that focuses on participation.
Purpose.
This paper reports the protocol for a proof-of-concept study to assess feasibility and preliminary evidence regarding efficacy of CO-OP for HMD following deep brain stimulation (DBS).
Method.
A series of N-of-1 trials with replications will be conducted with children, ages 6 and 21 years with HMD and DBS as indicated by the Manual Ability Classification System. Ten individualized CO-OP sessions, with multiple baselines before, during, and after, will be completed. The primary outcome measures are the Performance Quality Rating Scale and the Assessment of Motor and Process Skills. Outcome data will be plotted over time for each participant and supplemented with graph statistical analysis and effect size estimates.
Implications.
A written protocol will be developed based on evidence and feedback incorporating any changes to the CO-OP intervention for children and young people with HMD, as per the Medical Research Council’s Framework for Complex Interventions.
BarlowD.NockM.HersenM. (2009). Single case experimental designs: Strategies for studying behavior change (3rd ed.). Boston, MA: Pearson.
3.
BurkeR. E.FahnS.MarsdenD.BressmanS.MoskowitzC.FriedmanJ. (1985). Validity and reliability of a rating scale for the primary torsion dystonias. Neurology, 35, 73–77.
4.
CameronD.CraigT.EdwardsB.MissiunaC.SchwellnusH.PolatajkoH. J. (2017). Cognitive Orientation to daily Occupational Performance (CO-OP): A new approach for children with cerebral palsy. Physical and Occupational Therapy in Pediatrics, 37, 183–198. doi:10.1080/01942638.2016.1185500
5.
CloudL. J.JinnahH. A. (2010). Treatment strategies for dystonia. Expert Opinion on Pharmacotherapy, 11, 5–15. doi:10.1517/14656560903426171
CraigP.DieppeP.MacintyreS.MichieS.NazarethI.PetticrewM. (2008). Developing and evaluating complex interventions: The new Medical Research Council guidance. British Medical Journal, 337, a1655. doi:10.1136/bmj.a1655
8.
EliassonA. C.Krumlinde-SundholmL.RosbladB.BeckungE.ArnerM.OhrvallA. M.RosenbaumP. (2006). The Manual Ability Classification System (MACS) for children with cerebral palsy: Scale development and evidence of validity and reliability. Developmental Medicine & Child Neurology, 48, 549–554. doi:10.1017/S0012162206001162
9.
ElzeM. C.GimenoH.TustinK.BakerL.LumsdenD. E.HuttonJ. L.LinJ. P. (2016). Burke-Fahn-Marsden dystonia severity, gross motor, manual ability, and communication function classification scales in childhood hyperkinetic movement disorders including cerebral palsy: A “Rosetta Stone” study. Developmental Medicine & Child Neurology, 58, 145–153. doi:10.1111/dmcn.12965
10.
FioriS.CioniG.KlingelsK.OrtibusE.Van GestelL.RoseS.…GuzzettaA. (2014). Reliability of a novel, semi-quantitative scale for classification of structural brain magnetic resonance imaging in children with cerebral palsy. Developmental Medicine & Child Neurology, 56, 839–845. doi:10.1111/dmcn.12457
11.
FisherA. G. (2003). Assessment of Motor and Process Skills: Development, standardization and administration manual (Vol. 1). Fort Collins, CO: Three Star Press.
12.
GainsboroughM.SurmanG.MaestriG.ColverA.CansC. (2008). Validity and reliability of the guidelines of the Surveillance of Cerebral Palsy in Europe for the classification of cerebral palsy. Developmental Medicine & Child Neurology, 50, 828–831. doi:10.1111/j.1469-8749.2008.03141.x
13.
GimenoH.GordonA.TustinK.LinJ. P. (2013). Functional priorities in daily life for children and young people with dystonic movement disorders and their families. European Journal Paediatric Neurology, 17, 161–168. doi:10.1016/j.ejpn.2012.07.007
14.
GimenoH.LinJ.-P. (2017). The International Classification of Functioning (ICF) to evaluate deep brain stimulation neuromodulation in childhood dystonia-hyperkinesia informs future clinical and research priorities in a multidisciplinary model of care. European Journal Paediatric Neurology, 21, 147–167. doi:10.1016/j.ejpn.2016.08.016
15.
GimenoH.PolatajkoH. J.CorneliusV.LinJ.-P.BrownR. G. (2018). Protocol for N-of-1 trials with replications across therapists for childhood-onset dystonia rehabilitation: Study 2. Canadian Journal of Occupational Therapy, 85, 256–261. doi:10.1177/0008417417707734
16.
GioiaG. A.IsquithP. K.GuyS. C.KenworthyL. (2000). Behavior Rating Inventory of Executive Function (BRIEF) manual. Lutz, FL: Psychological Assessment Resources.
HaleyS. M.CosterW. J.DumasH. M.Fragala-PinkhamM. A.KramerJ.NiP.…LudlowL. H. (2011). Accuracy and precision of the Pediatric Evaluation of Disability Inventory computer-adaptive tests (PEDI-CAT). Developmental Medicine & Child Neurology, 53, 1100–1106. doi:10.1111/j.1469-8749.2011.04107.x
19.
HarrisonP.OaklandT. (2008). Adaptive Behavior Assessment System (2nd ed.). New York, NY: Elsevier.
20.
HenshawE.PolatajkoH.McEwenS.RyanJ. D.BaumC. M. (2011). Cognitive approach to improving participation after stroke: Two case studies. American Journal of Occupational Therapy, 65, 55–63. doi:10.5014/ajot.2011.09010
21.
HerdmanM.GudexC.LloydA.JanssenM.KindP.ParkinD.…BadiaX. (2011). Development and preliminary testing of the new five-level version of EQ-5D (EQ-5D-5 L). Qualitative Life Research, 20, 1727–1736. doi:10.1007/s11136-011-9903-x
22.
HimmelmannK.McManusV.HagbergG.UvebrantP.Krageloh-MannI.CansC. (2009). Dyskinetic cerebral palsy in Europe: Trends in prevalence and severity. Archives of Disease in Childhood, 94, 921–926. doi:10.1136/adc.2008.144014
23.
HoareB.ImmsC.RawickiB.CareyL. (2010). Modified constraint-induced movement therapy or bimanual occupational therapy following injection of Botulinum toxin-A to improve bimanual performance in young children with hemiplegic cerebral palsy: A randomised controlled trial methods paper. BMC Neurology, 10, Article 58. doi:10.1186/1471-2377-10-58
24.
JackmanM.NovakI.LanninN. (2014). Effectiveness of hand splints in children with cerebral palsy: A systematic review with meta-analysis. Developmental Medicine & Child Neurology, 56, 138–147. doi:10.1111/dmcn.12205
25.
JackmanM.NovakI.LanninN.FroudeE. (2017). Parents’ experience of undertaking an intensive Cognitive Orientation to daily Occupational Performance (CO-OP) group for children with cerebral palsy. Disability and Rehabilitation, 39, 1018–1024. doi:10.1080/09638288.2016.1179350
26.
JankovicJ. (2006). Treatment of dystonia. Lancet Neurology, 5, 864–872. doi:10.1016/S1474-4422(06)70574-9
JankovicJ. (2013). Medical treatment of dystonia. Movement Disorders, 28, 1001–1012. doi:10.1002/mds.25552
29.
KazdinA. E. (2011). Single-case research designs: Methods for clinical and applied settings (2nd ed.). New York, NY: Oxford University Press.
30.
KiresukT. J.SmithA.CardilloJ. E. (Eds.). (1994). Goal Attainment Scaling: Applications, theory, and measurement. Hillsdale, NJ: Lawrence Erlbaum.
31.
KoyA.LinJ.-P.SangerT. D.MarksW. A.MinkJ. W.TimmermannL. (2016). Advances in management of movement disorders in children. Lancet Neurology, 15, 719–735. doi:10.1016/s1474-4422(16)00132-0
32.
KratochwillT. R.HitchcockJ. H.HornerR. H.LevinJ. R.OdomS. L.RindskopfD. M.ShadishW. R. (2012). Single-case intervention research design standards. Remedial and Special Education, 34(1), 26–38. doi:10.1177/0741932512452794
33.
KratochwillT. R.LevinJ. R. (2010). Enhancing the scientific credibility of single-case intervention research: Randomization to the rescue. Psychological Methods, 15, 124–144. doi:10.1037/a0017736
34.
LancasterG. A. (2015). Pilot and feasibility studies come of age!Pilot and Feasibility Studies, 1, Article 1. doi: doi:10.1186/2055-5784-1-1
35.
LawM.BaptisteS.McCollM. A.OpzoomerA.PolatajkoH.PollockN. (2014). Canadian Occupational Performance Measure (5th ed.). Ottawa, ON: CAOT Publications ACE.
36.
LinJ. P.LumsdenD. E.GimenoH.KaminskaM. (2014). The impact and prognosis for dystonia in childhood including dystonic cerebral palsy: A clinical and demographic tertiary cohort study. Journal of Neurology, Neurosurgery, and Psychiatry, 85, 1239–1244. doi:10.1136/jnnp-2013-307041
37.
LumsdenD. E.GimenoH.TustinK.KaminskaM.LinJ.-P. (2015). Interventional studies in childhood dystonia do not address the concerns of children and their carers. European Journal of Paediatric Neurology, 19, 327–336. doi:10.1016/j.ejpn.2015.01.003
38.
LumsdenD. E.KaminskaM.TomlinS.LinJ.-P. (2016). Medication use in childhood dystonia. European Journal of Paediatric Neurology, 20, 625–629. doi:10.1016/j.ejpn.2016.02.003
39.
MajnemerA.ShevellM.HallN.PoulinC.LawM. (2010). Developmental and functional abilities in children with cerebral palsy as related to pattern and level of motor function. Journal of Child Neurology, 25, 1236–1241. doi:10.1177/0883073810363175
40.
MartiniR.RiosJ.PolatajkoH.WolfT.McEwenS. (2015). The Performance Quality Rating Scale (PQRS): Reliability, convergent validity, and internal responsiveness for two scoring systems. Disability and Rehabilitation, 37, 231–238. doi:10.3109/09638288.2014.913702
41.
McEwenS.PolatajkoH.BaumC.RiosJ.CironeD.DohertyM.WolfT. (2015). Combined cognitive-strategy and task-specific training improve transfer to untrained activities in subacute stroke: An exploratory randomized controlled trial. Neurorehabilitation and Neural Repair, 29, 526–536. doi:10.1177/1545968314558602
42.
McEwenS. E.PolatajkoH. J.HuijbregtsM. P.RyanJ. D. (2010). Inter-task transfer of meaningful, functional skills following a cognitive-based treatment: Results of three multiple baseline design experiments in adults with chronic stroke. Neuropsychological Rehabilitation, 20, 541–561. doi:10.1080/09602011003638194
MonbaliuE.OrtibusE. L. S.de CatJ. O. S.DanB.HeyrmanL.PrinzieP.…FeysH (2012). The Dyskinesia Impairment Scale: A new instrument to measure dystonia and choreoathetosis in dyskinetic cerebral palsy. Developmental Medicine & Child Neurology, 54, 278–283. doi:10.1111/j.1469-8749.2011.04209.x
45.
National Institute for Clinical Excellence. (2012). Spasticity in children and young people with non-progressive brain disorders: Management of spasticity and co-existing motor disorders and their early musculoskeletal complications. London, UK: Author.
46.
NorrisG.TateR. L. (2000). The Behavioural Assessment of the Dysexecutive Syndrome (BADS): Ecological, concurrent and construct validity. Neuropsychological Rehabilitation, 10, 33–45. doi:10.1080/096020100389282
47.
NourbakhshR.OttenbacherK. J. (1994). The statistical analysis of single-subject data: A comparative examination. Physical Therapy, 74, 768–776.
48.
NovakI.McIntyreS.MorganC.CampbellL.DarkL.MortonN.…GoldsmithS. (2013). A systematic review of interventions for children with cerebral palsy: State of the evidence. Developmental Medicine & Child Neurology, 55, 885–910. doi:10.1111/dmcn.12246
49.
PalisanoR. J.RosenbaumP.WalterS. D.RussellD. J.WoodE.GaluppiB. (1997). Development and realiability of a system to classify gross motor function in children with cerebral palsy. Developmental Medicine & Child Neurology, 39, 214–223. doi:10.1111/j.1469-8749.1997.tb07414.x
50.
ParkerR. I.VannestK. J.DavisJ. L.SauberS. B. (2011). Combining nonoverlap and trend for single-case research: Tau-U. Behavior Therapy, 42, 284–299. doi:10.1016/j.beth.2010.08.006
51.
PolatajkoH.MandichA. (2004). Enabling occupation in children: The Cognitive Orientation to daily Occupational Performance (CO-OP) Approach. Ottawa, ON: CAOT Publications ACE.
52.
RoubertieA.MarianiL. L.Fernandez-AlvarezE.DoummarD.RozeE. (2012). Treatment for dystonia in childhood. European Journal Neurology, 19, 1292–1299. doi:10.1111/j.1468-1331.2011.03649.x
53.
SangerT. D.ChenD.FehlingsD. L.HallettM.LangA. E.MinkJ. W.…Valero-CuevasF. (2010). Definition and classification of hyperkinetic movements in childhood. Movement Disorders, 25, 1538–1549. doi:10.1002/mds.23088
54.
ScammellE. M.BatesS. V.HouldinA.PolatajkoH. J. (2016). The Cognitive Orientation to daily Occupational Performance (CO-OP): A scoping review. Canadian Journal of Occupational Therapy, 83, 216–225. doi:10.1177/0008417416651277
55.
Shikako-ThomasK.Dahan-OlielN.ShevellM.LawM.BirnbaumR.RosenbaumP.…MajnemerA. (2012). Play and be happy? Leisure participation and quality of life in school-aged children with cerebral palsy. International Journal of Pediatrics, 2012, Article 387280. doi:10.1155/2012/387280
56.
SideridisG. D.GreenwoodC. R. (1996). Evaluating treatment effects in single-subject behavioral experiments using quality-control charts. Journal of Behavioral Education, 6, 203–211.
57.
SkidmoreE. R.McEwenS.GreenD.van den HoutenJ.DawsonD.PolatajkoH. (in press). Development and specification of the CO-OP Approach. In DawsonD.McEwenS.PolatajkoH. (Eds.), Enabling participation across the lifespan: Advancements, adaptations and extensions of the CO-OP Approach. Bethesda, MD: AOTA Press.
58.
SpenceS. H. (1998). A measure of anxiety symptoms among children. Behavior Research and Therapy, 36, 545–566.
59.
StamelouM.EdwardsM. J.HallettM.BhatiaK. P. (2012). The non-motor syndrome of primary dystonia: Clinical and pathophysiological implications. Brain, 135(Pt. 6), 1668–1681. doi:10.1093/brain/awr224
60.
SwaminathanH.RogersH. J.HornerR. H.SugaiG.SmolkowskiK. (2014). Regression models and effect size measures for single case designs. Neuropsychological Rehabilitation, 24, 554–571. doi:10.1080/09602011.2014.887586
61.
TateR. L.PerdicesM.RosenkoetterU.ShadishW.VohraS.BarlowD. H.…WilsonB. (2016). The Single-Case Reporting Guideline in Behavioural Interventions (SCRIBE) 2016 statement. Canadian Journal of Occupational Therapy, 83, 184–195. doi:10.1177/0008417416648124
62.
ThabaneL.MaJ.ChuR.ChengJ.IsmailaA.RiosL. P.…GoldsmithC. H. (2010). A tutorial on pilot studies: The what, why and how. BMC Medical Research Methodology, 10, Article 1. doi:10.1186/1471-2288-10 -1
63.
VohraS.ShamseerL.SampsonM.BukutuC.SchmidC. H.TateR.…CENT Group. (2015). CONSORT extension for reporting N-of-1 trials (CENT) 2015 statement. British Medical Journal, 350, h1738. doi:10.1136/bmj.h1738
64.
WallenM.O’FlahertyS. J.WaughM. C. (2007). Functional outcomes of intramuscular botulinum toxin type A and occupational therapy in the upper limbs of children with cerebral palsy: A randomized controlled trial. Archives of Physical Medicine and Rehabilitation, 88, 1–10. doi:10.1016/j.apmr.2006.10.017
65.
WechslerD. (2004). Wechsler Intelligence Scale for Children (4th ed.). London, UK: Pearson Education.
66.
World Health Organization. (2001). International Classification of Functioning, Disability and Health (ICF). Geneva, Switzerland: Author.
Supplementary Material
Please find the following supplemental material available below.
For Open Access articles published under a Creative Commons License, all supplemental material carries the same license as the article it is associated with.
For non-Open Access articles published, all supplemental material carries a non-exclusive license, and permission requests for re-use of supplemental material or any part of supplemental material shall be sent directly to the copyright owner as specified in the copyright notice associated with the article.
