Table 1

Study and participant characteristics

AuthorsStudy typeStudy designStudy design specifiedNAgeGender (male), n (%)Disease-specific characteristicsParents’ characteristics
James et al23 (CA), 59FGeneric qualitative research design (part of large RCT).
Interview study.
10M: 11 years 4 months (SD 2 years 6 months)5 (50.0)Spastic: 10 (100%)*
Hemiplegia: 10 (100%)
MACS:
I: 3 (30.0%)
II: 7 (70.0%)*
Gender: 1 male (10.0%)*
McBurney et al61FQualitative study (embedded in an RCT).11M: 12 years 9 months (SD 2 years 10 months)4 (36.4)*Spastic: 11 (100%)*
Diplegia: 11 (100%)*
GMFCS:
I: 2 (18.2%)
II: 2 (18.2%)
III: 7 (63.6%)*
Gender: 3 male (23.1%)*
Novak et al24 (CA), 11FQualitative study (embedded in an RCT).8Mdn: 6.5 years (range 5 years 5 months–12 years 8 months)*5 (62.5)*Spastic: 6 (75.0%)
Ataxic: 1 (12.5%)
Athetosis: 1 (12.5%)*
Hemiplegia: 1 (12.5%)
Bilateral: 5 (62.5%)
Unknown: 2 (25.0%)*
Gender: 2 male (20.0%)*
Taylor et al70FQualitative research design using indepth interviews, embedded in an RCT.11M: 12.7 years (SD 2.8 years)4 (36.4)*Spastic: 11 (100%)*
Diplegia: 11 (100%)*
GMFCS:
I: 2 (18.2%)
II: 2 (18.2%)
III: 7 (63.6%)*
Gender: 3 male (23.1%)*
Law and King15FFeasibility study, embedded in a clinical trial.72Range 18 months–8 yearsSpastic: 72 (100%)*
Lorentzen et al60FNon-randomised controlled clinical study, including a feasibility component.46M: 11 years (SD 2.6 years)*30 (65.2)*Spastic: 42 (91.3%)
Ataxic: 4 (8.7%)*
Hemiplegia: 38 (82.6%)
Bilateral: 4 (8.7%)
Unknown: 4 (8.7%)*
MACS:
I: 28 (60.9%)
II: 18 (39.1%)*
GMFCS:
I: 44 (95.7%)
II: 2 (4.3%)*
Psychouli and Kennedy65FUncontrolled clinical trial, using an A1-B-C-A2 design, with a feasibility component.9M: 6 years 9 months (range 5 years 1 months–11 years)6 (66.7)*Spastic: 9 (100%)*
Hemiplegia: 9 (100%)*
Ahl et al53FPilot study with feasibility component.14Mdn: 3 years 8 months (range 1 year 6 months–6 years)*11 (78.6)*Spastic: 14 (100%)
Diplegia: 12 (85.7%)
Quadriplegia: 2 (14.3%)*
GMFCS:
II: 1 (7.1%)
III: 8 (57.1%)
IV: 3 (21.4%)
V: 2 (14.3%)*
Novak et al14FPilot study (single-group, pretest–post-test design) with a feasibility component.20M: 3.8 years (range 2–7 years)16 (80)*Spastic: 20 (100%)*
Bilde et al71FPilot study including feasibility components.9M: 10 years 3 months5 (55.6)*Spastic: 9 (100%)*
MACS:
I: 4 (44.4%)
II: 5 (55.6%)*
GMFCS:
I: 8 (88.9%)
II: 1 (11.1%)*
Boyd et al25 (CA)FPre–post pilot study including a feasibility component.9Range 9–13 yearsSpastic: 9 (100%)*
Hemiplegia: 9 (100%)*
McCoy et al29 (CA)FPilot project.4Range 9–14 years3 (75)*Spastic: 4 (100%)*
Hemiplegia: 4 (100%)*
MACS:
I: 2 (50%)
III: 2 (50%)*
Farr et al99FTwo-group, parallel feasibility trial.30
Shierk et al108FEvaluated through a trial.65
Liu et al49 (CA)FSingle-group, pre–post intervention trial.15M: 94.2 months (SD 27.5 months)Hemiplegia: 15 (100%)*
Ferre et al22 (CA), 56FSingle-group design.11Mdn: 45 months (range 29–54 months)*6 (54.5)Spastic: 11 (100%)
Hemiplegia: 11 (100%)
MACS:
I: 2 (18.2%)
II: 5 (45.5%)
III: 3 (27.3%)
IV: 1 (9.1%)*
Chiu et al98FSingle-group, pre–post intervention group.20M: 8.7 years (SD 2.4 years)11 (55)*Hemiplegia: 8 (40%)
Diplegia: 10 (50%)
Quadriplegia: 2 (10%)*
MACS:
I and II: 17 (85%)
III: 3 (15%)*
GMFCS:
I and II: 17 (85%)
III: 3 (15%)*
Visser et al106FWithin-subjects, repeated-measures design.10Mdn: 14 years 3 months (range 6 years 2 months–16 years 6 months)*Spastic: 9 (90%)
Ataxic: 1 (10%)*
Diplegia: 5 (50%)
Triplegia: 3 (30%)
Quadriplegia: 1 (10%)
Unknown: 1 (10%)*
MACS:
I: 5 (50%)
II: 4 (40%)
III: 1 (10%)*
GMFCS:
II: 5 (50%)
III: 5 (50%)*
CFCS:
I: 7 (70%)
II: 1 (10%)
III: 1 (10%)
IV: 1 (10%)*
Fehlings et al27 (CA)FProspective intervention study design (case series), including a feasibility component.15M: 8.8 years (SD 2.3 years)Spastic: 15 (100%)*
Hemiplegia: 15 (100%)*
Kenyon et al105FCase series.3Mdn: 5 years 11 months (range 5 years 6 months–14 years 10 months)*3 (100)Spastic: 3 (100%)*
Diplegia: 1 (33.3%)
Triplegia: 1 (33.3%)
Quadriplegia: 1 (33.3%)*
MACS:
I: 1 (33.3%)
III: 1 (33.3%)
IV: 1 (33.3%)*
GMFCS:
III: 1 (33.3%)
IV: 2 (66.6%)*
CFCS:
I: 1 (33.3%)
IV: 2 (66.6%)*
Fergus et al55FCase report with feasibility component.113 months1 femaleSpastic: 1 (100%)*
Hemiplegia: 1 (100%)
Educational level: postgraduate
Reifenberg et al107FCase report.15 years1 (100)Spastic: 1 (100%)
Hemiplegia: 1 (100%)
Gender: 1 female
Hernandez Alvarado102FProspective case study with a single experimental group.5M: 15 years4 (80)*MACS:
I: 3 (60%)
II: 2 (40%)*
GMFCS:
III: 5 (100%)*
Jaber et al47 (CA)FMixed methods.15I: Mdn: 100 months*11 (73.3)
Basaran et al54FAdherence survey study (cross-sectional).147Range 2.5–18.0 years83 (56.5)*Spastic: 143 (97.3%)
Unspecified: 4 (2.7%)*
Hemiplegia: 39 (26.5%)
Diplegia: 54 (36.7%)
Quadriplegia: 50 (34%)
Unspecified: 4 (2.7%)*
GMFCS:
I: 37 (25.2%)
II: 21 (14.3%)
III: 32 (21.8%)
IV: 24 (16.3%)
V: 33 (22.4%)*
Age: range 20–57 years
Gender: 3 male (2.1%)*
Educational level:
Illiterate: 8 (5.4%)
Literate: 3 (2.0%)
Primary school: 68 (58.5%)
Secondary school: 23 (15.6)
High school: 23 (15.6%)
University: 4 (2.7%)*
Halvarsson et al57FQualitative study.15Range 3–19 yearsGMFCS:
II: 3 (30.0%)
III: 3 (30.0%)
IV: 4 (40.0%)
Gender: 5 male (33.3%)*
Hinojosa and Anderson58FQualitative study.9Mdn: 3 years (range 2–5 years)*5 (55.6)Spastic: 8 (88.9%)
Unspecified: 1 (11.1%)
Hemiplegia: 1 (11.1%)
Diplegia: 2 (22.2%)
Quadriplegia: 5 (55.6%)
Unspecified: 1 (11.1%)
Gender: 8 female
Peplow and Carpenter62FQualitative research design (with constructivist approach).4Gender: 1 male (25%)*
Piggot et al63FQualitative research project.7Range 2–10 yearsHemiplegia: 2 (28.6%)
Quadriplegia: 5 (71.4%)*
Age: range mid-20s to late 30s
Gender: 1 male (12.5%)*
Piggot et al64FGrounded theory study.
Ross and Thomson66FQuestionnaire study.23M: 27.6 months11 (47.8)*
Sandlund et al67FQualitative study.15M: 11 years (range 6–16 years)8 (53.3)*Gender: 6 male (31.6%)*
Gerhardy and Sandelance28 (CA)FA needs analysis was undertaken using semistructured interviews.17Range 2–7 years
Finet101FQualitative, phenomenological methodological design.9Range 1–12 yearsAge: range 32–53 years
Gender: 1 male (11.1%)*
Educational level:
Some college: 1 (11.1%)
High school: 2 (22.2%)
Bachelor’s degree: 5 (55.5%) Associate’s degree: 1 (11.1%)*
Sel et al50 (CA)FQuestionnaire study.118
Sandlund et al68F14M: 10 years 11 months (range 6–16 years)8 (57.1)*Spastic: 12 (85.7%)
Dyskinetic: 1 (7.1%)
Ataxic: 1 (7.1%)*
Hemiplegia: 7 (50.0%)
Bilateral: 5 (35.7%)
Unknown: 2 (14.3%)*
MACS:
I: 7 (50.0%)
II: 5 (35.7%)
III: 1 (7.1%)
IV: 1 (7.1%)*
GMFCS:
I: 10 (71.4%)
II: 2 (14.3%)
III: 2 (14.3%)*
Sevick et al69F4Mdn: 13.5 years (range 8–17 years)*2 (50.0)*Spastic: 4 (100%)*
Hemiplegia: 4 (100%)*
MACS:
II: 4 (100%)*
GMFCS:
I: 4 (100%)*
Dizmek et al26 (CA)F
Pasquet et al30 (CA)F28M: 11.9 years (SD 2.7 years)Spastic: 28 (100%)*
Hemiplegia: 28 (100%)*
Sisman Isik et al51 (CA)F6336 (57)*GMFCS:
I–III: 61.9%
IV–V: 38.1%
James et al31 (CA), 32 (CA), 76BEFLarge RCT (with narrow CI level I).Matched-pairs waitlist control RCT.102I: M: 11 years 8 months (SD 2 years 4 months)51 (50.5)*Spastic: 102 (100%)
Hemiplegia: 102 (100%)
MACS:
I: 24 (23.8%)
II: 76 (75.2%)
III: 1 (1.0%)*
GMFCS:
I: 45 (44.6%)
II: 56 (55.4%)*
Hoare et al75BEFSmaller RCT (with wider CI level II).Randomised, controlled, evaluator-blinded trial.35M: 35.8 months (SD 15.8 months)20 (58.8)*Spastic: 35 (100%)
Hemiplegia: 35 (100%)
Kirkpatrick et al77BEFSmaller RCT (with wider CI level II).Single-centre, single-blinded (outcomes assessor), parallel-group RCT with 1:1 allocation.70M: 5.6 years (SD 2.1 years)39 (55.7)*Spastic: 70 (100%)*
Hemiplegia: 70 (100%)
Gordon et al85BEFSmaller RCT (with wider CI level II).RCT including a feasibility component.44I: M: 6 years 3 months (SD 2 years 2 months)20 (47.6)*Spastic: 44 (100%)*
Hemiplegia: 44 (100%)
MACS:
I: 5 (11.9%)
II: 35 (83.3%)
III: 2 (4.8%)*
Wallen et al 33 (CA), 86BEFSmaller RCT (with wider CI level II).Pragmatic, randomised, assessor-blinded trial, including a feasibility component.50M: 48.6 months (SD 21.0 months)27 (54.0)*Spastic: 50 (100%)*
Hemiplegia: 50 (100%)
MACS:
I: 2 (4%)
II: 37 (77%)
III: 8 (17%)
IV: 1 (2%)
GMFCS:
I: 33 (67%)
II: 15 (31%)
III: 1 (2%)
Al-Oraibi and Eliasson72BEFSmaller RCT (with wider CI level II).20I: M: 47 months (SD 19 months)10 (71.4)*Spastic: 14 (100%)*
Hemiplegia: 14 (100%)
Educational level:
Diploma: 3 (21.4%)
Below high school: 3 (21.4%)
High school: 7 (50.0%)
Bachelor: 1 (7.1%)*
Eugster-Buesch et al73BEFSmaller RCT (with wider CI level II).Randomised, controlled, single-blinded pilot study including feasibility components.23I: M: 9.8 years (SD 3.5 years)12 (52.2)*Spastic: 23 (100%)*
Hemiplegia: 23 (100%)
GMFCS:
I: 20 (87.0%)
II: 3 (13.0%)*
Hsin et al74BEFSmaller RCT (with wider CI level II).12I: M: 6.9 years (SD 0.6 years)10 (45.5)*Spastic: 23 (100%)
Hemiplegia: 23 (100%)
Klingels et al78BEFSmaller RCT (with wider CI level II).Randomised, controlled and evaluator-blinded trial including a feasibility component.51M: 8 years 9 months (SD 2 years 2 months)28 (54.9)*Spastic: 51 (100%)*
Hemiplegia: 51 (100%)*
MACS:
I: 4 (7.8%)
II: 38 (74.5%)
III: 9 (17.6%)*
Lin et al79BEFSmaller RCT (with wider CI level II).RCT with feasibility component.22I: M: 76.7 months (SD 26.2 months)12 (57.1)*Hemiplegia: 11 (52.4%)
Quadriplegia: 10 (47.6%)*
Novak et al81BEFSmaller RCT (with wider CI level II).Double-blind RCT with a feasibility component.36M: 7.75 years (SD 2.02 years)25 (69.4)*Spastic: 30 (83.3%)
Dyskinetic: 3 (8.3%)
Ataxic: 1 (2.8%)
Athetosis: 2 (5.6%)*
Hemiplegia: 14 (38.9%)
Diplegia: 14 (38.9%)
Quadriplegia: 2 (5.6%)
Unknown: 6 (16.7%)*
MACS:
I: 17 (47.2%)
II: 9 (25.0%)
III: 2 (5.6%)
IV: 5 (13.9%)
V: 3 (8.3%)*
GMFCS:
I: 17 (47.2%)
II: 5 (13.9%)
III: 6 (16.7%)
IV: 2 (5.6%)
V: 6 (16.7%)*
Preston et al82BEFSmaller RCT (with wider CI level II).Pilot, single-blind, multicentre RCT, with a feasibility component.16M: 9 years 2 months (SD 2 years 5 months)9 (60.0)*Hemiplegia: 14 (93.3%)
Bilateral: 1 (6.7%)*
MACS:
II: 3 (20.0%)
III: 5 (33.3%)
IV: 7 (46.7%)*
Sakzewski et al83BEFSmaller RCT (with wider CI level II).Pragmatic, single-blind, matched-pairs RCT.53M: 7 years 10 months (SD 2 years 4 months)32 (68.1)*Spastic: 53 (100%)*
Hemiplegia: 46 (97.9%)
Unknown: 1 (2.1%)*
MACS:
I: 24 (51.1%)
II: 23 (48.9%)*
GMFCS:
I: 34 (72.3%)
II: 13 (27.7%)*
Charles et al84BEFSmaller RCT (with wider CI level II).Single-blinded RCT, including a feasibility component.33M: 6 years 8 months (SD 1 year 4 months)14 (63.6)*Spastic: 33 (100%)*
Hemiplegia: 33 (100%)
Chamudot et al44 (CA), 97BEFSmaller RCT (with wider CI level II).RCT including a feasibility component.36M corrected age 11.1 months (SD 2.2 months)19 (58)*Spastic: 33 (100%)*
Hemiplegia: 33 (100%)*
Ferre et al100 110BEFSmaller RCT (with wider CI level II).Randomised trial including a feasibility component.40I: M: 5.2 years (SD 2.7 years)10 (41.7)*Spastic: 24 (100%)*
Hemiplegia: 24 (100%)*
MACS:
I: 5 (20.8%)
II: 19 (79.2%)*
Fischer et al45 (CA)BEFSmaller RCT (with wider CI level II).Multisite RCT using a factorial design, including a feasibility component.55
Hobbs et al46 (CA)BEFSmaller RCT (with wider CI level II).Pilot RCT.18M: 10 years 8 months (SD 3 years 4 months)12 (66.7)*Hemiplegia: 13 (72.2%)
Diplegia: 5 (27.8%)*
MACS:
I: 2 (11.1%)
II: 10 (55.6%)
III: 3 (16.7%)
IV: 3 (16.7%)*
Hughes et al103BEFSmaller RCT (with wider CI level II).Non-blinded, randomised intervention study.28Range 18–68 months17 (60.7)*Educational level:
12 years of schooling or less
Kassee et al104BEFSmaller RCT (with wider CI level II).Pilot study employing pretest, post-test experimental design.6Mdn: 9 years (range 7–12 years)*6 (100)*Spastic: 6 (100%)*
Hemiplegia: 6 (100%)*
MACS:
I: 2 (33.3%)
II: 4 (66.7%)*
Law et al109BEFSmaller RCT (with wider CI level II).Two-by-two factorial design.7928 (39)*Spastic: 72 (100%)*
Hemiplegia: 28 (39%)
Quadriplegia: 44 (61%)*
Liang et al48 (CA)BEFSmaller RCT (with wider CI level II).Randomised trial.30
Hobbs et al52 (CA)BEFSmaller RCT (with wider CI level II).RCT.18M: 10 years 8 months (SD 3 years 4 months)12 (66.7)Hemiplegia: 13 (72.2%) Diplegia: 5 (27.8%)*
MACS:
I: 2 (11.1%)
II: 10 (55.6%)
III: 3 (16.7%)
IV: 3 (16.7%)*
Lowes et al80BEFPretest–post-test cohort design, with the participants serving as their own controls, including a feasibility component.7Mdn: 11.4 months (range 7.1–16.1 months)*3 (42.9)*Spastic: 7 (100%)*
Hemiplegia: 7 (100%)*
Facchin et al95ELarge RCT (with narrow CI level I).Multicentre, prospective, cluster-randomised controlled clinical trial.10553 (50.5)*Spastic: 105 (100%)*
Hemiplegia: 105 (100%)
Chen et al87ESmaller RCT (with wider CI level II).Single-blinded RCT.48I: M: 8.73 years (SD 1.9 years)21 (46.7)*Spastic: 45 (100%)
Hemiplegia: 45 (100%)
Chiu et al34 (CA), 88ESmaller RCT (with wider CI level II).Prospective, single-blind, randomised trial.62I: M: 9.4 years (SD 1.9 years)28 (45.2)*Spastic: 62 (100%)
Hemiplegia: 62 (100%)
MACS:
I–III: 42 (67.7%)
IV–V: 20 (32.3%)*
GMFCS:
I–III: 52 (83.9%)
IV–V: 10 (16.1%)*
Kim et al90ESmaller RCT (with wider CI level II).19I: M: 9.1 years (SD 1.8 years)10 (52.6)*Hemiplegia: 10 (52.6%)
Quadriplegia: 9 (47.4%)*
Xu et al92ESmaller RCT (with wider CI level II).Single-blinded RCT.75I: M: 56.8 months (SD 34.0 months)E: 25 (36.8)*Spastic: 75 (100%)*
Hemiplegia: 75 (100%)*
MACS:
I: 10 (14.7%)
II: 49 (72.1%)
III: 9 (13.2%)
GMFCS:
I: 60 (88.2%)
II: 8 (11.8%)
Abd El-Kafy et al93ESmaller RCT (with wider CI level II).30I: M: 6.0 years (SD 1.7 years)12 (44.4)*Spastic: 30 (100%)*
Hemiplegia: 30 (100%)
MACS:
II: 11 (40.7%)
III: 9 (33.3%)
IV: 7 (25.9%)*
Bagley et al35 (CA)ESmaller RCT (with wider CI level II).Prospective RCT with patient preference.38Range 5–15 yearsSpastic: 38 (100%)*
Hemiplegia: 38 (100%)*
Hoare et al36 37 (CA)ESmaller RCT (with wider CI level II).Randomised, controlled, assessor-blinded trial.34M: 3 years (SD 1 year 4 months)20 (58.8)*Spastic: 34 (100%)*
Hemiplegia: 34 (100%)*
Klingels et al38 (CA)ESmaller RCT (with wider CI level II).51M: 8 years 9 monthsSpastic: 51 (100%)*
Hemiplegia: 51 (100%)*
Koseotlu et al39 (CA)ESmaller RCT (with wider CI level II).32Spastic: 32 (100%)*
Hemiplegia: 32 (100%)*
Novak et al40 41 (CA)ESmaller RCT (with wider CI level II).Double-blind RCT.36
Sakzewski et al42 43 (CA)ESmaller RCT (with wider CI level II).Single-blind, matched-pairs, randomised comparison trial.48M: 7.9 years (SD 2.3 years)33 (68.8)*Spastic: 48 (100%)*
Hemiplegia: 48 (100%)*
MACS:
I: 25 (52.1%)
II: 23 (47.9%)*
Crocker et al89ESingle-subject design study
(level IV).
Single-subject, ABA experimental design.22 years and 3 years1 male and 1 femaleSpastic: 2 (100%)*
Hemiplegia: 2 (100%)
Naylor and Bower91ESingle-subject design study
(level IV).
Single-case, A–B–A experimental design.9Mdn: 31 months (range 21–61 months)*6 (66.7)*Spastic: 9 (100%)*
Hemiplegia: 9 (100%)*
Coker et al94ESingle-subject design study
(level IV).
Single-subject ABAB design with a 6-month follow-up evaluation.15 months1 (100)Spastic: 1 (100%)*
Hemiplegia: 1 (100%)
Gross et al96ESingle-subject design study
(level III).
Multiple-baseline, across-subjects design (A-B + follow up).3Mdn: 3 years 8 months (range 2 years 9 months–3 years 8 months)*2 (66.7)Spastic: 2 (66.7%)
Mixed: 1 (33.0%)*
Hemiplegia: 1 (33.3%)
Quadriplegia: 1 (33.3%)
Unspecified: 1 (33.3%)*
  • *Numbers and percentages were calculated by the authors of this review.

  • BEF, both efficacy/effectiveness; CA, conference abstract; CFCS, Communication Function Classification System; E, efficacy/effectiveness study; F, feasibility study; GMFCS, Gross Motor Function Classification System; I, intervention group; M, mean; MACS, Manual Ability Classification System; Mdn, median; RCT, randomised controlled trial.