Effect of Home-based Bimanual Training in Children with Unilateral Cerebral Palsy (The COAD-study): A Case Series

Figures & data

Figure 1. Participant flow.* One participant that was included in the analyses dropped out at follow-up (T3)

Table 1. Participant characteristics at study enrollment

Case	Home program	Allocation	Age (years)	Gender	Affected side	MACS level	GMFCS level	House Functional Classification type	BRIEF(-P)^a total score	Education	PS^b	NEO-FFI-3^c
1	Implicit	Random	3	M	L	N.A.^d	I	4	#	Mainstream nursery school/day-care	3	#; 38; 34; 35; 37
2	Implicit	Preferred	5	F	R	II	I	6	75	Mainstream primary school	#	29; 46; 39; 37; 37
3	Implicit	Preferred	3	M	R	N.A.^d	II	4	#	Mainstream nursery school/day-care	#	28; 40; 36; 31; #
4	Implicit	Random	4	M	R	II	I	5	115	Mainstream primary school	3	27; 36; 35; 34; 40
5	Implicit	Preferred	4	M	R	I	I	3	#	#	#	29; 43; 36; 36; 42
6	Explicit	Preferred	6	M	R	II	I	3	126	Mainstream primary school	#	30; 41; 38; 38; 42
7	Explicit	Random	4	M	L	II	I	5	104	Mainstream primary school	3	#; #; #; #; #
8	Explicit	Preferred	2	M	R	N.A.^d	I	7	#	N.A.	#	34; 39; 35; 31; 38
9	Explicit	Random	4	M	R	II	I	5	#	School for physically disabled children	3	25; 43; 35; 27; 35
10	Explicit	Preferred	3	F	R	N.A.^d	I	7	#	Mainstream nursery school/day-care	#	33; 38; 41; 36; 41
11	Explicit	Random	2	M	R	N.A.^d	I	7	#	Therapeutic toddler group	2	31; 37; 39; 34; 39
12	Explicit	Preferred	2	M	R	N.A.^d	I	5	#	Mainstream nursery school/day-care	#	36; 36; 34; 32; 39
13	Explicit	Preferred	7	F	L	II	I	5	110	School for physically disabled children	#	29; #; 36; 31; 41
14	Explicit	Preferred	4	M	R	III	I	3	83	Mainstream primary school	#	26; 44; 30; 31; 39

^aBehavior Rating Inventory of Executive Function (BRIEF) for children aged 5 through 7 years, and BRIEF Preschool Version (BRIEF-P) for children aged up to 5 years, to assess executive functioning of the child

^bParenting Scale (PS), to assess parenting style

^cNeuroticism-Extraversion-Openness Five-Factor Inventory (NEO-FFI-3), to assess the personality of the parent (neuroticism; extraversion; openness; agreeableness; conscientiousness)

^dN.A: not applicable; MACS is not validated for children <4 years of age. For younger children, the rehabilitation team judged that manual ability was comparable to MACS Levels I–III

M: male; F: female; L: left; R: right

# Missing value

Table 2. Parent-rated outcomes at child level

Case	Time point	COPM performance (0–10)	COPM satisfaction (0–10)	HUH logits (−4.69 – +5.17)	LAQ-CP
					Dimensional scores						Overall score (0–100)
					Physical independence (0–100)	Mobility (0–100)	Clinical burden (0–100)	Schooling (0–100)	Economic burden (0–100)	Social integration (0–100)
1^a	T0	4.2	4.4	−1.022	64.59	32.14	25.00	.00	29.17	18.75	43.20
	T1	6.8*	6.8*
	T2	6.8*	7.0*	−1.431	#	#	#	#	#	#	#
	T3	7.4*	6.6*	−.838	45.83	14.29	22.73	.00	25.00	15.63	30.57
2^a	T0	5.8	5.8	.376	16.67	14.29	20.46	.00	.00	18.75	18.73
	T1	7.0	7.4
	T2	6.8	7.0	.238	12.50	17.86	11.36	.00	.00	12.50	16.51
	T3	7.0	7.2	1.066	16.67	14.29	6.82	.00	.00	18.75	18.28
3^a	T0	5.2	5.6	−1.218	62.50	28.57	20.46	.00	.00	12.50	37.55
	T1	7.0	7.0
	T2	7.8*	8.4*	−.665	43.75	32.14	18.18	.00	.00	12.50	31.75
	T3	7.8*	8.4*	−.500	41.67	25.00	20.46	.00	.00	12.50	29.15
4^a	T0	2.8	4.6	.786	#	#	#	#	#	#	#
	T1	6.2*	7.2*
	T2	8.0*	9.0*	1.684	18.75	28.57	31.82	.00	45.83	28.13	29.57
	T3	7.8*	7.8*	1.684	#	#	#	#	#	#	#
5^a	T0	5.2	5.6	−.343	37.50	25.00	27.27	.00	16.67	21.88	31.35
	T1	6.2	7.0
	T2	7.8*	8.0*	.238	25.00	25.00	34.09	.00	25.00	15.63	26.40
	T3	7.0	8.0*	−.044	12.50	21.43	27.27	.00	16.67	12.50	19.37
6^b	T0	5.6	6.2	.098	18.75	50.00	22.73	.00	16.67	15.63	29.86
	T1	7.6*	8.2*
	T2	7.4	6.8	1.066	16.67	53.57	29.55	.00	25.00	28.13	33.79
	T3	7.7*	7.8	.925	16.67	46.43	27.27	.00	41.67	34.38	34.55
7^b	T0	5.0	10	−1.022	14.58	17.86	27.27	.00	8.33	25.00	21.26
	T1	6.0	10
	T2	7.2*	10	.376	20.83	17.86	25.00	.00	8.33	6.25	19.22
	T3	7.4*	10	.238	18.75	10.71	20.46	.00	8.33	15.63	18.50
8^b	T0	6.0	6.8	−1.927	45.83	25.00	27.27	.00	4.17	40.63	37.50
	T1	4.2	4.4^†
	T2	8.0*	8.2	−.665	56.25	32.14	27.27	.00	45.83	50.00	48.67
	T3	8.4*	8.6	−.343	41.67	21.43	31.82	.00	12.50	46.88	37.29
9^b	T0	2.2	3.2	−2.597	39.58	32.14	31.82	50.00	8.33	43.75	39.19
	T1	6.0*	6.4*
	T2	6.8*	7.4*	−1.431	39.58	32.14	31.82	37.50	25.00	68.75	45.96
	T3	6.6*	6.6*	−1.665	31.25	25.00	31.82	37.50	33.33	62.50	40.34
10^b	T0	4.8	8.0	.098	62.50	35.71	18.18	.00	8.33	25.00	42.89
	T1	6.4	6.4
	T2	8.0*	9.0	.376	52.09	25.00	25.00	.00	4.17	21.88	35.46
	T3	7.8*	8.4	1.066	39.58	28.57	13.64	.00	8.33	15.63	30.53
11^b	T0	3.0	3.0	.925	50.00	17.86	13.64	12.50	.00	12.50	30.17
	T1	7.6*	8.0*
	T2	8.8*	8.8*	1.211	20.83	10.71	13.64	12.50	.00	12.50	17.83
	T3	8.0*	8.0*	2.265	33.33	35.71	15.91	12.50	20.83	25.00	33.63
12^b	T0	4.2	5.0	−1.927	56.25	32.14	18.18	.00	25.00	25.00	41.06
	T1	6.4*	6.8
	T2	7.4*	7.6*	−.500	47.92	28.57	18.18	.00	16.67	25.00	36.44
	T3	7.0*	7.2*	−1.431	47.92	28.57	20.46	.00	16.67	18.75	35.11
13^b	T0	4.4	5.0	2.053	12.50	21.43	34.09	62.50	16.67	37.50	26.19
	T1	5.6	5.6
	T2	6.6*	7.0*	1.211	10.42	21.43	20.46	50.00	25.00	28.13	23.38
	T3	6.6*	6.8	3.134	6.25	7.14	20.46	62.50	8.33	40.63	19.67
14^b	T0	4.2	3.6	−1.218	12.50	21.43	27.27	.00	8.33	21.88	20.78
	T1	6.6*	6.0*
	T2	6.4*	6.6*	−1.022	25.00	21.43	38.64	.00	29.17	21.88	27.33
	T3	#	#	#	#	#	#	#	#	#	#

* Clinically relevant improvement compared to baseline

^†Clinically relevant decline compared to baseline

^aImplicit program

^bExplicit program

# Missing value

For COPM and HUH, higher score indicates better performance; for LAQ-CP, higher score indicates lower participation

Figure 2. COPM change scores

Table 3. Observer-rated outcomes at child level

Case	Time point	GAS (−3 – +2)	AHA-units (0–100)	OSAS^a
				Quality of use affected hand
				Reach (0/1)	Grasp fingers (1–3)	Grasp wrist (1–5)	Hold fingers (1–4)	Hold wrist (1–5)	Release (1–4)
1^b	T0	−2	50	0	2	1	2	1	2.5
				0	2	1	1	1	1
	T2	−2	50	0	2	1	2	1	3*
				0	2	1	2*	1	2.0*
	T3	−2	49	1	2	1	2	1	2.0^†
				0	1^†	1	1	1	1
2^b	T0	−2	55	0	2	1	2	1	3.0
				0	2	1	2	1	3
	T2	−2	57	0	2	1	2	1	3
				0	2	1	2	1	2.0^†
	T3	1	57	0	2	1	2	1	3.0
				0	2	3*	2	3*	3
3^b	T0	−2	48	0	2	1	2	1	2.0
				0	2	1	2	1	2
	T2	#	62*	0	2	1	2	1	3*
				0	2	1	2	1	2.0
	T3	#	52	0	2	1	2	1	2.0
				0	2	1	2	1	2
4^b	T0	−2	63	0	2	3	2	1	2.5
				0	2	1	3	1	2
	T2	−2	59	0	2	1^†	2	1	3*
				1	2	1	3	1	3.0*
	T3	1	61	0	1^†	2^†	3*	2*	1.0^†
				1	2	1	2^†	1	3*
5^b	T0	−2	52	0	2	3	2	3	3.0
				0	2	3	3	3	2
	T2	0	63*	0	2	3	2	3	3
				0	2	1^†	3	3	2.0
	T3	0	58*	0	2	3	2	3	2.0^†
				0	2	1^†	2^†	1^†	2
6^c	T0	−2	50	0	2	1	2	1	2.0
				0	2	1	2	1	2
	T2	0	49	0	2	1	2	1	2
				0	2	1	2	1	2.0
	T3	0	58*	0	2	1	2	1	2.0
				0	2	1	2	1	2
7^c	T0	−2	63	1	2	1	2	1	2.0
				0	2	1	2	1	1
	T2	2	64	0	2	1	2	1	3*
				0	2	1	2	1	2.5*
	T3	−1	65	0	2	1	2	1	3.0*
				0	2	1	2	1	3*
8^c	T0	−2	71	0	2	1	2	1	3.0
				0	2	1	3	1	2
	T2	0	73	0	2	1	2	1	3
				0	2	3*	3	1	3.0*
	T3	−2	79*	0	2	1	2	1	3.0
				0	2	1	3	1	3*
9^c	T0	−2	41	0	2	1	2	1	2.0
				0	2	3	1	3	2
	T2	−2	46*	1	2	1	2	1	2
				0	2	1^†	2*	3	2.0
	T3	−1	46*	0	2	1	2	1	2.5*
				0	2	3	2*	1^†	2
10^c	T0	−2	73	0	2	3	2	1	3.0
				0	2	1	3	1	2
	T2	−3	67^†	0	2	1^†	2	1	3
				0	2	1	3	1	3.0*
	T3	−1	63^†	0	2	1^†	2	1	3.0
				0	2	3*	3	1	3*
11^c	T0	−2	66	1	2	1	2	3	3.0
				0	2	3	3	1	3
	T2	#	71*	0	#	#	#	#	#
				0	#	#	#	#	#
	T3	#	69	0	#	#	#	#	#
				0	#	#	#	#	#
12^c	T0	−2	50	0	2	1	2	3	3.0
				0	2	3	3	3	2
	T2	1	47	0	2	4*	2	3	3
				0	2	3	3	3	3.0*
	T3	1	48	0	2	1	2	3	2.0^†
				0	2	2^†	3	3	2
13^c	T0	−2	64	1	2	1	2	1	3
				0	2	1	3	1	3
				0	2	1	2	1	3
	T2	2	68	0	2	1	2	1	3
				1	2	1	3	1	3
				0	2	1	2	1	3
	T3	−1	63	0	2	1	2	1	3
				0	2	1	3	1	3
				0	2	1	2	1	3
14^c	T0	−2	49	0	2	1	2	1	2.0
				0	2	1	2	1	2.0
	T2	−3	46	0	2	1	2	1	2
				0	2	1	1^†	1	1.0^†
	T3	#	#	0	#	#	#	#	#
				0	#	#	#	#	#

* Clinically relevant/statistically significant improvement compared to baseline

^†Clinically relevant/statistically significant decline compared to baseline

^aOSAS tasks are (from top to bottom) threading beads, and building with ‘Pop-Onz’ (Fisher Price®) for children aged 2.5 to 6 years, and buttering and cutting bread, small screw and nut construction, and large screw and nut construction for children from 7 years of age

^bImplicit program

^cExplicit program

# Missing value

For GAS, AHA, and OSAS, higher score indicates higher ability

Table 4. Parent-related outcomes

		VOST							OBVL^a (t-scores)						EMPO^b (t-scores)
Case	Time point	Overall (0–100)	Child (0–100)	Parent (0–100)	Family (0–100)	Environment (0–100)	Therapy (0–100)	Therapist (0–100)	Pcr (45–80)	Par (35–80)	Dep (46–80)	Rol (42–80)	Phy (46–80)	Total (30–80)	IP (20–80)	IA (20–80)	BC (20–80)	Total (20–80)
1^c	T0	21	35	11	#	5	5	#	57	63	54	46	48	52	57	34	33	36
	T2	2	20	20	20	30	20	10	57	63	56	50	57^†	55	#	#	#	#
	T3	10	17	17	16	16	14	#	53	63	60^†	50	57^†	55	50^†	39*	43*	40*
2^c	T0	NT	NT	NT	NT	NT	NT	NT	45	35	46	42	46	30	80	80	66	78
	T2	NT	#	#	#	#	#	#	45	35	46	42	46	30	80	80	48^†	80
	T3	#	#	#	#	#	#	#	45	35	46	42	46	30	80	80	75*	80
3^c	T0	0	#	#	#	#	#	#	57	57	48	46	52	48	45	45	48	43
	T2	NT	#	#	#	#	#	#	46*	45*	48	46	48	41	57*	52*	43^†	48*
	T3	#	#	#	#	#	#	#	46*	53	48	46	43	43	37^†	45	56*	40
4^c	T0	NT	NT	NT	NT	NT	NT	NT	61	66	66	60	73	70	33	45	43	34
	T2	NT	#	#	#	#	#	#	61	56*	66	49*	67	63	40*	52*	38^†	38
	T3	19	0	#	29	34	36	#	51*	52*	66	52*	56	55	45*	45	48*	43*
5^c	T0	0	#	#	#	#	#	#	51	50	50	60	61	53	45	72	48	57
	T2	4	#	#	#	#	#	#	45*	35*	46	56	46*	39	50*	62^†	56*	59
	T3	#	#	#	#	#	#	#	45*	41*	46	52*	46*	39	#	#	#	#
6^d	T0	13	13	16	#	16	#	#	56	47	60	73	62	62	40	61	38	43
	T2	91	0	0	1	0	2	1	56	50	53*	68	59	58	45*	52^†	48*	45
	T3	0	0	0	0	0	7	0	56	50	50*	75	61	61	40	58^†	48*	45
7^d	T0	NT	NT	NT	NT	NT	NT	NT	51	50	60	49	59	53	45	52	33	38
	T2	NT	#	#	#	#	#	#	45*	35*	46*	42	46*	30	73*	62*	66*	71*
	T3	#	#	#	#	#	#	#	45*	50	56	42	71^†	54	62*	61*	56*	61*
8^d	T0	60	56	16	44	#	12	#	53	67	52	50	60	55	50	58	38	45
	T2	13	19	18	22	12	#	#	53	63	48	60*	62	56	57*	52^†	33^†	43
	T3	28	46	10	16	#	#	#	53	72	56	56	60	61	50	52^†	33^†	40^†
9^d	T0	0	30	#	#	#	#	#	45	35	60	66	56	51	57	72	48	61
	T2	#	30	30	#	#	#	#	56^†	47^†	60	70	61	61	45^†	62^†	62*	59
	T3	#	40	#	#	#	#	#	67^†	52^†	56	79^†	61	68	45^†	62^†	43^†	51^†
10^d	T0	30	0	45	44	#	50	#	60	50	56	53	48	52	57	80	62	67
	T2	10	10	31	33	#	30	#	53*	48	52	50	48	45	57	61^†	56^†	59^†
	T3	9	10	9	0	0	0	0	53*	45	54	50	43	44	66*	72^†	66*	71
11^d	T0	0	0	0	0	0	0	0	46	41	52	46	62	45	66	52	48	57
	T2	0	0	0	4	#	3	0	46	41	48	46	52	41	80*	80*	48	71*
	T3	#	#	#	#	#	#	#	46	41	48	46	48	39	73*	62*	48	64*
12^d	T0	43	17	31	#	0	0	#	57	45	52	46	52	45	50	52	48	48
	T2	30	20	26	24	0	20	#	53	50	48	53	57	48	50	45^†	48	45
	T3	12	#	#	20	#	20	#	46*	57^†	48	50	48	46	45^†	52	48	45
13^d	T0	0	0	0	#	0	0	0	56	44	56	49	56	49	50	62	56	59
	T2	2	#	#	#	#	#	#	45*	44	50	46	56	42	62*	58^†	56	59
	T3	0	0	0	0	0	0	0	56	41	46*	49	56	44	57*	62	66*	64*
14^d	T0	NT	NT	NT	NT	NT	NT	NT	45	50	46	49	46	42	45	39	62	45
	T2	10	41	40	41	#	#	#	56^†	62^†	46	49	51	50	50*	52*	62	54*
	T3	#	#	#	#	#	#	#	#	#	#	#	#	#	#	#	#	#

* Statistically significant improvement compared to baseline

^†Statistically significant decline compared to baseline

^aPcr: Parent–child relationship problems; Par: Parenting problems; Dep: Depressive Mood; Rol: Parental role restriction; Phy: Physical health problems

^bIP: Intrapersonal; IA: Interactional; BC: Behavioral control

^cImplicit program

^dExplicit program

# Missing value

Italics represent participant included in the analysis of the VOST

For VOST and OBVL, higher score indicates higher stress; for EMPO, higher score indicates higher empowerment