Effect of targeted movement interventions on pain and quality of life in children with dyskinetic cerebral palsy: a pilot single subject research design to test feasibility of parent-reported assessments

Figures & data

Table 1. Description and psychometric properties of outcome measures used.

Measure	Description	Score	Psychometrics
^aPaediatric Pain Profile (PPP) [17] Parent proxy patient reported outcome measure (PROM)	Pain assessment for children aged 1–18 years with severe neurological disability and inability to communicate.	A score between 0 and 60 created from responses to a 20-item scale. ≥14/60 indicates clinically significant pain.	Interrater reliability – ICC 0.74–0.89 Internal consistency – Cronbach’s alpha 0.75–0.89
^aCare and Comfort Hypertonicity Questionnaire (CCHQ) [21]. Parent proxy patient reported outcome measure (PROM)	Questionnaire to document changes in care needs and health related quality of life for children with complex care needs.	27 Questions divided into 4 sections are rated on a 1–7 scale. The CCHQ summary score is the mean of the 4 section means.	Content and construct validity, and responsiveness were established.
^aSleep Disturbance Scale for Children (SDSC) [20] Parent proxy patient reported outcome measure (PROM)	Categorises sleep disturbance in children.	27 Item rating scale divided into 6 sections. A t-score is calculated from a raw score.	Internal consistency − 0.71 Test-retest reliability – r = 0.71
^bCanadian Occupational Performance Measure (COPM) [19] Parent proxy patient reported outcome measure (PROM)	Outcome measure used to set goals with clients and detect change in self-perceived occupational performance over time.	Performance and satisfaction are each rated out of 10 for up to 5 individual occupational performance problems.	Reliability, validity, responsiveness and utility have been established.
^bThe Cerebral Palsy Quality of Life (CP QoL) [22] Parent proxy patient reported outcome measure (PROM)	Quality of life questionnaire (primary caregiver or self-report), for children aged 4–12 years with CP, that assesses wellbeing across various domains of life.	Items are transformed to a scale with a range of 0–100. The mean of item values is computed for each domain.	Good reliability established [22]. Validity supported by correlations between CP QoL, KIDSCREEN and Gross Motor Function Classification System [22]

CP: Cerebral palsy; ICC: Intraclass Correlation Coefficients. ^aPrimary outcome measures (collected weekly). ^bProbe assessments.

Table 2. Participant characteristics.

	Participant 1	Participant 2	Participant 3
Age at start of baseline	12 y 1 month	14 y 2 months	13 y 10 months
Gender	Male	Male	Female
GMFCS	V	V	V
MACS	V	V	V
CFCS	IV	V	V
VFCS	IV	V	V
EDACS	IV	V	V
Motor type	Dyskinetic	Dyskinetic	Dyskinetic
BAD Scale	28	32	32
DIS (action/rest)	152/72	172/86	166/85
Topographical involvement	Quadriplegia	Quadriplegia	Quadriplegia
Associated impairments	Musculoskeletal deformity Cognitive impairment	Seizure disorder Musculoskeletal deformity Osteoporosis GOR Cognitive impairment	Seizure disorder Musculoskeletal deformity GOR Cognitive impairment
PPP “On a good day” Baseline “Most troublesome pain”	7 Stiffness in legs 31	11 Gastrointestinal 44	37 Scoliosis/Costopelvic impingement 40
Goal 1 (ICF code)	Ease of dressing (d5408)	Ease of dressing (d5408)	Ease of dressing (d5408)
Goal 2 (ICF code)	Improve sleep (b134)	Tolerate position change (e.g., side lying) (d4150)	Tolerate varied positions for spine health (d4108)
Goal 3 (ICF code)	Improve sit to stand (d4103)	Tolerating orthoses (s7302)	Tolerate movement opportunities (d4109)

BAD: Barry-Albright Dystonia Scale; CFCS: Communication Functional Classification System; CCHQ: Care and Comfort Hypertonicity Questionnaire; COPM: Canadian Occupational Performance Measure; CP Qol: Cerebral Palsy Quality of life Measure; DIS: Dyskinesia Impairment Scale; GMFCS: Gross Motor Function Classification System; EDACS: Eating & Drinking Classification System; GOR: Gastro-oesophageal reflux; ICF: International Classification of Functioning; MACS: Manual Ability Classification System; PPP: Paediatric Pain Profile; SCDC: Sleep Disturbance Scale for Children; VFCS: Visual Function Classification System.

Figure 1. Participant 1 results.

The figure displays the weekly results for participant one. The seven outcomes are represented in graphical form with the score on the vertical access, and the weekly timepoint on the horizontal axis.

Figure 2. Participant 2 results.

The figure displays the weekly results for participant two. The seven outcomes are represented in graphical form with the score on the vertical access, and the weekly timepoint on the horizontal axis.

Figure 3. Participant 3 results.

The figure displays the weekly results for participant three. The seven outcomes are represented in graphical form with the score on the vertical access, and the weekly timepoint on the horizontal axis.

Table 3. Results of weekly outcome measures.

ID	Outcome	Baseline (A)	Intervention (B)	Follow up (C)	Visual Analysis change	Change in means	2SD band	Non overlapping	NAP	Standard mean difference	Direction of treatment impact
		Mean (SD)	Mean (SD)	Mean (SD)	(Phase A to B)	(Phase A to B)	(Phase A to B)	(Phase A to B)	(Phase A to B)	(Phase A and B)
1	PPP	25.2 (9.2)	16.4 (8.6)	14.6 (4.5)	Yes	Yes	No	6/8	0.85	0.96	Positive
	CCHQ personal care	3.9 (0.0)	4.3 (0.7)	4.3 (0.4)	No	No	NA	2/8	0.25	NA	Nil
	CCHQ positioning	3.9 (0.2)	3.6 (0.6)	4.4 (0.4)	No	No	Yes	4/8	0.66	1.32	Unclear
	CCHQ comfort	5.2 (0.6)	4.0 (0.2)	4.6 (0.4)	Yes	Yes	No	7/8	0.98	2.10	Positive
	SDSC total	52.0 (2.4)	46.8 (7.7)	49.4 (7.9)	No	Yes	Yes	5/8	0.80	2.14	Unclear
	SDSC maintain	23.6 (0.5)	19.0 (3.4)	20.8 (4.5)	Yes	Yes	Yes	7/8	0.88	8.4	Positive
	SDSC somnolence	9.2 (2.4)	8.4 (2.5)	10.6 (3.3)	No	No	No	1/8	0.61	0.35	Nil
2	PPP	22.2 (12.3)	15.6 (6.1)	20.8 (8.2)	Yes	Yes	Yes	4/8	0.76	0.54	Positive
	CCHQ personal care	5.7 (0.6)	4.2 (0.7)	4.4 (0.2)	Yes	Yes	Yes	7/8	0.95	2.38	Positive
	CCHQ positioning	5.3 (0.3)	4.0 (0.5)	3.9 (0.2)	Yes	Yes	Yes	8/8	1	5.13	Positive
	CCHQ comfort	5.0 (0.9)	3.8 (1.3)	4.2 (1.0)	Yes	Yes	Yes	7/8	0.88	1.42	Positive
	SDSC total	50.8 (2.9)	46.5 (1.7)	46.6 (4.6)	No	Yes	No	4/8	0.91	1.50	Unclear
	SDSC maintain	9.8 (1.3)	9.0 (0.9)	9.6 (0.9)	No	No	No	2/8	0.71	0.61	Nil
	SDSC somnolence	18.8 (0.4)	17.4 (2.4)	17.4 (3.2)	Yes	Yes	Yes	4/8	0.66	3.19	Positive
3	PPP	43.0 (3.0)	31.0 (4.1)	28.4 (0.5)	Yes	Yes	Yes	8/8	1.00	4.00	Positive
	CCHQ personal care	6.6 (1.0)	6.4 (1.0)	4.4 (0.4)	No	No	No	1/7	0.62	0.19	Nil^a
	CCHQ positioning	5.7 (1.3)	4.5 (0.2)	4.9 (0.2)	Yes	Yes	No	0/8	0.80	0.92	Unclear
	CCHQ comfort	6.3 (0.2)	6.3 (0.5)	7.0 (0.0)	No	No	No	2/8	0.52	−0.09	Nil
	SDSC total	80.4 (6.9)	72.3 (6.2)	75.6 (2.2)	Yes	Yes	No	6/8	0.84	1.17	Positive
	SDSC maintain	29.6 (1.3)	26.9 (2.2)	25.8 (1.1)	Yes	Yes	Yes	4/8	0.85	2.03	Positive
	SDSC somnolence	17.4 (5.0)	17.8 (1.3)	21.8 (1.6)	No	No	No	0/8	0.40	−0.07	Nil

^aPhase change visual analysis and 2 SD band from b to c.

Note. CCHQ: Care and Comfort Hypertonicity Questionnaire; PPP: Pediatric Pain Profile; SCDC: Sleep Disturbance Scale for Children.

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