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Table 4 Outcome measures categorised according to pain, function and quality of life

From: Outcome measures for assessing change over time in studies of symptomatic children with hypermobility: a systematic review

Outcome measures Follow-up Timeframe d Baseline
Mean (SD)
Mean change in outcome at follow-up a 95% CI
Scale Test details Type     
PAIN (Intensity)
VAS [45, 46]
(Visual Analogue scale)
0-100
0 = no pain
100 = worst pain
PRO 2 months [42] Neutral treatment group: 40.0 (16.6) -19.9 NR
Hypermobility treatment group: 38.6 (16.9) -9.19 NR
Combined groups: 39.4 (14.2) -14.5 -5.2, -23.8
5 months e
[41]
Targeted Physiotherapy: 55.5 (21.3) -21.2 -38, -4.5
General Physiotherapy: 62.1 (24.1) -30.6 -50.16, -11.0
Combined groups: 57.6 (20.1) -25.8 -38.5, -13.1
WBFPS [47, 48]
(Wong-baker faces pain scale)
0-5
0 = no pain
5 = worst pain
PRO 12 months
[39]
Intervention: 2.2 (1.4) -1.6 -2.1, -1.1
Control: 2.5 (1.6) -1.6 -2.0, -1.2
PAIN (Intensity)
VAS-P [49]
(Visual Analogue scale-Parental)
0-100
0 = no pain
100 = worst pain
PRO b 5 months
[41]
Targeted Physiotherapy: 45.1 (23.0) -21.6 -33.2, -10.0
General Physiotherapy: 48.4 (22.9) -12. -23.3, 0.9
Combined groups:
46.7 (22.7)
-17.2 -25.3, -9.1
   12 months
[39]
Intervention: 33.8 (24.8) -6.8 -14.3, 0.7
Control: 40.6 (27.5) -7.3 -15.4, 0.8
FUNCTION
CHAQ [50] (Childhood Health Assessment Questionnaire) 0-3
0 = Without any difficulty
1 = With some difficulty
2 = With much difficulty
3 = Unable to do
PRO b 2 months
[42]
Neutral treatment group:
-0.13 (0.44)
0.12 NR
Hypermobility treatment group:
0.04 (0.71)
0.02 NR
Combined groups: -0.5 (0.6) 0.07 -0.1, 0.2
5 months
[41]
Targeted Physiotherapy: 0.62 (0.65) -0.15 -0.3, -0.02
General Physiotherapy: 0.76 (0.68) -0.16 ( -0.4, 0.1
Combined groups: 0.69 (0.66) -0.15 -0.3, -0.02
12 months
[39]
Intervention: 0.84 (0.62) 0.04 0.1, 0.2
Control: 0.86 (0.72) −0.02 -0.12, 0.08
Dynamometry [51]
Measurement of strength
Grip strength. Units: kilopascals CRO 12 months
[39]
Intervention: 57.0 (25.0) 4.7 0.1, 9.3
Control: 59.4 (31.7) 7.3 2.9, 11.7
Knee flexor and extensor strength. Units: Newtons 2 months
[42]
Neutral treatment group: 4.0 (1.7) 0.88 NR
Hypermobility treatment group: 4.4 (2.4) 1.21 NR
Combined groups: 4.2 (2.0) 1.1 0.4, 1.7
FUNCTION
M-ABC2 [52]
(Movement Assessment Battery for Children, 2nd Edition)
Measures coordination
Raw scores converted to centiles (0 – 100) with higher centiles indicating better performance compared to peers
CRO 12 months
[39]
Intervention: 33.4 (26.7) 3.8 -1.7, 9.3
Control: 35.6 (30.1) 10.8 5.4, 16.2
PODCI [53, 54]
(Pediatric Outcomes Data Collection Instrument)
0-100
100= highest functioning
PRO b 3 months
[40]
Transfer and basic mobility domain
Intervention: 82.1 (14.8)
11.8 0.30, 1.43
Control: 94.2 (14.8) 1.2 -0.46, 0.62
6 MWT [55]
(6 min walk test)
Maximum distance walked in 6 minutes (meters/leg length) CRO 36 months
[44]
Able/moderate: 7.3 (1.6) c -1.5 -1.3, -4.5
Severe: 5.3 (1.6) -2.3 -2.4, -2.7
No. of flights of stairs climbed in 2 min
[56]
Assesses functional ability in stairclimbing CRO 2 months
[42]
Neutral treatment group: 16.3 (5.0) 3.8 NR
Hypermobility treatment group: 20.9 (6.7) -0.33 NR
Combined groups: 18.6 (5.7) 1.7 -0.5, 3.9
Quality of Life
CHU9D [57]
(Child Health Utility 9D)
0-4
higher scores indicate poorer HRQoL
PRO 12 months
[39]
Intervention: 0.85 (0.10) 0.02 (0.09) -0.004, 0.04
Control: 0.85 (0.12) 0.00 (0.12) -0.03, 0.03
CHQ-PF50 [58]
(Child Health Questionnaire)
0-100
0 = worst QoL
100 = Best QoL
PRO b 2 months [42] Physical summary score
Neutral treatment group:
32.0 (11.9)
10.1 NR
Hypermobility treatment group: 41.6 (15.0) 2.3 NR
Combined groups: 38.0 (12.6) 5.3 1.7, 8.9
Psychological summary score
Neutral treatment group:
46.4 (12.3)
-0.9 NR
Hypermobility treatment group: 46.3 (9.0) 8.1 NR
Combined groups: 48 (10.3) 2.7 -0.3, 5.8
Quality of Life
PGIC [59, 60]
(Patient global impression of change)
1-7
1= very much improved
7= very much
worse
PRO 2 months
[42]
Neutral treatment group:
0.3 (1.1)
1.4 NR
Hypermobility treatment group: 0.2 (0.9) 1.6 NR
Combined groups: 0.2 (1.0) 1.5 1.0, 2.0
PODCI [53] (Pediatric Outcomes Data Collection Instrument)
Pain comfort
Happiness
0-100
Higher score means higher health-related quality
PRO b 3 months
[40]
1. Pain/comfort:
Intervention:
83.9 (16.2)
4.9 -0.22, 0.87
Control: 84.4 (17) -1.2 -0.61, 0.47
2. Happiness:
Intervention: 79.5 (18.7) -0.2 -0.55, 0.53
Control: 80.7 (15.9) -0.9 -0.60, 0.48
PedsQL parent proxy-reported format [61] and [62] (Pediatric Quality of Life Inventory-Generic Core Scale) 0-100
Lower score indicates lower quality of life
PRO b 3 months
[40]
1. Physical
Intervention:
62.3 (19.9)
3.9 -0.35, 0.73
Control: 79.2 (20.1) -8.1 -0.95, 0.14
2. Psychosocial   
Intervention:
65.6 (16.3)
0.9 -0.49, 0.59
Control: 73.8 (18.8) 0.3 -0.52, 0.56
Quality of Life
Global-VAS (parent’s global assessment) 0-100
0 = no impact of hypermobility
100 = high impact of hypermobility
PRO b 5 months
[41]
Targeted Physiotherapy:
36.1 (26.4)
General
-17.6 -31.1, -4.1
Physiotherapy:
37.2 (25.3)
3.7 -7.8, 15.3
Combined groups: 36.6 (25.7) n = 32 -7.6 -17.2, -2.0
  1. Abbreviations. 95% CI: 95% Confidence Interval; CRO: Clinician-reported outcome; GP: Generalised Physiotherapy; HRQOL=health-related quality of life; QoL: quality of life; PRO: Patient-reported outcome
  2. a difference in change score from baseline (outcome-baseline)
  3. b Indicates Parent reported outcomes
  4. c Data calculated by primary author to demonstrate the difference between children of different severity and supplied to the authorship team upon request. This was only able to be provided for 6MWT, not the other variables
  5. d Time points are when outcome measurements are reported
  6. e Authors converted the faces pain scale to a 0–100 scale to combine with VAS data