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Table 4 Test–retest reliability of the shortened HS-FOCUS questionnaire for HS-FOCUS total score and domains

From: Validation of the shortened Hunter Syndrome-Functional Outcomes for Clinical Understanding Scale (HS-FOCUS)

Stable patients based on GIS score

Caregiver-reported patients (3–26 years) n = 19

Self-reported patients (12–58 years) n = 14

Initial assessment at baseline

Follow-up at 2–4 weeks

ICC

Initial assessment at baseline

Follow-up at 2–4 weeks

ICC

Mean (SD) total score

1.02 (0.77)

1.00 (0.69)

0.97

0.48 (0.53)

0.46 (0.49)

0.93

 Lower body

0.84 (0.98)

0.95 (1.10)

0.95

0.59 (0.74)

0.48 (0.52)

0.90

 Upper body

1.58 (0.89)

1.61 (0.84)

0.91

0.58 (0.6)

0.48 (0.41)

0.83

 School/work

0.59 (0.48)

0.59 (0.54)

0.71

0.19 (0.53)

0.13 (0.35)

0.92

 Activities

1.05 (1.1)

0.84 (0.76)

0.68

0.49 (0.65)

0.51 (0.69)

0.79

 Breathing

0.92 (0.89)

0.93 (0.85)

0.86

0.52 (0.53)

0.52 (0.82)

0.73

  1. ICC was determined for stable caregiver- and self-reported patients based on GIS analysis; ICC > 0.70 was considered as an acceptable level of reliability
  2. Abbreviations: GIS Global Impression of Severity, HS-FOCUS Hunter Syndrome-Functional Outcomes for Clinical Understanding Scale, ICC intra-class coefficient, SD standard deviation