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Table 4 Key indicators of content validity for the BTHS-SA, adolescent version

From: Development and content validity of the Barth Syndrome Symptom Assessment (BTHS-SA) for adolescents and adults

Item

Interpreted as intended (n, %)

Relevance*

Mean response selected, range

Mean noticeable change rating (SD)

Mean important change rating§ (SD)

Experienced within the past 24 h

Experienced outside of the 24-h recall period

Item 1: Tiredness at rest

9/11 (81.8%)

8/10 (80.0%);

1/10 (10.0%)

1.00, 0–2

1.00 (0.00)

1.00 (0.00)

Item 2: Tiredness during activities

11/12 (91.7%)

8/12 (66.7%);

3/12 (25.0%)

1.42, 0–2

1.25 (0.46)

1.56 (0.53)

Item 3: Muscle weakness at rest

10/11 (90.9%)

4/12 (33.3%)

5/12 (41.7%)

1.17, 0–4

1.0 (0.00)

1.14 (0.38)

Item 4: Muscle weakness during activities

8/11 (72.7%)

8/11 (72.7%)

3/11 (27.3%)

1.64, 1–3

1.14 (0.38)

1.50 (0.53)

Item 5: Muscle pain at rest

9/11 (81.8%)

3/11 (27.3%)

3/11 (27.3%)

0.82, 0–2

1.25 (0.50)

1.50 (0.58)

Item 6: Muscle pain due to activities

10/11 (90.9%)

7/12 (58.3%)

2/12 (16.7%)

1.25, 0–3

1.00 (0.00)

1.43 (0.53)

Item 7: Feeling of early fullness

9/12 (75.0%)

7/11 (63.6%)

1/11 (9.1%)

0.82, 0–2

1.00 (0.00)

1.40 (0.55)

Item 8: Difficulty eating

11/11 (100.0%)

1/12 (8.3%)

5/12 (41.7%)

0.67, 0–2

1.00 (0.00)

1.00 (0.00)

Item 9: Headache

9/12 (75.0%)

4/12 (33.3%)

6/12 (50.0%)

1.00, 0–2

1.00 (0.00)

1.50 (0.76)

  1. *Number and percent of participants experiencing the symptom in the 24 h prior (and prior to the past 24 h) to assessment as reported during CDIs
  2. Severity as reported during CDIs on a five-point response scale (“No [CONCEPT] at all” to “Very severe [CONCEPT]”)
  3. The minimum change on a five-point response scale that would represent a noticeable improvement in the symptom concept
  4. §The minimum change on a five-point response scale that would represent an important improvement in the symptom concept