Abstract
Objective. To develop a composite DAS for JDM and provide preliminary evidence of its validity. Methods. The Juvenile DermatoMyositis Activity Index (JDMAI) is composed of four items: physician's global assessment of overall disease activity; parent's/child's global assessment of child's wellbeing; measurement of muscle strength; and assessment of skin disease activity. The score of the JDMAI is the arithmetic sum of the scores of each individual component. Six versions of the JDMAI were tested, which differed in the tools used to assess the third and fourth items. Validation procedures were conducted using three large multinational patient samples including a total of 627 patients. Results. The JDMAI was found to possess face and content validity, good construct validity, satisfactory internal consistency (Cronbach's alpha = 0.58-0.89), fair responsiveness to clinically important change (standardized response mean = 0.82-3.12 among patients improved) and strong capacity to discriminate patients judged as being in the state of inactive disease or low, moderate or high disease activity by the physician (P<0.001) or whose parents were satisfied or not satisfied with the course of their child's illness (P<0.001). Overall, the six versions of the JDMAI showed similar metrological performances in validation analyses. Conclusion. The JDMAI was found to possess good measurement properties in a large population of patients with a wide range of disease activity, and is, therefore, suitable for use in both clinical and research settings. The final version of the JDMAI will be selected after its prospective validation.
| Original language | English |
|---|---|
| Pages (from-to) | 1196-1205 |
| Number of pages | 10 |
| Journal | Rheumatology (United Kingdom) |
| Volume | 58 |
| Issue number | 7 |
| DOIs | |
| Publication status | Published - Jul 1 2019 |
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Keywords
- Composite disease activity scores
- Disease activity assessment
- Idiopathic inflammatory myositis
- JDM
- Muscle strength assessment
- Outcome measures
- Paediatric rheumatology
ASJC Scopus subject areas
- Rheumatology
- Pharmacology (medical)
Cite this
Development and validation of a composite disease activity score for measurement of muscle and skin involvement in juvenile dermatomyositis. / the Pediatric Rheumatology International Trials Organization (PRINTO).
In: Rheumatology (United Kingdom), Vol. 58, No. 7, 01.07.2019, p. 1196-1205.Research output: Contribution to journal › Article
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TY - JOUR
T1 - Development and validation of a composite disease activity score for measurement of muscle and skin involvement in juvenile dermatomyositis
AU - the Pediatric Rheumatology International Trials Organization (PRINTO)
AU - Rosina, Silvia
AU - Consolaro, Alessandro
AU - Van Dijkhuizen, Pieter
AU - Pistorio, Angela
AU - Varnier, Giulia Camilla
AU - Bovis, Francesca
AU - Nistala, Kiran
AU - Maillard, Susan
AU - Civino, Adele
AU - Tsitsami, Elena
AU - De Inocencio, Jaime
AU - Jelusic, Marija
AU - Vojinovic, Jelena
AU - Espada, Graciela
AU - Makay, Balahan
AU - Katsicas, Maria Martha
AU - Pratsidou-Gertsi, Polixeni
AU - Lazarevic, Dragana
AU - Rao, Anand Prahalad
AU - Pires Marafon, Denise
AU - Martini, Alberto
AU - Pilkington, Clarissa
AU - Ruperto, Nicolino
AU - Ravelli, Angelo
PY - 2019/7/1
Y1 - 2019/7/1
N2 - Objective. To develop a composite DAS for JDM and provide preliminary evidence of its validity. Methods. The Juvenile DermatoMyositis Activity Index (JDMAI) is composed of four items: physician's global assessment of overall disease activity; parent's/child's global assessment of child's wellbeing; measurement of muscle strength; and assessment of skin disease activity. The score of the JDMAI is the arithmetic sum of the scores of each individual component. Six versions of the JDMAI were tested, which differed in the tools used to assess the third and fourth items. Validation procedures were conducted using three large multinational patient samples including a total of 627 patients. Results. The JDMAI was found to possess face and content validity, good construct validity, satisfactory internal consistency (Cronbach's alpha = 0.58-0.89), fair responsiveness to clinically important change (standardized response mean = 0.82-3.12 among patients improved) and strong capacity to discriminate patients judged as being in the state of inactive disease or low, moderate or high disease activity by the physician (P<0.001) or whose parents were satisfied or not satisfied with the course of their child's illness (P<0.001). Overall, the six versions of the JDMAI showed similar metrological performances in validation analyses. Conclusion. The JDMAI was found to possess good measurement properties in a large population of patients with a wide range of disease activity, and is, therefore, suitable for use in both clinical and research settings. The final version of the JDMAI will be selected after its prospective validation.
AB - Objective. To develop a composite DAS for JDM and provide preliminary evidence of its validity. Methods. The Juvenile DermatoMyositis Activity Index (JDMAI) is composed of four items: physician's global assessment of overall disease activity; parent's/child's global assessment of child's wellbeing; measurement of muscle strength; and assessment of skin disease activity. The score of the JDMAI is the arithmetic sum of the scores of each individual component. Six versions of the JDMAI were tested, which differed in the tools used to assess the third and fourth items. Validation procedures were conducted using three large multinational patient samples including a total of 627 patients. Results. The JDMAI was found to possess face and content validity, good construct validity, satisfactory internal consistency (Cronbach's alpha = 0.58-0.89), fair responsiveness to clinically important change (standardized response mean = 0.82-3.12 among patients improved) and strong capacity to discriminate patients judged as being in the state of inactive disease or low, moderate or high disease activity by the physician (P<0.001) or whose parents were satisfied or not satisfied with the course of their child's illness (P<0.001). Overall, the six versions of the JDMAI showed similar metrological performances in validation analyses. Conclusion. The JDMAI was found to possess good measurement properties in a large population of patients with a wide range of disease activity, and is, therefore, suitable for use in both clinical and research settings. The final version of the JDMAI will be selected after its prospective validation.
KW - Composite disease activity scores
KW - Disease activity assessment
KW - Idiopathic inflammatory myositis
KW - JDM
KW - Muscle strength assessment
KW - Outcome measures
KW - Paediatric rheumatology
UR - http://www.scopus.com/inward/record.url?scp=85068485135&partnerID=8YFLogxK
UR - http://www.scopus.com/inward/citedby.url?scp=85068485135&partnerID=8YFLogxK
U2 - 10.1093/rheumatology/key421
DO - 10.1093/rheumatology/key421
M3 - Article
C2 - 30690571
AN - SCOPUS:85068485135
VL - 58
SP - 1196
EP - 1205
JO - Rheumatology
JF - Rheumatology
SN - 1462-0324
IS - 7
ER -