A novel assessment tool for clinical care of patients with autoinflammatory disease: juvenile autoinflammatory disease multidimensional assessment report

Clin Exp Rheumatol. 2016 Sep-Oct;34(6 Suppl 102):129-135. Epub 2016 Oct 25.

Abstract

Objectives: To develop and test a new multidimensional questionnaire for assessment of children with auto-inflammatory disease (AID) such as FMF, PFAPA, HIDS, TRAPS in standard clinical care.

Methods: The juvenile auto-inflammatory disease multidimensional assessment report (JAIMAR) includes 16 parent or patient-centered measures and four dimensions that assess functional status, pain, therapeutic compliance and health-related quality of life (physical, social, school, emotional status) with disease outcome. It is proposed for use as both a proxy-report and a patient self-report, with the suggested age range of 8-18 years for use as a self-report.

Results: 250 children with FMF were included in the study. Total of 179 forms were filled up by parents and patients, and 71 forms were filled up by parents having children less than 8 years. Completing and scoring the JAIMAR can be done in 15 minutes. For the JAIMAR's dimensions, the Cronbach's alpha coefficient for internal consistency was between 0.507-0.998. There was a significant and a positive correlation between the test-retest scale scores (ICC=0.607-0.966). Concerning construct validity, all factors loadings were above 0.30. For the criterion validity, the correlation level between each dimension and the related scale ranged from medium (r=0.329, p<0.0001) to large (r=0.894, p<0.0001). The parents' proxy-reported and children's self-reported data were outstandingly concordant (r=0.770-0.989).

Conclusions: The development of the JAIMAR introduces a new and multi-dimensional approach in paediatric rheumatology practice. It is a new tool for children with auto-inflammatory dis-ease and it may help enhance their quality of care.

Publication types

  • Validation Study

MeSH terms

  • Adolescent
  • Age Factors
  • Child
  • Child, Preschool
  • Cost of Illness
  • Familial Mediterranean Fever / diagnosis*
  • Familial Mediterranean Fever / physiopathology
  • Familial Mediterranean Fever / psychology
  • Familial Mediterranean Fever / therapy
  • Female
  • Fever / diagnosis*
  • Fever / physiopathology
  • Fever / psychology
  • Fever / therapy
  • Health Status
  • Hereditary Autoinflammatory Diseases / diagnosis*
  • Hereditary Autoinflammatory Diseases / physiopathology
  • Hereditary Autoinflammatory Diseases / psychology
  • Hereditary Autoinflammatory Diseases / therapy
  • Humans
  • Male
  • Predictive Value of Tests
  • Quality of Life
  • Reproducibility of Results
  • Severity of Illness Index
  • Surveys and Questionnaires*

Supplementary concepts

  • Periodic fever, familial, autosomal dominant