This was a two-centre validation study of two sets of patient reported outcome measures (PROMS) for adolescents (13-17- years-old) with inherited retinal diseases. The measures were the Michigan Retinal Degeneration Questionnaire (MRDQ) and the Michigan Vision-Related Anxiety Questionnaire (MVAQ). Both have previously been validated for use in adult patients. The authors note that many inherited retinal diseases may begin to manifest in childhood, and so PROMS validated for this period will be vital in assessing disease impact, and potentially treatment responses. Ninety-one patients took part over a 10-month period. Both the MRDQ and the MVAQ give an overall person score and individual domain scores. Neither the MRDQ nor the MVAQ demonstrated floor or ceiling effects, suggesting they are able to capture the full range of vision-related disability and anxiety. Eighteen patients went on to complete re-tests 14 days following the first questionnaire. This showed good test-retest correlation, in keeping with that found when the questionnaires were given to adults. This suggests that retests can be used to assess for meaningful changes over time. There was correlation between clinical characteristics of the patients involved, such as visual acuity, with domain scores of the PROMS. The MVAQ was further validated against the PHQ-4, a tool for detecting clinical depression and anxiety, and correlated well with it. The authors conclude that the MRDQ and the MVAQ are valid for assessing the impact of inherited retinal diseases in adolescents in terms of visual disability and vision related anxiety.
Patient reported outcomes in adolescents with inherited retinal diseases
Reviewed by Ian Reekie
The validation of inherited retinal disease-specific patient-reported outcome measures in adolescent patients.
CONTRIBUTOR
Ian Reekie
Sandwell and West Birmingham Hospitals NHS Trust, Birmingham, UK.
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