The development, content and response process validation of a caregiver-reported severity measure for CDKL5 deficiency disorder.

ABSTRACT:

Background

CDKL5 Deficiency Disorder (CDD) is a severe X-linked developmental and epileptic encephalopathy. Existing developmental outcome measures have floor effects and cannot capture incremental changes in symptoms. We modified the caregiver portion of a CDD clinical severity assessment (CCSA) and assessed content and response-process validity.

Methods

We conducted cognitive interviews with 15 parent caregivers of 1-39-year-old children with CDD. Caregivers discussed their understanding and concerns regarding appropriateness of both questions and answer options. Item wording and questionnaire structure were adjusted iteratively to ensure questions were understood as intended.

Results

The CCSA was refined during three rounds of cognitive interviews into two measures

SUBMITTER: Ziniel SI 

PROVIDER: S-EPMC10760432 | biostudies-literature | 2023 Nov

REPOSITORIES: biostudies-literature