Content Validation of Clinician-Reported Items for a Severity Measure for CDKL5 Deficiency Disorder-Reference-Cited by-同舟云学术

Content Validation of Clinician-Reported Items for a Severity Measure for CDKL5 Deficiency Disorder

Published:2021-08-11 Issue:11 Volume:36 Page:998-1006
ISSN:0883-0738
Container-title:Journal of Child Neurology
language:en
Short-container-title:J Child Neurol

Author:

Saldaris Jacinta¹,Weisenberg Judith²,Pestana-Knight Elia³,Marsh Eric D.⁴,Suter Bernhard⁵,Rajaraman Rajsekar⁶,Heidary Gena⁷,Olson Heather E.⁸,Devinsky Orrin⁹,Price Dana⁹,Jacoby Peter¹,Leonard Helen¹¹⁰,Benke Tim A.¹¹,Demarest Scott¹¹,Downs Jenny¹¹²^ORCID

Affiliation:

1. Telethon Kids Institute, Perth, Western Australia, Australia

2. St Louis Children’s Hospital and Washington University School of Medicine, St Louis, MO, USA

3. Cleveland Clinic, Neurological Institute, Cleveland, OH, USA

4. Division of Neurology, Children’s Hospital of Philadelphia and School of Medicine, University of Pennsylvania, Philadelphia, PA, USA

5. Texas Children’s Hospital and Baylor College of Medicine, Houston, TX, USA

6. UCLA Mattel Children’s Hospital, Los Angeles, CA, USA

7. Department of Ophthalmology, Boston Children’s Hospital, Boston, MA, USA

8. Department of Neurology, Boston Children’s Hospital, Boston, MA, USA

9. NYU Langone Health and Department of Neurology, New York University, NY, USA

10. The University of Western Australia, Perth, Western Australia, Australia

11. Children’s Hospital Colorado and University of Colorado School of Medicine, Aurora, CO, USA

12. Curtin School of Allied Health, Curtin University, Perth, Western Australia, Australia

Abstract

CDKL5 deficiency disorder (CDD) results in early-onset seizures and severe developmental impairments. A CDD clinical severity assessment (CCSA) was previously developed with clinician and parent-report items to capture information on a range of domains. Consistent with US Food and Drug Administration (FDA) guidelines, content validation is the first step in evaluating the psychometric properties of an outcome measure. The aim of this study was to validate the content of the clinician-reported items in the CCSA (CCSA-Clinician). Eight neurologists leading the USA CDD Center of Excellence clinics were interviewed using the “think aloud” technique to critique 26 clinician-reported items. Common themes were aggregated, and a literature search of related assessments informed item modifications. The clinicians then participated in 2 consensus meetings to review themes and finalize the items. A consensus was achieved for the content of the CCSA-Clinician. Eight of the original items were omitted, 11 items were added, and the remaining 18 items were revised. The final 29 items were classified into 2 domains: functioning and neurologic impairments. This study enabled refinement of the CCSA-Clinician and provided evidence for its content validity. This preliminary validation is essential before field testing and further validation, in order to advance the instrument toward clinical trial readiness.

Funder

NIH

Publisher

SAGE Publications

Subject

Clinical Neurology,Pediatrics, Perinatology, and Child Health

Link

http://journals.sagepub.com/doi/pdf/10.1177/08830738211019576

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