Development of a measure of genome sequencing knowledge for young people: The kids‐KOGS

Abstract

Genome sequencing (GS) is increasingly being used to diagnose rare diseases in paediatric patients; however, no measures exist to evaluate their knowledge of this technology. We aimed to develop a robust measure of knowledge of GS (the kids‐KOGS') suitable for use in the paediatric setting as well as for general public education. The target age was 11 to 15 year olds. An iterative process involving six sequential stages was conducted to develop a set of draft true/false items. These were then administered to 539 target‐age school pupils (mean 12.8; SD ± 1.3), from the United Kingdom. Item‐response theory was used to confirm the psychometric suitability of the candidate items. None of the Items was identified as misfits. All 10 items performed well under the two‐parameter logistic model. The internal consistency of the test was 0.84 (Cronbach alpha value) indicating excellent reliability. The mean kids‐KOGS score in the sample overall was 4.24 (SD; 2.49), where 0 = low knowledge and 10 = high knowledge. Age was positively associated with score in a multivariate linear regression. The kids‐KOGS is a short and reliable tool that can be used by researchers and healthcare professionals offering GS to paediatric patients. Further validation in a clinical setting is required.

Genomic Health Literacy Interventions in Pediatrics: Scoping Review

“…To chart and extract data from the articles selected for the scoping review, an extraction criterion was developed by 2 reviewers (AG and MT) to extract information from each publication about the study country, city, urban or not urban geography, population, sample size, age of participants, pediatric disease types, duration, demographic information, design, methodology, journal of publication, and results of the study (Multimedia Appendix 2 [3,12,16,[33][34][35][36][37][38][39][40][41][42][43]).…”

“…Of the 14 studies, 5 (36%) studies included populations where children were clinically diagnosed with various genetic conditions [16,[33][34][35][36]; 4 (29%) studies had populations that were not diagnosed with an illness [12,[37][38][39]; 1 (7%) study included children who were suspected to have a genetic condition but not diagnosed [40]; 1 (7%) study included children who were hospitalized for various reasons [3]; and the remaining 3 (21%) studies reported pediatric populations with illnesses such as cancer [41], congenital heart defects [42], and congenital lower limb deficiencies [43]. Most (11/14, 79%) of the studies selected for the review were from the United States [3,12,[33][34][35][36][38][39][40][41][42], 14% (2/14) from Canada [16,43] and 7% (1/14) from the United Kingdom [37]. Table 1 presents the overview of the included studies.…”

“…To develop and validate a robust kids-KOGS b suitable for use in the pediatric setting and for general public education Quantitative 539 Children Lewis et al [37] To determine the levels of knowledge about genetics and approaches to decisionmaking related to genetic testing among adolescents and parents Qualitative 33 (22 adolescents and 11 parents) Parents and adolescents Rew et al [39]…”

Development and mixed-methods evaluation of an online animation for young people about genome sequencing

“…Participants completed a quantitative survey which was implemented at two time-points; time 1 (T1) before watching the animation and time 2 (T2) after watching the animation (Supplementary Material 5). The survey was adapted from one developed by Sanderson et al [21] to evaluate the animation "Whole Genome Sequencing and You" [3] and included a new knowledge of genome sequencing measure for young people (the kids-KOGS) [22].…”

“…Responders were also asked "How would you describe your understanding of genetics" (None, Some, Good). Objective knowledge was assessed using the new 10-item kids-KOGS [22] which includes a series of statements about GS with responders asked to indicate whether the statement is true, false or don't know.…”