Misunderstood terms and concepts identified through user testing of educational materials for fragile X premutation: "Not weak or fragile?".

Complicated genetic mechanisms and unpredictable health risks associated with the FMR1 premutation can result in challenges for patient education when the diagnosis is made in a newborn. From October 15, 2018, to December 10, 2021, North Carolina parents could obtain FMR1 premutation results about t...

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Publicado en:Journal of Genetic Counseling Vol. 33; no. 2; pp. 341 - 352
Autores principales: Lincoln‐Boyea, Beth, Moultrie, Rebecca R., Biesecker, Barbara B., Underwood, Marcia, Duparc, Martin, Wheeler, Anne C., Peay, Holly L.
Formato: pictorial research tables/charts Journal Article
Publicado: Wiley-Blackwell Apr2024
Acceso en línea:Ver este registro en EBSCOhost
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      place: Malden, Massachusetts
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        atl: Misunderstood terms and concepts identified through user testing of educational materials for fragile X premutation: "Not weak or fragile?".
      aug:
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          Lincoln‐Boyea, Beth
          Moultrie, Rebecca R.
          Biesecker, Barbara B.
          Underwood, Marcia
          Duparc, Martin
          Wheeler, Anne C.
          Peay, Holly L.
        affil: Genomics, Bioinformatics, and Translational Research Center, RTI International, Research Triangle Park North Carolina,, USA
      sug:
        subj:
          Fragile X Syndrome Familial and Genetic
          Mutation
          Teaching Materials
          Health Information
          Health Knowledge Evaluation
          Human
          Male
          Female
          Funding Source
          North Carolina
          Content Analysis
          Genetic Counseling
          Interviews
          Concept Analysis
          Publication Bias
          Male
          Female
      ab: Complicated genetic mechanisms and unpredictable health risks associated with the FMR1 premutation can result in challenges for patient education when the diagnosis is made in a newborn. From October 15, 2018, to December 10, 2021, North Carolina parents could obtain FMR1 premutation results about their newborns through a voluntary expanded newborn screening research study. The study provided confirmatory testing, parental testing, and genetic counseling. We developed web‐based educational materials to augment information about fragile X premutation conveyed by a genetic counselor. Many genetics education materials are developed for the lay population. However, relatively little research is published on how well individuals understand these materials. We conducted three rounds of iterative user testing interviews to help refine web‐based educational materials that support understanding and self‐paced learning. The participants included 25 parents with a 2‐year college degree or less and without a child identified with fragile X syndrome, premutation, or gray‐zone allele. Content analysis of interview transcripts resulted in iterative changes and ultimately saturation of findings. Across all rounds of interviews, there were two terms that were commonly misunderstood (fragile and carrier) and two terms that elicited initial misconceptions that were overcome by participants. Many also had difficulty understanding the relationship between fragile X premutation and fragile X syndrome as well as appreciating the implications of having a "fragile X gene." Website layout, formatting, and graphics also influenced comprehension. Despite iterative changes to the content, certain issues with understandability persisted. The findings support the need for user testing to identify misconceptions that may interfere with understanding and using genetic information. Here, we describe a process used to develop and refine evidence‐based, understandable parental resources on fragile X premutation. Additionally, we provide recommendations to address ongoing educational challenges and discuss the potential impact of bias on the part of expert content developers.
      pubtype: Academic Journal
      doctype:
        pictorial
        research
        tables/charts
        Journal Article
      ougenre: Article
    language: English
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