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Developing and evaluating rare disease educational materials co-created by expert clinicians and patients: the paradigm of congenital hypogonadotropic hypogonadism

COST Action BM1105, ; Badiu, C; Bonomi, M; Borshchevsky, I; Cools, M; Craen, M; Ghervan, C; Hauschild, M; Hershkovitz, E; Hrabovszky, E; et al. COST Action BM1105; Badiu, C; Bonomi, M; Borshchevsky, I; Cools, M; Craen, M; Ghervan, C; Hauschild, M; Hershkovitz, E; Hrabovszky, E; Juul, A; Kim, S; Kumanov, P; Lecumberri, B; Lemos, MC; Neocleous, V; Niedziela, M; Pekic, S; Persani, L; Phan-Hug, F; Pignatelli, D; Pitteloud, N; Popovic, V; Quinton, R; Skordis, N; Smith, N; Stefanija, MA; Xu, C; Young, J; Dwyer, AA (2017) Developing and evaluating rare disease educational materials co-created by expert clinicians and patients: the paradigm of congenital hypogonadotropic hypogonadism. Orphanet Journal of Rare Diseases, 12. p. 57. ISSN 1750-1172 https://doi.org/10.1186/s13023-017-0608-2
SGUL Authors: Kim, Soo-Hyun

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Abstract

Background Patients with rare diseases face health disparities and are often challenged to find accurate information about their condition. We aimed to use the best available evidence and community partnerships to produce patient education materials for congenital hypogonadotropic hypogonadism (CHH) and the olfacto-genital (Kallmann) syndrome (i.e., CHH and defective sense of smell), and to evaluate end-user acceptability. Expert clinicians, researchers and patients co-created the materials in a multi-step process. Six validated algorithms were used to assess reading level of the final product. Comprehensibility and actionability were measured using the Patient Education Materials Assessment Tool via web-based data collection. Descriptive statistics were employed to summarize data and thematic analysis for analyzing open-ended responses. Subsequently, translation and cultural adaption were conducted by clinicians and patients who are native speakers. Results Co-created patient education materials reached the target 6th grade reading level according to 2/6 (33%) algorithms (range: grade 5.9–9.7). The online survey received 164 hits in 2 months and 63/159 (40%) of eligible patients completed the evaluation. Patients ranged in age from 18 to 66 years (median 36, mean 39 ± 11) and 52/63 (83%), had adequate health literacy. Patients scored understandability at 94.2% and actionability at 90.5%. The patient education materials were culturally adapted and translated into 20 languages (available in Additional file 1). Conclusions Partnering with patients enabled us to create patient education materials that met patient- identified needs as evidenced by high end-user acceptability, understandability and actionability. The web-based evaluation was effective for reaching dispersed rare disease patients. Combining dissemination via traditional healthcare professional platforms as well as patient-centric sites can facilitate broad uptake of culturally adapted translations. This process may serve as a roadmap for creating patient education materials for other rare diseases.

Item Type: Article
Additional Information: © The Author(s). 2017 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
Keywords: Genetics & Heredity, 1199 Other Medical And Health Sciences
SGUL Research Institute / Research Centre: Academic Structure > Molecular and Clinical Sciences Research Institute (MCS)
Journal or Publication Title: Orphanet Journal of Rare Diseases
ISSN: 1750-1172
Dates:
DateEvent
6 March 2017Accepted
20 March 2017Published
Publisher License: Creative Commons: Attribution 4.0
URI: https://openaccess.sgul.ac.uk/id/eprint/108663
Publisher's version: https://doi.org/10.1186/s13023-017-0608-2

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