TY - JOUR

T1 - Developing and evaluating rare disease educational materials co-created by expert clinicians and patients

T2 - the paradigm of congenital hypogonadotropic hypogonadism

AU - Badiu, Corin

AU - Bonomi, Marco

AU - Borshchevsky, Ivan

AU - Cools, Martine

AU - Craen, Margarita

AU - Ghervan, Cristina

AU - Hauschild, Michael

AU - Hershkovitz, Eli

AU - Hrabovszky, Erik

AU - Juul, Anders

AU - Kim, Soo-Hyun

AU - Kumanov, Phillip

AU - Lecumberri, Beatriz

AU - Lemos, Manuel C

AU - Neocleous, Vassos

AU - Niedziela, Marek

AU - Djurdjevic, Sandra Pekic

AU - Persani, Luca

AU - Phan-Hug, Franziska

AU - Pignatelli, Duarte

AU - Pitteloud, Nelly

AU - Popovic, Vera

AU - Quinton, Richard

AU - Skordis, Nicos

AU - Smith, Neil

AU - Stefanija, Magdalena Avbelj

AU - Xu, Cheng

AU - Young, Jacques

AU - Dwyer, Andrew A

AU - COST Action BM1105

PY - 2017/3/20

Y1 - 2017/3/20

N2 - 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.

AB - 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.

KW - Algorithms

KW - Health Literacy

KW - Humans

KW - Hypogonadism

KW - Kallmann Syndrome

KW - Nursing

KW - Patient Education as Topic/methods

KW - Rare Diseases

U2 - 10.1186/s13023-017-0608-2

DO - 10.1186/s13023-017-0608-2

M3 - Journal article

C2 - 28320476

VL - 12

JO - Orphanet Journal of Rare Diseases

JF - Orphanet Journal of Rare Diseases

SN - 1750-1172

M1 - 57

ER -