Navigating Disrupted Puberty: Development and Evaluation of a Mobile-Health Transition Passport for Klinefelter Syndrome.

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State: Public
Version: Final published version
License: CC BY 4.0
Serval ID
serval:BIB_3938769A5C18
Type
Article: article from journal or magazin.
Collection
Publications
Institution
Title
Navigating Disrupted Puberty: Development and Evaluation of a Mobile-Health Transition Passport for Klinefelter Syndrome.
Journal
Frontiers in endocrinology
Author(s)
Dwyer A.A., Héritier V., Llahana S., Edelman L., Papadakis G.E., Vaucher L., Pitteloud N., Hauschild M.
ISSN
1664-2392 (Print)
ISSN-L
1664-2392
Publication state
Published
Issued date
2022
Peer-reviewed
Oui
Volume
13
Pages
909830
Language
english
Notes
Publication types: Journal Article ; Systematic Review
Publication Status: epublish
Abstract
Klinefelter syndrome (KS) is the most common aneuploidy in men and has long-term sequelae on health and wellbeing. KS is a chronic, lifelong condition and adolescents/young adults (AYAs) with KS face challenges in transitioning from pediatric to adult-oriented services. Discontinuity of care contributes to poor outcomes for health and wellbeing and transition programs for KS are lacking. We aimed to develop and test a mobile health tool (KS Transition Passport) to educate patients about KS, encourage self-management and support successful transition to adult-oriented care. First, we conducted a retrospective chart review and patient survey to examine KS transition at a university hospital. Second, we conducted a systematic scoping review of the literature on AYAs with KS. Last, we developed a mobile health transition passport and evaluated it with patient support groups. Participants evaluated the tool using the System Usability Scale and Patient Education Materials Assessment Tool (PEMAT). Chart review identified 21 AYAs diagnosed between 3.9-16.8 years-old (median 10.2 years). The survey revealed only 4/10 (40%) were on testosterone therapy and fewer (3/10, 30%) had regular medical care. The scoping review identified 21 relevant articles highlighting key aspects of care for AYAs with KS. An interprofessional team developed the mobile-health KS transition passport using an iterative process. Support group members (n=35) rated passport usability as 'ok' to 'good' (70 ± 20, median 73.5/100). Of PEMAT dimensions, 5/6 were deemed 'high quality' (86-90/100) and participants knew what to do with the information (actionability = 83/100). In conclusion, many patients with KS appear to have gaps in transition to adult-oriented care. Iterative development of a KS transition passport produced a mobile health tool that was usable, understandable and had high ratings for actionability.
Keywords
Adolescent, Child, Child, Preschool, Chronic Disease, Health Transition, Humans, Klinefelter Syndrome/diagnosis, Klinefelter Syndrome/therapy, Male, Puberty, Retrospective Studies, Telemedicine, Young Adult, Klinefelter syndrome (KS), adolescent, continuity of care, puberty, transition
Pubmed
Web of science
Open Access
Yes
Create date
13/07/2022 15:14
Last modification date
06/07/2023 6:00
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