https://orcid.org/0000-0002-5926-0478
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ABSTRACT
Background
Objectives were to 1) assess COVID-19-associated medical and psychological challenges facing persons with inherited bleeding disorders (PIBD) and their parents/guardians (PG) in Germany, the US, and the UK; 2) describe similarities and differences among these countries; 3) identify needs and opportunities for intervention by patient advocacy organizations (PAGs).
Research Design & Methods
A cross-sectional, international survey was conducted in three countries using validated psychometric instruments and investigator-developed items.
Results
Five hundred and four surveys were included. Significant differences between countries were found including experiences with medical care, specific thoughts, and concerns about COVID-19, anxiety, and other mental health measures, as well as resources used to cope with stress. Age, education, income, race, IBD diagnosis, PIBD vs. BD group, and gender had moderating effects on resources used. Communication with friends/relatives and use of PAG and HTCs as resources for information/coping decreased in all countries during the pandemic.
Conclusions
There were similarities and differences between respondents across the country in the perceived impact of the pandemic, mental health scores, and strategies used to cope with stress. Recommendations: strategies to increase PAG access for PIBD and their PG during pandemics and natural disasters, ongoing assessment and adaptation to provide supportive resources to specific patient subgroups.
Plain Language Summary
Persons with inherited bleeding disorders (PIBD) and their parents faced many challenges during the COVID-19 pandemic. An online survey was conducted within three countries: Germany, the United States, and the United Kingdom to explore these challenges and address how patient advocacy organizations can better meet their needs. Areas explored included experiences with medical care, concerns, and thoughts expressed during the pandemic, and coping resources used before and during the pandemic. In addition, mental health issues were explored addressing anxiety, COVID-related fears, depression, and resilience. Differences were found regarding experiences with medical care, specific thoughts, and concerns about COVID-19, anxiety, and other mental health measures, as well as resources used to cope with stress. These results provide an opportunity for advocacy organizations for PIBD to develop appropriate assessment, adaptation, and education resources to help patients during pandemics and/or natural disasters in the future.
Declaration of interest
M Santaella is employed with NBDF. C Nichols is contracted by NBDF as a statistician. A Lambing was part of the NBDF speaker’s bureau at the time of this manuscript. M Wiktop was employed with NHF at the time of this manuscript. The remaining authors have no relevant affiliations or financial involvement with any organization or entity with a financial interest in or financial conflict with the subject matter or materials discussed in the manuscript. This includes employment, consultancies, honoraria, stock ownership or options, expert testimony, grants or patents received or pending, or royalties.
Reviewer disclosures
Peer reviewers in this manuscript have no relevant financial relationships or otherwise to disclose.
Ethics approval and consent to participate
This survey has been performed in accordance with the principles stated in the Declaration of Helsinki. Each country’s Internal Review Board determined the study to have exempt status. Although this survey was anonymous, PIBD and caregivers were asked to give their informed consent to participate in the research by ticking a box in the online survey after reading the study information.
Data availability statement
The analyzed data sets generated during the present study are available from the corresponding author on reasonable request.
Author contributions
S von Mackensen, K Khair, M Santaella, and M Witkop designed the study and developed the survey questionnaire. S von Mackensen, K Kair, M Santaella, and M Witkop recruited PIBD in the respective countries through the patient advocacy group (PAGs). C Nichols performed statistical analysis. All authors contributed to data analysis and interpretation. A Lambing drafted the initial manuscript. All authors agreed on the journal to which the article will be submitted, critically revised the manuscript, and all authors approved the final version of the manuscript for publication and any significant changes introduced in the proofing stage, and agree to take responsibility and accountability for the contents of the article and share responsibility to resolve any questions raised about the accuracy of integrity of the published work.
Acknowledgements
We thank Mabel Crescioni who had worked at Hemophilia Federation of America at the time of the design of the study for her contribution to develop the survey. We would also like to thank Yves Douma, who assisted us in the search and selection of the appropriate validated Mental Health Questionnaires which are included in this survey. We are grateful to all PAQs in Germany (Deutsche Hämophiliegesellschaft (DHG), Interessengemeinschaft Hämophiler (IGH) and Deutsche Bluthilfe (DBH)); the US (National Bleeding Disorders Foundation (NBDF), Hemophilia Federation of America (HFA) and chapter organizations) and the UK (The Haemophilia Society) who helped to recruit PIBD for this survey. Finally, we would like to thank all patients and caregivers of children with inherited bleeding disorders for their participation in this survey.
Supplementary material
Supplemental data for this article can be accessed online at https://doi.org/10.1080/17474086.2023.2277323