Advancing qualitative rare disease research methodology: a comparison of virtual and in-person focus group formats
| dc.contributor.author | Dwyer, Andrew A. | |
| dc.contributor.author | Uveges, Melissa | |
| dc.contributor.author | Dockray, Samantha | |
| dc.contributor.author | Smith, Neil | |
| dc.contributor.funder | National Institutes of Health | en |
| dc.date.accessioned | 2022-09-29T12:49:02Z | |
| dc.date.available | 2022-09-29T12:49:02Z | |
| dc.date.issued | 2022-09-11 | |
| dc.date.updated | 2022-09-27T15:59:42Z | |
| dc.description.abstract | Background: Rare disease research is hampered in part by the fact that patients are geographically dispersed. Rare disease patient communities are recognized for their use of the internet to learn about their condition and find peer-to-peer support. As such, web-based technologies offer promise for overcoming geographic barriers in rare disease research for many. Qualitative focus groups (FGs) are a widely used methodology used to understand patients and parents/families ‘lived experience’ and unmet needs is important to improve care for rare diseases. It is unclear if web-enabled (virtual) FGs are comparable to traditional in-person approaches. We conducted in-person (n = 3) and virtual (n = 3) FGs with rare disease patients to determine if virtual FGs produce similar results in-person FGs. Results: Three in-person (n = 33 participants) and three virtual (n = 25 participants) FGs were conducted examining attitudes and beliefs regarding genetic testing and family communication of risk. Participants included 30 males, 18 females, and 10 parents/guardians. Two independent investigators identified excerpts (meaningful sections of text) and coded themes/sub-themes using a codebook. Inter-coder agreement across identified excerpts (n = 530) in both FG formats was 844/875 (96.5%). Two additional investigators reviewed coded excerpts and did not identify additional themes/sub-themes—supporting data saturation across FG formats. Virtual FGs accounted for 303/530 (57.2%) of total excerpts and 957/1721 (55.7%) of all identified themes/sub-themes. Formats were similar in terms of overall number of excerpts (101 ± 7.8 vs. 75.7 ± 18.8, p = 0.26) and themes/sub-themes (319 ± 6.1 vs. 254.7 ± 103.6, p = 0.34) between virtual and in-person FGs. However, virtual FGs had significantly more coded excerpts specifically relating to sensitive/intimate topics including ‘attitudes and beliefs’ (n = 320 vs. n = 235, p < 0.001), ‘information and support’ (n = 184 vs. n = 99, p < 0.001), and ‘family communication’ (n = 208 vs. n = 114, p < 0.001). Conclusions: Virtual FGs yielded similar numbers of coded excerpts compared to traditional in-person FGs. Virtual FGs appear to support the relative anonymity of participants, resulting in richer discussion of highly sensitive, intimate topics. Findings support the validity and methodologic rigor of using web-enabled technologies for conducting FGs in rare diseases. | en |
| dc.description.sponsorship | National Institutes of Health (Eunice Kennedy Shriver National Institute of Child Health and Human Development 1P50HD104224-01) | en |
| dc.description.status | Peer reviewed | en |
| dc.description.version | Published Version | en |
| dc.format.mimetype | application/pdf | en |
| dc.identifier.articleid | 354 | en |
| dc.identifier.citation | Dwyer, A. A., Uveges, M., Dockray, S. and Smith, N. (2022) 'Advancing qualitative rare disease research methodology: a comparison of virtual and in-person focus group formats', Orphanet Journal of Rare Diseases, 17(1), 354 (8pp). doi: 10.1186/s13023-022-02522-3 | en |
| dc.identifier.doi | 10.1186/s13023-022-02522-3 | en |
| dc.identifier.eissn | 1750-1172 | |
| dc.identifier.endpage | 8 | en |
| dc.identifier.issued | 1 | en |
| dc.identifier.journaltitle | Orphanet Journal of Rare Diseases | en |
| dc.identifier.startpage | 1 | en |
| dc.identifier.uri | https://hdl.handle.net/10468/13700 | |
| dc.identifier.volume | 17 | en |
| dc.language.iso | en | en |
| dc.publisher | BioMed Central Ltd. | en |
| dc.rights | © The Authors, 2022. Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. The Creative Commons Public Domain Dedication waiver (http://creativecom mons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data. | en |
| dc.rights.uri | https://creativecommons.org/licenses/by/4.0/ | en |
| dc.subject | Community based participatory research | en |
| dc.subject | Genetic testing | en |
| dc.subject | Hypogonadotropic hypogonadism | en |
| dc.subject | Kallmann syndrome | en |
| dc.subject | Qualitative research methods | en |
| dc.subject | Rare disease | en |
| dc.title | Advancing qualitative rare disease research methodology: a comparison of virtual and in-person focus group formats | en |
| dc.type | Article (peer-reviewed) | en |
