Advancing qualitative rare disease research methodology: a comparison of virtual and in-person focus group formats

Andrew A Dwyer, Melissa Uveges, Samantha Dockray, Neil Smith, Andrew A Dwyer, Melissa Uveges, Samantha Dockray, Neil Smith

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.

Keywords: Community based participatory research; Genetic testing; Hypogonadotropic hypogonadism; Kallmann syndrome; Qualitative research methods; Rare disease.

Conflict of interest statement

The authors have no financial or non-financial competing interests to declare.

© 2022. The Author(s).

Figures

Fig. 1
Fig. 1
Percentage of total excerpts and codes by focus group. The first in-person FG (black bar) was used to create the code book and was set as the ‘standard’. A Excerpts from in person FGs (n = 113, 53, 61 respectively) did not differ from virtual FGs (n = 96, 110, 97 respectively). B Codes from in-person FGs (n = 374, 188, 202 respectively) did not differ from virtual FGs (n = 315, 326, 316 respectively)

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