William F. Connell School of Nursing, Boston College, Chestnut Hill, MA, USA.
Massachusetts General Hospital-Harvard Center for Reproductive Medicine, Boston, MA, USA.
Orphanet J Rare Dis. 2022 Sep 11;17(1):354. doi: 10.1186/s13023-022-02522-3.
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.
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).
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.
罕见病研究受到部分阻碍，原因是患者分布在不同的地理位置。罕见病患者群体以使用互联网来了解病情和寻找同病相怜的患者而闻名。因此，基于网络的技术有望为许多人克服罕见病研究中的地理障碍。定性焦点小组（FG）是一种广泛使用的方法，用于了解患者和父母/家庭的“生活体验”和未满足的需求，这对于改善罕见病的护理很重要。目前尚不清楚是否可以将基于网络的（虚拟）FG 与传统的面对面方法进行比较。我们对罕见病患者进行了面对面（n=3）和虚拟（n=3）FG，以确定虚拟 FG 是否能产生与面对面 FG 类似的结果。
进行了三次面对面（n=33 名参与者）和三次虚拟（n=25 名参与者）FG，以了解参与者对遗传检测和家庭风险沟通的态度和信念。参与者包括 30 名男性、18 名女性和 10 名父母/监护人。两名独立的研究人员使用代码簿确定了摘录（有意义的文本段）和编码主题/子主题。两种 FG 格式中，两个独立的研究人员识别的摘录（n=530）之间的编码一致性为 844/875（96.5%）。另外两名研究人员审查了编码摘录，并未在 FG 格式中发现其他主题/子主题，表明数据已经达到饱和。虚拟 FG 占 530 个摘录的 303/530（57.2%），占 1721 个已识别主题/子主题的 957/1721（55.7%）。虚拟 FG 和面对面 FG 在摘录总数（101±7.8 与 75.7±18.8，p=0.26）和主题/子主题（319±6.1 与 254.7±103.6，p=0.34）方面相似。然而，虚拟 FG 有明显更多的编码摘录专门涉及敏感/私密主题，包括“态度和信念”（n=320 与 n=235，p<0.001）、“信息和支持”（n=184 与 n=99，p<0.001）和“家庭沟通”（n=208 与 n=114，p<0.001）。
虚拟 FG 产生的编码摘录数量与传统的面对面 FG 相似。虚拟 FG 似乎支持参与者的相对匿名性，从而促进了对高度敏感和私密话题的更深入讨论。研究结果支持在罕见病中使用基于网络的技术进行 FG 的有效性和方法学严谨性。
