Family Meetings Increase Recruitment and Engagement of Genetically Defined Communities in Simons Variation in Individuals Project (Simons VIP)

Background:

The Simons Variation in Individuals Project (Simons VIP) was established in 2010 to study groups of individuals who share recurrent genetic variants known to increase the risk of autism spectrum disorder and other neurodevelopmental conditions. In 2014 Simons VIP launched an online registry, enabling longitudinal data collection. One way to enhance engagement is to collaborate with self-organized communities who share a genetic etiology ("family groups") to coordinate or attend family meetings, either in-person or virtually. Virtual meetings are interactive webinars that share study results, present on special interest topics, and include discussion time. Goals of family meetings include providing education on the genetic condition, returning findings from the registry, performing research, and family interaction time.

Objectives:

The aim of this study was to evaluate the impact of engagement with genetically defined communities, at both in-person and virtual family meetings, on the enrollment and participation in the Simons VIP online registry.

Methods:

The impact of engaging with family groups was evaluated in ten family meetings taking place between 2016 and early 2018. Six of these meetings occurred in person, while four were virtual meetings. Main outcomes assessed included study enrollment and measure completion. Measures included: medical history and Vineland Adaptive Behavior Scales Second Edition (phone interview), and standardized online behavioral questionnaires. The study period was four-months starting three months prior to and one month after the meeting date, which contains active preparation time with the family group and follow up after the event. An increase was calculated as the change in enrollment or measures completed compared to the number of enrollments or measures completed at the start of the study period. Changes in main outcome measures for groups with family meetings were compared to groups without family meetings during the same date-matched time frames.

Results:

Compared to genetic variant groups without family meetings, we found an increase in both enrollment and measure completion among those groups with whom Simons VIP actively engaged with over the study period. On average, there was a 44% increase in registry enrollment per group with engaged communities, compared to a 17% increase for groups without engagement. We also found a 192% average increase in measure completion with engaged communities, compared to a 16% increase for groups without engagement during the same time period. Note that the average increase in enrollment and measure completion is primarily driven by inaugural meetings for respective family groups.

Conclusions:

Based on findings from the initial ten family meetings, there is evidence of increased registry activity for genetically defined communities when Simons VIP engaged with those groups to participate in family meetings, irrespective of format (in-person or virtual). The virtual family meeting model is scalable to a broader range of genetically defined groups, and may be appropriate for groups just beginning to organize. The percentage increases in enrollment and measure completion varied across groups. Further research is necessary to demonstrate the impact of this type of engagement on cohort recruitment and retention.