Lessons learned about harmonizing survey measures for the CSER consortium

Abstract

Introduction: Implementation of genome-scale sequencing in clinical care has significant challenges: the technology is highly dimensional with many kinds of potential results, results interpretation and delivery require expertise and coordination across multiple medical specialties, clinical utility may be uncertain, and there may be broader familial or societal implications beyond the individual participant. Transdisciplinary consortia and collaborative team science are well poised to address these challenges. However, understanding the complex web of organizational, institutional, physical, environmental, technologic, and other political and societal factors that influence the effectiveness of consortia is understudied. We describe our experience working in the Clinical Sequencing Evidence-Generating Research (CSER) consortium, a multi-institutional translational genomics consortium. Methods: A key aspect of the CSER consortium was the juxtaposition of site-specific measures with the need to identify consensus measures related to clinical utility and to create a core set of harmonized measures. During this harmonization process, we sought to minimize participant burden, accommodate projectspecific choices, and use validated measures that allow data sharing. Results: Identifying platforms to ensure swift communication between teams and management of materials and data were essential to our harmonization efforts. Funding agencies can help consortia by clarifying key study design elements across projects during the proposal preparation phase and by providing a framework for data sharing data across participating projects. Conclusions: In summary, time and resources must be devoted to developing and implementing collaborative practices as preparatory work at the beginning of project timelines to improve the effectiveness of research consortia.

Examining access to care in clinical genomic research and medicine: Experiences from the CSER Consortium

“…CSER collected participant data through surveys with measures harmonized across projects where possible. 27,28 Baseline surveys asked about access-related sociodemographic factors, and about barriers to care directly through a two-item harmonized measure adapted from the Agency for Healthcare Research and Quality's Medical Expenditure Panel Survey. 29 The CSER measure asked whether respondents experienced barriers to accessing clinical care for themselves (adult patients) or for their children (pediatric patients) in the past year with multiple response options; respondents could select all that applied.…”

“…Evaluating Access to Care in CSER CSER collected participant data through surveys with measures harmonized across projects where possible [27][28][29]. Baseline surveys asked about access-related sociodemographic factors and about barriers to care directly through a two-item harmonized measure adapted from the Agency for Healthcare Research and Quality's Medical Expenditure Panel Survey [30].…”

“…Most participants had realistic expectations of sequencing and high levels of optimism, openness, and resilience. Our experience suggests that groups who have faced historical discrimination and exploitation in scientific research, and thus have reasons to mistrust researchers, may be more skeptical when weighing the risks Adapted from Amendola et al [18] and Goddard et al [27]. c ClinSeq completed enrollment at the start of the extramural studies and thus did not assess the access to care or other non-access-related variables as in other CSER projects.…”

“…Effective access also includes elements of patient satisfaction and health outcomes, which encompass the understudied topic of culturally competent and language concordant genomic communication. CSER has begun to fill these gaps through multiple avenues [27,87,88], including evaluating novel language-tailored genomics content and communication strategies like NYCKidSeq's GUÍA tool [61]. Continued research utilizing stakeholder engagement and community-based participatory approaches [48,64,89,90] is needed to provide further insight into the experience of LEP populations, the impact of language barriers on quality of care, and the relationship between language and literacy barriers in genomics.…”

Integration of stakeholder engagement from development to dissemination in genomic medicine research: Approaches and outcomes from the CSER Consortium

“…Each CSER site is unique with different aims, processes, and targeted enrollment populations as described in Table 1. 10,11 Therefore, each project planned and carried out different stakeholder engagement activities with differing goals and expected outcomes (Figure 2). Approaches were selected from each of the active enrollment sites to demonstrate a variety of methods.…”