Enhancing Value and Uptake for Whole-Population Cohorts of Children and Parents: Methods to Integrate Registries into the Generation Victoria Cohort

Abstract

Health registries are critical to understanding, benchmarking and improving quality of care for specific diseases and conditions, but face hurdles including funding, bias towards clinical rather than population samples, lack of pre-morbid and outcomes data, and absent cross-registry harmonisation and coordination. Children are particularly under-represented in registry research. This paper lays out novel principles, methods and governance to integrate diverse registries within or alongside a planned children's mega-cohort to rapidly generate translatable evidence. GenV (Generation Victoria) will approach for recruitment parents of all newborns (estimated 150,000) over two years from mid-2021 in the state of Victoria (population 6.5 million), Australia. Its sample size and population denominator mean it will contain almost all children with uncommon or co-morbid conditions as they emerge over time. By design, it will include linked datasets, biosamples (including from pregnancy), phenotypes and participant-reported measures, all of which will span pre-morbid to long-term outcomes. We provide a vignette of a planned new registry for high-risk pregnancies to illustrate the possibilities. To our knowledge, this is the first paper to describe such a methodology designed prospectively to enhance both the clinical relevance of a large multipurpose cohort and the value and inclusivity of registries in a population.

Keywords: GenV (Generation Victoria); children; population studies; registries; registry trials; research methodology.

Figure 1

Principles for registries collaborating with GenV.

Figure 2

Conceptual relationship of hypothetical registries/depth sub-cohorts to children born in the GenV window. SCN = special care nursery; ASD = autism spectrum disorder; MFM = maternal fetal medicine; DDH = developmental dysplasia of the hip; Ax = assessment. * = existing registries. Refer to Figure 3 regarding Models 1 & 2.

Figure 3

Process flowchart for registries within and alongside GenV. Note: further information on data sharing is captured in Figure 5. PII = personal identifying information required for registry to approach participants to invite into registry.

Figure 4

Benefits of registries sharing data with GenV and of GenV sharing data with registries. Applies to children born in the GenV window (2021–2023) and their parents.

Figure 5

GenV data sharing models. This figure illustrates flow from GenV to registries. The reverse is also expected; that is, the figure can be mirrored to illustrate flow from registries to GenV.