Genetically modified animals as models of neurodevelopmental conditions: A review of systematic review reporting quality.

Abstract

Using genetically modified animals to model neurodevelopmental conditions helps better our understanding of biology underlying these conditions. Animal research has unique characteristics not shared with clinical research, meaning systematic review methods must be adapted to this context. We aim to evaluate the quantity, characteristics, and reporting quality of systematic reviews which synthesise research using genetically modified animals to model neurodevelopmental conditions. On 23 January 2023, we searched PubMed, Embase, and the Web of Science Core Collection to identify systematic reviews of genetic neurodevelopmental condition animal research where the modified gene was one in a list of 102 genes associated with neurodevelopmental conditions identified through large-scale exome sequencing or Fmr1, Mecp2, or Ube3a. Two independent reviewers screened studies based on full text and assessed the reporting quality of relevant reviews using an adapted version of the PRISMA checklist (PRISMA-Pre). Twelve review publications met our criteria. We found mixed levels of reporting: items such as identifying the publication as a systematic review in the title, search strategies, and funding sources being well reported, and others such as protocol registration and data sharing less well reported. We also identified 19 review registrations via PROSPERO, most of which remain unpublished after their anticipated end dates. Systematic reviews are limited by lack of reporting. Increased awareness of reporting guidelines may help authors increase the transparency and reproducibility, and therefore the reliability, of their systematic reviews.