Table of Contents
- Fruit flies on the front line: the translational impact of Drosophila
Drosophila Collection: This Editorial introduces a new Special Collection, ‘Spotlight on Drosophila: Translational Impact’, providing a summary of its contents so far and highlighting the impact of Drosophila as a model for human diseases.
AT A GLANCE
- Drosophila tools and assays for the study of human diseases
Editors' choice - Drosophila Collection: In this review and accompanying poster, we highlight the physiological and molecular parallels between fly and human organs that validate the use of Drosophila to study the molecular pathogenesis underlying human diseases.
- FlyBase portals to human disease research using Drosophila models
Drosophila Collection: Drosophila melanogaster is well established as a model for studying human disease. Here, we highlight recent efforts to enhance the availability and accessibility of disease model data in FlyBase, the model organism database for Drosophila.
- Modeling congenital disease and inborn errors of development in Drosophila melanogaster
Drosophila Collection: This Review highlights several Drosophila models of human inborn errors of development, with particular emphasis on the strategies geneticists have developed to break through the current genotype-to-phenotype bottleneck.
- The interplay between intestinal bacteria and host metabolism in health and disease: lessons from Drosophila melanogaster
Drosophila Collection: Drosophila melanogaster is an ideal host in which to dissect the impact of intestinal microbes on host metabolism.
- Glial expression of Swiss cheese (SWS), the Drosophila orthologue of neuropathy target esterase (NTE), is required for neuronal ensheathment and function
Drosophila Collection: Loss of sws in glia results in locomotion deficits, suggesting that glial changes contribute to the paralysis and spastic paraplegia in humans carrying mutations in its orthologue, NTE.
- The calcineurin inhibitor Sarah (Nebula) exacerbates Aβ42 phenotypes in a Drosophila model of Alzheimer's disease
Drosophila Collection: Chronically increased levels of Sarah (Nebula), a calcineurin inhibitor, cause mitochondria dysfunction and subsequently increased Aβ42-induced cytotoxicity in Drosophila.
- Tau excess impairs mitosis and kinesin-5 function, leading to aneuploidy and cell death
Drosophila Collection: We show that Tau, a microtubule-binding protein involved in many neurodegenerative diseases, impairs mitosis when in excess. We show that this occurs via the inhibition of the kinesin-5 mitotic motor.
- Assessing a peptidylic inhibitor-based therapeutic approach that simultaneously suppresses polyglutamine RNA- and protein-mediated toxicities in patient cells and Drosophila
Drosophila Collection: We identified a novel peptidylic inhibitor against expanded CAG RNA toxicity. Our study highlights the importance of simultaneously targeting both toxic RNA and protein species in treating polyQ degeneration.
- Cardiomyocyte-specific conditional knockout of the histone chaperone HIRA in mice results in hypertrophy, sarcolemmal damage and focal replacement fibrosis
Summary: Deletion of HIRA, a gene encoding a histone chaperone that lies within the critical region of 22q11.2 deletion syndrome, alters cardiac gene expression and leads to cardiomyopathy.
- Mouse myofibers lacking the SMYD1 methyltransferase are susceptible to atrophy, internalization of nuclei and myofibrillar disarray
Summary: Elimination of the lysine methyltransferase SMYD1 from mouse skeletal muscle caused myopathy with excessive internal nuclei, atrophy, myofibrillar disorganization and broad upregulation of muscle gene expression.