ArticleDisease models & mechanisms
UDP-galactose 4' epimerase (GALE) is essential for development of Drosophila melanogaster.
Article in Disease models & mechanisms. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 29 papers, 1 of them a synthesis that pooled it.
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Who cites it
29 citing papers in PubMed, 1 synthesis or guideline pooled it, 53 citations in OpenAlex.
- Pathophysiology and targets for treatment in hereditary galactosemia: A systematic review of animal and cellular models.Journal of inherited metabolic disease · 2020Pooled it
- Design rules for bioorthogonal glycan-based tools.The Biochemical journal · 2026Review
- An in vivo chemical genetic approach for targeted glycoproteome analysis inbioRxiv : the preprint server for biology · 2026Article
- Tissue and cellular spatiotemporal dynamics in colon aging.Nature biotechnology · 2026Article
- Comparative transcriptome analysis of midgut of Bombyx mori under a sucrose-supplemented diet.Scientific reports · 2025Article
- Enhancing mitochondrial one-carbon metabolism is neuroprotective in Alzheimer's disease models.Cell death & disease · 2024Article
- Restoring galactose metabolism without restoring GALT rescues both compromised survival in larvae and an adult climbing deficit in a GALT-null D. Melanogaster model of classic galactosemia.Journal of inherited metabolic disease · 2024Article
- GALDAR: A genetically encoded galactose sensor for visualizing sugar metabolism in vivo.PLoS biology · 2024Article
- Rare sugar L-sorbose exerts antitumor activity by impairing glucose metabolism.Communications biology · 2023Article
- Bombyx mori β1,4-N-acetylgalactosaminyltransferase possesses relaxed donor substrate specificity in N-glycan synthesis.Scientific reports · 2021Article
- Galactosemia: Towards Pharmacological Chaperones.Journal of personalized medicine · 2021Review
- RNAi-Mediated Silencing of Pgants Shows Core 1Frontiers in physiology · 2021Article
- Association of UDP-galactose-4-epimerase with milk protein concentration in the Chinese Holstein population.Asian-Australasian journal of animal sciences · 2020Article
- Metabolic precision labeling enables selective probing of O-linkedProceedings of the National Academy of Sciences of the United States of America · 2020Article
- A galactose-1-phosphate uridylyltransferase-null rat model of classic galactosemia mimics relevant patient outcomes and reveals tissue-specific and longitudinal differences in galactose metabolism.Journal of inherited metabolic disease · 2020Article
- Human UDP-galactose 4'-epimerase (GALE) is required for cell-surface glycome structure and function.The Journal of biological chemistry · 2020Article
- Drosophila melanogaster Models of Galactosemia.Current topics in developmental biology · 2017Review
- Acute and long-term outcomes in a Drosophila melanogaster model of classic galactosemia occur independently of galactose-1-phosphate accumulation.Disease models & mechanisms · 2016Article
- Coordinated movement, neuromuscular synaptogenesis and trans-synaptic signaling defects in Drosophila galactosemia models.Human molecular genetics · 2016Article
- Disruption of Methionine Metabolism in Drosophila melanogaster Impacts Histone Methylation and Results in Loss of Viability.G3 (Bethesda, Md.) · 2015Article
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4 authors at 1 institution in 1 country.
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Abstract
UDP-galactose 4' epimerase (GALE) catalyzes the interconversion of UDP-galactose and UDP-glucose in the final step of the Leloir pathway; human GALE (hGALE) also interconverts UDP-N-acetylgalactosamine and UDP-N-acetylglucosamine. GALE therefore plays key roles in the metabolism of dietary galactose, in the production of endogenous galactose, and in maintaining the ratios of key substrates for glycoprotein and glycolipid biosynthesis. Partial impairment of hGALE results in the potentially lethal disorder epimerase-deficiency galactosemia. We report here the generation and initial characterization of a first whole-animal model of GALE deficiency using the fruit fly Drosophila melanogaster. Our results confirm that GALE function is essential in developing animals; Drosophila lacking GALE die as embryos but are rescued by the expression of a human GALE transgene. Larvae in which GALE has been conditionally knocked down die within days of GALE loss. Conditional knockdown and transgene expression studies further demonstrate that GALE expression in the gut primordium and Malpighian tubules is both necessary and sufficient for survival. Finally, like patients with generalized epimerase deficiency galactosemia, Drosophila with partial GALE loss survive in the absence of galactose but succumb in development if exposed to dietary galactose. These data establish the utility of the fly model of GALE deficiency and set the stage for future studies to define the mechanism(s) and modifiers of outcome in epimerase deficiency galactosemia.
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