ArticleThe Journal of biological chemistry2010
Deficiency of a transmembrane prolyl 4-hydroxylase in the zebrafish leads to basement membrane defects and compromised kidney function.
Article in The Journal of biological chemistry, 2010. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 16 papers, 1 of them a synthesis that pooled it.
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Who cites it
16 citing papers in PubMed, 1 synthesis or guideline pooled it, 42 citations in OpenAlex.
- Genetic association for renal traits among participants of African ancestry reveals new loci for renal function.PLoS genetics · 2011Pooled it
- Dangguibuxue Decoction Attenuated AA I-Induced Renal Fibrosis: Integrating Network Pharmacology and Experimental Validation.Pharmaceuticals (Basel, Switzerland) · 2026Article
- Recent Advances and Therapeutic Implications of 2-Oxoglutarate-Dependent Dioxygenases in Ischemic Stroke.Molecular neurobiology · 2024Review
- Article
- Loss of the collagen IV modifier prolyl 3-hydroxylase 2 causes thin basement membrane nephropathy.The Journal of clinical investigation · 2022Article
- High-calorie diet results in reversible obesity-related glomerulopathy in adult zebrafish regardless of dietary fat.American journal of physiology. Renal physiology · 2022Article
- Oxygen-sensing mechanisms in development and tissue repair.Development (Cambridge, England) · 2021Review
- Effects of dietary hydroxyproline on collagen metabolism, proline 4-hydroxylase activity, and expression of related gene in swim bladder of juvenile Nibea diacanthus.Fish physiology and biochemistry · 2019Article
- Biallelic loss-of-function P4HTM gene variants cause hypotonia, hypoventilation, intellectual disability, dysautonomia, epilepsy, and eye abnormalities (HIDEA syndrome).Genetics in medicine : official journal of the American College of Medical Genetics · 2019Article
- von Hippel-Lindau gene plays a role during zebrafish pronephros development.In vitro cellular & developmental biology. Animal · 2015Article
- Anaemia in kidney disease: harnessing hypoxia responses for therapy.Nature reviews. Nephrology · 2015Review
- Using zebrafish to study podocyte genesis during kidney development and regeneration.Genesis (New York, N.Y. : 2000) · 2014Review
- Molecular Mechanisms of Podocyte Development Revealed by Zebrafish Kidney Research.Cell & developmental biology · 2014Article
- "Zebrafishing" for novel genes relevant to the glomerular filtration barrier.BioMed research international · 2013Review
- Regulation of erythropoiesis by hypoxia-inducible factors.Blood reviews · 2013Review
- Structure of transmembrane prolyl 4-hydroxylase reveals unique organization of EF and dioxygenase domains.The Journal of biological chemistryArticle
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Authors and funding
8 authors at 4 institutions in 2 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Prolyl 4-hydroxylases (P4Hs) catalyze the hydroxylation of collagens and hypoxia-inducible factor (HIF)-α subunits. We studied the zebrafish homologue of the recently characterized human transmembrane P4H (P4H-TM) that can hydroxylate HIF-α, but not collagens, in vitro and influence HIF-α levels in cellulo. The zebrafish P4H-TM mRNA had its highest expression in the eye and brain and lower levels in other tissues, including the kidney. Morpholino knockdown of P4H-TM in embryos resulted in a reduction in the size of the eye and head and morphological alterations in the head from 2 days postfertilization onward. In addition, pericardial edema, regarded as a sign of kidney dysfunction, developed from 3 days postfertilization onward. The phenotype was dependent on the P4H-TM catalytic activity because similar results were obtained with morpholinos targeting either translation initiation or catalytic residues of the enzyme. Structural and functional analyses of the morphant pronephric kidneys revealed fragmented glomerular basement membranes (BMs), disorganized podocyte foot processes, and severely compromised pronephric kidney function leading to proteinuria. The opacity of the eye lens was increased due to the presence of extra nuclei and deposits, and the structure of the lens capsule BM was altered. Our data suggest that P4H-TM catalytic activity is required for the proper development of the glomerular and lens capsule BMs. Many HIF target genes were induced in the P4H-TM-deficient morphants, but the observed phenotype is not likely to be mediated at least solely via the HIF pathway, and thus P4H-TM probably has additional, as yet unknown, substrates.
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Registered trials
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