ArticleProceedings of the National Academy of Sciences of the United States of America2008
A role for a lithium-inhibited Golgi nucleotidase in skeletal development and sulfation.
Article in Proceedings of the National Academy of Sciences of the United States of America, 2008. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 43 papers.
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Who cites it
43 citing papers in PubMed, 66 citations in OpenAlex.
- MESH1 functions as a metazoan PAPS phosphatase to regulate sulfation.Nature chemical biology · 2026Article
- Proteoglycan inhibition of canonical BMP-dependent cartilage maturation delays endochondral ossification.Development (Cambridge, England) · 2024Article
- Impad1 and Syt11 work in an epistatic pathway that regulates EMT-mediated vesicular trafficking to drive lung cancer invasion and metastasis.Cell reports · 2022Article
- N7-methylguanosine modification of lncRNAs in a rat model of hypoxic pulmonary hypertension: a comprehensive analysis.BMC genomics · 2022Article
- Bisphosphate nucleotidase 2 (BPNT2), a molecular target of lithium, regulates chondroitin sulfation patterns in the cerebral cortex and hippocampus.Advances in biological regulation · 2022Article
- Roles of Chondroitin Sulfate Proteoglycans as Regulators of Skeletal Development.Frontiers in cell and developmental biology · 2022Review
- Sulfation of glycosaminoglycans depends on the catalytic activity of lithium-inhibited phosphatase BPNT2 in vitro.The Journal of biological chemistry · 2021Article
- Molecular signature of extracellular matrix pathology in schizophrenia.The European journal of neuroscience · 2021Article
- An Overview ofFrontiers in cell and developmental biology · 2021Review
- Congenital Disorders of Deficiency in Glycosaminoglycan Biosynthesis.Frontiers in genetics · 2021Review
- Chondrodysplasias With Multiple Dislocations Caused by Defects in Glycosaminoglycan Synthesis.Frontiers in genetics · 2021Review
- IMPAD1 and KDELR2 drive invasion and metastasis by enhancing Golgi-mediated secretion.Oncogene · 2020Article
- Modulation of sulfur assimilation metabolic toxicity overcomes anemia and hemochromatosis in mice.Advances in biological regulation · 2020Article
- Skeletal Dysplasias Caused by Sulfation Defects.International journal of molecular sciences · 2020Review
- Regulation of coenzyme A levels by degradation: the 'Ins and Outs'.Progress in lipid research · 2020Review
- Sulfation pathways from red to green.The Journal of biological chemistry · 2019Review
- Suppressing UPR-dependent overactivation of FGFR3 signaling ameliorates SLC26A2-deficient chondrodysplasias.EBioMedicine · 2019Article
- Testing the Cre-mediated genetic switch for the generation of conditional knock-in mice.PloS one · 2019Article
- Glycerophosphatidylcholine PC(36:1) absence and 3'-phosphoadenylate (pAp) accumulation are hallmarks of the human glioma metabolome.Scientific reports · 2018Article
- Modulation of intestinal sulfur assimilation metabolism regulates iron homeostasis.Proceedings of the National Academy of Sciences of the United States of America · 2018Article
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Authors and funding
7 authors at 3 institutions in 1 country.
Funding
Abstract
Sulfation is an important biological process that modulates the function of numerous molecules. It is directly mediated by cytosolic and Golgi sulfotransferases, which use 3'-phosphoadenosine 5'-phosphosulfate to produce sulfated acceptors and 3'-phosphoadenosine 5'-phosphate (PAP). Here, we identify a Golgi-resident PAP 3'-phosphatase (gPAPP) and demonstrate that its activity is potently inhibited by lithium in vitro. The inactivation of gPAPP in mice led to neonatal lethality, lung abnormalities resembling atelectasis, and dwarfism characterized by aberrant cartilage morphology. The phenotypic similarities of gPAPP mutant mice to chondrodysplastic models harboring mutations within components of the sulfation pathway lead to the discovery of undersulfated chondroitin in the absence of functional enzyme. Additionally, we observed loss of gPAPP leads to perturbations in the levels of heparan sulfate species in lung tissue and whole embryos. Our data are consistent with a model that clearance of the nucleotide product of sulfotransferases within the Golgi plays an important role in glycosaminoglycan sulfation, provide a unique genetic basis for chondrodysplasia, and define a function for gPAPP in the formation of skeletal elements derived through endochondral ossification.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.