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PMID:18213582

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Citation

Ratzka, A, Kalus, I, Moser, M, Dierks, T, Mundlos, S and Vortkamp, A (2008) Redundant function of the heparan sulfate 6-O-endosulfatases Sulf1 and Sulf2 during skeletal development. Dev. Dyn. 237:339-53

Abstract

Modification of the sulfation pattern of heparan sulfate (HS) during organ development is thought to regulate binding and signal transduction of several growth factors. The secreted sulfatases, Sulf1 and Sulf2, desulfate HS on 6-O-positions extracellularly. We show that both sulfatases are expressed in overlapping patterns during embryonic skeletal development. Analysis of compound mutants of Sulf1 and Sulf2 derived from gene trap insertions and targeted null alleles revealed subtle but distinct skeletal malformations including reduced bone length, premature vertebrae ossification and fusions of sternebrae and tail vertebrae. Molecular analysis of endochondral ossification points to a function of Sulf1 and Sulf2 in delaying the differentiation of endochondral bones. Penetrance and severity of the phenotype increased with reduced numbers of functional alleles indicating redundant functions of both sulfatases. The mild skeletal phenotype of double mutants suggests a role for extracellular modification of 6-O-sulfation in fine-tuning rather than regulating the development of skeletal structures.

Links

PubMed Online version:10.1002/dvdy.21423

Keywords

Animals; Bone and Bones/embryology; DNA Primers/genetics; Heparitin Sulfate/metabolism; Immunohistochemistry; In Situ Hybridization; Mice; Mutation/genetics; Osteogenesis/physiology; Phenotype; Reverse Transcriptase Polymerase Chain Reaction; Signal Transduction/physiology; Sulfatases/genetics; Sulfatases/metabolism; Sulfotransferases/genetics; Sulfotransferases/metabolism

Significance

Annotations

Gene product Qualifier GO Term Evidence Code with/from Aspect Extension Notes Status

MOUSE:SULF1

involved_in

GO:0048706: embryonic skeletal system development

ECO:0000316: genetic interaction evidence used in manual assertion

UniProtKB:Q8CFG0

P

Seeded From UniProt

complete

MOUSE:SULF2

involved_in

GO:0048706: embryonic skeletal system development

ECO:0000316: genetic interaction evidence used in manual assertion

UniProtKB:Q8CFG0

P

Seeded From UniProt

complete


See also

References

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