Redundant function of the heparan sulfate 6-O-endosulfatases Sulf1 and Sulf2 during skeletal development

Ratzka A, Kalus I, Moser M, Dierks T, Mundlos S, Vortkamp A (2008)
DEVELOPMENTAL DYNAMICS 237(2): 339-353.

Zeitschriftenaufsatz | Veröffentlicht | Englisch
 
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Abstract / Bemerkung
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.
Stichworte
heparan sulfate; ossification; endochondral; chondrocyte differentiation; bone; sulfatase; cartilage; Sulf2; Sulf1; mouse
Erscheinungsjahr
2008
Zeitschriftentitel
DEVELOPMENTAL DYNAMICS
Band
237
Ausgabe
2
Seite(n)
339-353
ISSN
1058-8388
Page URI
https://pub.uni-bielefeld.de/record/1592682

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Ratzka A, Kalus I, Moser M, Dierks T, Mundlos S, Vortkamp A. Redundant function of the heparan sulfate 6-O-endosulfatases Sulf1 and Sulf2 during skeletal development. DEVELOPMENTAL DYNAMICS. 2008;237(2):339-353.
Ratzka, A., Kalus, I., Moser, M., Dierks, T., Mundlos, S., & Vortkamp, A. (2008). Redundant function of the heparan sulfate 6-O-endosulfatases Sulf1 and Sulf2 during skeletal development. DEVELOPMENTAL DYNAMICS, 237(2), 339-353. doi:10.1002/dvdy.21423
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. DEVELOPMENTAL DYNAMICS 237, 339-353.
Ratzka, A., et al., 2008. Redundant function of the heparan sulfate 6-O-endosulfatases Sulf1 and Sulf2 during skeletal development. DEVELOPMENTAL DYNAMICS, 237(2), p 339-353.
A. Ratzka, et al., “Redundant function of the heparan sulfate 6-O-endosulfatases Sulf1 and Sulf2 during skeletal development”, DEVELOPMENTAL DYNAMICS, vol. 237, 2008, pp. 339-353.
Ratzka, A., Kalus, I., Moser, M., Dierks, T., Mundlos, S., Vortkamp, A.: Redundant function of the heparan sulfate 6-O-endosulfatases Sulf1 and Sulf2 during skeletal development. DEVELOPMENTAL DYNAMICS. 237, 339-353 (2008).
Ratzka, Andreas, Kalus, Ina, Moser, Markus, Dierks, Thomas, Mundlos, Stefan, and Vortkamp, Andrea. “Redundant function of the heparan sulfate 6-O-endosulfatases Sulf1 and Sulf2 during skeletal development”. DEVELOPMENTAL DYNAMICS 237.2 (2008): 339-353.

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