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The BMP antagonist noggin regulates cranial suture fusion.
Nature. 2003 Apr 10; 422(6932):625-9.Nat

Abstract

During skull development, the cranial connective tissue framework undergoes intramembranous ossification to form skull bones (calvaria). As the calvarial bones advance to envelop the brain, fibrous sutures form between the calvarial plates. Expansion of the brain is coupled with calvarial growth through a series of tissue interactions within the cranial suture complex. Craniosynostosis, or premature cranial suture fusion, results in an abnormal skull shape, blindness and mental retardation. Recent studies have demonstrated that gain-of-function mutations in fibroblast growth factor receptors (fgfr) are associated with syndromic forms of craniosynostosis. Noggin, an antagonist of bone morphogenetic proteins (BMPs), is required for embryonic neural tube, somites and skeleton patterning. Here we show that noggin is expressed postnatally in the suture mesenchyme of patent, but not fusing, cranial sutures, and that noggin expression is suppressed by FGF2 and syndromic fgfr signalling. Since noggin misexpression prevents cranial suture fusion in vitro and in vivo, we suggest that syndromic fgfr-mediated craniosynostoses may be the result of inappropriate downregulation of noggin expression.

Authors+Show Affiliations

Department of Surgery, Stanford University School of Medicine, Stanford, California 94305-5148, USA.No affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info available

Pub Type(s)

Journal Article
Research Support, Non-U.S. Gov't
Research Support, U.S. Gov't, P.H.S.

Language

eng

PubMed ID

12687003

Citation

Warren, Stephen M., et al. "The BMP Antagonist Noggin Regulates Cranial Suture Fusion." Nature, vol. 422, no. 6932, 2003, pp. 625-9.
Warren SM, Brunet LJ, Harland RM, et al. The BMP antagonist noggin regulates cranial suture fusion. Nature. 2003;422(6932):625-9.
Warren, S. M., Brunet, L. J., Harland, R. M., Economides, A. N., & Longaker, M. T. (2003). The BMP antagonist noggin regulates cranial suture fusion. Nature, 422(6932), 625-9.
Warren SM, et al. The BMP Antagonist Noggin Regulates Cranial Suture Fusion. Nature. 2003 Apr 10;422(6932):625-9. PubMed PMID: 12687003.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - The BMP antagonist noggin regulates cranial suture fusion. AU - Warren,Stephen M, AU - Brunet,Lisa J, AU - Harland,Richard M, AU - Economides,Aris N, AU - Longaker,Michael T, PY - 2002/10/02/received PY - 2003/03/07/accepted PY - 2003/4/11/pubmed PY - 2003/5/6/medline PY - 2003/4/11/entrez SP - 625 EP - 9 JF - Nature JO - Nature VL - 422 IS - 6932 N2 - During skull development, the cranial connective tissue framework undergoes intramembranous ossification to form skull bones (calvaria). As the calvarial bones advance to envelop the brain, fibrous sutures form between the calvarial plates. Expansion of the brain is coupled with calvarial growth through a series of tissue interactions within the cranial suture complex. Craniosynostosis, or premature cranial suture fusion, results in an abnormal skull shape, blindness and mental retardation. Recent studies have demonstrated that gain-of-function mutations in fibroblast growth factor receptors (fgfr) are associated with syndromic forms of craniosynostosis. Noggin, an antagonist of bone morphogenetic proteins (BMPs), is required for embryonic neural tube, somites and skeleton patterning. Here we show that noggin is expressed postnatally in the suture mesenchyme of patent, but not fusing, cranial sutures, and that noggin expression is suppressed by FGF2 and syndromic fgfr signalling. Since noggin misexpression prevents cranial suture fusion in vitro and in vivo, we suggest that syndromic fgfr-mediated craniosynostoses may be the result of inappropriate downregulation of noggin expression. SN - 0028-0836 UR - https://www.unboundmedicine.com/medline/citation/12687003/The_BMP_antagonist_noggin_regulates_cranial_suture_fusion_ L2 - https://doi.org/10.1038/nature01545 DB - PRIME DP - Unbound Medicine ER -