Literature DB >> 14749384

Matrilin-3 is dispensable for mouse skeletal growth and development.

Yaping Ko1, Birgit Kobbe, Claudia Nicolae, Nicolai Miosge, Mats Paulsson, Raimund Wagener, Attila Aszódi.   

Abstract

Matrilin-3 belongs to the matrilin family of extracellular matrix (ECM) proteins and is primarily expressed in cartilage. Mutations in the gene encoding human matrilin-3 (MATN-3) lead to autosomal dominant skeletal disorders, such as multiple epiphyseal dysplasia (MED), which is characterized by short stature and early-onset osteoarthritis, and bilateral hereditary microepiphyseal dysplasia, a variant form of MED characterized by pain in the hip and knee joints. To assess the function of matrilin-3 during skeletal development, we have generated Matn-3 null mice. Homozygous mutant mice appear normal, are fertile, and show no obvious skeletal malformations. Histological and ultrastructural analyses reveal endochondral bone formation indistinguishable from that of wild-type animals. Northern blot, immunohistochemical, and biochemical analyses indicated no compensatory upregulation of any other member of the matrilin family. Altogether, our findings suggest functional redundancy among matrilins and demonstrate that the phenotypes of MED disorders are not caused by the absence of matrilin-3 in cartilage ECM.

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Year:  2004        PMID: 14749384      PMCID: PMC344189          DOI: 10.1128/MCB.24.4.1691-1699.2004

Source DB:  PubMed          Journal:  Mol Cell Biol        ISSN: 0270-7306            Impact factor:   4.272


  32 in total

1.  Organization of the myotendinous junction is dependent on the presence of alpha7beta1 integrin.

Authors:  N Miosge; C Klenczar; R Herken; M Willem; U Mayer
Journal:  Lab Invest       Date:  1999-12       Impact factor: 5.662

2.  Molecular structure, processing, and tissue distribution of matrilin-4.

Authors:  A R Klatt; D P Nitsche; B Kobbe; M Macht; M Paulsson; R Wagener
Journal:  J Biol Chem       Date:  2001-02-14       Impact factor: 5.157

3.  Structure and mapping of the mouse matrilin-3 gene (Matn3), a member of a gene family containing a U12-type AT-AC intron.

Authors:  R Wagener; B Kobbe; A Aszódi; Z Liu; D R Beier; M Paulsson
Journal:  Mamm Genome       Date:  2000-02       Impact factor: 2.957

4.  Mutations in the region encoding the von Willebrand factor A domain of matrilin-3 are associated with multiple epiphyseal dysplasia.

Authors:  K L Chapman; G R Mortier; K Chapman; J Loughlin; M E Grant; M D Briggs
Journal:  Nat Genet       Date:  2001-08       Impact factor: 38.330

5.  Expression of matrilins during maturation of mouse skeletal tissues.

Authors:  Andreas R Klatt; Mats Paulsson; Raimund Wagener
Journal:  Matrix Biol       Date:  2002-04       Impact factor: 11.583

6.  Early expression of endomucin on endothelium of the mouse embryo and on putative hematopoietic clusters in the dorsal aorta.

Authors:  G Brachtendorf; A Kuhn; U Samulowitz; R Knorr; E Gustafsson; A J Potocnik; R Fässler; D Vestweber
Journal:  Dev Dyn       Date:  2001-11       Impact factor: 3.780

7.  Molecular properties of matrilin-3 isolated from human growth cartilage.

Authors:  D Kleemann-Fischer; G R Kleemann; D Engel; J R Yates; J J Wu; D R Eyre
Journal:  Arch Biochem Biophys       Date:  2001-03-15       Impact factor: 4.013

8.  Molecular structure and tissue distribution of matrilin-3, a filament-forming extracellular matrix protein expressed during skeletal development.

Authors:  A R Klatt; D P Nitsche; B Kobbe; M Mörgelin; M Paulsson; R Wagener
Journal:  J Biol Chem       Date:  2000-02-11       Impact factor: 5.157

9.  Matrilin-3 in human articular cartilage: increased expression in osteoarthritis.

Authors:  O Pullig; G Weseloh; A R Klatt; R Wagener; B Swoboda
Journal:  Osteoarthritis Cartilage       Date:  2002-04       Impact factor: 6.576

10.  Normal skeletal development of mice lacking matrilin 1: redundant function of matrilins in cartilage?

Authors:  A Aszódi; J F Bateman; E Hirsch; M Baranyi; E B Hunziker; N Hauser; Z Bösze; R Fässler
Journal:  Mol Cell Biol       Date:  1999-11       Impact factor: 4.272

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  18 in total

1.  Unique matrix structure in the rough endoplasmic reticulum cisternae of pseudoachondroplasia chondrocytes.

Authors:  Thomas M Merritt; Roger Bick; Brian J Poindexter; Joseph L Alcorn; Jacqueline T Hecht
Journal:  Am J Pathol       Date:  2007-01       Impact factor: 4.307

2.  Functional knockout of the matrilin-3 gene causes premature chondrocyte maturation to hypertrophy and increases bone mineral density and osteoarthritis.

Authors:  Louise van der Weyden; Lei Wei; Junming Luo; Xu Yang; David E Birk; David J Adams; Allan Bradley; Qian Chen
Journal:  Am J Pathol       Date:  2006-08       Impact factor: 4.307

3.  Derlin-2-deficient mice reveal an essential role for protein dislocation in chondrocytes.

Authors:  Stephanie K Dougan; Chih-Chi Andrew Hu; Marie-Eve Paquet; Matthew B Greenblatt; Jun Kim; Brendan N Lilley; Nicki Watson; Hidde L Ploegh
Journal:  Mol Cell Biol       Date:  2011-01-10       Impact factor: 4.272

Review 4.  Extracellular matrix and developing growth plate.

Authors:  Johanna Myllyharju
Journal:  Curr Osteoporos Rep       Date:  2014-12       Impact factor: 5.096

5.  Altered integration of matrilin-3 into cartilage extracellular matrix in the absence of collagen IX.

Authors:  Bastian Budde; Katrin Blumbach; Joni Ylöstalo; Frank Zaucke; Harald W A Ehlen; Raimund Wagener; Leena Ala-Kokko; Mats Paulsson; Peter Bruckner; Susanne Grässel
Journal:  Mol Cell Biol       Date:  2005-12       Impact factor: 4.272

6.  Matrilin-3 inhibits chondrocyte hypertrophy as a bone morphogenetic protein-2 antagonist.

Authors:  Xu Yang; Samir K Trehan; Yingjie Guan; Changqi Sun; Douglas C Moore; Chathuraka T Jayasuriya; Qian Chen
Journal:  J Biol Chem       Date:  2014-10-20       Impact factor: 5.157

7.  WISP3, the gene responsible for the human skeletal disease progressive pseudorheumatoid dysplasia, is not essential for skeletal function in mice.

Authors:  Wendy E Kutz; Yaoqin Gong; Matthew L Warman
Journal:  Mol Cell Biol       Date:  2005-01       Impact factor: 4.272

8.  Matrilin-1 is essential for zebrafish development by facilitating collagen II secretion.

Authors:  Cristian Dan Neacsu; Ya-Ping Ko; Andreas Tagariello; Kristina Røkenes Karlsen; Wolfram Friedrich Neiss; Mats Paulsson; Raimund Wagener
Journal:  J Biol Chem       Date:  2013-11-29       Impact factor: 5.157

9.  Neutral sphingomyelinase (SMPD3) deficiency causes a novel form of chondrodysplasia and dwarfism that is rescued by Col2A1-driven smpd3 transgene expression.

Authors:  Wilhelm Stoffel; Britta Jenke; Barbara Holz; Erika Binczek; Robert Heinz Günter; Jutta Knifka; Jürgen Koebke; Anja Niehoff
Journal:  Am J Pathol       Date:  2007-07       Impact factor: 4.307

10.  Decreased chondrocyte proliferation and dysregulated apoptosis in the cartilage growth plate are key features of a murine model of epiphyseal dysplasia caused by a matn3 mutation.

Authors:  Matthew P Leighton; Seema Nundlall; Tobias Starborg; Roger S Meadows; Farhana Suleman; Lynette Knowles; Raimund Wagener; David J Thornton; Karl E Kadler; Raymond P Boot-Handford; Michael D Briggs
Journal:  Hum Mol Genet       Date:  2007-05-21       Impact factor: 6.150

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