The Transcription Factors L-Sox5 and Sox6 Are Essential for Cartilage Formation
L-Sox5 and Sox6 are highly identical Sry-related transcription factors coexpressed in cartilage. Whereas Sox5 and Sox6 single null mice are born with mild skeletal abnormalities, Sox5; Sox6 double null fetuses die with a severe, generalized chondrodysplasia. In these double mutants, chondroblasts po...
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| Published in: | Developmental cell Vol. 1; no. 2; pp. 277 - 290 |
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| Main Authors: | , , , , , , , |
| Format: | Journal Article |
| Language: | English |
| Published: |
United States
Elsevier Inc
01-08-2001
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| Abstract | L-Sox5 and Sox6 are highly identical Sry-related transcription factors coexpressed in cartilage. Whereas
Sox5 and
Sox6 single null mice are born with mild skeletal abnormalities,
Sox5; Sox6 double null fetuses die with a severe, generalized chondrodysplasia. In these double mutants, chondroblasts poorly differentiate. They express the genes for all essential cartilage extracellular matrix components at low or undetectable levels and initiate proliferation after a long delay. All cartilages are thus extracellular matrix deficient and remain rudimentary. While chondroblasts in the center of cartilages ultimately activate prehypertrophic chondrocyte markers, epiphyseal chondroblasts ectopically activate hypertrophic chondrocyte markers. Thick intramembranous bone collars develop, but the formation of cartilage growth plates and endochondral bones is disrupted. L-Sox5 and Sox6 are thus redundant, potent enhancers of chondroblast functions, thereby essential for endochondral skeleton formation. |
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| AbstractList | L-Sox5 and Sox6 are highly identical Sry-related transcription factors coexpressed in cartilage. Whereas Sox5 and Sox6 single null mice are born with mild skeletal abnormalities, Sox5; Sox6 double null fetuses die with a severe, generalized chondrodysplasia. In these double mutants, chondroblasts poorly differentiate. They express the genes for all essential cartilage extracellular matrix components at low or undetectable levels and initiate proliferation after a long delay. All cartilages are thus extracellular matrix deficient and remain rudimentary. While chondroblasts in the center of cartilages ultimately activate prehypertrophic chondrocyte markers, epiphyseal chondroblasts ectopically activate hypertrophic chondrocyte markers. Thick intramembranous bone collars develop, but the formation of cartilage growth plates and endochondral bones is disrupted. L-Sox5 and Sox6 are thus redundant, potent enhancers of chondroblast functions, thereby essential for endochondral skeleton formation. L-Sox5 and Sox6 are highly identical Sry-related transcription factors coexpressed in cartilage. Whereas Sox5 and Sox6 single null mice are born with mild skeletal abnormalities, Sox5; Sox6 double null fetuses die with a severe, generalized chondrodysplasia. In these double mutants, chondroblasts poorly differentiate. They express the genes for all essential cartilage extracellular matrix components at low or undetectable levels and initiate proliferation after a long delay. All cartilages are thus extracellular matrix deficient and remain rudimentary. While chondroblasts in the center of cartilages ultimately activate prehypertrophic chondrocyte markers, epiphyseal chondroblasts ectopically activate hypertrophic chondrocyte markers. Thick intramembranous bone collars develop, but the formation of cartilage growth plates and endochondral bones is disrupted. L-Sox5 and Sox6 are thus redundant, potent enhancers of chondroblast functions, thereby essential for endochondral skeleton formation. |
| Author | Lefebvre, Véronique Deng, Jian Ming Behringer, Richard R Li, Ping Smits, Patrick Mandel, Jennifer de Crombrugghe, Benoit Zhang, Zhaoping |
| Author_xml | – sequence: 1 givenname: Patrick surname: Smits fullname: Smits, Patrick – sequence: 2 givenname: Ping surname: Li fullname: Li, Ping – sequence: 3 givenname: Jennifer surname: Mandel fullname: Mandel, Jennifer – sequence: 4 givenname: Zhaoping surname: Zhang fullname: Zhang, Zhaoping – sequence: 5 givenname: Jian Ming surname: Deng fullname: Deng, Jian Ming – sequence: 6 givenname: Richard R surname: Behringer fullname: Behringer, Richard R – sequence: 7 givenname: Benoit surname: de Crombrugghe fullname: de Crombrugghe, Benoit – sequence: 8 givenname: Véronique surname: Lefebvre fullname: Lefebvre, Véronique email: lefebvrv@bme.ri.ccf.org |
| BackLink | https://www.ncbi.nlm.nih.gov/pubmed/11702786$$D View this record in MEDLINE/PubMed |
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| Snippet | L-Sox5 and Sox6 are highly identical Sry-related transcription factors coexpressed in cartilage. Whereas
Sox5 and
Sox6 single null mice are born with mild... L-Sox5 and Sox6 are highly identical Sry-related transcription factors coexpressed in cartilage. Whereas Sox5 and Sox6 single null mice are born with mild... |
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| SubjectTerms | Animals Bone and Bones - abnormalities Bone Development Cartilage - embryology Cartilage - physiology Cell Differentiation Chondrocytes - cytology Chondrocytes - metabolism DNA-Binding Proteins - metabolism DNA-Binding Proteins - physiology Exostoses, Multiple Hereditary - genetics High Mobility Group Proteins - metabolism High Mobility Group Proteins - physiology In Situ Hybridization L-Sox5 protein Mice Microscopy, Fluorescence Models, Biological Models, Genetic Mutation Nuclear Proteins - metabolism Nuclear Proteins - physiology Phenotype Sox6 protein SOXD Transcription Factors Transcription Factors |
| Title | The Transcription Factors L-Sox5 and Sox6 Are Essential for Cartilage Formation |
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