spacer gif spacer gif spacer gif spacer gif spacer gif
QUICK SEARCH:   [advanced]


spacer gif
     Home     Help     Feedback     Subscriptions     Archive     Search     Table of Contents    

First published online June 6, 2008
doi: 10.1242/10.1242/dev.019950

Development 135, 2215-2220 (2008)
Published by The Company of Biologists 2008
This Article
Right arrow Figures Only
Right arrow Full Text
Right arrow Full Text (PDF)
Right arrow Supplementary Material
Right arrow Alert me when this article is cited
Right arrow Alert me if a correction is posted
Services
Right arrow Email this article to a friend
Right arrow Related articles in Development
Right arrow Similar articles in this journal
Right arrow Similar articles in PubMed
Right arrow Alert me to new issues of the journal
Right arrow Download to citation manager
Right arrow reprints & permissions
Citing Articles
Right arrow Citing Articles via HighWire
Google Scholar
Right arrow Articles by Sohaskey, M. L.
Right arrow Articles by Harland, R. M.
PubMed
Right arrow PubMed Citation
Right arrow Articles by Sohaskey, M. L.
Right arrow Articles by Harland, R. M.

Research Report

JAWS coordinates chondrogenesis and synovial joint positioning

Michael L. Sohaskey1,*, Jane Yu1, Michael A. Diaz2, Anna H. Plaas2 and Richard M. Harland1,*

1 Division of Genetics, Genomics and Development, Department of Molecular and Cell Biology, Center for Integrative Genomics, University of California, Berkeley, CA 94720, USA.
2 Departments of Internal Medicine (Rheumatology) and Biochemistry, Rush University Medical Center, 1725 Harrison Street, Chicago, IL 60612, USA.

* Authors for correspondence (e-mail: sohaskey{at}berkeley.edu; harland{at}berkeley.edu)

Accepted 27 April 2008

SUMMARY

Properly positioned synovial joints are crucial to coordinated skeletal movement. Despite their importance for skeletal development and function, the molecular mechanisms that underlie joint positioning are not well understood. We show that mice carrying an insertional mutation in a previously uncharacterized gene, which we have named Jaws (joints abnormal with splitting), die perinatally with striking skeletal defects, including ectopic interphalangeal joints. These ectopic joints develop along the longitudinal axis and persist at birth, suggesting that JAWS is uniquely required for the orientation and consequent positioning of interphalangeal joints within the endochondral skeleton. Jaws mutant mice also exhibit severe chondrodysplasia characterized by delayed and disorganized maturation of growth plate chondrocytes, together with impaired chondroitin sulfation and abnormal metabolism of the chondroitin sulfate proteoglycan aggrecan. Our findings identify JAWS as a key regulator of chondrogenesis and synovial joint positioning required for the restriction of joint formation to discrete stereotyped locations in the embryonic skeleton.

Key words: Chondrogenesis, Synovial joints, Interzone, Gdf5, Chondroitin sulfate, Extracellular matrix, Mouse embryo


Related articles in Development:

Out of joint with JAWS

Development 2008 135: e1303. [Full Text]  



This article has been cited by other articles:


Home page
 Proc. Natl. Acad. Sci. USAHome page
J. P. Frederick, A. T. Tafari, S.-M. Wu, L. C. Megosh, S.-T. Chiou, R. P. Irving, and J. D. York
From the Cover: A role for a lithium-inhibited Golgi nucleotidase in skeletal development and sulfation
PNAS, August 19, 2008; 105(33): 11605 - 11612.
[Abstract] [Full Text] [PDF]


© The Company of Biologists Ltd 2008