Human Molecular Genetics, Vol 6, 831-841, Copyright © 1997 by Oxford University Press
RA Williamson, MD Henry, KJ Daniels, RF Hrstka, JC Lee, Y Sunada, O Ibraghimov- Beskrovnaya and KP Campbell
Dystroglycan is a central component of the dystrophin-glycoprotein complex
(DGC), a protein assembly that plays a critical role in a variety of
muscular dystrophies. In order to better understand the function of
dystroglycan in development and disease, we have generated a null allele of
dystroglycan (Dag1neo2) in mice. Heterozygous Dag1neo2 mice are viable and
fertile. In contrast, homozygous Dag1neo2 embryos exhibit gross
developmental abnormalities beginning around 6.5 days of gestation.
Analysis of the mutant phenotype indicates that an early defect in the
development of homozygous Dag1neo2 embryos is a disruption of Reichert's
membrane, an extra-embryonic basement membrane. Consistent with the
functional defects observed in Reichert's membrane, dystroglycan protein is
localized in apposition to this structure in normal egg cylinder stage
embryos. We also show that the localization of two critical structural
elements of Reichert's membrane- -laminin and collagen IV--are specifically
disrupted in the homozygous Dag1neo2 embryos. Taken together, the data
indicate that dystroglycan is required for the development of Reichert's
membrane. Furthermore, these results suggest that disruption of basement
membrane organization might be a common feature of muscular dystrophies
linked to the DGC.
ARTICLES
Dystroglycan is essential for early embryonic development: disruption of Reichert's membrane in Dag1-null mice
Department of Obstetrics and Gynecology, University of Iowa Hospitals and Clinics, Iowa City 52242, USA.
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C. J. I. Raats, J. van den Born, M. A. H. Bakker, B. Oppers-Walgreen, B. J. M. Pisa, H. B. P. M. Dijkman, K. J. M. Assmann, and J. H. M. Berden Expression of Agrin, Dystroglycan, and Utrophin in Normal Renal Tissue and in Experimental Glomerulopathies Am. J. Pathol., May 1, 2000; 156(5): 1749 - 1765. [Abstract] [Full Text] [PDF]
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H. M. REGELE, E. FILLIPOVIC, B. LANGER, H. POCZEWKI, I. KRAXBERGER, R. E. BITTNER, and D. KERJASCHKI Glomerular Expression of Dystroglycans Is Reduced in Minimal Change Nephrosis But Not in Focal Segmental Glomerulosclerosis J. Am. Soc. Nephrol., March 1, 2000; 11(3): 403 - 412. [Abstract] [Full Text] [PDF]
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M James, A Nuttall, J. Ilsley, K Ottersbach, J. Tinsley, M Sudol, and S. Winder Adhesion-dependent tyrosine phosphorylation of (beta)-dystroglycan regulates its interaction with utrophin J. Cell Sci., January 5, 2000; 113(10): 1717 - 1726. [Abstract] [PDF]
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M Aumailley, M Pesch, L Tunggal, F Gaill, and R Fassler Altered synthesis of laminin 1 and absence of basement membrane component deposition in (beta)1 integrin-deficient embryoid bodies J. Cell Sci., January 1, 2000; 113(2): 259 - 268. [Abstract] [PDF]
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M. Durbeej and K. P. Campbell Biochemical Characterization of the Epithelial Dystroglycan Complex J. Biol. Chem., September 10, 1999; 274(37): 26609 - 26616. [Abstract] [Full Text] [PDF]
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K. M. D. Bushby Making sense of the limb-girdle muscular dystrophies Brain, August 1, 1999; 122(8): 1403 - 1420. [Abstract] [Full Text] [PDF]
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F. Montanaro, M. Lindenbaum, and S. Carbonetto {alpha}-Dystroglycan Is a Laminin Receptor Involved in Extracellular Matrix Assembly on Myotubes and Muscle Cell Viability J. Cell Biol., June 14, 1999; 145(6): 1325 - 1340. [Abstract] [Full Text] [PDF]
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H. Colognato, D. A. Winkelmann, and P. D. Yurchenco Laminin Polymerization Induces a Receptor-Cytoskeleton Network J. Cell Biol., May 3, 1999; 145(3): 619 - 631. [Abstract] [Full Text] [PDF]
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N. Smyth, H. S. Vatansever, P. Murray, M. Meyer, C. Frie, M. Paulsson, and D. Edgar Absence of Basement Membranes after Targeting the LAMC1 Gene Results in Embryonic Lethality Due to Failure of Endoderm Differentiation J. Cell Biol., January 11, 1999; 144(1): 151 - 160. [Abstract] [Full Text] [PDF]
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U Muller and A. Brandli Cell adhesion molecules and extracellular-matrix constituents in kidney development and disease J. Cell Sci., January 11, 1999; 112(22): 3855 - 3867. [Abstract] [PDF]
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M Ferletta and P Ekblom Identification of laminin-10/11 as a strong cell adhesive complex for a normal and a malignant human epithelial cell line J. Cell Sci., January 1, 1999; 112(1): 1 - 10. [Abstract] [PDF]
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 A. Rambukkana, H. Yamada, G. Zanazzi, T. Mathus, J. L. Salzer, P. D. Yurchenco, K. P. Campbell, and V. A. Fischetti Role of -Dystroglycan as a Schwann Cell Receptor for Mycobacterium leprae Science, December 11, 1998; 282(5396): 2076 - 2079. [Abstract] [Full Text]
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W. Cao, M. D. Henry, P. Borrow, H. Yamada, J. H. Elder, E. V. Ravkov, S. T. Nichol, R. W. Compans, K. P. Campbell, and M. B. Oldstone Identification of -Dystroglycan as a Receptor for Lymphocytic Choriomeningitis Virus and Lassa Fever Virus Science, December 11, 1998; 282(5396): 2079 - 2081. [Abstract] [Full Text]
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N. R. Smalheiser, S. M. Haslam, M. Sutton-Smith, H. R. Morris, and A. Dell Structural Analysis of Sequences O-Linked to Mannose Reveals a Novel Lewis X Structure in Cranin (Dystroglycan) Purified from Sheep Brain J. Biol. Chem., September 11, 1998; 273(37): 23698 - 23703. [Abstract] [Full Text] [PDF]
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A. A. Hack, C. T. Ly, F. Jiang, C. J. Clendenin, K. S. Sigrist, R. L. Wollmann, and E. M. McNally {gamma}-Sarcoglycan Deficiency Leads to Muscle Membrane Defects and Apoptosis Independent of Dystrophin J. Cell Biol., September 7, 1998; 142(5): 1279 - 1287. [Abstract] [Full Text] [PDF]
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M. J. Cullen, J. Walsh, S. A. Stevenson, S. Rothery, and N. J. Severs Co-localization of Dystrophin and ß-Dystroglycan Demonstrated in En Face View by Double Immunogold Labeling of Freeze-fractured Skeletal Muscle J. Histochem. Cytochem., August 1, 1998; 46(8): 945 - 954. [Abstract] [Full Text]
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M. Durbeej, M. D. Henry, M. Ferletta, K. P. Campbell, and P. Ekblom Distribution of Dystroglycan in Normal Adult Mouse Tissues J. Histochem. Cytochem., April 1, 1998; 46(4): 449 - 458. [Abstract] [Full Text]
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S. Stevenson, S. Rothery, M. J. Cullen, and N. J. Severs Spatial Relationship of the C-Terminal Domains of Dystrophin and ß-Dystroglycan in Cardiac Muscle Support a Direct Molecular Interaction at the Plasma Membrane Interface Circ. Res., January 23, 1998; 82(1): 82 - 93. [Abstract] [Full Text] [PDF]
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M. Gesemann, A. Brancaccio, B. Schumacher, and M. A. Ruegg Agrin Is a High-affinity Binding Protein of Dystroglycan in Non-muscle Tissue J. Biol. Chem., January 2, 1998; 273(1): 600 - 605. [Abstract] [Full Text] [PDF]
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A. J. Ettinger, G. Feng, and J. R. Sanes epsilon -Sarcoglycan, a Broadly Expressed Homologue of the Gene Mutated in Limb-Girdle Muscular Dystrophy 2D J. Biol. Chem., December 19, 1997; 272(51): 32534 - 32538. [Abstract] [Full Text] [PDF]
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