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Dev Biol February 15, 2006; 290 (2): 411-20.

Dystroglycan is required for proper retinal layering.

Dystroglycan (DG) is a transmembrane receptor linking the extracellular matrix to the internal cytoskeleton. Its structural function has been mainly characterized in muscle fibers, but DG plays signaling and developmental roles also in different tissues and cell types. We have investigated the effects of dystroglycan depletion during eye development of Xenopus laevis. We have injected a specific morpholino (Mo) antisense oligonucleotide in the animal pole of one dorsal blastomere of embryos at four cells stage. Mo-mediated loss of DG function caused disruption of the basal lamina layers, increased apoptosis and reduction of the expression domains of specific retinal markers, at early stages. Later in development, morphants displayed unilateral ocular malformations, such as microphtalmia and retinal delayering with photoreceptors and ganglion cells scattered throughout the retina or aggregated in rosette-like structures. These results recall the phenotypes observed in specific human diseases and suggest that DG presence is crucial at early stages for the organization of retinal architecture.

PubMed ID: 16406325
Article link: Dev Biol

Species referenced: Xenopus laevis
Genes referenced: crx crybb1 dag1 gal.2 itgb1 lama1 nppa nrp1 otx2 prkci prkcz rax rbpms rbpms2 tubb2b zic1
Antibodies: BrdU Ab2 Dag1 Ab2 Itgb1 Ab1 Lama1 Ab1 Prkcz Ab1 Tuba4b Ab5
Morpholinos: dag1 MO3

Disease Ontology terms: congenital muscular dystrophy [+]

Article Images: [+] show captions