Impaired retinal differentiation and maintenance in zebrafish laminin mutants

Biehlmaier, O. and Makhankov, Y. and Neuhauss, S. C. F.. (2007) Impaired retinal differentiation and maintenance in zebrafish laminin mutants. Investigative Ophthalmology & Visual Science, 48 (6). pp. 2887-2894.

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Official URL: http://edoc.unibas.ch/45817/

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PURPOSE: To characterize morphologic and physiological alterations in the retina of three laminin mutant zebrafish, bashful (bal, lama1), grumpy (gup, lamb1), and sleepy (sly, lamc1), which were identified in forward genetic screens and were found to be impaired in visual functions. METHODS: Mutant larvae were observed for defects in visual behavior by testing their optokinetic response (OKR). In addition, electroretinograms (ERG) were measured and retinal morphology was examined by standard histology, immunocytochemistry, TUNEL assay, and electron microscopy. RESULTS: Both, gup and sly showed no OKR at any light intensity tested, whereas bal embryos showed some remaining OKR behavior at more than 40% of contrast. Consistent with the OKR result, gup and sly did not show an ERG response at any light intensity tested, whereas bal mutants exhibited small a- and b-waves at high light intensities. All three laminin mutants showed altered ganglion cell layers, optic nerve fasciculations, and lens defects. Again, bal showed the least severe morphologic phenotype with no additional defects. In contrast, both, gup and sly, showed severe photoreceptor outer segment shortening and synapse alteration (floating ribbons) as well as increased cell death. CONCLUSIONS: Lamb1 and lamc1 chains play an important role in the morphogenesis of photoreceptors and their synapses. In contrast, lama1 is not involved in outer retina development.
Faculties and Departments:05 Faculty of Science > Departement Biozentrum > Services Biozentrum > Imaging Core Facility (Biehlmaier)
UniBasel Contributors:Biehlmaier, Oliver
Item Type:Article, refereed
Article Subtype:Research Article
Publisher:Association for Research in Vision and Ophthalmology
Note:Publication type according to Uni Basel Research Database: Journal article
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Last Modified:28 Nov 2017 07:56
Deposited On:28 Nov 2017 07:56

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