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The Journal of Neuroscience, August 1, 1999, 19(15):6267-6274

Myosin VIIa Participates in Opsin Transport through The Photoreceptor Cilium

Xinran Liu1, Igor P. Udovichenko1, Stephen D.M. Brown2, Karen P. Steel3, and David S. Williams1

1 Departments of Pharmacology and Neurosciences, University of California at San Diego School of Medicine, La Jolla, California 92093-0983, 2 Medical Research Council Mammalian Genetics Unit and Mouse Genome Centre, Harwell OX11 0RD, United Kingdom, and 3 Medical Research Council Institute of Hearing Research, University Park, Nottingham NG7 2RD, United Kingdom

Two types of Usher syndrome, a blindness-deafness disorder, result from mutations in the myosin VIIa gene. As for most other unconventional myosins, little is known about the function or functions of myosin VIIa. Here, we studied the photoreceptor cells of mice with mutant myosin VIIa by electron immunomicroscopy and microscopic autoradiography. We found evidence that myosin VIIa functions in the connecting cilium of each photoreceptor cell and participates in the transport of opsin through this structure. These findings provide the first direct evidence that opsin travels along the connecting cilium en route to the outer segment. They demonstrate that a myosin may function in a cilium and suggest that abnormal opsin transport might contribute to blindness in Usher syndrome.

Key words: myosin; molecular motor; cilium; photoreceptor; opsin; Usher syndrome


Copyright © 1999 Society for Neuroscience  0270-6474/99/19156267-08$05.00/0


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