原创力文档roles of the espin actin-bundling proteins in the morphogenesis and stabilization of hair cell stereocilia revealed in cbacaj congenic jerker mice角色的espin actin-bundling蛋白质形态发生和稳定的毛细胞静纤毛在cbacaj句老鼠肌肉抽搐.pdfVIP
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roles of the espin actin-bundling proteins in the morphogenesis and stabilization of hair cell stereocilia revealed in cbacaj congenic jerker mice角色的espin actin-bundling蛋白质形态发生和稳定的毛细胞静纤毛在cbacaj句老鼠肌肉抽搐
Roles of the Espin Actin-Bundling Proteins in the
Morphogenesis and Stabilization of Hair Cell Stereocilia
Revealed in CBA/CaJ Congenic Jerker Mice
´ 1 2,3 1,3
Gabriella Sekerkova , Claus-Peter Richter , James R. Bartles *
1 Department of Cell and Molecular Biology, Northwestern University Feinberg School of Medicine, Chicago, Illinois, United States of America, 2 Department of
Otolaryngology, Northwestern University Feinberg School of Medicine, Chicago, Illinois, United States of America, 3 Hugh Knowles Center for Clinical and Basic Science in
Hearing and Its Disorders, Northwestern University Feinberg School of Medicine, Chicago, Illinois, United States of America
Abstract
Hearing and vestibular function depend on mechanosensory staircase collections of hair cell stereocilia, which are produced
from microvillus-like precursors as their parallel actin bundle scaffolds increase in diameter and elongate or shorten. Hair cell
stereocilia contain multiple classes of actin-bundling protein, but little is known about what each class contributes. To
investigate the roles of the espin class of actin-bundling protein, we used a genetic approach that benefited from a
judicious selection of mouse background strain and an examination of the effects of heterozygosity. A congenic jerker
mouse line was prepared by repeated backcrossing into the inbred CBA/CaJ strain, which is known for excellent hearing and
minimal age-related hearing loss. We compared stereocilia in wild-type CBA/CaJ mice, jerker homozygotes that lack espin
proteins owing to a frameshift mutation in the espin gene, and jerker heterozygotes that contain reduced espin levels. The
lack of espins radically impaired stereociliary morphogenesis, resu
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