Hearing in Drosophila requires TilB, a conserved protein associated with ciliary motility

Ryan G Kavlie; Maurice J Kernan; Daniel F Eberl

doi:10.1534/genetics.110.114009

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Hearing in Drosophila requires TilB, a conserved protein associated with ciliary motility

Journal article

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Hearing in Drosophila requires TilB, a conserved protein associated with ciliary motility

Ryan G Kavlie, Maurice J Kernan and Daniel F Eberl

Genetics (Austin), Vol.185(1), pp.177-188

05/2010

DOI: 10.1534/genetics.110.114009

PMCID: PMC2870953

PMID: 20215474

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Abstract

Cilia were present in the earliest eukaryotic ancestor and underlie many biological processes ranging from cell motility and propulsion of extracellular fluids to sensory physiology. We investigated the contribution of the touch insensitive larva B (tilB) gene to cilia function in Drosophila melanogaster. Mutants of tilB exhibit dysfunction in sperm flagella and ciliated dendrites of chordotonal organs that mediate hearing and larval touch sensitivity. Mutant sperm axonemes as well as sensory neuron dendrites of Johnston's organ, the fly's auditory organ, lack dynein arms. Through deficiency mapping and sequencing candidate genes, we identified tilB mutations in the annotated gene CG14620. A genomic CG14620 transgene rescued deafness and male sterility of tilB mutants. TilB is a 395-amino-acid protein with a conserved N-terminal leucine-rich repeat region at residues 16-164 and a coiled-coil domain at residues 171-191. A tilB-Gal4 transgene driving fluorescently tagged TilB proteins elicits cytoplasmic expression in embryonic chordotonal organs, in Johnston's organ, and in sperm flagella. TilB does not appear to affect tubulin polyglutamylation or polyglycylation. The phenotypes and expression of tilB indicate function in cilia construction or maintenance, but not in intraflagellar transport. This is also consistent with phylogenetic association of tilB homologs with presence of genes encoding axonemal dynein arm components. Further elucidation of tilB functional mechanisms will provide greater understanding of cilia function and will facilitate understanding ciliary diseases.

Dyneins - metabolism

Axoneme - metabolism

Drosophila melanogaster - cytology

Glycosylation

Drosophila melanogaster - ultrastructure

Drosophila Proteins - metabolism

Mutation - genetics

Recombinant Fusion Proteins - metabolism

Cilia - metabolism

Genetic Vectors - genetics

Drosophila melanogaster - metabolism

Tubulin - metabolism

Animals

Movement - physiology

Peptides - metabolism

Proteins - metabolism

Animal Structures - ultrastructure

Axoneme - ultrastructure

Cilia - ultrastructure

Conserved Sequence

Dyneins - ultrastructure

Hearing - physiology

Details

Title: Subtitle: Hearing in Drosophila requires TilB, a conserved protein associated with ciliary motility
Creators: Ryan G Kavlie - Interdisciplinary Graduate Program in Genetics and Department of Biology, University of Iowa, Iowa City, IA 52242, USA
Maurice J Kernan
Daniel F Eberl
Resource Type: Journal article
Publication Details: Genetics (Austin), Vol.185(1), pp.177-188
Publisher: United States
DOI: 10.1534/genetics.110.114009
PMID: 20215474
PMCID: PMC2870953
ISSN: 0016-6731
eISSN: 1943-2631
Grant note: T32 GM008629 / NIGMS NIH HHS R01 DC004848 / NIDCD NIH HHS R01 DC002780 / NIDCD NIH HHS DC 004848 / NIDCD NIH HHS DC 002780 / NIDCD NIH HHS
Language: English
Date published: 05/2010
Academic Unit: Iowa Neuroscience Institute; Biology
Record Identifier: 9983991996902771

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