RRC ID |
79040
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Author |
Sutton DC, Andrews JC, Dolezal DM, Park YJ, Li H, Eberl DF, Yamamoto S, Groves AK.
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Title |
Comparative exploration of mammalian deafness gene homologues in the Drosophila auditory organ shows genetic correlation between insect and vertebrate hearing.
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Journal |
PLoS One
|
Abstract |
Johnston's organ, the Drosophila auditory organ, is anatomically very different from the mammalian organ of Corti. However, recent evidence indicates significant cellular and molecular similarities exist between vertebrate and invertebrate hearing, suggesting that Drosophila may be a useful platform to determine the function of the many mammalian deafness genes whose underlying biological mechanisms are poorly characterized. Our goal was a comprehensive screen of all known orthologues of mammalian deafness genes in the fruit fly to better understand conservation of hearing mechanisms between the insect and the fly and ultimately gain insight into human hereditary deafness. We used bioinformatic comparisons to screen previously reported human and mouse deafness genes and found that 156 of them have orthologues in Drosophila melanogaster. We used fluorescent imaging of T2A-GAL4 gene trap and GFP or YFP fluorescent protein trap lines for 54 of the Drosophila genes and found 38 to be expressed in different cell types in Johnston's organ. We phenotypically characterized the function of strong loss-of-function mutants in three genes expressed in Johnston's organ (Cad99C, Msp-300, and Koi) using a courtship assay and electrophysiological recordings of sound-evoked potentials. Cad99C and Koi were found to have significant courtship defects. However, when we tested these genes for electrophysiological defects in hearing response, we did not see a significant difference suggesting the courtship defects were not caused by hearing deficiencies. Furthermore, we used a UAS/RNAi approach to test the function of seven genes and found two additional genes, CG5921 and Myo10a, that gave a statistically significant delay in courtship but not in sound-evoked potentials. Our results suggest that many mammalian deafness genes have Drosophila homologues expressed in the Johnston's organ, but that their requirement for hearing may not necessarily be the same as in mammals.
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Volume |
19(2)
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Pages |
e0297846
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Published |
2024-1-1
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DOI |
10.1371/journal.pone.0297846
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PII |
PONE-D-23-22910
|
PMID |
38412189
|
PMC |
PMC10898740
|
MeSH |
Animals
Deafness*
Drosophila* / genetics
Drosophila melanogaster / genetics
Hearing / genetics
Humans
Mammals
Mice
Vertebrates
|
IF |
2.74
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Resource |
Drosophila |
DGRC#115004
DGRC#115061
DGRC#115075
DGRC#115234
DGRC#115256
DGRC#115261
DGRC#115339
DGRC#115365
DGRC#115366
DGRC#115395
DGRC#115428
DGRC#115434
DGRC#115453
DGRC#115521
DGRC#115585
DGRC#115586
DGRC#115590 |