Journal article
Comparative exploration of mammalian deafness gene homologues in the Drosophila auditory organ shows genetic correlation between insect and vertebrate hearing
PloS one, Vol.19(2), e0297846
02/27/2024
DOI: 10.1371/journal.pone.0297846
PMCID: PMC10898740
PMID: 38412189
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.
Details
- Title: Subtitle
- Comparative exploration of mammalian deafness gene homologues in the Drosophila auditory organ shows genetic correlation between insect and vertebrate hearing
- Creators
- Daniel C Sutton - Baylor College of MedicineJonathan C Andrews - Baylor College of MedicineDylan M Dolezal - University of IowaYe Jin Park - Baylor College of MedicineHongjie Li - Baylor College of MedicineDaniel F Eberl - University of IowaShinya Yamamoto - Baylor College of MedicineAndrew K Groves - Baylor College of Medicine
- Resource Type
- Journal article
- Publication Details
- PloS one, Vol.19(2), e0297846
- DOI
- 10.1371/journal.pone.0297846
- PMID
- 38412189
- PMCID
- PMC10898740
- ISSN
- 1932-6203
- eISSN
- 1932-6203
- Grant note
- name: NIH, award: R01 DC014932; name: NIH, award: R00 AG062746; name: CPRIT Scholar in Cancer Research, award: RR200063; name: NSF, award: R00AG062746; name: Baylor College of Medicine Intellectual and Developmental Disabilities Research Center, award: U54HD083092
- Language
- English
- Date published
- 02/27/2024
- Academic Unit
- Iowa Neuroscience Institute; Biology
- Record Identifier
- 9984562590802771
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