Development of Johnston's organ in Drosophila

Eberl, Daniel F.; Boekhoff-Falk, Grace

doi:10.1387/ijdb.072364de

Cited by 63 publications

(56 citation statements)

References 81 publications

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“…Because Drosophila has only one diaphanous gene, these crosses serve as models for gainof-function mutations in both DIAPH3 and DIAPH1. The JO consists of an array of mechanosensory neurons that share many developmental and functional similarities with mammalian inner hair cells (30,31). There are five neuronal subgroups in the JO: neurons of subgroups A and B are activated by sound vibrations and are essential for hearing, whereas neurons in subgroups C and E respond to gravity and wind movements (32,33).…”

Section: Expression Of Diaph3 Protein Is Significantly Increased Inmentioning

confidence: 99%

Increased activity of Diaphanous homolog 3 ( DIAPH3 )/ diaphanous causes hearing defects in humans with auditory neuropathy and in Drosophila

Schoen

Emery²,

Thorne³

et al. 2010

Proc. Natl. Acad. Sci. U.S.A.

107

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Auditory neuropathy is a rare form of deafness characterized by an absent or abnormal auditory brainstem response with preservation of outer hair cell function. We have identified Diaphanous homolog 3 (DIAPH3) as the gene responsible for autosomal dominant nonsyndromic auditory neuropathy (AUNA1), which we previously mapped to chromosome 13q21-q24. Genotyping of additional family members narrowed the interval to an 11-Mb, 3.28-cM gene-poor region containing only four genes, including DIAPH3. DNA sequencing of DIAPH3 revealed a c.-172G > A, g. 48G > A mutation in a highly conserved region of the 5′ UTR. The c.-172G > A mutation occurs within a GC box sequence element and was not found in 379 controls. Using genome-wide expression arrays and quantitative RT-PCR, we demonstrate a 2-to 3-fold overexpression of DIAPH3 mRNA in lymphoblastoid cell lines from affected individuals. Likewise, a significant increase (≈1.5-fold) in DIAPH3 protein was found by quantitative immunoblotting of lysates from lymphoblastoid cell lines derived from affected individuals in comparison with controls. In addition, the c.-172G > A mutation is sufficient to drive overexpression of a luciferase reporter. Finally, the expression of a constitutively active form of diaphanous protein in the auditory organ of Drosophila melanogaster recapitulates the phenotype of impaired response to sound. To date, only two genes, the otoferlin gene OTOF and the pejvakin gene PJVK, are known to underlie nonsyndromic auditory neuropathy. Genetic testing for DIAPH3 may be useful for individuals with recessive as well as dominant inheritance of nonsyndromic auditory neuropathy.

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Section: Expression Of Diaph3 Protein Is Significantly Increased Inmentioning

confidence: 99%

Increased activity of Diaphanous homolog 3 ( DIAPH3 )/ diaphanous causes hearing defects in humans with auditory neuropathy and in Drosophila

Schoen

Emery²,

Thorne³

et al. 2010

Proc. Natl. Acad. Sci. U.S.A.

107

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“…Starting with a brief description of JO structure, this review focuses on these recent findings in the study of antennal hearing and tries to identify pending questions in the field. Additional information on insect auditory and mechanosensory systems, including tympanal hearing organs, can be found in Albert et al (2007b), Kernan (2007), Eberl and Boekhoff-Falk (2007), Bechstedt and Howard (2008), Hedwig and Pollack (2008), Göpfert (2008), Göpfert and Robert (2008), Robert (2008), Yack and Dawson (2008, and Lu et al (2009).…”

Section: Introductionmentioning

confidence: 99%

Antennal hearing in insects – New findings, new questions

et al. 2011

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“…Like vertebrate hair cells, JO neurons are ciliated and respond to mechanical stimulation. Although JO has morphologically diverged from hair cells in the human inner ear, the genetic program for its development shares a strong homology (19,20). For example, the Atoh1 gene required for vertebrate auditory hair cell specification was found by direct homology to the fly atonal gene required for JO specification and atonal/Atoh1 genes can be functionally exchanged between mice and flies (21,22).…”

mentioning

confidence: 99%

Physiological, anatomical, and behavioral changes after acoustic trauma in Drosophila melanogaster

Christie

Sivan-Loukianova

Smith

et al. 2013

Proc. Natl. Acad. Sci. U.S.A.

Self Cite

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Noise-induced hearing loss (NIHL) is a growing health issue, with costly treatment and lost quality of life. Here we establish Drosophila melanogaster as an inexpensive, flexible, and powerful genetic model system for NIHL. We exposed flies to acoustic trauma and quantified physiological and anatomical effects. Trauma significantly reduced sound-evoked potential (SEP) amplitudes and increased SEP latencies in control genotypes. SEP amplitude but not latency effects recovered after 7 d. Although trauma produced no gross morphological changes in the auditory organ (Johnston's organ), mitochondrial cross-sectional area was reduced 7 d after exposure. In nervana 3 heterozygous flies, which slightly compromise ion homeostasis, trauma had exaggerated effects on SEP amplitude and mitochondrial morphology, suggesting a key role for ion homeostasis in resistance to acoustic trauma. Thus, Drosophila exhibit acoustic trauma effects resembling those found in vertebrates, including inducing metabolic stress in sensory cells. This report of noise trauma in Drosophila is a foundation for studying molecular and genetic sequelae of NIHL. mitochondria | Na/K ATPase | locomotion | auditory courtship behavior N oise-induced hearing loss (NIHL) is a pervasive and growing health issue arising from occupational and recreational hazards, with significant costs in health care and personal quality of life. Despite this, the molecular and physiological mechanisms involved in the etiology or recovery from injury are not yet fully understood. Importantly, intense acoustic trauma can induce permanent damage-unlike other vertebrates, mammals cannot regenerate auditory hair cells (1, 2). NIHL associated with permanent changes in auditory sensitivity causes multiple consistent effects: stereocilia bundle disruption, inner (IHC) and outer hair cell (OHC) death or damage, supporting cell tissue disruption, and eventual spiral ganglion cell damage or loss (3-7). Most studies to date used mammalian model organisms such as mice (8, 9), rats (10), and guinea pigs (11)(12)(13)(14). These animals have difficult access to the inner ear inside the temporal bone and high maintenance costs coupled with relatively long generation times.Drosophila is a compelling alternative model system with strong genetic tools, inexpensive production of large numbers of animals, and an accessible auditory system that is becoming better understood genetically and physiologically. During courtship, Drosophila males vibrate their wings to produce a courtship song composed of pulse and sinusoidal components (15,16). This song facilitates species identification and mate selection (16,17). Drosophila males and females detect airborne vibrations via Johnston's organ (JO) in the second antennal segment (18). The JO is an array of chordotonal mechanoreceptors (or scolopidia; Fig. 1 A-C). Via the aristae, acoustic energy is transformed to rotational movement of the third antennal segment, activating mechanosensitive channels on JO neuron dendrites. Like vertebrate hair cells, JO ne...

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Development of Johnston's organ in Drosophila

Cited by 63 publications

References 81 publications

Increased activity of Diaphanous homolog 3 ( DIAPH3 )/ diaphanous causes hearing defects in humans with auditory neuropathy and in Drosophila

Increased activity of Diaphanous homolog 3 ( DIAPH3 )/ diaphanous causes hearing defects in humans with auditory neuropathy and in Drosophila

Antennal hearing in insects – New findings, new questions

Physiological, anatomical, and behavioral changes after acoustic trauma in Drosophila melanogaster

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