The miR-183/ItgA3 axis is a key regulator of prosensory area during early inner ear development

Ackerveken, Priscilla Van den; Mounier, Anaïs; Huyghe, Aurélia; Sacheli, Rosalie; Vanlerberghe, Pierre-Bernard; Volvert, Marie-Laure; Delacroix, Laurence; Nguyen, Laurent; Malgrange, Brigitte

doi:10.1038/cdd.2017.127

Cited by 12 publications

(3 citation statements)

References 58 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…The absence of a retinal phenotype in any of the mutants indicates that, at least in zebrafish, these miRNAs are collectively dispensable for that tissue. Our study is consistent with previous reports describing their role in hair cells 24–26,31,61,62 , and expands on those findings by shedding light on their relative contribution to the support of those cells. Because neither the mir-183 lri70 , mir-96 lri60 , nor mir-182 lri61 mutants had compromised hair cells, we can also conclude that there is no absolute requirement for any one of the three miRNAs.…”

Section: Discussionsupporting

confidence: 93%

Genomic non-redundancy of the mir-183/96/182 cluster and its requirement for hair cell maintenance

Fogerty

Stepanyan

Cianciolo

et al. 2019

Sci Rep

View full text Add to dashboard Cite

microRNAs are important regulators of gene expression. In the retina, the mir-183/96/182 cluster is of particular interest due to its robust expression and studies in which loss of the cluster caused photoreceptor degeneration. However, it is unclear which of the three miRNAs in the cluster are ultimately required in photoreceptors, whether each may have independent, contributory roles, or whether a single miRNA from the cluster compensates for the loss of another. These are important questions that will not only help us to understand the role of these particular miRNAs in the retina, but will deepen our understanding of how clustered microRNAs evolve and operate. To that end, we have developed a complete panel of single, double, and triple mir-183/96/182 mutant zebrafish. While the retinas of all mutant animals were normal, the triple mutants exhibited acute hair cell degeneration which corresponded with impaired swimming and death at a young age. By measuring the penetrance of this phenotype in each mutant line, we determine which of the three miRNAs in the cluster are necessary and/or sufficient to ensure normal hair cell development and function.

show abstract

Section: Discussionsupporting

confidence: 93%

Genomic non-redundancy of the mir-183/96/182 cluster and its requirement for hair cell maintenance

Fogerty

Stepanyan

Cianciolo

et al. 2019

Sci Rep

View full text Add to dashboard Cite

show abstract

“…A previous study in the mouse inner ear has shown that progressive reduction of miR-183 expression after Pax2 -Cre conditional Dicer knockout (KO) results in progressive loss of neurosensory gene expression, arrested neurosensory development and loss of CVGs with an associated disruption of morphogenesis [ 4 ]. More recently, a Dicer1 conditional KO line ( Atoh1-cre;Dicer1 flox/flox and Foxg1-cre; Dicer1 flox/flox ) showed defects in proliferation in the prosensory domain of the cochlea including CVGs [ 7 , 8 , 58 ]. Our observations coupled with the findings in mice suggest that Dicer pathway interactions with the miR-183 family play an essential role in the regulation of early mice inner ear development.…”

Section: Discussion/conclusionmentioning

confidence: 99%

Developmental profiling of microRNAs in the human embryonic inner ear

et al. 2018

View full text Add to dashboard Cite

Due to the extreme inaccessibility of fetal human inner ear tissue, defining of the microRNAs (miRNAs) that regulate development of the inner ear has relied on animal tissue. In the present study, we performed the first miRNA sequencing of otic precursors in human specimens. Using HTG miRNA Whole Transcriptome assays, we examined miRNA expression in the cochleovestibular ganglion (CVG), neural crest (NC), and otic vesicle (OV) from paraffin embedded (FFPE) human specimens in the Carnegie developmental stages 13–15. We found that in human embryonic tissues, there are different patterns of miRNA expression in the CVG, NC and OV. In particular, members of the miR-183 family (miR-96, miR-182, and miR-183) are differentially expressed in the CVG compared to NC and OV at Carnegie developmental stage 13. We further identified transcription factors that are differentially targeted in the CVG compared to the other tissues from stages 13–15, and we performed gene set enrichment analyses to determine differentially regulated pathways that are relevant to CVG development in humans. These findings not only provide insight into the mechanisms governing the development of the human inner ear, but also identify potential signaling pathways for promoting regeneration of the spiral ganglion and other components of the inner ear.

show abstract

“…In the inner ear, RNA-sequencing experiments have led to the identification of hundreds of miRNAs ( 143 ). Global abolishment of miRNA expression in the mouse inner ear was achieved by conditional knockout of Dicer at different stages of development ( 47 , 73 , 154 , 166 ). All such mouse lines exhibit severe cochlear defects, such as thinner cochlear ducts or disorganized stereocilia bundles.…”

Section: The Noncoding Genome and The Inner Earmentioning

confidence: 99%

The Genomics of Auditory Function and Disease

Taiber

Gwilliam

Hertzano

et al. 2022

Annu. Rev. Genom. Hum. Genet.

View full text Add to dashboard Cite

Current estimates suggest that nearly half a billion people worldwide are affected by hearing loss. Because of the major psychological, social, economic, and health ramifications, considerable efforts have been invested in identifying the genes and molecular pathways involved in hearing loss, whether genetic or environmental, to promote prevention, improve rehabilitation, and develop therapeutics. Genomic sequencing technologies have led to the discovery of genes associated with hearing loss. Studies of the transcriptome and epigenome of the inner ear have characterized key regulators and pathways involved in the development of the inner ear and have paved the way for their use in regenerative medicine. In parallel, the immense preclinical success of using viral vectors for gene delivery in animal models of hearing loss has motivated the industry to work on translating such approaches into the clinic. Here, we review the recent advances in the genomics of auditory function and dysfunction, from patient diagnostics to epigenetics and gene therapy. Expected final online publication date for the Annual Review of Genomics and Human Genetics, Volume 23 is October 2022. Please see http://www.annualreviews.org/page/journal/pubdates for revised estimates.

show abstract

The miR-183/ItgA3 axis is a key regulator of prosensory area during early inner ear development

Cited by 12 publications

References 58 publications

Genomic non-redundancy of the mir-183/96/182 cluster and its requirement for hair cell maintenance

Genomic non-redundancy of the mir-183/96/182 cluster and its requirement for hair cell maintenance

Developmental profiling of microRNAs in the human embryonic inner ear

The Genomics of Auditory Function and Disease

Contact Info

Product

Resources

About