Spatiotemporal expression of zic genes during vertebrate inner ear development

Chervenak, Andrew P.; Hakim, Ibrahim; Barald, Kate F.

doi:10.1002/dvdy.23978

Cited by 9 publications

(15 citation statements)

References 64 publications

(108 reference statements)

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“…Each of the Zic genes is expressed in the dorsal hindbrain and periotic mesenchyme (POM) adjacent to the developing inner ear, but not in the developing otic epithelium, in either mouse or chick embryos. Similar to findings for other regions where the Zic genes are expressed (Elms et al, 2004), each Zic gene has a unique spatiotemporal expression pattern during inner ear development, but the spatio-temporal expression of any individual Zic gene partially overlaps with another/others (Chervenak et al, 2013). Moreover, the Zic genes have been proposed to interact with the SHH, BMP, and WNT signaling pathways (Rohr et al, 1999;Nyholm et al, 2007), each of which is implicated in otic vesicle development.…”

Section: Introductionmentioning

confidence: 57%

“…Despite the long‐term availability of Zic mouse mutants and a growing list of Zic ‐dependent biological processes (Houtmeyers et al, ), the molecular basis of Zic requirements is generally unknown and it is likely that further Zic ‐dependent processes remain to be discovered. To determine whether the Zic genes may be involved in inner ear development, we recently characterized the expression of Zic1–5 (mouse) and Zic1–4 (chick) in the region of the developing inner ear of chick and mouse embryos (Chervenak et al, ). Each of the Zic genes is expressed in the dorsal hindbrain and periotic mesenchyme (POM) adjacent to the developing inner ear, but not in the developing otic epithelium, in either mouse or chick embryos.…”

Section: Introductionmentioning

confidence: 99%

“…Each of the gene pairs appears to share some regulatory elements, such that Zic1 and Zic4 have highly overlapping mRNA expression patterns as do Zic2 and Zic5 (Houtmeyers et al, 2013). Furthermore, in some cases the expression of all five Zic genes overlaps, such as during inner ear development in both mouse and chick (Chervenak et al, 2013), raising the possibility that the Zic genes could act redundantly during development. Mutation of individual Zic genes does, however, produce exclusive phenotypes indicating partial functional divergence (Grinberg and Millen, 2005;Houtmeyers et al, 2013).…”

Section: Introductionmentioning

confidence: 99%

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The role of Zic genes in inner ear development in the mouse: Exploring mutant mouse phenotypes

Chervenak

Bank

Thomsen

et al. 2014

Developmental Dynamics

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Background Murine Zic genes (Zic1-5) are expressed in the dorsal hindbrain and in periotic mesenchyme (POM) adjacent to the developing inner ear. Zic genes are involved in developmental signaling pathways in many organ systems, including the ear, although their exact roles haven't been fully elucidated. This report examines the role of Zic1, Zic2, and Zic4 during inner ear development in mouse mutants in which these Zic genes are affected Results Zic1/Zic4 double mutants don't exhibit any apparent defects in inner ear morphology. By contrast, inner ears from Zic2kd/kd and Zic2Ku/Ku mutants have severe but variable morphological defects in endolymphatic duct/sac and semicircular canal formation and in cochlear extension in the inner ear. Analysis of otocyst patterning in the Zic2Ku/Ku mutants by in situ hybridization showed changes in the expression patterns of Gbx2 and Pax2. Conclusions The experiments provide the first genetic evidence that the Zic genes are required for morphogenesis of the inner ear. Zic2 loss-of-function doesn't prevent initial otocyst patterning but leads to molecular abnormalities concomitant with morphogenesis of the endolymphatic duct. Functional hearing deficits often accompany inner ear dysmorphologies, making Zic2 a novel candidate gene for ongoing efforts to identify the genetic basis of human hearing loss.

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Section: Introductionmentioning

confidence: 57%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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The role of Zic genes in inner ear development in the mouse: Exploring mutant mouse phenotypes

Chervenak

Bank

Thomsen

et al. 2014

Developmental Dynamics

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“…8A-E, 9A-E; (Lillevali et al, 2007)); Pax2 positive but Gata3, Crabp1 , and Tbx3 negative cells defined the SM (Fig. 8A-E, 9A-E; (Chervenak et al, 2013; Li et al, 2004)); Gata3 positive but Crabp1 and Tbx3 negative cells defined the basilar papilla (BP, Fig. 8A-E, 9A-E).…”

Section: Resultsmentioning

confidence: 99%

Temporal coupling between specifications of neuronal and macular fates of the inner ear

Deng

2016

Developmental Biology

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The inner ear is a complex organ comprised of various specialized sensory organs for detecting sound and head movements. The timing of specification for these sensory organs, however, is not clear. Previous fate mapping results of the inner ear indicate that vestibular and auditory ganglia and two of the vestibular sensory organs, the utricular macula (UM) and saccular macula (SM), are lineage related. Based on the medial-lateral relationship where respective auditory and vestibular neuroblasts exit from the otic epithelium and the subsequent formation of the medial SM and lateral UM in these regions, we hypothesized that specification of the two lateral structures, the vestibular ganglion and the UM are coupled and likewise for the two medial structures, the auditory ganglion and the SM. We tested this hypothesis by surgically inverting the primary axes of the otic cup in ovo and investigating the fate of the vestibular neurogenic region, which had been spotted with a lipophilic dye. Our results showed that the laterally-positioned, dye-associated, vestibular ganglion and UM were largely normal in transplanted ears, whereas both auditory ganglion and SM showed abnormalities suggesting the lateral but not the medial-derived structures were mostly specified at the time of transplantation. Both of these results are consistent with a temporal coupling between neuronal and macular fate specifications.

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“…It is one of the most important factors affecting neural tube development, cell differentiation and proliferation. ZIC2 alterations have been connected to pathogenesis and progression of some tumours [7,8,13,24,30]. Another candidate gene, located at chromosome 13q and hence notoriously affected by GCTs, is FGF9.…”

Section: Discussionmentioning

confidence: 99%

Hedgehog signalling network gene status analysis in paediatric intracranial germ cell tumours

Kuleszo

Lipska‐Ziętkiewicz

Koczkowska

et al. 2019

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Introduction: Germ cell tumours (GCTs) in the children comprise a group of tumours that originate from primordial germ cells but their pathogenesis is not clear. Intracranial GCTs represent a special subset of these paediatric neoplasms. Hedgehog (Hh) pathway gene status in GCTs is generally unexplored, while Hh signalling is involved in germ cell biology. Material and methods: Comparative genomic profiling analysis with a microarray-comparative genomic hybridization (CGH) + single nucleotide polymorphism (SNP) technique in a group of intracranial paediatric GCTs was performed. The analysis included evaluation of genes being ligands, receptors, regulators, effectors, and targets of Hh signalling. Results: Chromosomal aberrations were found in 62% of examined tumours, showing their heterogeneity. A number of private genomic imbalances were observed, but only a few recurrent ones. The most common numerical changes were trisomies 19, 21 and monosomies 13, 18 while the most frequent structural aberration was gain/ amplification of the chromosome 12p. The analysis of the gene status of Hh network elements showed imbalances in a proportion of tumours. PTCH1, GLI2, IHH and ZIC2 gene aberrations occurred most frequently. Moreover, six tumours had various copy gains or losses of several other genes involved in the pathway, including HHIP, GLI1, GLI3, DHH, SHH, SMO, PTCH2, and several genes from the WNT group. Interestingly, four cases showed losses of pathway repressors, with parallel gains of activators in two of them. Correlations with patho-clinical tumour features were not found, most probably due to the heterogeneity of the examined limited group. Conclusions: Our results show few genomic alterations related to the Hh signalling pathway genes in paediatric intracranial GCTs. Further analysis of Hedgehog pathway alterations can potentially disclose its biological significance and define new prognostic factors and/or therapeutic targets for high-risk patients.

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Spatiotemporal expression of zic genes during vertebrate inner ear development

Cited by 9 publications

References 64 publications

The role of Zic genes in inner ear development in the mouse: Exploring mutant mouse phenotypes

The role of Zic genes in inner ear development in the mouse: Exploring mutant mouse phenotypes

Temporal coupling between specifications of neuronal and macular fates of the inner ear

Hedgehog signalling network gene status analysis in paediatric intracranial germ cell tumours

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