Role of chromatin remodeling gene <i>Cecr2</i> in neurulation and inner ear development

Dawe, Christine E.; Kooistra, Megan; Fairbridge, Nicholas A.; Pisio, Amanda C; McDermid, Heather E.

doi:10.1002/dvdy.22547

Cited by 26 publications

(34 citation statements)

References 47 publications

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“…CECR2 contributes to sperm fertility and may regulate Dlx5 in the testis CECR2 plays important roles in neurulation and inner ear development, 10,19 but its functional roles in adult tissues have not been explored. In the present study, we found that CECR2 influences the ability of spermatozoa to fertilize oocytes.…”

Section: Discussionmentioning

confidence: 99%

“…The Cecr2 Gt45Bic gene trap mutation on BALB/c background (Cecr2 Gt ) was generated previously and genotyped by multiplex PCR. 10,19 For our initial fertility testing (see Fig. 1), 11 BALB/c Cecr2 Gt45Bic mutant males and 23 wild-type males between ages of 10 and 24 weeks were each set up in mating pairs to wild-type females and left together for 3 months.…”

Section: Methodsmentioning

confidence: 99%

“…17 The SNF2L-containing complex CERF was purified from human embryonic kidney cells (HEK293) and includes the bromodomain-containing protein CECR2. 10 CECR2-containing complexes play important roles in embryogenesis, since mutations in Cecr2 causes neural tube and inner ear defects; 10,18,19 however, the role of CECR2 in adult tissues has not been investigated. In the present study, we investigated the role of CECR2 in the male germ line and report that CECR2 is involved in spermatogenesis.…”

Section: Introductionmentioning

confidence: 99%

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CECR2 Is Involved in Spermatogenesis and Forms a Complex with SNF2H in the Testis

Thompson

Norton

Niri

et al. 2012

Journal of Molecular Biology

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Section: Discussionmentioning

confidence: 99%

Section: Methodsmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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CECR2 Is Involved in Spermatogenesis and Forms a Complex with SNF2H in the Testis

Thompson

Norton

Niri

et al. 2012

Journal of Molecular Biology

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“…17,18 SLC25A18 catalyzes unidirectional transport of glutamate in the inner membranes of mitochondria. 19 ATP6V1E1 codes for the regulatory accessory subunit E of the vacuolar-type H(+)-ATPase, which is responsible for acidification of intracellular organelles and involved in hydrogen ion transport across the plasma membrane.…”

Section: Discussionmentioning

confidence: 99%

A 600 kb triplication in the cat eye syndrome critical region causes anorectal, renal and preauricular anomalies in a three-generation family

et al. 2012

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Cat eye syndrome (CES) is caused by a gain of the proximal part of chromosome 22. Usually, a supernumerary marker chromosome is present, containing two extra copies of the chromosome 22q11.1q11.21 region. More sporadically, the gain is present intrachromosomally. The critical region for CES is currently estimated to be about 2.1 Mb and to contain at least 14 RefSeq genes. Gain of this region may cause ocular coloboma, preauricular, anorectal, urogenital and congenital heart malformations. We describe a family in which a 600 kb intrachromosomal triplication is present in at least three generations. The copy number alteration was detected using MLPA and further characterized with interphase and metaphase FISH and SNP-array. The amplified fragment is located in the distal part of the CES region. The family members show anal atresia and preauricular tags or pits, matching part of the phenotype of this syndrome. This finding suggests that amplification of the genes CECR2, SLC25A18 and ATP6V1E1, mapping within the critical region for CES, may be responsible for anorectal, renal and preauricular anomalies in patients with CES.

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“…A recent report has shown that the ATPdependent chromatin-remodeling enzyme CHD7 regulates proneural gene expression and neurogenesis in the inner ear but might function in parallel with Eya1 (Hurd et al, 2010). Another ATP-dependent chromatin-remodeling protein CECR2 has also been reported to be involved in inner ear development (Dawe et al, 2011). It will be interesting to test whether there is functional redundancy or crosstalk between these different ATP-dependent chromatin-remodeling complexes in mediating neuronal differentiation in the inner ear.…”

Section: Requirement Of the Swi/snf Complex For Mediating The Transcrmentioning

confidence: 99%

EYA1 and SIX1 drive the neuronal developmental program in cooperation with the SWI/SNF chromatin-remodeling complex and SOX2 in the mammalian inner ear

2012

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SUMMARYInner ear neurogenesis depends upon the function of the proneural basic helix-loop-helix (bHLH) transcription factors NEUROG1 and NEUROD1. However, the transcriptional regulation of these factors is unknown. Here, using loss-and gain-of-function models, we show that EYA1 and SIX1 are crucial otic neuronal determination factors upstream of NEUROG1 and NEUROD1. Overexpression of both Eya1 and Six1 is sufficient to convert non-neuronal epithelial cells within the otocyst and cochlea as well as the 3T3 fibroblast cells into neurons. Strikingly, all the ectopic neurons express not only Neurog1 and Neurod1 but also mature neuronal markers such as neurofilament, indicating that Eya1 and Six1 function upstream of, and in the same pathway as, Neurog1 and Neurod1 to not only induce neuronal fate but also regulate their differentiation. We demonstrate that EYA1 and SIX1 interact directly with the SWI/SNF chromatin-remodeling subunits BRG1 and BAF170 to drive neurogenesis cooperatively in 3T3 cells and cochlear nonsensory epithelial cells, and that SOX2 cooperates with these factors to mediate neuronal differentiation. Importantly, we show that the ATPase BRG1 activity is required for not only EYA1-and SIX1-induced ectopic neurogenesis but also normal neurogenesis in the otocyst. These findings indicate that EYA1 and SIX1 are key transcription factors in initiating the neuronal developmental program, probably by recruiting and interacting with the SWI/SNF chromatinremodeling complex to specifically mediate Neurog1 and Neurod1 transcription. Moreover, many genes have been shown to require SWI/SNF complexes for activation in yeast, fruit flies and mammals (Armstrong et al., 2002;Krebs et al., 2000;Liu et al., 2001;Ng et al., 2002;Sudarsanam et al., 2000). Recently, the ATP-dependent chromatin-remodeling enzyme CHD7 has been shown to regulate neurogenesis in the inner ear (Hurd et al., 2010). However, whether the SWI/SNF complexes play a role in mammalian inner ear neurogenesis and whether they interact with other transcription factors to regulate the transcriptional activities of Neurog1 and Neurod1 are not understood. The murine eyes absent (Eya) and homeobox Six gene families, homologous to Drosophila eyes absent and sine oculis, respectively, play essential roles for inner ear development. Haploinsufficiency for human EYA1 or SIX1 leads to branchio-oto-renal syndrome (Abdelhak et al., 1997;Ruf et al., 2004), and genetic deletion of either gene in mice results in early arrest of inner ear development at the otocyst stage (Xu et al., 1999;Zheng et al., 2003;Zou et al., 2006). We have shown that Six1 functions downstream of and interacts genetically with Eya1 during inner ear development (Xu et al., 1999;Zheng et al., 2003;Zou et al., 2006), and their gene products participate in protein-protein interaction (Buller et al., 2001). In Eya1-or Six1-null mutants, neurogenesis is initiated normally but the neuroblast cells fail to form a morphologically detectable ganglion owing to abnormal apoptosis (Zou et al., 2004), ind...

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Role of chromatin remodeling gene Cecr2 in neurulation and inner ear development

Cited by 26 publications

References 47 publications

CECR2 Is Involved in Spermatogenesis and Forms a Complex with SNF2H in the Testis

CECR2 Is Involved in Spermatogenesis and Forms a Complex with SNF2H in the Testis

A 600 kb triplication in the cat eye syndrome critical region causes anorectal, renal and preauricular anomalies in a three-generation family

EYA1 and SIX1 drive the neuronal developmental program in cooperation with the SWI/SNF chromatin-remodeling complex and SOX2 in the mammalian inner ear

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