EYA1 Expression in the Developing Inner Ear

Bane, Brian C.; Rybroek, Jana M. Van; Kolker, Sandra J.; Weeks, Daniel L.; Manaligod, José R.

doi:10.1177/000348940511401108

Cited by 10 publications

(10 citation statements)

References 21 publications

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“…Like the Six genes, vertebrate Eya genes are expressed in numerous embryonic tissues, including the eyes, muscles, kidneys, the PPE and placodes (Duncan et al, 1997; Xu et al, 1997; Sahly et al, 1999; David et al, 2001; Ishihara et al, 2008; Neilson et al, 2010; Modrell and Baker, 2012). In Xenopus , chick and fish, the expression patterns of Eya1 and Eya2 are nearly identical to that of Six1 (Sahly et al, 1999; David et al, 2001; McLarren et al, 2003; Bane et al, 2005; Neilson et al, 2010), suggesting that they have important roles in PPE and placode development. Eya proteins contain a highly conserved protein-binding domain called the Eya domain (ED) located at the C-terminus.…”

Section: Induction and Specification Of The Pre-placodal Fieldmentioning

confidence: 97%

Transcriptional Regulation of Cranial Sensory Placode Development

Moody

LaMantia

2015

Current Topics in Developmental Biology

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Cranial sensory placodes derive from discrete patches of the head ectoderm, and give rise to numerous sensory structures. During gastrulation, a specialized “neural border zone” forms around the neural plate in response to interactions between the neural and non-neural ectoderm and signals from adjacent mesodermal and/or endodermal tissues. This zone subsequently gives rise to two distinct precursor populations of the peripheral nervous system: the neural crest and the pre-placodal ectoderm (PPE). The PPE is a common field from which all cranial sensory placodes arise (adenohypophyseal, olfactory, lens, trigeminal, epibranchial, otic). Members of the Six family of transcription factors are major regulators of PPE specification, in partnership with co-factor proteins such as Eya. Six gene activity also maintains tissue boundaries between the PPE, neural crest and epidermis by repressing genes that specify the fates of those adjacent ectodermally-derived domains. As the embryo acquires anterior-posterior identity, the PPE becomes transcriptionally regionalized, and it subsequently subdivides into specific placodes with distinct developmental fates in response to signaling from adjacent tissues. Each placode is characterized by a unique transcriptional program that leads to the differentiation of highly specialized cells, such as neurosecretory cells, somatic sensory receptor cells, chemosensory neurons, peripheral glia and supporting cells. In this review, we summarize the transcriptional and signaling factors that regulate key steps of placode development, influence subsequent sensory neuron specification, and discuss what is known about mutations in some of the essential PPE genes that underlie human congenital syndromes.

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Section: Induction and Specification Of The Pre-placodal Fieldmentioning

confidence: 97%

Transcriptional Regulation of Cranial Sensory Placode Development

Moody

LaMantia

2015

Current Topics in Developmental Biology

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“…It contains two putative classical nuclear localization signals, at residues 18–23 and 457–465, which are absent in vertebrate EYA family proteins [6]. In contrast, the vertebrate EYA is strongly distributed in the cytoplasm in vivo [47] and in cultured cells. Studies in cultured cells revealed that vertebrate EYA1–4 require coexpression of SIX1/2 or SIX4/5 subgroups to translocate into the nucleus [36,48, 49], which has not been demonstrated in vivo during development.…”

Section: Nuclear and Cytoplasmic Roles Of Eyamentioning

confidence: 99%

The EYA-SO/SIX complex in development and disease

2012

Pediatr Nephrol

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Eyes absent (EYA) and Sine oculis (SO/SIX) proteins function as transcriptional activation complexes and play essential roles in organogenesis during embryonic development in regulating cell proliferation and survival and coordination of particular differentiation programs. Mutations of the Eya and So/Six genes cause profound developmental defects in organisms as diverse as flies, frogs, fish, mice, and humans. EYA proteins also possess an intrinsic phosphatase activity, which is essential for normal development. Here, we review crucial roles of EYA and SO/SIX in development and disease in mice and humans.

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“…The focus of SO/Six co-factor research has been primarily on Eya (Eyes absent) and Gro (Groucho)/Grg (Groucho-related) family members because they are known to bind to Six proteins, affect their transcriptional activity and are expressed in the same tissues as many Six genes (Bajoghli et al, 2005; Bane et al, 2005; Brugmann and Moody, 2005; Brugmann et al, 2004; Kobayashi et al, 2001; Li et al, 2010; Neilson et al, 2010; Ohto et al, 1999; Ozaki et al, 2002; Zhu et al, 2002). But, SO/Six1 proteins also can interact with other proteins.…”

Section: Introductionmentioning

confidence: 99%

Pa2G4 is a novel Six1 co-factor that is required for neural crest and otic development

Neilson

Abbruzzesse

Kenyon

et al. 2017

Developmental Biology

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Mutations in SIX1 and in its co-factor, EYA1, underlie Branchiootorenal Spectrum disorder (BOS), which is characterized by variable branchial arch, otic and kidney malformations. However, mutations in these two genes are identified in only half of patients. We screened for other potential co-factors, and herein characterize one of them, Pa2G4 (aka Ebp1/Plfap). In human embryonic kidney cells, Pa2G4 binds to Six1 and interferes with the Six1-Eya1 complex. In Xenopus embryos, knock-down of Pa2G4 leads to down-regulation of neural border zone, neural crest and cranial placode genes, and concomitant expansion of neural plate genes. Gain-of-function leads to a broader neural border zone, expanded neural crest and altered cranial placode domains. In loss-of-function assays, the later developing otocyst is reduced in size, which impacts gene expression. In contrast, the size of the otocyst in gain-of-function assays is not changed but the expression domains of several otocyst genes are reduced. Together these findings establish an interaction between Pa2G4 and Six1, and demonstrate that it has an important role in the development of tissues affected in BOS. Thereby, we suggest that pa2g4 is a potential candidate gene for BOS.

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EYA1 Expression in the Developing Inner Ear

Cited by 10 publications

References 21 publications

Transcriptional Regulation of Cranial Sensory Placode Development

Transcriptional Regulation of Cranial Sensory Placode Development

The EYA-SO/SIX complex in development and disease

Pa2G4 is a novel Six1 co-factor that is required for neural crest and otic development

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