Esco2 regulates <i>cx43</i> expression during skeletal regeneration in the zebrafish fin

Banerji, Rajeswari; Eble, Diane M.; Iovine, M. Kathryn; Skibbens, Robert V.

doi:10.1002/dvdy.24354

Cited by 36 publications

(93 citation statements)

References 70 publications

(152 reference statements)

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“…However, a subset of mutant cells exhibit normal mitoses, suggesting the potential for multiple mechanisms underlying RBS phenotypes (Percival et al, ). Moreover, an apoptotic‐independent mechanism underlying bone and tissue growth defects was reported in the regenerating‐fin Zebrafish model (Banerji et al, ).…”

Section: Part Ii: State Of Understanding—suggested Mechanisms Of Diseasementioning

confidence: 99%

“…Zebrafish is a powerful and penetrant model in which to study human disease, including cohesinopathies (Horsfield et al, ). For instance, the Zebrafish CNS, heart, gut, cephalic structures, and skeleton appear exquisitely sensitive to cohesins, Nipbl, and Esco2 levels (Muto et al, ; Pistocchi et al, ; Muto et al, ; Banerji et al, ; Fazio et al, ). Similar to that of humans, the Zebrafish genome harbors orthologues of most cohesion genes ( nipbla and nipblb; esco1 and esco2; smc1a and smc1al; smc3; rad21a and rad21b; stag1a/b and stag2a/b and stag3 ; pds5a and pds5b ).…”

Section: Part I: Area Of Study and Significancementioning

confidence: 99%

“…Fin regeneration is a powerful model system from which to distinguish between different RBS models. For example, morpholino‐mediated esco2 knockdown phenotypes include reduced regenerate length, bone segment length, and cell proliferation (Banerji et al, ). It is important to note that apoptosis levels are not increased in esco2 morphant regenerating tails, in contrast to the embryonic model of RBS (Monnich et al, ; Morita et al, ; Percival et al, ).…”

Section: Part Ii: State Of Understanding—suggested Mechanisms Of Diseasementioning

confidence: 99%

“…Intriguingly, Esco2‐dependent phenotypes mimic those of the cx43 mutant, short fin ( sof b123 ) (Iovine et al, ; Hoptak‐Solga et al, ; Sims et al, ), suggesting that Esco2 may directly regulate cx43 expression. Remarkably, Esco2 knockdown indeed reduces expression of the cx43 gene as well as cx43 ‐dependent genes (Banerji et al, ). Overexpression of cx43 rescues the Esco2‐dependent phenotypes, providing compelling support for the hypothesis that esco2 and cx43 function in a linear pathway in which cx43 is downstream of Esco2 function.…”

Section: Part Ii: State Of Understanding—suggested Mechanisms Of Diseasementioning

confidence: 99%

“…There is compelling evidence that cohesion pathway factors regulate gene transcription (Dorsett and Krantz, ; Cucco and Musio, ; Dorsett, ). While microarray analyses of individual cohesion pathway gene knockdowns often reveal limited overlap in gene deregulation (Rhodes et al, ; Monnich et al, ; Muto et al, ; Muto et al, ), new studies provide a growing body of evidence that RBS models, similar to those of CdLS, exhibit significant changes in gene expression (Banerji et al, ; Choi et al, ; Leem et al, ; Monnich et al, ; Xu et al, ; Rahman et al, ). This unifying model may appear to be challenged by disparate findings regarding NIPBL, cohesin, and CTCF residency on chromatin (Misulovin et al, ; Kagey et al, ; Muto et al, ; Zuin et al, ; Minamino et al, ; Rahman et al, ).…”

Section: Part Iii: Major Questions To Be Addressedmentioning

confidence: 99%

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How many roads lead to cohesinopathies?

Banerji

Skibbens

Iovine

2017

Developmental Dynamics

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Genetic mapping studies reveal that mutations in cohesion pathways are responsible for multispectrum developmental abnormalities termed cohesinopathies. These include Roberts syndrome (RBS), Cornelia de Lange Syndrome (CdLS), and Warsaw Breakage Syndrome (WABS). The cohesinopathies are characterized by overlapping phenotypes ranging from craniofacial deformities, limb defects, and mental retardation. Though these syndromes share a similar suite of phenotypes and arise due to mutations in a common cohesion pathway, the underlying mechanisms are currently believed to be distinct. Defects in mitotic failure and apoptosis i.e. trans DNA tethering events are believed to be the underlying cause of RBS, whereas the underlying cause of CdLS is largely modeled as occurring through defects in transcriptional processes i.e. cis DNA tethering events. Here, we review recent findings described primarily in zebrafish, paired with additional studies in other model systems, including human patient cells, which challenge the notion that cohesinopathies represent separate syndromes. We highlight numerous studies that illustrate the utility of zebrafish to provide novel insights into the phenotypes, genes affected and the possible mechanisms underlying cohesinopathies. We propose that transcriptional deregulation is the predominant mechanism through which cohesinopathies arise. Developmental Dynamics 246:881-888, 2017. V C 2017 Wiley Periodicals, Inc.

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Section: Part Ii: State Of Understanding—suggested Mechanisms Of Diseasementioning

confidence: 99%

Section: Part I: Area Of Study and Significancementioning

confidence: 99%

Section: Part Ii: State Of Understanding—suggested Mechanisms Of Diseasementioning

confidence: 99%

Section: Part Ii: State Of Understanding—suggested Mechanisms Of Diseasementioning

confidence: 99%

Section: Part Iii: Major Questions To Be Addressedmentioning

confidence: 99%

See 3 more Smart Citations

How many roads lead to cohesinopathies?

Banerji

Skibbens

Iovine

2017

Developmental Dynamics

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Gap junctional signaling in pattern regulation: Physiological network connectivity instructs growth and form

Mathews

Levin

2016

Developmental Neurobiology

121

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Gap junctions (GJs) are aqueous channels that allow cells to communicate via physiological signals directly. The role of gap junctional connectivity in determining single-cell functions has long been recognized. However, GJs have another important role: the regulation of large-scale anatomical pattern. GJs are not only versatile computational elements that allow cells to control which small molecule signals they receive and emit, but also establish connectivity patterns within large groups of cells. By dynamically regulating the topology of bioelectric networks in vivo, GJs underlie the ability of many tissues to implement complex morphogenesis. Here, a review of recent data on patterning roles of GJs in growth of the zebrafish fin, the establishment of left-right patterning, the developmental dysregulation known as cancer, and the control of large-scale head-tail polarity, and head shape in planarian regeneration has been reported. A perspective in which GJs are not only molecular features functioning in single cells, but also enable global neural-like dynamics in non-neural somatic tissues has been proposed. This view suggests a rich program of future work which capitalizes on the rapid advances in the biophysics of GJs to exploit GJ-mediated global dynamics for applications in birth defects, regenerative medicine, and morphogenetic bioengineering. © 2016 Wiley Periodicals, Inc. Develop Neurobiol 77: 643-673, 2017.

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Cx43 suppresses evx1 expression to regulate joint initiation in the regenerating fin

Dardis

Tryon

Ton

et al. 2017

Developmental Dynamics

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Background How joints are correctly positioned in the vertebrate skeleton remains poorly understood. From our studies on the regenerating fin, we have evidence that the gap junction protein Cx43 suppresses joint formation by suppressing the expression of the evx1 transcription factor. Joint morphogenesis proceeds through at least two discrete stages. First, cells that will produce the joint condense in a single row on the bone matrix (“initiation”). Second, these cells separate coincident with articulation of the bone matrix. We propose that Cx43 activity is transiently reduced prior to joint initiation. Results We first define the timing of joint initiation with respect to regeneration. We next correlate reduced cx43 expression and increased evx1 expression with initiation. Through manipulation of cx43 expression we demonstrate that Cx43 negatively influences evx1 expression and joint formation. We further demonstrate that Cx43 activity in the dermal fibroblasts is required to rescue joint formation in the cx43 mutant, short fin b123. Conclusions We conclude that Cx43 activity in the dermal fibroblasts influences the expression of evx1, and therefore the differentiation of the precursor cells that give rise to the joint-forming osteoblasts.

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Esco2 regulates cx43 expression during skeletal regeneration in the zebrafish fin

Cited by 36 publications

References 70 publications

How many roads lead to cohesinopathies?

How many roads lead to cohesinopathies?

Gap junctional signaling in pattern regulation: Physiological network connectivity instructs growth and form

Cx43 suppresses evx1 expression to regulate joint initiation in the regenerating fin

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