From Cloning Neural Development Genes to Functional Studies in Mice, 30 Years of Advancements

Joyner, Alexandra L.

doi:10.1016/bs.ctdb.2015.11.035

Cited by 7 publications

(4 citation statements)

References 78 publications

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“…Later, cloning of the causative genes was facilitated by the availability of the mouse genome sequence, revealing the first molecular insights into the genetic programs driving these events. The advent and rapid evolution of genome engineering technologies (Singh et al, 2015; Joyner, 2016) has since enabled the generation of a multitude of new mouse models to specifically assess the function of any gene expressed during cerebellar development. Further, mouse mutants directly modelling human cerebellar disease mutations are readily constructed.…”

Section: Introductionmentioning

confidence: 99%

What cerebellar malformations tell us about cerebellar development

Haldipur¹,

Millen

2019

Neuroscience Letters

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Structural birth defects of the cerebellum, or cerebellar malformations, in humans, have long been recognized. However, until recently there has been little progress in elucidating their developmental pathogenesis. Innovations in brain imaging and human genetic technologies over the last 2 decades have led to better classifications of these disorders and identification of several causative genes. In contrast, cerebellar malformations in model organisms, particularly mice, have been the focus of intense study for more than 70 years. As a result, many of the molecular, genetic and cellular programs that drive formation of the cerebellum have been delineated in mice. In this review, we overview the basic epochs and key molecular regulators of the developmental programs that build the structure of the mouse cerebellum. This mouse-centric approach has been a useful to interpret the developmental pathogenesis of human cerebellar malformations. However, it is becoming apparent that we actually know very little regarding the specifics of human cerebellar development beyond what is inferred from mice. A better understanding of human cerebellar development will not only facilitate improved diagnosis of human cerebellar malformations, but also lead to the development of treatment paradigms for these important neurodevelopmental disorders.

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Section: Introductionmentioning

confidence: 99%

What cerebellar malformations tell us about cerebellar development

Haldipur¹,

Millen

2019

Neuroscience Letters

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“…The Cre recombinases can mediate specific recombination of gene sequences riveted by loxp. The Cre/loxp system was first used in vivo cell lineage tracing studies by Joyner et al [ 5 ]. They constructed two kinds of transgenic mice in the process of studying brain development.…”

Section: Lineage Tracingmentioning

confidence: 99%

Lineage Tracing and Molecular Real-Time Imaging of Cancer Stem Cells

et al. 2022

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The cancer stem cells (CSC) are the roots of cancer. The CSC hypothesis may provide a model to explain the tumor cell heterogeneity. Understand the biological mechanism of CSC will help the early detection and cure of cancer. The discovery of the dynamic changes in CSC will be possible by the using of bio-engineering techniques-lineage tracing. However, it is difficult to obtain real-time, continuous, and dynamic live-imaging information using the traditional approaches that take snapshots of time points from different animals. The goal of molecular imaging is to monitor the in situ, continuous molecular changes of cells in vivo. Therefore, the most advanced bioengineering lineage tracing approach, while using a variety of molecular detection methods, will maximize the presentation of CSC. In this review, we first introduce the method of lineage tracing, and then introduce the various components of molecular images to dynamic detect the CSC. Finally, we analyze the current situation and look forward the future of CSC detection.

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“…More than a hundred genes are known to regulate cerebellar development. Since the genetic regulation of cerebellar development has been the focus of several recent reviews [11,21,22], this topic will only be briefly touched in this paper. Probably the most wellknown transcription factors that regulate cerebellar development are Atoh1, Pax6 and Zic1.…”

Section: Cerebellar Developmentmentioning

confidence: 99%