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Neural Crest Development in Health and Disease

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A special issue of International Journal of Molecular Sciences (ISSN 1422-0067). This special issue belongs to the section "Molecular Neurobiology".

Deadline for manuscript submissions: closed (31 March 2022) | Viewed by 39063

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Prof. Dr. Nicolas Pilon

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Département des sciences biologiques, Université du Québec à Montréal, Montreal, QC H3C 3J7, Canada
Interests: developmental biology; neural crest cells; neurocristopathies; enteric nervous system; molecular genetics; mouse models
Special Issues, Collections and Topics in MDPI journals

Special Issue Information

Dear Colleagues,

There are several reasons why so many researchers remain fascinated by neural crest cells more than a century after their first description. One reason is the central role these vertebrate-specific cells played during evolution. A second is the unique ability of these cells to migrate over very long distances during a relatively short time window. A third reason is the multipotency of these cells, which is so extensive that the neural crest is even considered a germ layer on its own, just like the ectoderm, mesoderm, and endoderm. Neural crest cells not only contribute a wide array of specialized cell types (e.g., neurons, glia, melanocytes, neuroendocrine cells, craniofacial osteoblasts, and vascular smooth muscle cells), but can also indirectly affect tissue morphogenesis by influencing the behavior and/or function of adjacent non-neural crest cells. A fourth reason is the apparent propensity of some neural crest cells to persist as tissue-resident stem cells in many adult tissues. A fifth reason is their involvement in a large number of human diseases, collectively referred to as neurocristopathies, an expanding research field that now benefits from decades of fundamental research on neural crest development.

The main objective of this Special Issue entitled “Neural Crest Development in Health and Disease” is to gather biologists from all backgrounds with a common interest in the fascinating neural crest cells. There is no particular limitation to the type of manuscripts or research questions (a list of potential topics is provided in the Keywords section below).

Prof. Dr. Nicolas Pilon
Guest Editor

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Manuscripts should be submitted online at www.mdpi.com by registering and logging in to this website. Once you are registered, click here to go to the submission form. Manuscripts can be submitted until the deadline. All submissions that pass pre-check are peer-reviewed. Accepted papers will be published continuously in the journal (as soon as accepted) and will be listed together on the special issue website. Research articles, review articles as well as short communications are invited. For planned papers, a title and short abstract (about 100 words) can be sent to the Editorial Office for announcement on this website.

Submitted manuscripts should not have been published previously, nor be under consideration for publication elsewhere (except conference proceedings papers). All manuscripts are thoroughly refereed through a single-blind peer-review process. A guide for authors and other relevant information for submission of manuscripts is available on the Instructions for Authors page. International Journal of Molecular Sciences is an international peer-reviewed open access semimonthly journal published by MDPI.

Please visit the Instructions for Authors page before submitting a manuscript. There is an Article Processing Charge (APC) for publication in this open access journal. For details about the APC please see here. Submitted papers should be well formatted and use good English. Authors may use MDPI's English editing service prior to publication or during author revisions.

Keywords

Animal models
Cellular models
Cell fate determination
Cell metabolism
Cell migration
Cell signaling
Disease modeling
Evolution
Gene–environment interactions
Gene regulatory networks
Neural-crest-related congenital malformations
Neural-crest-related cancers
Molecular genetics
Molecular therapies
Pathogenic mechanisms
Stem-cell-based therapies

Published Papers (14 papers)

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Editorial

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2 pages, 186 KiB

Open AccessEditorial

Neural Crest Development in Health and Disease

by Nicolas Pilon

Int. J. Mol. Sci. 2022, 23(22), 13684; https://0-doi-org.brum.beds.ac.uk/10.3390/ijms232213684 - 08 Nov 2022

Viewed by 919

Abstract

The first volume of this Special Issue met its goal of covering several aspects regarding both the normal and abnormal development of neural crest cells, which form a truly unique multipotent and highly migratory cell population that only exists in vertebrates [...] Full article

(This article belongs to the Special Issue Neural Crest Development in Health and Disease)

Research

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16 pages, 2971 KiB

Open AccessArticle

A New Transgenic Tool to Study the Ret Signaling Pathway in the Enteric Nervous System

by Ashoka Bandla, Ellie Melancon, Charlotte R. Taylor, Ann E. Davidson, Judith S. Eisen and Julia Ganz

Int. J. Mol. Sci. 2022, 23(24), 15667; https://0-doi-org.brum.beds.ac.uk/10.3390/ijms232415667 - 10 Dec 2022

Cited by 3 | Viewed by 1296

Abstract

The receptor tyrosine kinase Ret plays a critical role in regulating enteric nervous system (ENS) development. Ret is important for proliferation, migration, and survival of enteric progenitor cells (EPCs). Ret also promotes neuronal fate, but its role during neuronal differentiation and in the adult ENS is less well understood. Inactivating RET mutations are associated with ENS diseases, e.g., Hirschsprung Disease, in which distal bowel lacks ENS cells. Zebrafish is an established model system for studying ENS development and modeling human ENS diseases. One advantage of the zebrafish model system is that their embryos are transparent, allowing visualization of developmental phenotypes in live animals. However, we lack tools to monitor Ret expression in live zebrafish. Here, we developed a new BAC transgenic line that expresses GFP under the ret promoter. We find that EPCs and the majority of ENS neurons express ret:GFP during ENS development. In the adult ENS, GFP⁺ neurons are equally present in females and males. In homozygous mutants of ret and sox10—another important ENS developmental regulator gene—GFP⁺ ENS cells are absent. In summary, we characterize a ret:GFP transgenic line as a new tool to visualize and study the Ret signaling pathway from early development through adulthood. Full article

(This article belongs to the Special Issue Neural Crest Development in Health and Disease)

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18 pages, 4598 KiB

Open AccessArticle

Craniofacial Defects in Embryos with Homozygous Deletion of Eftud2 in Their Neural Crest Cells Are Not Rescued by Trp53 Deletion

by Marie-Claude Beauchamp, Alexia Boucher, Yanchen Dong, Rachel Aber and Loydie A. Jerome-Majewska

Int. J. Mol. Sci. 2022, 23(16), 9033; https://doi.org/10.3390/ijms23169033 - 12 Aug 2022

Cited by 2 | Viewed by 1529

Abstract

Embryos with homozygous mutation of Eftud2 in their neural crest cells (Eftud2^ncc−/−) have brain and craniofacial malformations, hyperactivation of the P53-pathway and die before birth. Treatment of Eftud2^ncc−/− embryos with pifithrin-α, a P53-inhibitor, partly improved brain and craniofacial development. To uncover if craniofacial malformations and death were indeed due to P53 hyperactivation we generated embryos with homozygous loss of function mutations in both Eftud2 and Trp53 in the neural crest cells. We evaluated the molecular mechanism underlying craniofacial development in pifithrin-α-treated embryos and in Eftud2; Trp53 double homozygous (Eftud2^ncc−/−; Trp53^ncc−/−) mutant embryos. Eftud2^ncc−/− embryos that were treated with pifithrin-α or homozygous mutant for Trp53 in their neural crest cells showed reduced apoptosis in their neural tube and reduced P53-target activity. Furthermore, although the number of SOX10 positive cranial neural crest cells was increased in embryonic day (E) 9.0 Eftud2^ncc−/−; Trp53^ncc−/− embryos compared to Eftud2^ncc−/− mutants, brain and craniofacial development, and survival were not improved in double mutant embryos. Furthermore, mis-splicing of both P53-regulated transcripts, Mdm2 and Foxm1, and a P53-independent transcript, Synj2bp, was increased in the head of Eftud2^ncc−/−; Trp53^ncc−/− embryos. While levels of Zmat3, a P53- regulated splicing factor, was similar to those of wild-type. Altogether, our data indicate that both P53-regulated and P53-independent pathways contribute to craniofacial malformations and death of Eftud2^ncc−/− embryos. Full article

(This article belongs to the Special Issue Neural Crest Development in Health and Disease)

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14 pages, 4025 KiB

Open AccessArticle

Genetic Background Influences Severity of Colonic Aganglionosis and Response to GDNF Enemas in the Holstein Mouse Model of Hirschsprung Disease

by Rodolphe Soret, Nejia Lassoued, Grégoire Bonnamour, Guillaume Bernas, Aurélie Barbe, Mélanie Pelletier, Manon Aichi and Nicolas Pilon

Int. J. Mol. Sci. 2021, 22(23), 13140; https://0-doi-org.brum.beds.ac.uk/10.3390/ijms222313140 - 05 Dec 2021

Cited by 3 | Viewed by 2417

Abstract

Hirschsprung disease is a congenital malformation where ganglia of the neural crest-derived enteric nervous system are missing over varying lengths of the distal gastrointestinal tract. This complex genetic condition involves both rare and common variants in dozens of genes, many of which have been functionally validated in animal models. Modifier loci present in the genetic background are also believed to influence disease penetrance and severity, but this has not been frequently tested in animal models. Here, we addressed this question using Holstein mice in which aganglionosis is due to excessive deposition of collagen VI around the developing enteric nervous system, thereby allowing us to model trisomy 21-associated Hirschsprung disease. We also asked whether the genetic background might influence the response of Holstein mice to GDNF enemas, which we recently showed to have regenerative properties for the missing enteric nervous system. Compared to Holstein mice in their original FVB/N genetic background, Holstein mice maintained in a C57BL/6N background were found to have a less severe enteric nervous system defect and to be more responsive to GDNF enemas. This change of genetic background had a positive impact on the enteric nervous system only, leaving the neural crest-related pigmentation phenotype of Holstein mice unaffected. Taken together with other similar studies, these results are thus consistent with the notion that the enteric nervous system is more sensitive to genetic background changes than other neural crest derivatives. Full article

(This article belongs to the Special Issue Neural Crest Development in Health and Disease)

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22 pages, 5398 KiB

Open AccessArticle

SUMOylation Potentiates ZIC Protein Activity to Influence Murine Neural Crest Cell Specification

by Helen M. Bellchambers, Kristen S. Barratt, Koula E. M. Diamand and Ruth M. Arkell

Int. J. Mol. Sci. 2021, 22(19), 10437; https://0-doi-org.brum.beds.ac.uk/10.3390/ijms221910437 - 28 Sep 2021

Cited by 4 | Viewed by 2840

Abstract

The mechanisms of neural crest cell induction and specification are highly conserved among vertebrate model organisms, but how similar these mechanisms are in mammalian neural crest cell formation remains open to question. The zinc finger of the cerebellum 1 (ZIC1) transcription factor is considered a core component of the vertebrate gene regulatory network that specifies neural crest fate at the neural plate border. In mouse embryos, however, Zic1 mutation does not cause neural crest defects. Instead, we and others have shown that murine Zic2 and Zic5 mutate to give a neural crest phenotype. Here, we extend this knowledge by demonstrating that murine Zic3 is also required for, and co-operates with, Zic2 and Zic5 during mammalian neural crest specification. At the murine neural plate border (a region of high canonical WNT activity) ZIC2, ZIC3, and ZIC5 function as transcription factors to jointly activate the Foxd3 specifier gene. This function is promoted by SUMOylation of the ZIC proteins at a conserved lysine immediately N-terminal of the ZIC zinc finger domain. In contrast, in the lateral regions of the neurectoderm (a region of low canonical WNT activity) basal ZIC proteins act as co-repressors of WNT/TCF-mediated transcription. Our work provides a mechanism by which mammalian neural crest specification is restricted to the neural plate border. Furthermore, given that WNT signaling and SUMOylation are also features of non-mammalian neural crest specification, it suggests that mammalian neural crest induction shares broad conservation, but altered molecular detail, with chicken, zebrafish, and Xenopus neural crest induction. Full article

(This article belongs to the Special Issue Neural Crest Development in Health and Disease)

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18 pages, 4494 KiB

Open AccessArticle

Ptk7 Is Dynamically Localized at Neural Crest Cell–Cell Contact Sites and Functions in Contact Inhibition of Locomotion

by Anita Grund, Katharina Till, Klaudia Giehl and Annette Borchers

Int. J. Mol. Sci. 2021, 22(17), 9324; https://0-doi-org.brum.beds.ac.uk/10.3390/ijms22179324 - 28 Aug 2021

Cited by 5 | Viewed by 2269

Abstract

Neural crest (NC) cells are highly migratory cells that contribute to various vertebrate tissues, and whose migratory behaviors resemble cancer cell migration and invasion. Information exchange via dynamic NC cell–cell contact is one mechanism by which the directionality of migrating NC cells is controlled. One transmembrane protein that is most likely involved in this process is protein tyrosine kinase 7 (PTK7), an evolutionary conserved Wnt co-receptor that is expressed in cranial NC cells and several tumor cells. In Xenopus, Ptk7 is required for NC migration. In this study, we show that the Ptk7 protein is dynamically localized at cell–cell contact zones of migrating Xenopus NC cells and required for contact inhibition of locomotion (CIL). Using deletion constructs of Ptk7, we determined that the extracellular immunoglobulin domains of Ptk7 are important for its transient accumulation and that they mediate homophilic binding. Conversely, we found that ectopic expression of Ptk7 in non-NC cells was able to prevent NC cell invasion. However, deletion of the extracellular domains of Ptk7 abolished this effect. Thus, Ptk7 is sufficient at protecting non-NC tissue from NC cell invasion, suggesting a common role of PTK7 in contact inhibition, cell invasion, and tissue integrity. Full article

(This article belongs to the Special Issue Neural Crest Development in Health and Disease)

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17 pages, 5270 KiB

Open AccessArticle

Conditional Deletion of AP-2β in the Periocular Mesenchyme of Mice Alters Corneal Epithelial Cell Fate and Stratification

by Haydn Walker, Aftab Taiyab, Paula Deschamps, Trevor Williams and Judith A. West-Mays

Int. J. Mol. Sci. 2021, 22(16), 8730; https://0-doi-org.brum.beds.ac.uk/10.3390/ijms22168730 - 13 Aug 2021

Cited by 4 | Viewed by 2351

Abstract

The cornea is an anterior eye structure specialized for vision. The corneal endothelium and stroma are derived from the periocular mesenchyme (POM), which originates from neural crest cells (NCCs), while the stratified corneal epithelium develops from the surface ectoderm. Activating protein-2β (AP-2β) is highly expressed in the POM and important for anterior segment development. Using a mouse model in which AP-2β is conditionally deleted in the NCCs (AP-2β NCC KO), we investigated resulting corneal epithelial abnormalities. Through PAS and IHC staining, we observed structural and phenotypic changes to the epithelium associated with AP-2β deletion. In addition to failure of the mutant epithelium to stratify, we also observed that Keratin-12, a marker of the differentiated epithelium, was absent, and Keratin-15, a limbal and conjunctival marker, was expanded across the central epithelium. Transcription factors PAX6 and P63 were not observed to be differentially expressed between WT and mutant. However, growth factor BMP4 was suppressed in the mutant epithelium. Given the non-NCC origin of the epithelium, we hypothesize that the abnormalities in the AP-2β NCC KO mouse result from changes to regulatory signaling from the POM-derived stroma. Our findings suggest that stromal pathways such as Wnt/β-Catenin signaling may regulate BMP4 expression, which influences cell fate and stratification. Full article

(This article belongs to the Special Issue Neural Crest Development in Health and Disease)

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19 pages, 28163 KiB

Open AccessArticle

5-HT3 Signaling Alters Development of Sacral Neural Crest Derivatives That Innervate the Lower Urinary Tract

by K. Elaine Ritter, Dennis P. Buehler, Stephanie B. Asher, Karen K. Deal, Shilin Zhao, Yan Guo and E Michelle Southard-Smith

Int. J. Mol. Sci. 2021, 22(13), 6838; https://0-doi-org.brum.beds.ac.uk/10.3390/ijms22136838 - 25 Jun 2021

Cited by 4 | Viewed by 2220

Abstract

The autonomic nervous system derives from the neural crest (NC) and supplies motor innervation to the smooth muscle of visceral organs, including the lower urinary tract (LUT). During fetal development, sacral NC cells colonize the urogenital sinus to form pelvic ganglia (PG) flanking the bladder neck. The coordinated activity of PG neurons is required for normal urination; however, little is known about the development of PG neuronal diversity. To discover candidate genes involved in PG neurogenesis, the transcriptome profiling of sacral NC and developing PG was performed, and we identified the enrichment of the type 3 serotonin receptor (5-HT3, encoded by Htr3a and Htr3b). We determined that Htr3a is one of the first serotonin receptor genes that is up-regulated in sacral NC progenitors and is maintained in differentiating PG neurons. In vitro cultures showed that the disruption of 5-HT3 signaling alters the differentiation outcomes of sacral NC cells, while the stimulation of 5-HT3 in explanted fetal pelvic ganglia severely diminished neurite arbor outgrowth. Overall, this study provides a valuable resource for the analysis of signaling pathways in PG development, identifies 5-HT3 as a novel regulator of NC lineage diversification and neuronal maturation in the peripheral nervous system, and indicates that the perturbation of 5-HT3 signaling in gestation has the potential to alter bladder function later in life. Full article

(This article belongs to the Special Issue Neural Crest Development in Health and Disease)

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Review

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11 pages, 1310 KiB

Open AccessReview

How to Heal the Gut’s Brain: Regeneration of the Enteric Nervous System

by Helen Rueckert and Julia Ganz

Int. J. Mol. Sci. 2022, 23(9), 4799; https://0-doi-org.brum.beds.ac.uk/10.3390/ijms23094799 - 27 Apr 2022

Cited by 5 | Viewed by 2847

Abstract

The neural-crest-derived enteric nervous system (ENS) is the intrinsic nervous system of the gastrointestinal (GI) tract and controls all gut functions, including motility. Lack of ENS neurons causes various ENS disorders such as Hirschsprung Disease. One treatment option for ENS disorders includes the activation of resident stem cells to regenerate ENS neurons. Regeneration in the ENS has mainly been studied in mammalian species using surgical or chemically induced injury methods. These mammalian studies showed a variety of regenerative responses with generally limited regeneration of ENS neurons but (partial) regrowth and functional recovery of nerve fibers. Several aspects might contribute to the variety in regenerative responses, including observation time after injury, species, and gut region targeted. Zebrafish have recently emerged as a promising model system to study ENS regeneration as larvae possess the ability to generate new neurons after ablation. As the next steps in ENS regeneration research, we need a detailed understanding of how regeneration is regulated on a cellular and molecular level in animal models with both high and low regenerative capacity. Understanding the regulatory programs necessary for robust ENS regeneration will pave the way for using neural regeneration as a therapeutic approach to treating ENS disorders. Full article

(This article belongs to the Special Issue Neural Crest Development in Health and Disease)

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17 pages, 2298 KiB

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Roles of Enteric Neural Stem Cell Niche and Enteric Nervous System Development in Hirschsprung Disease

by Yue Ji, Paul Kwong-Hang Tam and Clara Sze-Man Tang

Int. J. Mol. Sci. 2021, 22(18), 9659; https://0-doi-org.brum.beds.ac.uk/10.3390/ijms22189659 - 07 Sep 2021

Cited by 18 | Viewed by 3657

Abstract

The development of the enteric nervous system (ENS) is highly modulated by the synchronized interaction between the enteric neural crest cells (ENCCs) and the neural stem cell niche comprising the gut microenvironment. Genetic defects dysregulating the cellular behaviour(s) of the ENCCs result in incomplete innervation and hence ENS dysfunction. Hirschsprung disease (HSCR) is a rare complex neurocristopathy in which the enteric neural crest-derived cells fail to colonize the distal colon. In addition to ENS defects, increasing evidence suggests that HSCR patients may have intrinsic defects in the niche impairing the extracellular matrix (ECM)-cell interaction and/or dysregulating the cellular niche factors necessary for controlling stem cell behaviour. The niche defects in patients may compromise the regenerative capacity of the stem cell-based therapy and advocate for drug- and niche-based therapies as complementary therapeutic strategies to alleviate/enhance niche-cell interaction. Here, we provide a summary of the current understandings of the role of the enteric neural stem cell niche in modulating the development of the ENS and in the pathogenesis of HSCR. Deciphering the contribution of the niche to HSCR may provide important implications to the development of regenerative medicine for HSCR. Full article

(This article belongs to the Special Issue Neural Crest Development in Health and Disease)

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13 pages, 1171 KiB

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Endocytic Protein Defects in the Neural Crest Cell Lineage and Its Pathway Are Associated with Congenital Heart Defects

by Angelo B. Arrigo and Jiuann-Huey Ivy Lin

Int. J. Mol. Sci. 2021, 22(16), 8816; https://0-doi-org.brum.beds.ac.uk/10.3390/ijms22168816 - 16 Aug 2021

Cited by 4 | Viewed by 2479

Abstract

Endocytic trafficking is an under-appreciated pathway in cardiac development. Several genes related to endocytic trafficking have been uncovered in a mutagenic ENU screen, in which mutations led to congenital heart defects (CHDs). In this article, we review the relationship between these genes (including LRP1 and LRP2) and cardiac neural crest cells (CNCCs) during cardiac development. Mice with an ENU-induced Lrp1 mutation exhibit a spectrum of CHDs. Conditional deletion using a floxed Lrp1 allele with different Cre drivers showed that targeting neural crest cells with Wnt1-Cre expression replicated the full cardiac phenotypes of the ENU-induced Lrp1 mutation. In addition, LRP1 function in CNCCs is required for normal OFT lengthening and survival/expansion of the cushion mesenchyme, with other cell lineages along the NCC migratory path playing an additional role. Mice with an ENU-induced and targeted Lrp2 mutation demonstrated the cardiac phenotype of common arterial trunk (CAT). Although there is no impact on CNCCs in Lrp2 mutants, the loss of LRP2 results in the depletion of sonic hedgehog (SHH)-dependent cells in the second heart field. SHH is known to be crucial for CNCC survival and proliferation, which suggests LRP2 has a non-autonomous role in CNCCs. In this article, other endocytic trafficking proteins that are associated with CHDs that may play roles in the NCC pathway during development, such as AP1B1, AP2B1, FUZ, MYH10, and HECTD1, are reviewed. Full article

(This article belongs to the Special Issue Neural Crest Development in Health and Disease)

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16 pages, 1024 KiB

Open AccessReview

Toward Understanding the Mechanisms of Malignant Peripheral Nerve Sheath Tumor Development

by Teddy Mohamad, Camille Plante and Jean-Philippe Brosseau

Int. J. Mol. Sci. 2021, 22(16), 8620; https://0-doi-org.brum.beds.ac.uk/10.3390/ijms22168620 - 10 Aug 2021

Cited by 13 | Viewed by 3750

Abstract

Malignant peripheral nerve sheath tumors (MPNSTs) originate from the neural crest lineage and are associated with the neurofibromatosis type I syndrome. MPNST is an unmet clinical need. In this review article, we summarize the knowledge and discuss research perspectives related to (1) the natural history of MPNST development; (2) the mouse models recapitulating the progression from precursor lesions to MPNST; (3) the role of the tumor microenvironment in MPNST development, and (4) the signaling pathways linked to MPNST development. Full article

(This article belongs to the Special Issue Neural Crest Development in Health and Disease)

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34 pages, 2244 KiB

Open AccessReview

The Mandibular and Hyoid Arches—From Molecular Patterning to Shaping Bone and Cartilage

by Jaroslav Fabik, Viktorie Psutkova and Ondrej Machon

Int. J. Mol. Sci. 2021, 22(14), 7529; https://0-doi-org.brum.beds.ac.uk/10.3390/ijms22147529 - 14 Jul 2021

Cited by 9 | Viewed by 4586

Abstract

The mandibular and hyoid arches collectively make up the facial skeleton, also known as the viscerocranium. Although all three germ layers come together to assemble the pharyngeal arches, the majority of tissue within viscerocranial skeletal components differentiates from the neural crest. Since nearly one third of all birth defects in humans affect the craniofacial region, it is important to understand how signalling pathways and transcription factors govern the embryogenesis and skeletogenesis of the viscerocranium. This review focuses on mouse and zebrafish models of craniofacial development. We highlight gene regulatory networks directing the patterning and osteochondrogenesis of the mandibular and hyoid arches that are actually conserved among all gnathostomes. The first part of this review describes the anatomy and development of mandibular and hyoid arches in both species. The second part analyses cell signalling and transcription factors that ensure the specificity of individual structures along the anatomical axes. The third part discusses the genes and molecules that control the formation of bone and cartilage within mandibular and hyoid arches and how dysregulation of molecular signalling influences the development of skeletal components of the viscerocranium. In conclusion, we notice that mandibular malformations in humans and mice often co-occur with hyoid malformations and pinpoint the similar molecular machinery controlling the development of mandibular and hyoid arches. Full article

(This article belongs to the Special Issue Neural Crest Development in Health and Disease)

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19 pages, 1719 KiB

Open AccessReview

From Neural Crest to Definitive Roof Plate: The Dynamic Behavior of the Dorsal Neural Tube

by Dina Rekler and Chaya Kalcheim

Int. J. Mol. Sci. 2021, 22(8), 3911; https://0-doi-org.brum.beds.ac.uk/10.3390/ijms22083911 - 10 Apr 2021

Cited by 9 | Viewed by 4007

Abstract

Research on the development of the dorsal neural tube is particularly challenging. In this highly dynamic domain, a temporal transition occurs between early neural crest progenitors that undergo an epithelial-to-mesenchymal transition and exit the neural primordium, and the subsequent roof plate, a resident epithelial group of cells that constitutes the dorsal midline of the central nervous system. Among other functions, the roof plate behaves as an organizing center for the generation of dorsal interneurons. Despite extensive knowledge of the formation, emigration and migration of neural crest progenitors, little is known about the mechanisms leading to the end of neural crest production and the transition into a roof plate stage. Are these two mutually dependent or autonomously regulated processes? Is the generation of roof plate and dorsal interneurons induced by neural tube-derived factors throughout both crest and roof plate stages, respectively, or are there differences in signaling properties and responsiveness as a function of time? In this review, we discuss distinctive characteristics of each population and possible mechanisms leading to the shift between the above cell types. Full article

(This article belongs to the Special Issue Neural Crest Development in Health and Disease)

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