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Conference Paper: Sox10 regulates enteric neural crest cell migration in the developing gut

TitleSox10 regulates enteric neural crest cell migration in the developing gut
Authors
Issue Date2013
PublisherThe International Society of Developmental Biologists (ISDB).
Citation
The 17th International Congress of Developmental Biology of the International Society of Developmental Biologists (ISDB), Cancun, Mexico, 16-20 June 2013. In 17th ISDB 2013 Abstracts, 2013, p. 5, abstract no. 14 How to Cite?
AbstractSox10 is a HMG-domain containing transcription factor which plays important roles in neural crest cell survival and differentiation. Mutations of Sox10 have been identified in patients with Waardenburg-Hirschsprung syndrome, who suffer from deafness, pigmentation defects and intestinal aganglionosis. Enteric neural crest cells (ENCCs) with Sox10 mutation undergo premature differentiation and fail to colonize the distal hindgut. It is unclear, however, whether Sox10 plays a role in the migration of ENCCs. To visualize the migration behaviour of mutant ENCCs, we generated a Sox10NGFP mouse model where EGFP is fused to the N-terminal domain of Sox10. Using time-lapse imaging, we found that ENCCs in Sox10NGFP/+ mutants displays lower migration speed and altered trajectories compared to normal controls. This behaviour was cell-autonomous, as shown by organotypic grafting of Sox10NGFP/+ gut segments onto control guts and vice versa. ENCCs encounter different extracellular matrix (ECM) molecules along the developing gut. We performed gut explant culture on various ECM and found that Sox10NGFP/+ ENCCs tend to form aggregates, particularly on fibronectin. Time-lapse imaging of single cells in gut explant culture indicated that the tightly-packed Sox10 mutant cells failed to exhibit contact inhibition of locomotion. We determined the expression of adhesion molecule families by qPCR analysis, and found integrin expression unaffected while L1-cam and selected cadherins were altered, suggesting that Sox10 mutation affects cell adhesion properties of ENCCs. Our findings identify a de novo role of Sox10 in regulating the migration behaviour of ENCCs, which has important implications for the treatment of Hirschsprung disease.
Description17th ISDB 2013 cum 72nd Annual Meeting of the Society for Developmental Biology, VII Latin American Society of Developmental Biology Meeting and XI Congreso de la Sociedad Mexicana de Biologia del Desarrollo. The Conference's web site is located at http://www.inb.unam.mx/isdb/
Concurrent Sessions 1: 1.3 - Organs to organisms: Models of Human Diseases: abstract no. 14
Persistent Identifierhttp://hdl.handle.net/10722/184926

 

DC FieldValueLanguage
dc.contributor.authorSham, MHen_US
dc.contributor.authorLeung, Cen_US
dc.contributor.authorZhang, Men_US
dc.contributor.authorSit, HMen_US
dc.date.accessioned2013-07-15T10:18:26Z-
dc.date.available2013-07-15T10:18:26Z-
dc.date.issued2013en_US
dc.identifier.citationThe 17th International Congress of Developmental Biology of the International Society of Developmental Biologists (ISDB), Cancun, Mexico, 16-20 June 2013. In 17th ISDB 2013 Abstracts, 2013, p. 5, abstract no. 14en_US
dc.identifier.urihttp://hdl.handle.net/10722/184926-
dc.description17th ISDB 2013 cum 72nd Annual Meeting of the Society for Developmental Biology, VII Latin American Society of Developmental Biology Meeting and XI Congreso de la Sociedad Mexicana de Biologia del Desarrollo. The Conference's web site is located at http://www.inb.unam.mx/isdb/-
dc.descriptionConcurrent Sessions 1: 1.3 - Organs to organisms: Models of Human Diseases: abstract no. 14-
dc.description.abstractSox10 is a HMG-domain containing transcription factor which plays important roles in neural crest cell survival and differentiation. Mutations of Sox10 have been identified in patients with Waardenburg-Hirschsprung syndrome, who suffer from deafness, pigmentation defects and intestinal aganglionosis. Enteric neural crest cells (ENCCs) with Sox10 mutation undergo premature differentiation and fail to colonize the distal hindgut. It is unclear, however, whether Sox10 plays a role in the migration of ENCCs. To visualize the migration behaviour of mutant ENCCs, we generated a Sox10NGFP mouse model where EGFP is fused to the N-terminal domain of Sox10. Using time-lapse imaging, we found that ENCCs in Sox10NGFP/+ mutants displays lower migration speed and altered trajectories compared to normal controls. This behaviour was cell-autonomous, as shown by organotypic grafting of Sox10NGFP/+ gut segments onto control guts and vice versa. ENCCs encounter different extracellular matrix (ECM) molecules along the developing gut. We performed gut explant culture on various ECM and found that Sox10NGFP/+ ENCCs tend to form aggregates, particularly on fibronectin. Time-lapse imaging of single cells in gut explant culture indicated that the tightly-packed Sox10 mutant cells failed to exhibit contact inhibition of locomotion. We determined the expression of adhesion molecule families by qPCR analysis, and found integrin expression unaffected while L1-cam and selected cadherins were altered, suggesting that Sox10 mutation affects cell adhesion properties of ENCCs. Our findings identify a de novo role of Sox10 in regulating the migration behaviour of ENCCs, which has important implications for the treatment of Hirschsprung disease.-
dc.languageengen_US
dc.publisherThe International Society of Developmental Biologists (ISDB).-
dc.relation.ispartofISDB 17th International Congress of Developmental Biology 2013 Abstractsen_US
dc.rightsCreative Commons: Attribution 3.0 Hong Kong License-
dc.titleSox10 regulates enteric neural crest cell migration in the developing guten_US
dc.typeConference_Paperen_US
dc.identifier.emailSham, MH: mhsham@hku.hken_US
dc.identifier.emailLeung, C: leungcar@hkucc.hku.hken_US
dc.identifier.emailZhang, M: zhangmei@hkusua.hku.hken_US
dc.identifier.emailSit, HM: briansit@hku.hk-
dc.identifier.authoritySham, MH=rp00380en_US
dc.description.naturepostprint-
dc.identifier.hkuros216497en_US
dc.identifier.spage5-
dc.identifier.epage5-
dc.publisher.placeMexico-

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