ArticleHuman molecular genetics2010
Genetic background impacts developmental potential of enteric neural crest-derived progenitors in the Sox10Dom model of Hirschsprung disease.
Article in Human molecular genetics, 2010. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 26 papers.
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Who cites it
26 citing papers in PubMed, 47 citations in OpenAlex.
- SOX10 ablation severely impairs the generation of postmigratory neural crest from human pluripotent stem cells.Cell death & disease · 2021Article
- 5-HT3 Signaling Alters Development of Sacral Neural Crest Derivatives That Innervate the Lower Urinary Tract.International journal of molecular sciences · 2021Article
- Genetic background-dependent abnormalities of the enteric nervous system and intestinal function in Kif26a-deficient mice.Scientific reports · 2021Article
- Dlx1/2 mice have abnormal enteric nervous system function.JCI insight · 2020Article
- Down syndrome mouse models have an abnormal enteric nervous system.JCI insight · 2019Article
- DRG-Derived Neural Progenitors Differentiate into Functional Enteric Neurons Following Transplantation in the Postnatal Colon.Cell transplantation · 2019Article
- Acquired isolated hypoganglionosis as a distinct entity: results from a nationwide survey.Pediatric surgery international · 2019Article
- Mouse models of Hirschsprung disease and other developmental disorders of the enteric nervous system: Old and new players.Developmental biology · 2016Review
- White paper on guidelines concerning enteric nervous system stem cell therapy for enteric neuropathies.Developmental biology · 2016Review
- Hirschsprung disease is associated with an L286P mutation in the fifth transmembrane domain of the endothelin-B receptor in the N-ethyl-N-nitrosourea-induced mutant line.Experimental animals · 2016Article
- Enteric neuron imbalance and proximal dysmotility in ganglionated intestine of theCellular and molecular gastroenterology and hepatology · 2015Article
- Article
- Up-regulated FHL1 expression maybe involved in the prognosis of Hirschsprung's disease.International journal of medical sciences · 2014Article
- Balancing on the crest - Evidence for disruption of the enteric ganglia via inappropriate lineage segregation and consequences for gastrointestinal function.Developmental biology · 2013Review
- Simple rules for a "simple" nervous system? Molecular and biomathematical approaches to enteric nervous system formation and malformation.Developmental biology · 2013Review
- Enteric nervous system development: migration, differentiation, and disease.American journal of physiology. Gastrointestinal and liver physiology · 2013Review
- Article
- Development and developmental disorders of the enteric nervous system.Nature reviews. Gastroenterology & hepatology · 2013Review
- An optimized procedure for fluorescence-activated cell sorting (FACS) isolation of autonomic neural progenitors from visceral organs of fetal mice.Journal of visualized experiments : JoVE · 2012Article
- Immunophenotypic characterization of enteric neural crest cells in the developing avian colorectum.Developmental dynamics : an official publication of the American Association of Anatomists · 2012Article
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5 authors at 2 institutions in 1 country.
Funding
Abstract
Abnormalities in the development of enteric neural crest-derived progenitors (ENPs) that generate the enteric nervous system (ENS) can lead to aganglionosis in a variable portion of the distal gastrointestinal tract. Cumulative evidence suggests that variation of aganglionosis is due to gene interactions that modulate the ability of ENPs to populate the intestine; however, the developmental processes underlying this effect are unknown. We hypothesized that differences in enteric ganglion deficits could be attributable to the effects of genetic background on early developmental processes, including migration, proliferation, or lineage divergence. Developmental processes were investigated in congenic Sox10(Dom) mice, an established Hirschsprung disease (HSCR) model, on distinct inbred backgrounds, C57BL/6J (B6) and C3HeB/FeJ (C3Fe). Immuno-staining on whole-mount fetal gut tissue and dissociated cell suspensions was used to assess migration and proliferation. Flow cytometry utilizing the cell surface markers p75 and HNK-1 was used to isolate live ENPs for analysis of developmental potential. Frequency of ENPs was reduced in Sox10(Dom) embryos relative to wild-type embryos, but was unaffected by genetic background. Both migration and developmental potential of ENPs in Sox10(Dom) embryos were altered by inbred strain background with the most highly significant differences seen for developmental potential between strains and genotypes. In vivo imaging of fetal ENPs and postnatal ganglia demonstrates that altered lineage divergence impacts ganglia in the proximal intestine. Our analysis demonstrates that genetic background alters early ENS development and suggests that abnormalities in lineage diversification can shift the proportions of ENP populations and thus may contribute to ENS deficiencies in vivo.
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