Development of the Enteric Nervous System
The ENS is a unique part of the nervous system
Quail-chick interspecies chimeras reveal the migration pathways of crest-derived cells
DiI-labeled sacral crest cells colonize the post-umbilical bowel
Slide 5
Microenvironmental signals determine the fates of crest-derived cells
Slide 7
Congenital aganglionosis causes pseudoobstruction
Crest-derived cells require Edn3 (ET-3) and Ednrb (ETB) to complete their colonization of the gut
Megacolon occurs in mice that lack edn3 (ET-3)
The terminal colon of ET-3-deficient mice is aganglionic
Co-cultured sources of crest fail to colonize presumptive end3ls gut
Presumptive aganglionic gut from edn3ls mice cannot be entered by quail crest cells
The terminal colon is normally colonized in end3ls <> WT chimeric mice
Edn3 inhibits the development of neurons from crest-derived precursors
Crest-derived cells are present in the proximal bowel of edn3-deficient mice but do not enter the terminal gut
Exogenous Edn3 enables crest-derived cells to enter the terminal colon of Edn3-deficient mice
Exogenous ET-3 allows crest-derived cells to colonize the entire colon in vitro
Ectopic ganglia develop in the pelvis of endls mice
Ret and Ednrb interact in humans and in mice (mice tested to verify human data)
Enteric neurons are Ret-dependent
The GDNF family of growth factors activate Ret
Specific transcription and growth factors define stages in ENS development
The earlier a gene acts in development, the more massive the defect that follows its deletion
Genes associated with Hirschsprung’s disease
Crest-derived cells are isolated by immunoselection.
Neurons develop in cultures of isolated crest-derived cells.
GDNF is mitogenic and promotes neurogenesis at E12
Promotion of neurogenesis by GDNF decreases at E14; neurogenic response to NT-3 is acquired
Slide 30
Crest-derived cells colonize the bowel and then migrate from the gut to the pancreas.
Crest-derived cells migrate in the outer gut mesenchyme; the submucosal plexus forms secondarily.
mRNA encoding netrin-1 is found in E13 mouse gut and pancreas.
Transcripts encoding netrin receptors are expressed in the developing gut and pancreas
Crest-derived cells migrate from explants of bowel toward transfected cells expressing netrin-1.
Crest-derived cells migrate toward cells that secrete netrin-1.
Crest-derived cells isolated from chick gut migrate toward co-cultured netrin-1-expressing cells.
Slide 38
Antibodies to DCC block the inward migration of crest-derived cells in chick gut explants.
Antibodies to DCC inhibit the migration of crest-derived cells from gut toward pancreas.
The pancreas of DCC -/- mice is aganglionic
Vagal axons entering the stomach  at E13 are DCC-immunoreactive
Summary & Conclusions