ArticleNeurogastroenterology and motility2016
Isogenic enteric neural progenitor cells can replace missing neurons and glia in mice with Hirschsprung disease.
Article in Neurogastroenterology and motility, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 39 papers.
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Who cites it
39 citing papers in PubMed, 66 citations in OpenAlex.
- Dual roles of GDNF in enteric glial cell plasticity: direct transdifferentiation via the CaMKII/NeuroD1 pathway and cooperative regulation in a neural stem cell-inducing medium.Stem cell research & therapy · 2026Article
- Embryonic Gut Enteric Neural Crest Cells Derived Neurospheres.Methods in molecular biology (Clifton, N.J.) · 2026Article
- Current and future state of the management of Hirschsprung disease.World journal of pediatric surgery · 2025Review
- Cecal growth factors promote enteric neurosphere formation and hindgut colonization in the avian model.Frontiers in cell and developmental biology · 2025Article
- Cellular trafficking and fate mapping of cells within the nervous system after in utero hematopoietic cell transplantation.Communications biology · 2024Article
- Highly neurogenic glia from human and mouse myenteric ganglia generate functional neurons following culture and transplantation into the gut.Cell reports · 2024Article
- Mature enteric neurons have the capacity to reinnervate the intestine with glial cells as their guide.Neuron · 2024Article
- Cutting-edge regenerative therapy for Hirschsprung disease and its allied disorders.Surgery today · 2024Review
- A novel method for culturing enteric neurons generates neurospheres containing functional myenteric neuronal subtypes.Journal of neuroscience methods · 2024Article
- Agrin Inhibition in Enteric Neural Stem Cells Enhances Their Migration Following Colonic Transplantation.Stem cells translational medicine · 2024Article
- Autologous cell transplantation for treatment of colorectal aganglionosis in mice.Nature communications · 2024Article
- Updates and Challenges in ENS Cell Therapy for the Treatment of Neurointestinal Diseases.Biomolecules · 2024Review
- Intra-arterial delivery of neurospheres into isolated perfused porcine colons: a proof of concept.Biology methods & protocols · 2024Article
- Activation of mechanoreceptor Piezo1 inhibits enteric neuronal growth and migrationFrontiers in molecular neuroscience · 2024Article
- Oxidative Stress and Neural Dysfunction in Gastrointestinal Diseases: Can Stem Cells Offer a Solution?Stem cells translational medicine · 2023Article
- Endothelin signaling in development.Development (Cambridge, England) · 2023Review
- Enteric neurospheres retain the capacity to assemble neural networks with motile and metamorphic gliocytes and ganglia.Stem cell research & therapy · 2023Article
- Harnessing the Power of Enteric Glial Cells' Plasticity and Multipotency for Advancing Regenerative Medicine.International journal of molecular sciences · 2023Review
- Bone Marrow Stem Cells Derived from Nerves Have Neurogenic Properties and Potential Utility for Regenerative Therapy.International journal of molecular sciences · 2023Article
- Schwann Cells in the Aganglionic Colon of Hirschsprung Disease Can Generate Neurons for Regenerative Therapy.Stem cells translational medicine · 2022Article
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Authors and funding
7 authors at 3 institutions in 3 countries.
Funding
Abstract
backgroundTransplanting autologous patient-derived enteric neuronal stem/progenitor cells (ENSCs) is an innovative approach to replacing missing enteric neurons in patients with Hirschsprung disease (HSCR). Using autologous cells eliminates immunologic and ethical concerns raised by other cell sources. However, whether postnatal aganglionic bowel is permissive for transplanted ENSCs and whether ENSCs from HSCR patients can be successfully isolated, cultured, and transplanted in vivo remains unknown.
methodsENSCs isolated from the ganglionic intestine of Ednrb(-/-) mice (HSCR-ENSCs) were characterized immunohistochemically and evaluated for their capacity to proliferate and differentiate in vitro. Fluorescently labeled ENSCs were co-cultured ex vivo with aganglionic Ednrb(-/-) colon. For in vivo transplantation, HSCR-ENSCs were labeled with lentivirus expressing green fluorescent protein (GFP) and implanted into aganglionic embryonic chick gut in ovo and postnatal aganglionic Ednrb(-/-) rectum in vivo. KEY
resultsHSCR-ENSCs maintain normal capacity self-renewal and neuronal differentiation. Moreover, the Ednrb(-/-) aganglionic environment is permissive to engraftment by wild-type ENSCs ex vivo and supports migratrion and neuroglial differentiation of these cells following transplantation in vivo. Lentiviral GFP-labeled HSCR-ENSCs populated embryonic chick hindgut and postnatal colon of Ednrb(-/-) HSCR, with cells populating the intermuscular layer and forming enteric neurons and glia. CONCLUSIONS & INFERENCES: ENSCs can be isolated and cultured from mice with HSCR, and transplanted into the aganglionic bowel of HSCR littermates to generate enteric neuronal networks. These results in an isogenic model establish the potential of using autologous-derived stem cells to treat HSCR and other intestinal neuropathies.
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