TY - JOUR
AU - Nouri, Parivash
AU - Götz, Sebastian
AU - Rauser, Benedict
AU - Irmler, Martin
AU - Peng, Changgeng
AU - Trümbach, Dietrich
AU - Kempny, Christian
AU - Lechermeier, Carina G
AU - Bryniok, Agnes
AU - Dlugos, Andrea
AU - Euchner, Ellen
AU - Beckers, Johannes
AU - Brodski, Claude
AU - Klümper, Claudia
AU - Wurst, Wolfgang
AU - Prakash, Nilima
TI - Dose-Dependent and Subset-Specific Regulation of Midbrain Dopaminergic Neuron Differentiation by LEF1-Mediated WNT1/b-Catenin Signaling.
JO - Frontiers in cell and developmental biology
VL - 8
SN - 2296-634X
CY - Lausanne
PB - Frontiers Media
M1 - DZNE-2021-00120
SP - 587778
PY - 2020
N1 - ISSN 2296-634X not unique: **3 hits**.
AB - The mesodiencephalic dopaminergic (mdDA) neurons, including the nigrostriatal subset that preferentially degenerates in Parkinson's Disease (PD), strongly depend on an accurately balanced Wingless-type MMTV integration site family member 1 (WNT1)/beta-catenin signaling pathway during their development. Loss of this pathway abolishes the generation of these neurons, whereas excessive WNT1/b-catenin signaling prevents their correct differentiation. The identity of the cells responding to this pathway in the developing mammalian ventral midbrain (VM) as well as the precise progression of WNT/b-catenin action in these cells are still unknown. We show that strong WNT/b-catenin signaling inhibits the differentiation of WNT/b-catenin-responding mdDA progenitors into PITX3+ and TH+ mdDA neurons by repressing the Pitx3 gene in mice. This effect is mediated by RSPO2, a WNT/b-catenin agonist, and lymphoid enhancer binding factor 1 (LEF1), an essential nuclear effector of the WNT/b-catenin pathway, via conserved LEF1/T-cell factor binding sites in the Pitx3 promoter. LEF1 expression is restricted to a caudolateral mdDA progenitor subset that preferentially responds to WNT/b-catenin signaling and gives rise to a fraction of all mdDA neurons. Our data indicate that an attenuation of WNT/b-catenin signaling in mdDA progenitors is essential for their correct differentiation into specific mdDA neuron subsets. This is an important consideration for stem cell-based regenerative therapies and in vitro models of neuropsychiatric diseases.
KW - Parkinson’s disease (Other)
KW - dopamine (Other)
KW - mouse (Other)
KW - nerve cell (Other)
KW - regenerative therapy (Other)
LB - PUB:(DE-HGF)16
C6 - pmid:33195246
C2 - pmc:PMC7649324
DO - DOI:10.3389/fcell.2020.587778
UR - https://pub.dzne.de/record/154266
ER -