2021
DOI: 10.1002/dneu.22824
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Engrailed‐2 is a cell autonomous regulator of neurogenesis in cultured hippocampal neural stem cells

Abstract: Abnormalities in genes that regulate early brain development are known risk factors for neurodevelopmental disorders. Engrailed-2 (En2) is a homeodomain transcription factor with established roles in cerebellar patterning. En2 is highly expressed in the developing mid-hindbrain region, and En2 knockout (KO) mice exhibit major deficits in mid-hindbrain structures. However, En2 is also expressed in forebrain regions including the hippocampus, but its function is unknown. Previous studies have shown that the hipp… Show more

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Cited by 4 publications
(2 citation statements)
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“…Notably, we observed down-regulation of a transcription factors, such as En2 and Sox5, in Kmt2a +/LSL mNPCs compared to the Kmt2a +/+ mNPCs. Disruption of these transcription factors has previously been linked to defects in hippocampal neurogenesis [26], [27] as well as being associated with intellectual disability and autism-spectrum-disorder (ASD) [28][29]. Since up to 50% of WDSTS patients have ASD [30], we compared the rescued genes to known ASD-risk genes [31] and discovered a significant overlap of 26 genes (p-value < 0.0001, Fisher's exact test, Figure 3F).…”
Section: Resultsmentioning
confidence: 99%
“…Notably, we observed down-regulation of a transcription factors, such as En2 and Sox5, in Kmt2a +/LSL mNPCs compared to the Kmt2a +/+ mNPCs. Disruption of these transcription factors has previously been linked to defects in hippocampal neurogenesis [26], [27] as well as being associated with intellectual disability and autism-spectrum-disorder (ASD) [28][29]. Since up to 50% of WDSTS patients have ASD [30], we compared the rescued genes to known ASD-risk genes [31] and discovered a significant overlap of 26 genes (p-value < 0.0001, Fisher's exact test, Figure 3F).…”
Section: Resultsmentioning
confidence: 99%
“…The En2 -KO mice displayed reductions in hippocampal volume and cell numbers due to aberrant neurogenesis characterized by excess proliferation in the early Sox2 + /Tbr2 + progenitors, increased apoptosis in differentiating neuroblasts, and reduced newborn neuron survival [ 108 ]. Notably, En2 functions in hippocampal NPCs by inhibiting proliferation and promoting survival and differentiation in a cell-autonomous manner [ 109 ].…”
Section: Utilizing Mouse Models To Understand the Impact Of Asd On Hi...mentioning
confidence: 99%