ArticleNature communications2019
Precisely controlling endogenous protein dosage in hPSCs and derivatives to model FOXG1 syndrome.
Article in Nature communications, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 34 papers.
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Who cites it
34 citing papers in PubMed, 48 citations in OpenAlex.
- Brain organoids and genome editing: A new era in understanding human brain development and disorders.Neural regeneration research · 2026Article
- Non-coding structural variants disrupt FOXG1 transcriptional regulation in early neurodevelopment.Nature communications · 2026Article
- Single-dose genome editing therapy rescues auditory and vestibular functions in adult mice with DFNA41 deafness.The Journal of clinical investigation · 2025Article
- Recapitulating dengue virus infection with human pluripotent stem cell-derived liver organoids for antiviral screening.Nature communications · 2025Article
- Leveraging CRISPR activation for rapid assessment of gene editing products in human pluripotent stem cells.Stem cell reports · 2025Article
- The Interface of Gene Editing with Regenerative Medicine.Engineering (Beijing, China) · 2025Article
- A compact, versatile drug-induced splicing switch system with minimal background expression.Cell reports methods · 2024Article
- Foxg1 regulates translation of neocortical neuronal genes, including the main NMDA receptor subunit gene, Grin1.BMC biology · 2024Article
- Targeted genome editing restores auditory function in adult mice with progressive hearing loss caused by a human microRNA mutation.Science translational medicine · 2024Article
- Foxg1 bimodally tunes L1-mRNA and -DNA dynamics in the developing murine neocortex.Development (Cambridge, England) · 2024Article
- Degron tagging for rapid protein degradation in mice.Disease models & mechanisms · 2024Article
- Identification and characterization of human hematopoietic mesoderm.Science China. Life sciences · 2024Article
- Induction of local immunosuppression in allogeneic cell transplantation by cell-type-specific expression of PD-L1 and CTLA4Ig.Stem cell reports · 2023Article
- METTL3-Dependent N6-Methyladenosine Modification Programs Human Neural Progenitor Cell Proliferation.International journal of molecular sciences · 2023Article
- Treatment of monogenic and digenic dominant genetic hearing loss by CRISPR-Cas9 ribonucleoprotein delivery in vivo.Nature communications · 2023Article
- Cooperative regulation of Zhx1 and hnRNPA1 drives the cardiac progenitor-specific transcriptional activation during cardiomyocyte differentiation.Cell death discovery · 2023Article
- A Human-Specific De Novo Gene Promotes Cortical Expansion and Folding.Advanced science (Weinheim, Baden-Wurttemberg, Germany) · 2023Article
- Review
- A degron system targeting endogenous PD-1 inhibits the growth of tumor cells in mice.NAR cancer · 2022Article
- FOXG1 dose tunes cell proliferation dynamics in human forebrain progenitor cells.Stem cell reports · 2022Article
Corrections and comments
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Authors and funding
10 authors at 2 institutions in 2 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Dosage of key regulators impinge on developmental disorders such as FOXG1 syndrome. Since neither knock-out nor knock-down strategy assures flexible and precise protein abundance control, to study hypomorphic or haploinsufficiency expression remains challenging. We develop a system in human pluripotent stem cells (hPSCs) using CRISPR/Cas9 and SMASh technology, with which we can target endogenous proteins for precise dosage control in hPSCs and at multiple stages of neural differentiation. We also reveal FOXG1 dose-dependently affect the cellular constitution of human brain, with 60% mildly affect GABAergic interneuron development while 30% thresholds the production of MGE derived neurons. Abnormal interneuron differentiation accounts for various neurological defects such as epilepsy or seizures, which stimulates future innovative cures of FOXG1 syndrome. By means of its robustness and easiness, dosage-control of proteins in hPSCs and their derivatives will update the understanding and treatment of additional diseases caused by abnormal protein dosage.
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