ArticleNature communications2022
CHD7 regulates otic lineage specification and hair cell differentiation in human inner ear organoids.
Article in Nature communications, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 30 papers.
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Who cites it
30 citing papers in PubMed, 43 citations in OpenAlex.
- Functionalized Biomimetic Scaffolds for Human-Derived Auditory Neural Circuit Construction.Advanced science (Weinheim, Baden-Wurttemberg, Germany) · 2026Article
- Role of CHD chromatin remodelers in heart development.World journal of pediatrics : WJP · 2026Review
- Human induced pluripotent stem cell-derived inner ear organoids reveal hair cell damage and plasticity after cisplatin and gentamicin exposure.Disease models & mechanisms · 2026Article
- Recent Progress in Mechanism-Based Therapies forInternational journal of molecular sciences · 2026Review
- Generation and characterization of vestibular inner ear organoids from human pluripotent stem cells.Nature protocols · 2026Review
- Bioactive hydrogels for the reconstruction of sensorineural hearing loss.Bioactive materials · 2026Review
- Inner Ear Organoids: A Hydrogel-Based Platform for Drug Screening and Deafness Modeling.Neuroscience bulletin · 2026Review
- Advances and prospects of 3D air-liquid interface cultured middle ear epithelial cells and inner ear organoids in otologic diseases.Frontiers in bioengineering and biotechnology · 2026Review
- Inner ear organoids: Recent progress and potential applications.Fundamental research · 2025Review
- Chemically defined and dynamic click hydrogels support hair cell differentiation in human inner ear organoids.Stem cell reports · 2025Article
- Review
- Timing and Graded BMP Signalling Determines Fate of Neural Crest and Ectodermal Placode Derivatives from Pluripotent Stem Cells.Biomedicines · 2024Review
- Metabolic Profiling of Cochlear Organoids Identifies α-Ketoglutarate and NADAdvanced science (Weinheim, Baden-Wurttemberg, Germany) · 2024Article
- Advance and Application of Single-cell Transcriptomics in Auditory Research.Neuroscience bulletin · 2024Review
- Article
- Mapping the developmental potential of mouse inner ear organoids at single-cell resolution.iScience · 2024Article
- A novel CHD7 variant in a chinese family with CHARGE syndrome.Genes & genomics · 2024Article
- Inner Ear Organoids: Strengths and Limitations.Journal of the Association for Research in Otolaryngology : JARO · 2024Article
- In vitro modeling of cranial placode differentiation: Recent advances, challenges, and perspectives.Developmental biology · 2024Review
- Genetic correction of induced pluripotent stem cells from a DFNA36 patient results in morphologic and functional recovery of derived hair cell-like cells.Stem cell research & therapy · 2024Article
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Authors and funding
4 authors at 1 institution in 1 country.
Funding
Abstract
Mutations in CHD7 cause CHARGE syndrome, affecting multiple organs including the inner ear in humans. We investigate how CHD7 mutations affect inner ear development using human pluripotent stem cell-derived organoids as a model system. We find that loss of CHD7 or its chromatin remodeling activity leads to complete absence of hair cells and supporting cells, which can be explained by dysregulation of key otic development-associated genes in mutant otic progenitors. Further analysis of the mutant otic progenitors suggests that CHD7 can regulate otic genes through a chromatin remodeling-independent mechanism. Results from transcriptome profiling of hair cells reveal disruption of deafness gene expression as a potential underlying mechanism of CHARGE-associated sensorineural hearing loss. Notably, co-differentiating CHD7 knockout and wild-type cells in chimeric organoids partially rescues mutant phenotypes by restoring otherwise severely dysregulated otic genes. Taken together, our results suggest that CHD7 plays a critical role in regulating human otic lineage specification and hair cell differentiation.
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Registered trials
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