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10 sources retrieved Β· Most recent: April 2026 Β· Index updated 15 days ago
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ATOH7
atonal bHLH transcription factor 7
Chromosome 10 Β· 10q21.3|10q21.3-q22.1
NCBI Gene: 220202Ensembl: ENSG00000179774.9HGNC: HGNC:13907UniProt: F1T0H4
36PubMed Papers
21Diseases
0Drugs
5Pathogenic Variants
FUNCTIONAL ROLE
Transcription Factor
CLINICAL
OMIM Disease Gene
DATA QUALITY
βœ“ Experimental GO Evidenceβœ“ Swiss-Prot Reviewed
nucleusoptic nerve developmentprotein bindingnucleoplasmpersistent hyperplastic primary vitreous, autosomal recessiveopen-angle glaucomaglaucomapersistent hyperplastic primary vitreous
✦AI Summary

ATOH7 is a basic helix-loop-helix transcription factor that plays a crucial role in retinal ganglion cell (RGC) development and specification 1. The protein functions as a proneural transcription factor expressed in early retinal progenitors, where it is essential for RGC fate determination and survival 1. ATOH7 regulates transcription by binding to specific DNA sequences and controlling expression of RGC-specific genes while inhibiting alternative cell fates 1. The gene's expression is controlled by dual enhancer mechanisms that ensure robust transcriptional output necessary for proper optic nerve development 2. Loss-of-function mutations in ATOH7 lead to severe developmental abnormalities including optic nerve hypoplasia and persistent hyperplastic primary vitreous 31. Genome-wide association studies have identified ATOH7 variants as major determinants of human optic disc size, highlighting its quantitative role in normal retinal development 3. The gene has also been implicated as a susceptibility factor for primary open-angle glaucoma 4. ATOH7's function extends beyond RGC specification, with recent evidence suggesting roles in photoreceptor development during late retinogenesis 5. The high evolutionary conservation of ATOH7 across species underscores its fundamental importance in vertebrate visual system development.

Sources cited
1
ATOH7 is a proneural transcription factor essential for RGC development, regulates RGC-specific genes, and mutations cause optic nerve hypoplasia
PMID: 38994994
2
ATOH7 expression is controlled by dual enhancer mechanisms ensuring robust transcriptional output for optic nerve development
PMID: 32817515
3
ATOH7 variants are major determinants of human optic disc size and mutations found in optic nerve hypoplasia patients
PMID: 20395239
4
ATOH7 is a susceptibility gene for primary open-angle glaucoma
PMID: 26497787
5
ATOH7 has roles in photoreceptor specification during late retinogenesis
PMID: 32386599
Disease Associationsβ“˜21
persistent hyperplastic primary vitreous, autosomal recessiveOpen Targets
0.76Strong
open-angle glaucomaOpen Targets
0.50Moderate
glaucomaOpen Targets
0.48Moderate
persistent hyperplastic primary vitreousOpen Targets
0.46Moderate
neurodegenerative diseaseOpen Targets
0.45Moderate
Autosomal dominant optic atrophy, classic typeOpen Targets
0.43Moderate
foveal hypoplasiaOpen Targets
0.42Moderate
Hypoplasia of the foveaOpen Targets
0.42Moderate
smoking initiationOpen Targets
0.41Moderate
Congenital cataract microcornea with corneal opacityOpen Targets
0.37Weak
gastrointestinal diseaseOpen Targets
0.16Weak
X-linked retinal dysplasiaOpen Targets
0.10Weak
retinitis pigmentosaOpen Targets
0.09Suggestive
choroidal dystrophy, central areolar, 1Open Targets
0.09Suggestive
Leber hereditary optic neuropathyOpen Targets
0.09Suggestive
severe early-childhood-onset retinal dystrophyOpen Targets
0.09Suggestive
Stargardt diseaseOpen Targets
0.09Suggestive
age-related macular degenerationOpen Targets
0.08Suggestive
Familial exudative vitreoretinopathyOpen Targets
0.08Suggestive
Leber congenital amaurosis 13Open Targets
0.08Suggestive
Persistent hyperplastic primary vitreous, autosomal recessiveUniProt
Pathogenic Variants5
NM_145178.4(ATOH7):c.154C>G (p.Arg52Gly)Likely pathogenic
Persistent hyperplastic primary vitreous, autosomal recessive
β˜…β˜†β˜†β˜†2023β†’ Residue 52
NM_145178.4(ATOH7):c.175G>A (p.Ala59Thr)Pathogenic
Foveal hypoplasia;Optic nerve hypoplasia
β˜†β˜†β˜†β˜†2019β†’ Residue 59
NC_000010.11:g.68247368_68253890delPathogenic
Persistent hyperplastic primary vitreous, autosomal recessive
β˜†β˜†β˜†β˜†2018
NM_145178.4(ATOH7):c.136A>C (p.Asn46His)Pathogenic
Persistent hyperplastic primary vitreous, autosomal recessive
β˜†β˜†β˜†β˜†2012β†’ Residue 46
NM_145178.4(ATOH7):c.146A>T (p.Glu49Val)Pathogenic
Persistent hyperplastic primary vitreous, autosomal recessive
β˜†β˜†β˜†β˜†2012β†’ Residue 49
View on ClinVar β†—
Related Genes
GDF11Protein interaction91%POU4F2Protein interaction82%VSX2Protein interaction82%VSX1Protein interaction78%ATOH1Shared pathway36%OLIG3Shared pathway25%
Tissue Expression6 tissues
Liver
100%
Brain
39%
Ovary
4%
Lung
1%
Bone Marrow
1%
Heart
0%
Gene Interaction Network
Click a node to explore
ATOH7GDF11POU4F2VSX2VSX1ATOH1OLIG3
PROTEIN STRUCTURE
Preparing viewer…
AlphaFoldAI-predicted Β· UniProt Q8N100
View on AlphaFold β†—
Constraintβ“˜
LOEUFβ“˜
1.76LoF Tolerant
pLIβ“˜
0.00Tolerant
Observed/Expected LoF1.09 [0.67–1.76]
RankingsWhere ATOH7 stands among ~20K protein-coding genes
  • #10,735of 20,598
    Most Researched36
  • #3,610of 5,498
    Most Pathogenic Variants5
  • #16,376of 17,882
    Most Constrained (LOEUF)1.76
Genes detectedATOH7
Sources retrieved10 papers
Response timeβ€”
πŸ“„ Sources
10β–Ό
1
Single-Cell Analysis of Human Retina Identifies Evolutionarily Conserved and Species-Specific Mechanisms Controlling Development.
PMID: 32386599
Dev Cell Β· 2020
1.00
2
Identification and Characterization of ATOH7-Regulated Target Genes and Pathways in Human Neuroretinal Development.
PMID: 38994994
Cells Β· 2024
0.90
3
Genome-wide association identifies ATOH7 as a major gene determining human optic disc size.
PMID: 20395239
Hum Mol Genet Β· 2010
0.80
4
Elevated expression of human bHLH factor ATOH7 accelerates cell cycle progression of progenitors and enhances production of avian retinal ganglion cells.
PMID: 29717171
Sci Rep Β· 2018
0.70
5
Human retinal ganglion cell neurons generated by synchronous BMP inhibition and transcription factor mediated reprogramming.
PMID: 37773257
NPJ Regen Med Β· 2023
0.60