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10 sources retrieved Β· Most recent: April 2026 Β· Index updated 15 days ago
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SAMD11
sterile alpha motif domain containing 11
Chromosome 1 Β· 1p36.33
NCBI Gene: 148398Ensembl: ENSG00000187634.13HGNC: HGNC:28706UniProt: A0A087WU74
21PubMed Papers
21Diseases
0Drugs
1Pathogenic Variants
FUNCTIONAL ROLE
Transcription Factor
CLINICAL
OMIM Disease Gene
DATA QUALITY
βœ“ Experimental GO Evidenceβœ“ Swiss-Prot Reviewed
protein bindingchromatin bindinghistone bindingDNA-binding transcription repressor activity, RNA polymerase II-specificretinitis pigmentosaAbnormal erythrocyte morphologyRetinal dystrophyoptic atrophy
✦AI Summary

SAMD11 (sterile alpha motif domain containing 11) is a transcriptional co-repressor that plays critical roles in retinal development and photoreceptor function. The protein is strongly expressed in human retina with differential localization between cone and rod photoreceptors 1. SAMD11 functions as a component of Polycomb group (PcG) multiprotein complexes and interacts with the photoreceptor transcription factor CRX to regulate gene expression in retinal cells 1. The gene exhibits extensive alternative splicing, with up to 45 splice variants identified in humans, though functional differences between species exist as human SAMD11 shows broader tissue expression and is not directly regulated by retinal transcription factors like mouse Samd11 2. Beyond retinal function, SAMD11 regulates collagen production in fibroblasts through upregulation of PLOD1, contributing to skin rejuvenation processes 3. Disease-wise, homozygous nonsense mutations in SAMD11 cause autosomal recessive retinitis pigmentosa, confirming its essential role in photoreceptor survival 1. The gene has also been implicated in autism spectrum disorders, with rare variants on chromosome 1.33 showing transmission patterns in multiple affected families 4. Additionally, SAMD11 shows differential methylation patterns across breast cancer subtypes 5.

Sources cited
1
SAMD11 is a transcriptional co-repressor strongly expressed in human retina, interacts with CRX, and causes retinitis pigmentosa when mutated
PMID: 27734943
2
SAMD11 has extensive alternative splicing with up to 45 variants in humans and broader expression compared to mouse ortholog
PMID: 23978614
3
SAMD11 regulates collagen production in fibroblasts through PLOD1 upregulation for skin rejuvenation
PMID: 40074205
4
Rare SAMD11 variants on 1p36.33 are transmitted with autism spectrum disorders in multiple families
PMID: 30276537
5
SAMD11 shows differential methylation patterns across breast cancer subtypes
PMID: 28912426
Disease Associationsβ“˜21
retinitis pigmentosaOpen Targets
0.45Moderate
Abnormal erythrocyte morphologyOpen Targets
0.24Weak
Retinal dystrophyOpen Targets
0.18Weak
Neutrophil inclusion bodiesOpen Targets
0.11Weak
optic atrophyOpen Targets
0.11Weak
attention deficit hyperactivity disorderOpen Targets
0.06Suggestive
Griscelli diseaseOpen Targets
0.05Suggestive
hereditary attention deficit-hyperactivity disorderOpen Targets
0.04Suggestive
uncombable hair syndromeOpen Targets
0.04Suggestive
neoplasmOpen Targets
0.04Suggestive
schizophrenia 15Open Targets
0.04Suggestive
subarachnoid hemorrhageOpen Targets
0.04Suggestive
Griscelli disease type 3Open Targets
0.04Suggestive
Griscelli syndrome type 3Open Targets
0.04Suggestive
Griscelli disease type 1Open Targets
0.04Suggestive
Griscelli syndrome type 1Open Targets
0.04Suggestive
Phelan-McDermid syndromeOpen Targets
0.04Suggestive
nerve plexus diseaseOpen Targets
0.03Suggestive
hypotrichosis 8Open Targets
0.03Suggestive
isolated familial wooly hair disorderOpen Targets
0.03Suggestive
Retinitis pigmentosaUniProt
Pathogenic Variants1
NM_001385641.1(SAMD11):c.2377C>T (p.Arg793Ter)Pathogenic
not provided
β˜…β˜†β˜†β˜†2025β†’ Residue 793
View on ClinVar β†—
Related Genes
NRLShared pathway50%RPGRIP1Shared pathway50%SAMD7Shared pathway33%RPGRIP1LShared pathway20%RP1Shared pathway10%RORBShared pathway10%
Tissue Expression6 tissues
Liver
100%
Bone Marrow
69%
Heart
67%
Lung
35%
Ovary
31%
Brain
20%
Gene Interaction Network
Click a node to explore
SAMD11NRLRPGRIP1SAMD7RPGRIP1LRP1RORB
PROTEIN STRUCTURE
Preparing viewer…
AlphaFoldAI-predicted Β· UniProt Q96NU1
View on AlphaFold β†—
Constraintβ“˜
LOEUFβ“˜
1.76LoF Tolerant
pLIβ“˜
0.00Tolerant
Observed/Expected LoF1.46 [1.20–1.76]
RankingsWhere SAMD11 stands among ~20K protein-coding genes
  • #14,003of 20,598
    Most Researched21
  • #5,367of 5,498
    Most Pathogenic Variants1
  • #16,398of 17,882
    Most Constrained (LOEUF)1.76
Genes detectedSAMD11
Sources retrieved10 papers
Response timeβ€”
πŸ“„ Sources
10β–Ό
1
Increasing collagen synthesis in fibroblasts: The roles of PCL microspheres and the SAMD11-PLOD1 axis in skin rejuvenation.
PMID: 40074205
Biochim Biophys Acta Mol Cell Res Β· 2025
1.00
2
Identification and characterization of novel alternative splice variants of human SAMD11.
PMID: 23978614
Gene Β· 2013
0.90
3
Identification of the Photoreceptor Transcriptional Co-Repressor SAMD11 as Novel Cause of Autosomal Recessive Retinitis Pigmentosa.
PMID: 27734943
Sci Rep Β· 2016
0.80
4
Whole exome sequencing in extended families with autism spectrum disorder implicates four candidate genes.
PMID: 26204995
Hum Genet Β· 2015
0.70
5
Founder Mutations for Early Onset Melanoma as Revealed by Whole Exome Sequencing Suggests That This is Not Associated with the Increasing Incidence of Melanoma in Poland.
PMID: 29764119
Cancer Res Treat Β· 2019
0.60