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10 sources retrieved Β· Most recent: April 2026 Β· Index updated 14 days ago
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CWC27
CWC27 spliceosome associated cyclophilin
Chromosome 5 Β· 5q12.3
NCBI Gene: 10283Ensembl: ENSG00000153015.17HGNC: HGNC:10664UniProt: A0A8I5KQK1
64PubMed Papers
21Diseases
0Drugs
45Pathogenic Variants
FUNCTIONAL ROLE
Hub Gene
CLINICAL
OMIM Disease Gene
DATA QUALITY
βœ“ Experimental GO Evidenceβœ“ Swiss-Prot Reviewed
peptidyl-prolyl cis-trans isomerase activitynucleoplasmU2-type precatalytic spliceosomecatalytic step 2 spliceosomeMetaphyseal chondrodysplasia - retinitis pigmentosametaphyseal chondrodysplasia-retinitis pigmentosa syndromeAbnormality of the skeletal systemRetinal dystrophy
✦AI Summary

CWC27 is a spliceosome-associated cyclophilin that functions as a core component of the pre-mRNA splicing machinery 1. Unlike typical cyclophilins, CWC27 is a probable inactive peptidyl-prolyl cis-trans isomerase lacking enzymatic PPIase activity 2. Instead, it acts as a structural protein within the spliceosome, participating in catalytic splicing steps and potentially in U12-type minor spliceosome function. CWC27 forms a functional heterodimer with CWC22 that links the exon junction complex to spliceosomes, serving as an intermediate landing platform during spliceosome assembly 3. Biallelic CWC27 mutations cause CWC27-related spliceosomopathy, a rare autosomal recessive disorder 4. Disease pathophysiology involves disrupted pre-mRNA splicing, with particular sensitivity in neural crest cell-derived tissues, likely through p53-mediated cell death pathways 5. Clinical manifestations include early-onset retinitis pigmentosa with chorioretinal atrophy 4, skeletal anomalies, short stature, and distinctive ectodermal features (sparse hair, nail dysplasia, dental anomalies) 6. Additional systemic involvement may include craniofacial malformations, neurological defects, and immune abnormalities 7. Gene augmentation therapy via AAV8-mediated CWC27 delivery successfully rescued retinal degeneration in Cwc27 mutant mice 8, suggesting therapeutic potential for this severe spliceosomopathy.

Sources cited
1
CWC27 plays a role in pre-mRNA splicing as part of the spliceosome
PMID: 29360106
2
CWC27 is a probable inactive PPIase with no peptidyl-prolyl cis-trans isomerase activity
PMID: 20676357
3
CWC27/CWC22 heterodimer structure and function linking exon junction complex to spliceosomes
PMID: 32329775
4
CWC27 mutations associated with craniofacial spliceosomopathies and neural crest cell sensitivity to splicing defects
PMID: 32315467
5
CWC27-related spliceosomopathy presents with early-onset retinitis pigmentosa, short stature, skeletal anomalies, and systemic features
PMID: 38956876
6
Ectodermal involvement in CWC27 spliceosomopathy including sparse hair, nail dysplasia, and dental anomalies
PMID: 36718996
7
CWC27 variants lead to retinal degeneration, skeletal anomalies, short stature, and neurological defects
PMID: 31481716
8
AAV8-mediated CWC27 gene therapy rescues retinal degeneration in Cwc27 mutant mice
PMID: 37479075
Disease Associationsβ“˜21
Metaphyseal chondrodysplasia - retinitis pigmentosaOpen Targets
0.77Strong
metaphyseal chondrodysplasia-retinitis pigmentosa syndromeOpen Targets
0.76Strong
Abnormality of the skeletal systemOpen Targets
0.45Moderate
Retinal dystrophyOpen Targets
0.43Moderate
diverticular diseaseOpen Targets
0.43Moderate
osteoarthritis, hipOpen Targets
0.39Weak
dengue diseaseOpen Targets
0.37Weak
osteoarthritisOpen Targets
0.36Weak
osteoarthritis, kneeOpen Targets
0.36Weak
ventral herniaOpen Targets
0.34Weak
retinitis pigmentosaOpen Targets
0.34Weak
urinary bladder carcinomaOpen Targets
0.33Weak
albuminuriaOpen Targets
0.33Weak
obesityOpen Targets
0.33Weak
nephritisOpen Targets
0.29Weak
NephropathyOpen Targets
0.29Weak
total joint arthroplastyOpen Targets
0.29Weak
Hammer Toe SyndromeOpen Targets
0.29Weak
pyogenic granulomaOpen Targets
0.29Weak
heart failureOpen Targets
0.27Weak
Retinitis pigmentosa with or without skeletal anomaliesUniProt
Pathogenic Variants45
NM_005869.4(CWC27):c.1156dup (p.Leu386fs)Pathogenic
not provided|Retinal dystrophy
β˜…β˜…β˜†β˜†2026β†’ Residue 386
NM_005869.4(CWC27):c.666_669del (p.Ser222fs)Pathogenic
not provided
β˜…β˜…β˜†β˜†2025β†’ Residue 222
NM_005869.4(CWC27):c.1002dup (p.Val335fs)Pathogenic
Metaphyseal chondrodysplasia-retinitis pigmentosa syndrome|not provided|Retinal dystrophy
β˜…β˜…β˜†β˜†2025β†’ Residue 335
NM_005869.4(CWC27):c.495G>A (p.Glu165=)Pathogenic
Metaphyseal chondrodysplasia-retinitis pigmentosa syndrome|not provided|Retinitis pigmentosa
β˜…β˜…β˜†β˜†2025β†’ Residue 165
NM_005869.4(CWC27):c.756del (p.Gly253fs)Pathogenic
not provided|Metaphyseal chondrodysplasia-retinitis pigmentosa syndrome
β˜…β˜…β˜†β˜†2024β†’ Residue 253
NM_005869.4(CWC27):c.938+2T>CLikely pathogenic
not provided|Metaphyseal chondrodysplasia-retinitis pigmentosa syndrome|Familial pancreatic carcinoma
β˜…β˜…β˜†β˜†2024
NM_005869.4(CWC27):c.427C>T (p.Arg143Ter)Pathogenic
Metaphyseal chondrodysplasia-retinitis pigmentosa syndrome|not provided
β˜…β˜…β˜†β˜†2024β†’ Residue 143
NM_005869.4(CWC27):c.397-1G>ALikely pathogenic
Metaphyseal chondrodysplasia-retinitis pigmentosa syndrome
β˜…β˜…β˜†β˜†2024
NM_005869.4(CWC27):c.1066_1070del (p.Ala356fs)Pathogenic
Metaphyseal chondrodysplasia-retinitis pigmentosa syndrome|not provided
β˜…β˜…β˜†β˜†2023β†’ Residue 356
NM_005869.4(CWC27):c.1122_1125del (p.Ser375fs)Pathogenic
Retinitis pigmentosa
β˜…β˜†β˜†β˜†2026β†’ Residue 375
NM_005869.4(CWC27):c.1117del (p.Gln373fs)Pathogenic
not provided
β˜…β˜†β˜†β˜†2025β†’ Residue 373
NM_005869.4(CWC27):c.94G>T (p.Glu32Ter)Pathogenic
not provided
β˜…β˜†β˜†β˜†2025β†’ Residue 32
NM_005869.4(CWC27):c.1152+2T>ALikely pathogenic
not provided
β˜…β˜†β˜†β˜†2025
NM_005869.4(CWC27):c.326del (p.Asn109fs)Pathogenic
not provided
β˜…β˜†β˜†β˜†2025β†’ Residue 109
NM_005869.4(CWC27):c.676A>T (p.Lys226Ter)Pathogenic
not provided
β˜…β˜†β˜†β˜†2024β†’ Residue 226
NM_005869.4(CWC27):c.829del (p.Glu277fs)Likely pathogenic
Metaphyseal chondrodysplasia-retinitis pigmentosa syndrome
β˜…β˜†β˜†β˜†2024β†’ Residue 277
NM_005869.4(CWC27):c.139+2T>CLikely pathogenic
Metaphyseal chondrodysplasia-retinitis pigmentosa syndrome
β˜…β˜†β˜†β˜†2024
NM_005869.4(CWC27):c.872del (p.Lys291fs)Likely pathogenic
Metaphyseal chondrodysplasia-retinitis pigmentosa syndrome
β˜…β˜†β˜†β˜†2024β†’ Residue 291
NM_005869.4(CWC27):c.1033dup (p.Arg345fs)Pathogenic
not provided
β˜…β˜†β˜†β˜†2024β†’ Residue 345
NM_005869.4(CWC27):c.355C>T (p.Arg119Ter)Pathogenic
not provided
β˜…β˜†β˜†β˜†2023β†’ Residue 119
View on ClinVar β†—
Related Genes
XAB2Protein interaction100%SNRPA1Protein interaction100%PRPF19Protein interaction100%SNRPB2Protein interaction100%PRPF3Protein interaction100%PRPF31Protein interaction100%
Tissue Expression6 tissues
Heart
100%
Bone Marrow
98%
Brain
93%
Ovary
63%
Lung
47%
Liver
31%
Gene Interaction Network
Click a node to explore
CWC27XAB2SNRPA1PRPF19SNRPB2PRPF3PRPF31
PROTEIN STRUCTURE
Preparing viewer…
PDB2HQ6 Β· 1.75 Γ… Β· X-ray
View on RCSB β†—
Constraintβ“˜
LOEUFβ“˜
1.18LoF Tolerant
pLIβ“˜
0.00Tolerant
Observed/Expected LoF0.80 [0.56–1.18]
RankingsWhere CWC27 stands among ~20K protein-coding genes
  • #7,255of 20,598
    Most Researched64
  • #1,428of 5,498
    Most Pathogenic Variants45
  • #12,386of 17,882
    Most Constrained (LOEUF)1.18
Genes detectedCWC27
Sources retrieved10 papers
Response timeβ€”
πŸ“„ Sources
10β–Ό
1
Further delineation of the CWC27-associated spliceosomeopathy: Case report and review of the literature.
PMID: 36718996
Am J Med Genet A Β· 2023
1.00
2
Spliceosomopathies and neurocristopathies: Two sides of the same coin?
PMID: 32315467
Dev Dyn Β· 2020
0.90
3
A novel small deletion in
PMID: 38956876
Ophthalmic Genet Β· 2024
0.80
4
Gene augmentation therapy to rescue degenerative photoreceptors in a Cwc27 mutant mouse model.
PMID: 37479075
Exp Eye Res Β· 2023
0.70
5
Structural and Functional Insights into Human Nuclear Cyclophilins.
PMID: 30518120
Biomolecules Β· 2018
0.60