SNUPN

Chr 15AR

snurportin 1

Also known as: KPNBL, LGMDR29, RNUT1, Snurportin1

The protein functions as a nuclear import receptor that specifically recognizes the trimethylguanosine cap structure of spliceosomal U snRNPs and facilitates their transport into the nucleus. Mutations cause limb-girdle muscular dystrophy type 29, which follows autosomal recessive inheritance. The gene shows extreme intolerance to loss-of-function variants (pLI ~1.0), indicating that biallelic variants are required for disease manifestation.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismARLOEUF 1.491 OMIM phenotype
Clinical SummarySNUPN
🧬
Gene-Disease Validity (ClinGen)
SNUPN-related muscular dystrophy with or without multi-system involvement · ARStrong

Strong evidence — appropriate for clinical testing

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
33 unique Pathogenic / Likely Pathogenic· 56 VUS of 105 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.49LOEUF
pLI 0.000
Z-score -0.16
OE 1.04 (0.731.49)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.61Z-score
OE missense 0.88 (0.781.00)
175 obs / 199.2 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE1.04 (0.731.49)
00.351.4
Missense OE0.88 (0.781.00)
00.61.4
Synonymous OE0.84
01.21.6
LoF obs/exp: 21 / 20.2Missense obs/exp: 175 / 199.2Syn Z: 1.09

ClinVar Variant Classifications

105 submitted variants in ClinVar

Classification Summary

Pathogenic28
Likely Pathogenic5
VUS56
Likely Benign3
Conflicting1
28
Pathogenic
5
Likely Pathogenic
56
VUS
3
Likely Benign
1
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
28
0
28
Likely Pathogenic
2
0
3
0
5
VUS
0
46
10
0
56
Likely Benign
0
3
0
0
3
Benign
0
0
0
0
0
Conflicting
1
Total24941093

LoF = frameshift, stop gained/lost, canonical splice · Counts from ClinVar esearch · Updated hourly

View in ClinVar →

Protein Context — Lollipop Plot

SNUPN · protein map & ClinVar variants

Showing all ClinVar variants across the protein. Search a specific variant to highlight its position.

3D Protein StructureAlphaFold

Clinical Trials

Active and recruiting trials from ClinicalTrials.gov

No active trials found for this gene.

Search ClinicalTrials.gov →
Clinical Literature
Open Research Assistant →
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC