SEPHS1

Chr 10AD

selenophosphate synthetase 1

Also known as: SELD, SPS, SPS1, VERBRAS2

This protein functions as a core component of the zincore complex that stabilizes zinc finger transcription factors at DNA-binding sites to control gene expression, and plays an essential role in cellular redox homeostasis. Mutations cause Ververi-Brady syndrome 2, inherited in an autosomal dominant pattern. The gene is highly constrained against loss-of-function variants, indicating that functional copies are critical for normal development.

Summary from RefSeq, OMIM, UniProt
Research Assistant →

Primary Disease Associations & Inheritance

Ververi-Brady syndrome 2MIM #621325
AD
0
Active trials
7
Pubs (1 yr)
25
P/LP submissions
14%
P/LP missense
0.30
LOEUF· LoF intol.
LOF
Mechanism· predicted
Clinical SummarySEPHS1
Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 0.98). One damaged copy is likely sufficient to cause disease.
📋
ClinVar Variants
22 unique Pathogenic / Likely Pathogenic· 43 VUS of 89 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Dual constrained — LoF & missense intolerant
LoF Constraint
0.30LOEUF
pLI 0.976
Z-score 3.46
OE 0.06 (0.020.30)
Highly constrained

Highly LoF-intolerant (top ~10% of genes)

Missense Constraint
3.24Z-score
OE missense 0.41 (0.350.49)
99 obs / 240.3 exp
Constrained

Highly missense-constrained (top ~0.1%)

Observed / Expected Ratios
LoF OE0.06 (0.020.30)
00.351.4
Missense OE0.41 (0.350.49)
00.61.4
Synonymous OE1.10
01.21.6
LoF obs/exp: 1 / 15.9Missense obs/exp: 99 / 240.3Syn Z: -0.78
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
moderateSEPHS1-related neurodevelopmental disorderOTHERAD
DN
0.3991th %ile
GOF
0.3590th %ile
LOF
0.68top 10%

The highest-scoring mechanism for this gene is loss-of-function (haploinsufficiency).

LOFprediction above median · LOEUF 0.30

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

89 submitted variants in ClinVar

Classification Summary

Pathogenic20
Likely Pathogenic2
VUS43
Likely Benign5
Benign2
Conflicting1
20
Pathogenic
2
Likely Pathogenic
43
VUS
5
Likely Benign
2
Benign
1
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
2
18
0
20
Likely Pathogenic
0
1
1
0
2
VUS
1
37
5
0
43
Likely Benign
0
0
1
4
5
Benign
0
0
1
1
2
Conflicting
1
Total14026573

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

View in ClinVar →

Protein Context — Lollipop Plot

SEPHS1 · protein map & ClinVar variants

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

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