SOX5

Chr 12AD

SRY-box transcription factor 5

Also known as: L-SOX5, L-SOX5B, L-SOX5F, LAMSHF

The SOX5 protein is a transcription factor that regulates embryonic development and cell fate determination, with particular involvement in chondrogenesis. Loss-of-function mutations cause Lamb-Shaffer syndrome, an autosomal dominant neurodevelopmental disorder. The gene is highly constrained against loss-of-function variants, indicating that haploinsufficiency is the mechanism underlying this condition.

OMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
LOFmechanismADLOEUF 0.191 OMIM phenotype
Clinical SummarySOX5
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Gene-Disease Validity (ClinGen)
Lamb-Shaffer syndrome · ADDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 1.00). One damaged copy is likely sufficient to cause disease.
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ClinVar Variants
80 unique Pathogenic / Likely Pathogenic· 167 VUS of 300 total submissions
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Clinical Trials
1 active or recruiting trial — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Dual constrained — LoF & missense intolerant
LoF Constraint
0.19LOEUF
pLI 1.000
Z-score 5.52
OE 0.07 (0.030.19)
Highly constrained

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

Missense Constraint
3.15Z-score
OE missense 0.57 (0.510.64)
244 obs / 427.3 exp
Constrained

Highly missense-constrained (top ~0.1%)

Observed / Expected Ratios
LoF OE0.07 (0.030.19)
00.351.4
Missense OE0.57 (0.510.64)
00.61.4
Synonymous OE1.02
01.21.6
LoF obs/exp: 3 / 41.2Missense obs/exp: 244 / 427.3Syn Z: -0.21
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongSOX5-related 12p12.5 intragenic deletions associated with intellectual disabilityLOFAD
DN
0.3296th %ile
GOF
0.3193th %ile
LOF
0.84top 5%

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

LOFprediction above median · 1 literature citation · 53% of P/LP variants are LoF · LOEUF 0.19

Literature Evidence

LOFClinical and genetic characterization of a patient with SOX5 haploinsufficiency caused by a de novo balanced reciprocal translocation.PMID:29477873

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

300 submitted variants in ClinVar

Classification Summary

Pathogenic56
Likely Pathogenic24
VUS167
Likely Benign31
Benign3
Conflicting3
56
Pathogenic
24
Likely Pathogenic
167
VUS
31
Likely Benign
3
Benign
3
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
30
2
24
0
56
Likely Pathogenic
12
9
3
0
24
VUS
3
139
22
3
167
Likely Benign
2
17
5
7
31
Benign
0
0
2
1
3
Conflicting
3
Total471675611284

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

View in ClinVar →

Protein Context — Lollipop Plot

SOX5 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
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