SMAD6

Chr 15AD

SMAD family member 6

Also known as: AOVD2, HsT17432, MADH6, MADH7

SMAD6 encodes an inhibitory SMAD protein that negatively regulates BMP and TGF-beta signaling pathways, which are critical for bone, cartilage, and cardiovascular development. Mutations cause autosomal dominant craniosynostosis, radioulnar synostosis, and aortic valve disease. The gene is not highly constrained against loss-of-function variants, consistent with its role in developmental disorders affecting specific tissue types rather than causing severe multisystem disease.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismADLOEUF 1.983 OMIM phenotypes
Clinical SummarySMAD6
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Gene-Disease Validity (ClinGen)
syndromic craniosynostosis · ADDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
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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

Tolerant — LoF & missense variants common in population
LoF Constraint
1.98LOEUF
pLI 0.000
Z-score -3.07
OE 1.97 (1.311.98)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.59Z-score
OE missense 1.11 (1.001.23)
249 obs / 224.1 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE1.97 (1.311.98)
00.351.4
Missense OE1.11 (1.001.23)
00.61.4
Synonymous OE0.96
01.21.6
LoF obs/exp: 23 / 11.7Missense obs/exp: 249 / 224.1Syn Z: 0.37
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
limitedSMAD6-related non-syndromic craniosynostosisLOFAD
DN
0.3693th %ile
GOF
0.3888th %ile
LOF
0.66top 25%

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

LOFprediction above median · 1 literature citation

Literature Evidence

LOFIn addition, none of the 18 family members who carried only the rs1884302 risk allele, including 2 homozygotes, had craniosynostosis. Segregation analysis in a parametric 2-locus linkage model yielded a 7.37 maximum lod score. Of the 13 SMAD6 variants, 8 were frameshift or premature termination mutaPMID:27606499

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

SMAD6 · 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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Full-Text Mentions
NLP-detected gene mentions in article bodies · via PubTator3
PubTator3
Top 5 full-text resultsSearch PubTator3 ↗