WAC

Chr 10AD

WW domain containing adaptor with coiled-coil

Also known as: BM-016, DESSH, PRO1741, Wwp4

The WAC protein contains a WW domain that mediates protein-protein interactions by binding proline-rich peptide motifs and localizes to nuclear speckles. Loss-of-function mutations cause Desanto-Shinawi syndrome, an autosomal dominant neurodevelopmental disorder. The gene is highly intolerant to loss-of-function variants, indicating haploinsufficiency as the likely pathogenic mechanism.

OMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
LOFmechanismADLOEUF 0.081 OMIM phenotype
Clinical SummaryWAC
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Gene-Disease Validity (ClinGen)
DeSanto-Shinawi 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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Clinical Trials
2 active or recruiting trials — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.08LOEUF
pLI 1.000
Z-score 5.53
OE 0.00 (0.000.08)
Highly constrained

Among the most LoF-intolerant genes (~top 3%)

Missense Constraint
1.51Z-score
OE missense 0.77 (0.690.85)
262 obs / 340.5 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.00 (0.000.08)
00.351.4
Missense OE0.77 (0.690.85)
00.61.4
Synonymous OE1.05
01.21.6
LoF obs/exp: 0 / 35.6Missense obs/exp: 262 / 340.5Syn Z: -0.42
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveWAC-related Desanto-Shinawi syndromeLOFAD
DN
0.2299th %ile
GOF
0.1799th %ile
LOF
0.90top 5%

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

LOFprediction above median · 1 literature citation · LOEUF 0.08

Literature Evidence

LOFThe clinical features associated with WAC haploinsufficiency include recognizable dysmorphic facial features that were recently delineated as DeSanto-Shinawi syndrome (DESSH; OMIM 616708).PMID:29928181

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

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

WAC · 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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