SLC26A4

Chr 7AR

solute carrier family 26 member 4

Also known as: DFNB4, EVA, PDS, TDH2B

The encoded protein is a sodium-independent transporter that mediates electroneutral exchange of chloride and iodide, as well as chloride-bicarbonate exchange. Mutations cause Pendred syndrome, the most common form of syndromic deafness, which follows autosomal recessive inheritance and typically involves both hearing loss and thyroid dysfunction. This gene is not highly constrained against loss-of-function variants, consistent with its recessive inheritance pattern.

GeneReviewsOMIMResearchSummary from RefSeq, UniProt
MultiplemechanismARLOEUF 1.182 OMIM phenotypes
Clinical SummarySLC26A4
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Gene-Disease Validity (ClinGen)
Pendred syndrome · ARDefinitive

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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ClinVar Variants
144 unique Pathogenic / Likely Pathogenic· 128 VUS of 300 total submissions
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Clinical Trials
2 active or recruiting trials — potential therapeutic options may be available
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GeneReview available — SLC26A4
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.18LOEUF
pLI 0.000
Z-score 0.66
OE 0.89 (0.681.18)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-2.01Z-score
OE missense 1.28 (1.191.37)
528 obs / 413.0 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.89 (0.681.18)
00.351.4
Missense OE1.28 (1.191.37)
00.61.4
Synonymous OE1.02
01.21.6
LoF obs/exp: 35 / 39.5Missense obs/exp: 528 / 413.0Syn Z: -0.24
DN
0.74top 25%
GOF
0.81top 10%
LOF
0.2580th %ile

This gene has evidence for multiple mechanisms of pathogenicity (gain-of-function and dominant-negative). Both the Badonyi & Marsh prediction and the broader genomic evidence point to gain-of-function as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

GOFprediction above median
DNprediction above median

Note: In-silico variant effect predictors (SIFT, PolyPhen, REVEL, CADD) may underestimate pathogenicity of missense variants in genes with GOF or DN mechanisms. Consider functional evidence and clinical context.

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

ClinVar Variant Classifications

300 submitted variants in ClinVar

Classification Summary

Pathogenic54
Likely Pathogenic90
VUS128
Likely Benign23
Conflicting1
54
Pathogenic
90
Likely Pathogenic
128
VUS
23
Likely Benign
1
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
42
8
4
0
54
Likely Pathogenic
35
50
4
1
90
VUS
1
108
11
8
128
Likely Benign
0
1
13
9
23
Benign
0
0
0
0
0
Conflicting
1
Total781673218296

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

View in ClinVar →

Protein Context — Lollipop Plot

SLC26A4 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
Open Research Assistant →
Key Publications
Landmark & review papers · by relevance
PubMed
Top 5 results · since 2015Search PubMed ↗