ABCC4

Chr 13

ATP binding cassette subfamily C member 4 (PEL blood group)

Also known as: MOAT-B, MOATB, MRP4

ABCC4 encodes an ATP-binding cassette transporter that actively pumps cyclic nucleotides (cAMP, cGMP), prostaglandins, bile acids, and other signaling molecules out of cells. Mutations cause autosomal recessive pseudohypoaldosteronism type 1 with salt-wasting, hyperkalemia, and metabolic acidosis typically presenting in infancy. The gene is highly constrained against loss-of-function mutations (LOEUF 0.492), indicating that complete protein loss is likely incompatible with normal development.

OMIMResearchSummary from RefSeq, UniProt
MultiplemechanismLOEUF 0.49
Clinical SummaryABCC4
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Gene-Disease Validity (ClinGen)
qualitative platelet defect · ARModerate

Moderate evidence — consider for supplementary testing

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
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ClinVar Variants
78 unique Pathogenic / Likely Pathogenic· 149 VUS of 313 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

Moderate LoF intolerance
LoF Constraint
0.49LOEUF
pLI 0.000
Z-score 5.12
OE 0.35 (0.260.49)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
1.70Z-score
OE missense 0.82 (0.770.88)
591 obs / 719.4 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.35 (0.260.49)
00.351.4
Missense OE0.82 (0.770.88)
00.61.4
Synonymous OE1.06
01.21.6
LoF obs/exp: 26 / 73.3Missense obs/exp: 591 / 719.4Syn Z: -0.75
DN
0.76top 25%
GOF
0.73top 25%
LOF
0.2581th %ile

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

DNprediction above median
GOFprediction 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

313 submitted variants in ClinVar

Classification Summary

Pathogenic78
VUS149
Likely Benign23
Benign25
78
Pathogenic
149
VUS
23
Likely Benign
25
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
78
0
78
Likely Pathogenic
0
0
0
0
0
VUS
1
135
13
0
149
Likely Benign
0
9
4
10
23
Benign
0
6
8
11
25
Total115010321275

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

View in ClinVar →

Protein Context — Lollipop Plot

ABCC4 · 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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