ABCC8

Chr 11ADAR

ATP binding cassette subfamily C member 8

The ABCC8 protein is a regulatory subunit of pancreatic ATP-sensitive potassium channels that controls insulin release by forming KATP channels with KCNJ11. Mutations cause a spectrum of glucose homeostasis disorders including neonatal hyperinsulinemic hypoglycemia, transient and permanent neonatal diabetes, and maturity-onset diabetes of the young type 12. Inheritance can be either autosomal dominant or autosomal recessive depending on the specific condition and mutation.

OMIMResearchSummary from RefSeq, OMIM, UniProt
MultiplemechanismAD/ARLOEUF 0.776 OMIM phenotypes
Clinical SummaryABCC8
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Gene-Disease Validity (ClinGen)
monogenic diabetes · SDDefinitive

Definitive — sufficient evidence for diagnostic panels

4 total gene-disease associations curated

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
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Clinical Trials
2 active or recruiting trials — potential therapeutic options may be available
Some data sources returned errors (1)

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Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.77LOEUF
pLI 0.000
Z-score 3.35
OE 0.61 (0.480.77)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
1.91Z-score
OE missense 0.82 (0.770.87)
752 obs / 914.9 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.61 (0.480.77)
00.351.4
Missense OE0.82 (0.770.87)
00.61.4
Synonymous OE1.06
01.21.6
LoF obs/exp: 51 / 84.2Missense obs/exp: 752 / 914.9Syn Z: -0.85
DN
0.78top 25%
GOF
0.80top 10%
LOF
0.2288th %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 · 1 literature citation
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.

Literature Evidence

GOFHeterogeneous nature of diabetes in a family with a gain-of-function mutation in the ATP-binding cassette subfamily C member 8 (ABCC8) genePMID:30068891

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

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

ABCC8 · 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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