GRAMD1B

Chr 11

GRAM domain containing 1B

Also known as: LINC01059

The protein functions as a cholesterol transporter that mediates non-vesicular cholesterol transfer from the plasma membrane to the endoplasmic reticulum at membrane contact sites, playing a crucial role in cellular cholesterol homeostasis. Mutations in this highly constrained gene (pLI=1.0, LOEUF=0.25) cause autosomal recessive congenital adrenal insufficiency with distinctive clinical features. The gene's critical role in adrenal steroidogenesis and its extreme intolerance to loss-of-function variants make it an important consideration in patients presenting with primary adrenal insufficiency.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
16
Pubs (1 yr)
52
P/LP submissions
0%
P/LP missense
0.24
LOEUF· LoF intol.
Mechanism
Clinical SummaryGRAMD1B
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.
📋
ClinVar Variants
52 unique Pathogenic / Likely Pathogenic· 82 VUS of 167 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Dual constrained — LoF & missense intolerant
LoF Constraint
0.24LOEUF
pLI 0.999
Z-score 5.37
OE 0.12 (0.060.24)
Highly constrained

Highly LoF-intolerant (top ~10% of genes)

Missense Constraint
3.23Z-score
OE missense 0.57 (0.510.63)
249 obs / 439.6 exp
Constrained

Highly missense-constrained (top ~0.1%)

Observed / Expected Ratios
LoF OE0.12 (0.060.24)
00.351.4
Missense OE0.57 (0.510.63)
00.61.4
Synonymous OE1.00
01.21.6
LoF obs/exp: 5 / 43.0Missense obs/exp: 249 / 439.6Syn Z: -0.01

ClinVar Variant Classifications

167 submitted variants in ClinVar

Classification Summary

Pathogenic49
Likely Pathogenic3
VUS82
Likely Benign14
Benign7
49
Pathogenic
3
Likely Pathogenic
82
VUS
14
Likely Benign
7
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
49
0
49
Likely Pathogenic
0
0
3
0
3
VUS
0
75
7
0
82
Likely Benign
0
6
4
4
14
Benign
0
2
2
3
7
Total083657155

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

View in ClinVar →

Protein Context — Lollipop Plot

GRAMD1B · protein map & ClinVar variants

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

Clinical Trials

Active and recruiting trials from ClinicalTrials.gov

No active trials found for this gene.

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Clinical Literature
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