The STBD1 protein acts as a cargo receptor that delivers glycogen to lysosomes through an autophagic pathway called glycophagy. Biallelic mutations cause polyglucosan body myopathy 1, an autosomal recessive disorder characterized by adult-onset progressive muscle weakness and cardiomyopathy. The gene is not constrained against loss-of-function variants, consistent with the recessive inheritance pattern observed in affected individuals.

OMIMResearchSummary from RefSeq, UniProt
LOEUF 1.85
Clinical SummarySTBD1
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.85LOEUF
pLI 0.000
Z-score -0.96
OE 1.31 (0.871.85)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.23Z-score
OE missense 0.95 (0.841.08)
182 obs / 190.9 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE1.31 (0.871.85)
00.351.4
Missense OE0.95 (0.841.08)
00.61.4
Synonymous OE1.04
01.21.6
LoF obs/exp: 15 / 11.5Missense obs/exp: 182 / 190.9Syn Z: -0.30

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

STBD1 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold

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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