WIPI2

Chr 7AR

WD repeat domain, phosphoinositide interacting 2

Also known as: ATG18B, Atg21, CGI-50, IDDSSA, WIPI-2

WIPI2 encodes a component of the autophagy machinery that controls intracellular degradation by packaging cytoplasmic materials into autophagosomes for delivery to lysosomes, and is involved in early formation of preautophagosomal structures. Autosomal recessive mutations cause intellectual developmental disorder with short stature and variable skeletal anomalies. The gene shows tolerance to loss-of-function variants (pLI 0.006, LOEUF 0.619), consistent with the recessive inheritance pattern.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismARLOEUF 0.621 OMIM phenotype
Clinical SummaryWIPI2
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.34) despite low pLI — interpret in context.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.62LOEUF
pLI 0.006
Z-score 2.94
OE 0.34 (0.200.62)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
1.66Z-score
OE missense 0.72 (0.640.81)
204 obs / 282.6 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.34 (0.200.62)
00.351.4
Missense OE0.72 (0.640.81)
00.61.4
Synonymous OE1.11
01.21.6
LoF obs/exp: 8 / 23.3Missense obs/exp: 204 / 282.6Syn Z: -0.99

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

WIPI2 · 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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