PACS2

Chr 14AD

phosphofurin acidic cluster sorting protein 2

Also known as: DEE66, EIEE66, PACS-2, PACS1L

PACS2 encodes a protein that maintains endoplasmic reticulum calcium homeostasis and tethers mitochondria to the endoplasmic reticulum. Loss-of-function mutations cause developmental and epileptic encephalopathy 66, inherited in an autosomal dominant pattern. The pathogenic mechanism involves disrupted cellular calcium regulation and impaired mitochondrial-ER communication.

OMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
LOFmechanismADLOEUF 0.281 OMIM phenotype
Clinical SummaryPACS2
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Gene-Disease Validity (ClinGen)
developmental and epileptic encephalopathy · ADDefinitive

Definitive — sufficient evidence for diagnostic panels

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.
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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

LoF intolerant — likely haploinsufficient
LoF Constraint
0.28LOEUF
pLI 0.996
Z-score 5.41
OE 0.15 (0.080.28)
Highly constrained

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

Missense Constraint
2.24Z-score
OE missense 0.73 (0.680.80)
414 obs / 563.3 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.15 (0.080.28)
00.351.4
Missense OE0.73 (0.680.80)
00.61.4
Synonymous OE1.11
01.21.6
LoF obs/exp: 7 / 47.1Missense obs/exp: 414 / 563.3Syn Z: -1.41
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongPACS2-related neurodevelopmental disorderOTHERAD
DN
0.4587th %ile
GOF
0.3887th %ile
LOF
0.76top 10%

The highest-scoring mechanism for this gene is loss-of-function (haploinsufficiency).

LOFprediction above median · LOEUF 0.28

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

PACS2 · 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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