DDHD1

Chr 14AR

DDHD domain containing 1

Also known as: PA-PLA1, PAPLA1, SPG28, iPLA1I, iPLA1alpha

The protein functions as an intracellular phospholipase A1 that preferentially hydrolyzes phosphatidic acid and regulates mitochondrial dynamics through its cytosolic and mitochondrial localization. Mutations cause autosomal recessive spastic paraplegia 28 (SPG28), characterized by progressive weakness and spasticity of the legs. The pathogenic mechanism involves loss of function leading to disrupted mitochondrial organization and dynamics.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
LOFmechanismARLOEUF 0.341 OMIM phenotype
Clinical SummaryDDHD1
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Gene-Disease Validity (ClinGen)
hereditary spastic paraplegia · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 0.90). One damaged copy is likely sufficient to cause disease.
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ClinVar Variants
27 unique Pathogenic / Likely Pathogenic· 259 VUS of 500 total submissions
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GeneReview available — DDHD1
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.34LOEUF
pLI 0.902
Z-score 4.93
OE 0.19 (0.110.34)
Highly constrained

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

Missense Constraint
1.18Z-score
OE missense 0.85 (0.790.92)
427 obs / 501.4 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.19 (0.110.34)
00.351.4
Missense OE0.85 (0.790.92)
00.61.4
Synonymous OE1.02
01.21.6
LoF obs/exp: 8 / 42.8Missense obs/exp: 427 / 501.4Syn Z: -0.25
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveDDHD1-related spastic paraplegiaLOFAR

Predictions shown for reference only — model trained on dominant genes, not applicable to AR conditions.

DN
0.3694th %ile
GOF
0.4579th %ile
LOF
0.65top 25%

The Badonyi & Marsh prediction model was trained exclusively on dominant disease genes. Predictions are not reliable for genes with autosomal recessive inheritance and are shown at reduced opacity for reference only.

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

500 submitted variants in ClinVar

Classification Summary

Pathogenic20
Likely Pathogenic7
VUS259
Likely Benign163
Benign27
Conflicting10
20
Pathogenic
7
Likely Pathogenic
259
VUS
163
Likely Benign
27
Benign
10
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
12
0
8
0
20
Likely Pathogenic
5
1
1
0
7
VUS
1
245
11
2
259
Likely Benign
0
9
54
100
163
Benign
0
5
18
4
27
Conflicting
10
Total1826092106486

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

View in ClinVar →

Protein Context — Lollipop Plot

DDHD1 · 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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