PDGFRA

Chr 4Isolated casesSomatic

platelet derived growth factor receptor alpha

Also known as: CD140A, PDGFR-2, PDGFR2

The protein functions as a cell surface tyrosine kinase receptor for platelet-derived growth factors and is essential for embryonic development, gastrointestinal tract development, and mesenchymal cell differentiation. Mutations cause gastrointestinal stromal tumors (GIST), GIST-plus syndrome, and idiopathic hypereosinophilic syndrome through constitutive activation of downstream signaling pathways including PI3K/AKT, MAPK, and STAT cascades. These conditions typically occur as isolated cases or result from somatic mutations.

Summary from RefSeq, OMIM, UniProt
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Primary Disease Associations & Inheritance

Gastrointestinal stromal tumor/GIST-plus syndrome, somatic or familialMIM #175510
Hypereosinophilic syndrome, idiopathic, resistant to imatinibMIM #607685
Isolated casesSomatic
3
Active trials
325
Pubs (1 yr)
2
P/LP submissions
P/LP missense
0.17
LOEUF· LoF intol.
GOF*
Mechanism· G2P
Clinical SummaryPDGFRA
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Gene-Disease Validity (ClinGen)
gastrointestinal stromal tumor · ADDefinitive

Definitive — sufficient evidence for diagnostic panels

2 total gene-disease associations curated

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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ClinVar Variants
2 unique Pathogenic / Likely Pathogenic· 503 VUS of 900 total submissions
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Clinical Trials
3 active or recruiting trials — potential therapeutic options may be available
Some data sources returned errors (1)

ensembl: TimeoutError: The operation was aborted due to timeout

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.17LOEUF
pLI 1.000
Z-score 6.31
OE 0.07 (0.040.17)
Highly constrained

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

Missense Constraint
1.94Z-score
OE missense 0.78 (0.720.84)
458 obs / 590.7 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.07 (0.040.17)
00.351.4
Missense OE0.78 (0.720.84)
00.61.4
Synonymous OE1.01
01.21.6
LoF obs/exp: 4 / 54.1Missense obs/exp: 458 / 590.7Syn Z: -0.10
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitivePDGFRA-related gastrointestinal stromal tumor/GIST-plus syndrome, somatic or familialGOFAD
DN
0.5279th %ile
GOF
0.6931th %ile
LOF
0.52top 25%

This gene has evidence for multiple mechanisms of pathogenicity (loss-of-function and gain-of-function). The Badonyi & Marsh model scores gain-of-function highest among its predictions, but genomic evidence (constraint, ClinVar variant spectrum, and literature) most strongly supports loss-of-function (haploinsufficiency). Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

LOFLOEUF 0.17
GOFprediction above median

Note: In-silico variant effect predictors (SIFT, PolyPhen, REVEL, CADD) may underestimate pathogenicity of missense variants in genes with GOF or DN mechanisms. Consider functional evidence and clinical context.

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

900 submitted variants in ClinVar

Classification Summary

Pathogenic2
VUS503
Likely Benign331
Benign4
Conflicting3
2
Pathogenic
503
VUS
331
Likely Benign
4
Benign
3
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
2
0
2
Likely Pathogenic
0
0
0
0
0
VUS
28
429
37
9
503
Likely Benign
1
6
131
193
331
Benign
1
0
3
0
4
Conflicting
3
Total30435173202843

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

View in ClinVar →

Protein Context — Lollipop Plot

PDGFRA · protein map & ClinVar variants

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

Clinical Literature
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Key Publications
Landmark & review papers · by relevance
PubMed
Top 5 results · since 2015Search PubMed ↗