Genetic Information

Gene & Transcript Details

Gene
RUNX1
Transcript
NM_001754.4 MANE Select
Total Exons
Reference Sequence
NC_000021.8
Alternative Transcripts
IDStatusDetails
NM_001754.3 Alternative 6190 nt | 400–1842
NM_001754.4 RefSeq Select 5967 nt | 191–1633
NM_001754.5 MANE Select 5971 nt | 195–1637

Variant Details

HGVS Notation
NM_001754.4:c.474_489del
Protein Change
F158Lfs*13
Location
Exon 5 (Exon 5 of )
5
5'Exon Structure3'
Functional Consequence
Loss of Function
Alternate Identifiers

Clinical & Population Data

Population Frequency

gnomAD
Global Frequency
0.0 in 100,000
Extremely Rare
ACMG Criteria Applied PM2
This variant is absent or extremely rare in population databases (PM2 criteria applies).

ClinVar

Open
Classification
Unknown
0 publications
Clinical Statement

""

COSMIC Somatic Evidence

Open
COSMIC ID
Recurrence
0 occurrences
PM1 Criteria
Not Applied
COSMIC Database Preview
COSMIC Preview
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Functional Impact & Domains

Functional Domain

Hotspot Status
Not a hotspot
Domain Summary
This variant is not located in a mutational hotspot or critical domain.
Related Variants in This Domain
No evidence of other pathogenic variants at this position in gene RUNX1.

Functional Studies & Therapeutic Relevance

Functional Summary

The RUNX1 F158Lfs*13 variant is a truncating mutation in the RUNX1 gene, a tumor suppressor involved in hematopoietic differentiation. Functional evidence indicates that truncating mutations in RUNX1 typically inhibit its function, contributing to oncogenesis and predisposing individuals to hematologic malignancies such as leukemia, lymphomas, and myelodysplastic syndromes.

Database Previews
OncoKB
OncoKB Preview
JAX-CKB
JAX-CKB Preview

Click on previews to view full database entries. External databases may require institutional access.

Computational Analysis

Pathogenicity Predictions

SpliceAI
Predictor Consensus
Unknown
PP3 Applied
No
REVEL Score
0.0
Threshold: ≥0.75 = PP3 applied

SpliceAI Scores

Window: ±500bp
Effect Type Score Position
- Acceptor Loss (AL) 0.0 4 bp
- Donor Loss (DL) 0.18 5 bp
+ Acceptor Gain (AG) 0.0 138 bp
+ Donor Gain (DG) 0.04 465 bp
High impact (≥0.5) Medium impact (0.2-0.49) Low impact (<0.2)

VCEP Guidelines

Applied ACMG/AMP Criteria (VCEP Specific)

Filter Criteria:
PVS1

PVS1 (Very Strong)

According to VCEP guidelines, the rule for PVS1 is: "Very Strong Strength: Very Strong Per modified RUNX1 PVS1 decision tree for SNVs and CNVs and table of splicing effects." The evidence for this variant shows: NM_001754.4:c.474_489delTAATGACCTCAGGTTT (F158Lfs*13) is a frameshift truncating variant upstream of the last exon in RUNX1, a gene where loss of function is a known mechanism of disease. Therefore, this criterion is applied at Very Strong strength because it meets the VCEP PVS1 rule for a null variant in RUNX1.

PS1

PS1 (Not Applied)

According to standard ACMG guidelines, the rule for PS1 is: "Same amino acid change as a previously established pathogenic variant regardless of nucleotide change." The evidence for this variant shows: F158Lfs*13 is a novel frameshift, not a recurrent amino acid substitution. Therefore, PS1 is not applied.

PS2

PS2 (Not Applied)

According to standard ACMG guidelines, the rule for PS2 is: "De novo (both maternity and paternity confirmed) in a patient with the disease and no family history." The evidence for this variant shows: no de novo status or parental testing data available. Therefore, PS2 is not applied.

PS3

PS3 (Not Applied)

According to VCEP guidelines, the rule for PS3 is: "Strong Transactivation assays demonstrating altered transactivation (<20% of wt) AND data from a secondary assay demonstrating altered function. Not applicable if variant meets PVS1. If variant meets PVS1_strong, either apply PS3_moderate or upgrade to PVS1." The evidence for this variant shows: it meets PVS1. Therefore, PS3 is not applied.

PS4

PS4 (Not Applied)

According to VCEP guidelines, the rule for PS4 is: "Strong: ≥4 probands meeting at least one of the RUNX1-phenotypic criteria; Moderate: 2-3 probands; Supporting: 1 proband." The evidence for this variant shows: no case-level proband data reported. Therefore, PS4 is not applied.

PM1

PM1 (Not Applied)

According to VCEP guidelines, the rule for PM1 is: "Supporting Strength: Variant affecting one of the other amino acid residues 89-204 within the RHD (excluding key residues)." The evidence for this variant shows: it is a frameshift deletion, not a missense/in-frame alteration at a specific RHD residue. Therefore, PM1 is not applied.

PM2

PM2 (Supporting)

According to VCEP guidelines, the rule for PM2 is: "Supporting Strength: Variant must be completely absent from all population databases." The evidence for this variant shows: it is absent from gnomAD and other population datasets. Therefore, PM2 is applied at Supporting strength because the variant is absent from population databases.

PM3

PM3 (Not Applied)

According to standard ACMG guidelines, the rule for PM3 is: "For recessive disorders, detected in trans with a pathogenic variant." The evidence for this variant shows: no evidence of trans phase or recessive inheritance. Therefore, PM3 is not applied.

PM4

PM4 (Not Applied)

According to VCEP guidelines, the rule for PM4 is: "Moderate Strength: In-frame deletion/insertion impacting key RHD residues; Supporting Strength: in-frame deletion/insertion impacting other RHD residues." The evidence for this variant shows: it is a frameshift, not an in-frame indel. Therefore, PM4 is not applied.

PM5

PM5 (Supporting)

According to VCEP guidelines, the rule for PM5 is: "Supporting Strength: PM5_supporting is also applied to nonsense/frameshift variants that are downstream of c.98." The evidence for this variant shows: F158Lfs*13 is a frameshift truncation downstream of c.98. Therefore, PM5 is applied at Supporting strength.

PM6

PM6 (Not Applied)

According to standard ACMG guidelines, the rule for PM6 is: "Assumed de novo, but without confirmation of paternity and maternity." The evidence for this variant shows: no de novo data available. Therefore, PM6 is not applied.

PP1

PP1 (Not Applied)

According to VCEP guidelines, the rule for PP1 is: "Supporting Strength: 3-4 meioses co-segregation; Moderate: 5-6 meioses; Strong: ≥7 meioses." The evidence for this variant shows: no family segregation data. Therefore, PP1 is not applied.

PP2

PP2 (Not Applied)

According to standard ACMG guidelines, the rule for PP2 is: "Missense variant in a gene with low rate of benign missense variation and where missense is a common mechanism of disease." The evidence for this variant shows: it is a frameshift, not a missense. Therefore, PP2 is not applied.

PP3

PP3 (Not Applied)

According to VCEP guidelines, the rule for PP3 is: "Supporting Strength: For missense variants, REVEL ≥0.88; for splice, SpliceAI ≥0.38." The evidence for this variant shows: it is a frameshift, and in silico splicing impact is below threshold. Therefore, PP3 is not applied.

PP4

PP4 (Not Applied)

According to standard ACMG guidelines, the rule for PP4 is: "Patient’s phenotype or family history is highly specific for a disease with a single genetic etiology." The evidence for this variant shows: no detailed phenotypic specificity provided. Therefore, PP4 is not applied.

PP5

PP5 (Not Applied)

According to standard ACMG guidelines, the rule for PP5 is: "Reputable source reports variant as pathogenic without evidence available." The evidence for this variant shows: not reported in ClinVar or other databases. Therefore, PP5 is not applied.

BA1

BA1 (Not Applied)

According to VCEP guidelines, the rule for BA1 is: "Stand Alone Strength: Minor allele frequency ≥0.15%." The evidence for this variant shows: MAF = 0%. Therefore, BA1 is not applied.

BS1

BS1 (Not Applied)

According to VCEP guidelines, the rule for BS1 is: "Strong Strength: MAF between 0.015% and 0.15%." The evidence for this variant shows: absent from population databases. Therefore, BS1 is not applied.

BS2

BS2 (Not Applied)

According to standard ACMG guidelines, the rule for BS2 is: "Observed in a healthy adult individual for a fully penetrant dominant disorder." The evidence for this variant shows: no healthy individuals reported. Therefore, BS2 is not applied.

BS3

BS3 (Not Applied)

According to VCEP guidelines, the rule for BS3 is: "Strong Strength: Transactivation assays demonstrating normal function; Supporting Strength: normal transactivation only." The evidence for this variant shows: functional data indicate loss of function. Therefore, BS3 is not applied.

BS4

BS4 (Not Applied)

According to standard ACMG guidelines, the rule for BS4 is: "Lack of segregation in affected members of a family." The evidence for this variant shows: no segregation data available. Therefore, BS4 is not applied.

BP1

BP1 (Not Applied)

According to standard ACMG guidelines, the rule for BP1 is: "Missense variant in a gene where only truncating variants cause disease." The evidence for this variant shows: it is truncating. Therefore, BP1 is not applied.

BP2

BP2 (Not Applied)

According to standard ACMG guidelines, the rule for BP2 is: "Observed in trans with a pathogenic variant for a dominant gene or in cis with a pathogenic variant." The evidence for this variant shows: no such data. Therefore, BP2 is not applied.

BP3

BP3 (Not Applied)

According to standard ACMG guidelines, the rule for BP3 is: "In-frame indel in a repetitive region without a known function." The evidence for this variant shows: it is a frameshift not in a repetitive region. Therefore, BP3 is not applied.

BP4

BP4 (Not Applied)

According to VCEP guidelines, the rule for BP4 is: "Supporting Strength: REVEL <0.50 and SpliceAI ≤0.20 for missense/synonymous/intronic." The evidence for this variant shows: it is a frameshift. Therefore, BP4 is not applied.

BP5

BP5 (Not Applied)

According to standard ACMG guidelines, the rule for BP5 is: "Variant found in a case with an alternate molecular basis for disease." The evidence for this variant shows: no alternate molecular basis reported. Therefore, BP5 is not applied.

BP6

BP6 (Not Applied)

According to standard ACMG guidelines, the rule for BP6 is: "Reputable source reports variant as benign without evidence." The evidence for this variant shows: no such report. Therefore, BP6 is not applied.

BP7

BP7 (Not Applied)

According to VCEP guidelines, the rule for BP7 is: "Supporting Strength: Synonymous or intronic variant with SpliceAI ≤0.20 and phyloP ≤2.0." The evidence for this variant shows: it is a frameshift. Therefore, BP7 is not applied.