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.964dup
Protein Change
S322Ffs*278
Location
Exon 8 (Exon 8 of )
8
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
COSM8341346
Recurrence
1 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 S322Ffs*278 variant is a truncating mutation in the RUNX1 gene, a known tumor suppressor involved in hematopoietic differentiation. Functional evidence indicates that truncating mutations in RUNX1 can inhibit its function, contributing to oncogenesis and predisposing to hematologic malignancies such as leukemia, lymphomas, and myelodysplastic syndromes.

Database Previews
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 None bp
- Donor Loss (DL) 0.0 None bp
+ Acceptor Gain (AG) 0.0 None bp
+ Donor Gain (DG) 0.0 None 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 Per modified RUNX1 PVS1 decision tree for SNVs and CNVs and table of splicing effects." The evidence for this variant shows: c.964dupT causes a frameshift leading to a premature stop codon (S322Ffs*278), a null variant in RUNX1 not in the last exon, where loss‐of‐function is a known disease mechanism. Therefore, this criterion is applied at Very Strong strength because it is a truncating variant meeting the VCEP PVS1 decision tree.

PS1

PS1 (Not Applied)

According to VCEP guidelines, PS1 applies when the same amino acid change as a previously established pathogenic variant occurs. The evidence for this variant shows: S322Ffs*278 is a novel frameshift, not a recurrence of a known pathogenic amino acid change. Therefore, this criterion is not applied.

PS2

PS2 (Not Applied)

According to VCEP guidelines, PS2 (de novo) requires proven or assumed de novo occurrence. The evidence for this variant shows: parental origin data are unavailable. Therefore, this criterion is not applied.

PS3

PS3 (Not Applied)

According to VCEP guidelines, PS3 is not applicable if a variant meets PVS1. The evidence for this variant shows: PVS1 has been applied at Very Strong. Therefore, PS3 is not applied.

PS4

PS4 (Not Applied)

According to VCEP guidelines, PS4 requires ≥4 probands (Strong), 2–3 probands (Moderate), or 1 proband (Supporting) meeting RUNX1-phenotypic criteria. The evidence for this variant shows: no proband data reported. Therefore, this criterion is not applied.

PM1

PM1 (Not Applied)

According to VCEP guidelines, PM1 applies to variants affecting specific residues within the RHD. The evidence for this variant shows: S322Ffs*278 lies outside the RHD (residues 50–182). Therefore, this criterion is not applied.

PM2

PM2 (Supporting)

According to VCEP guidelines, the rule for PM2 is: "Supporting Variant must be completely absent from all population databases." The evidence for this variant shows: c.964dupT is absent from gnomAD and other population databases. Therefore, this criterion is applied at Supporting strength because the variant is completely absent.

PM3

PM3 (Not Applied)

According to standard ACMG guidelines, PM3 applies when a variant occurs in trans with a pathogenic variant for a recessive disorder. The evidence for this variant shows: no data on trans observations or recessive context. Therefore, this criterion is not applied.

PM4

PM4 (Not Applied)

According to VCEP guidelines, PM4 applies to in-frame indels in the RHD. The evidence for this variant shows: c.964dupT is a frameshift, not an in-frame indel. Therefore, this criterion is not applied.

PM5

PM5 (Supporting)

According to VCEP guidelines, the rule for PM5 is: "PM5_supporting is also applied to nonsense/frameshift variants that are downstream of c.98 (in transcript NM_001754.4)." The evidence for this variant shows: c.964dupT is downstream of c.98 and causes a frameshift. Therefore, this criterion is applied at Supporting strength because it meets the VCEP PM5_supporting rule.

PM6

PM6 (Not Applied)

According to VCEP guidelines, PM6 (assumed de novo) requires de novo occurrence data. The evidence for this variant shows: no parental data available. Therefore, this criterion is not applied.

PP1

PP1 (Not Applied)

According to VCEP guidelines, PP1 requires co-segregation with disease in families. The evidence for this variant shows: no segregation data. Therefore, this criterion is not applied.

PP2

PP2 (Not Applied)

According to standard ACMG guidelines, PP2 applies to missense variants in genes with low benign missense variation. The evidence for this variant shows: it is a frameshift, not a missense. Therefore, this criterion is not applied.

PP3

PP3 (Not Applied)

According to VCEP guidelines, PP3 applies to computational evidence for missense or splice-impact. The evidence for this variant shows: it is a truncating frameshift, not applicable for computational missense/splice prediction. Therefore, this criterion is not applied.

PP4

PP4 (Not Applied)

According to standard ACMG guidelines, PP4 requires a highly specific phenotype. The evidence for this variant shows: detailed clinical phenotype data are not provided. Therefore, this criterion is not applied.

PP5

PP5 (Not Applied)

According to standard ACMG guidelines, PP5 applies when a reputable source reports the variant as pathogenic. The evidence for this variant shows: no ClinVar or similar submission. Therefore, this criterion is not applied.

BA1

BA1 (Not Applied)

According to standard ACMG guidelines, BA1 requires MAF ≥0.15% in any population. The evidence for this variant shows: absent from population databases. Therefore, this criterion is not applied.

BS1

BS1 (Not Applied)

According to standard ACMG guidelines, BS1 requires MAF between 0.015% and 0.15%. The evidence for this variant shows: absent from databases. Therefore, this criterion is not applied.

BS2

BS2 (Not Applied)

According to standard ACMG guidelines, BS2 applies when variant observed in healthy adults. The evidence for this variant shows: no such observations. Therefore, this criterion is not applied.

BS3

BS3 (Not Applied)

According to VCEP guidelines, BS3 requires normal functional assay results. The evidence for this variant shows: functional studies demonstrate damaging effect. Therefore, this criterion is not applied.

BS4

BS4 (Not Applied)

According to standard ACMG guidelines, BS4 requires lack of segregation in affected members. The evidence for this variant shows: no segregation data. Therefore, this criterion is not applied.

BP1

BP1 (Not Applied)

According to standard ACMG guidelines, BP1 applies to missense in genes where only LOF is disease mechanism. The evidence for this variant shows: it is LOF. Therefore, this criterion is not applied.

BP2

BP2 (Not Applied)

According to standard ACMG guidelines, BP2 applies when observed in trans with a pathogenic variant. The evidence for this variant shows: no such data. Therefore, this criterion is not applied.

BP3

BP3 (Not Applied)

According to standard ACMG guidelines, BP3 applies to in-frame indels in repetitive regions. The evidence for this variant shows: frameshift, not in repetitive region. Therefore, this criterion is not applied.

BP4

BP4 (Not Applied)

According to VCEP guidelines, BP4 requires computational evidence of benign impact. The evidence for this variant shows: frameshift causing LOF. Therefore, this criterion is not applied.

BP5

BP5 (Not Applied)

According to standard ACMG guidelines, BP5 applies when variant found in a case with an alternate molecular cause. The evidence for this variant shows: no alternate cause reported. Therefore, this criterion is not applied.

BP6

BP6 (Not Applied)

According to standard ACMG guidelines, BP6 applies when a reputable source reports variant as benign. The evidence for this variant shows: no such reports. Therefore, this criterion is not applied.

BP7

BP7 (Not Applied)

According to VCEP guidelines, BP7 applies to synonymous or intronic variants with no splicing impact. The evidence for this variant shows: it is a frameshift, not synonymous. Therefore, this criterion is not applied.