Genetic Information

Gene & Transcript Details

Gene
FLT3
Transcript
NM_004119.2 MANE Select
Total Exons
Reference Sequence
NC_000013.10
Alternative Transcripts
IDStatusDetails
NM_004119.3 MANE Select 3826 nt | 67–3048
NM_004119.2 RefSeq Select 3848 nt | 83–3064
NM_004119.1 Alternative 3475 nt | 58–3039

Variant Details

HGVS Notation
NM_004119.2:c.1794_1795insACCATTGATTTCAGAGAATATGAA
Protein Change
E598_Y599insTIDFREYE
Location
Exon 14 (Exon 14 of )
14
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 FLT3.

Functional Studies & Therapeutic Relevance

Functional Summary

The FLT3 E598_Y599insTIDFREYE variant has been functionally characterized as activating. It is located in the juxtamembrane domain of the FLT3 protein and has been shown to increase growth factor-independent proliferation and induce a myeloproliferative phenotype in various cell lines and an in vivo bone marrow transplant model, indicating a damaging effect.

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) 0.06 131 bp
- Donor Loss (DL) 0.02 165 bp
+ Acceptor Gain (AG) 0.04 10 bp
+ Donor Gain (DG) 0.0 -37 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 (Not Applied)

According to standard ACMG guidelines, the rule for PVS1 is: 'Null variant (nonsense, frameshift, canonical ±1 or 2 splice sites, initiation codon, single exon or multiexon deletion) in a gene where loss of function is a known mechanism of disease.' The evidence for this variant shows: it is an in‐frame insertion, not predicted to result in loss of function. Therefore, this criterion is not applied.

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: no known variant produces the identical E598_Y599insTIDFREYE amino acid change. Therefore, this criterion 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 parental testing or segregation data are available. Therefore, this criterion is not applied.

PS3

PS3 (Strong)

According to standard ACMG guidelines, the rule for PS3 is: 'Well-established in vitro or in vivo functional studies supportive of a damaging effect on the gene or gene product.' The evidence for this variant shows: functional studies demonstrate that E598_Y599insTIDFREYE increases growth factor-independent proliferation and induces a myeloproliferative phenotype. Therefore, this criterion is applied at Strong strength because well-established assays show a damaging activating effect.

PS4

PS4 (Not Applied)

According to standard ACMG guidelines, the rule for PS4 is: 'Prevalence of the variant in affected individuals is significantly increased compared with controls.' The evidence for this variant shows: no case–control or large cohort data are available. Therefore, this criterion is not applied.

PM1

PM1 (Moderate)

According to standard ACMG guidelines, the rule for PM1 is: 'Located in a mutational hot spot and/or critical and well-established functional domain without benign variation.' The evidence for this variant shows: the insertion lies within the juxtamembrane domain of FLT3, a known hotspot for activating insertions. Therefore, this criterion is applied at Moderate strength because the variant precisely falls in a functional hot spot.

PM2

PM2 (Moderate)

According to standard ACMG guidelines, the rule for PM2 is: 'Absent from controls (or at extremely low frequency if recessive) in population databases.' The evidence for this variant shows: it is not found in gnomAD or other population databases (MAF = 0%). Therefore, this criterion is applied at Moderate strength because the variant is absent from controls.

PM3

PM3 (Not Applied)

According to standard ACMG guidelines, the rule for PM3 is: 'Detected in trans with a pathogenic variant for recessive disorders.' The evidence for this variant shows: no data on trans configuration in a recessive context. Therefore, this criterion is not applied.

PM4

PM4 (Moderate)

According to standard ACMG guidelines, the rule for PM4 is: 'Protein length changes due to in-frame deletions/insertions in a non-repeat region or stop-loss variants.' The evidence for this variant shows: it is an in-frame insertion of eight amino acids in a non-repetitive region. Therefore, this criterion is applied at Moderate strength because the insertion alters protein length.

PM5

PM5 (Not Applied)

According to standard ACMG guidelines, the rule for PM5 is: 'Novel missense change at an amino acid residue where a different pathogenic missense change has been seen before.' The evidence for this variant shows: it is not a missense change and no pathogenic missense variants at these positions have been reported. Therefore, this criterion is not applied.

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 parental information is available. Therefore, this criterion is not applied.

PP1

PP1 (Not Applied)

According to standard ACMG guidelines, the rule for PP1 is: 'Co‐segregation with disease in multiple affected family members.' The evidence for this variant shows: no family segregation data. Therefore, this criterion is not applied.

PP2

PP2 (Not Applied)

According to standard ACMG guidelines, the rule for PP2 is: 'Missense variant in a gene that has a low rate of benign missense variation and in which missense variants are a common mechanism of disease.' The evidence for this variant shows: it is an in-frame insertion, not a missense change. Therefore, this criterion is not applied.

PP3

PP3 (Not Applied)

According to standard ACMG guidelines, the rule for PP3 is: 'Multiple lines of computational evidence support a deleterious effect on the gene or gene product.' The evidence for this variant shows: in silico tools including SpliceAI predict no impact. Therefore, this criterion 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 phenotype or family history data provided. Therefore, this criterion is not applied.

PP5

PP5 (Not Applied)

According to standard ACMG guidelines, the rule for PP5 is: 'Reputable source reports the variant as pathogenic, but the evidence is not available to the laboratory.' The evidence for this variant shows: not present in ClinVar or other reputable variant databases. Therefore, this criterion is not applied.

BA1

BA1 (Not Applied)

According to standard ACMG guidelines, the rule for BA1 is: 'Allele frequency is greater than expected for disorder.' The evidence for this variant shows: allele frequency is zero. Therefore, this criterion is not applied.

BS1

BS1 (Not Applied)

According to standard ACMG guidelines, the rule for BS1 is: 'Allele frequency is greater than expected for disorder.' The evidence for this variant shows: allele frequency is zero. Therefore, this criterion is not applied.

BS2

BS2 (Not Applied)

According to standard ACMG guidelines, the rule for BS2 is: 'Observed in healthy adult individuals for a recessive (homozygous) or dominant (heterozygous) disorder when full penetrance is expected at an early age.' The evidence for this variant shows: no observations in healthy individuals. Therefore, this criterion is not applied.

BS3

BS3 (Not Applied)

According to standard ACMG guidelines, the rule for BS3 is: 'Well-established functional studies show no damaging effect on protein function or splicing.' The evidence for this variant shows: functional studies demonstrate a damaging activating effect. Therefore, this criterion is not applied.

BS4

BS4 (Not Applied)

According to standard ACMG guidelines, the rule for BS4 is: 'Lack of segregation in affected family 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, the rule for BP1 is: 'Missense variant in a gene for which only loss of function causes disease.' The evidence for this variant shows: it is an in-frame insertion, not missense. Therefore, this criterion 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 disorder or in cis with a pathogenic variant.' The evidence for this variant shows: no data on cis/trans configuration. Therefore, this criterion is not applied.

BP3

BP3 (Not Applied)

According to standard ACMG guidelines, the rule for BP3 is: 'In-frame deletions/insertions in a repetitive region without a known function.' The evidence for this variant shows: the insertion is not in a repetitive region. Therefore, this criterion is not applied.

BP4

BP4 (Supporting)

According to standard ACMG guidelines, the rule for BP4 is: 'Multiple lines of computational evidence suggest no impact on gene or gene product.' The evidence for this variant shows: in silico predictions including SpliceAI show minimal impact on splicing and no predicted deleterious effect. Therefore, this criterion is applied at Supporting strength because computational evidence indicates likely benign impact.

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 such alternate molecular basis reported. Therefore, this criterion is not applied.

BP6

BP6 (Not Applied)

According to standard ACMG guidelines, the rule for BP6 is: 'Reputable source reports variant as benign, but the evidence is not available.' The evidence for this variant shows: no such report. Therefore, this criterion is not applied.

BP7

BP7 (Not Applied)

According to standard ACMG guidelines, the rule for BP7 is: 'Synonymous variant with no predicted impact on splicing.' The evidence for this variant shows: it is not a synonymous change. Therefore, this criterion is not applied.