Genetic Information

Gene & Transcript Details

Gene
DNMT3A
Transcript
NM_022552.4 MANE Select
Total Exons
Reference Sequence
NC_000002.11
Alternative Transcripts
IDStatusDetails
NM_022552.4 RefSeq Select 4324 nt | 268–3006
NM_022552.5 MANE Select 9421 nt | 278–3016
NM_022552.3 Alternative 4314 nt | 258–2996

Variant Details

HGVS Notation
NM_022552.4:c.2536del
Protein Change
Q846Sfs*7
Location
Exon 22 (Exon 22 of )
22
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 DNMT3A.

Functional Studies & Therapeutic Relevance

Functional Summary

The DNMT3A Q846Sfs*7 variant is a truncating mutation in the DNMT3A gene, a known tumor suppressor and DNA methyltransferase. Functional evidence indicates that such truncating mutations in DNMT3A are likely to lead to nonsense-mediated decay and haploinsufficiency. This results in disturbed methylation patterns and global hypomethylation, which can affect transcription of lineage-specific programs in myeloid development. Therefore, the functional evidence supports a damaging effect of this variant.

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) 0.05 53 bp
- Donor Loss (DL) 0.02 -60 bp
+ Acceptor Gain (AG) 0.0 48 bp
+ Donor Gain (DG) 0.0 -266 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 standard ACMG guidelines, the rule for PVS1 is: "Null variant in a gene where loss of function (LoF) is a known mechanism of disease (e.g., nonsense, frameshift, canonical ±1 or 2 splice sites, initiation codon, single or multi-exon deletion in a LoF gene)". The evidence for this variant shows: it is a deletion causing a frameshift and premature stop (Q846Sfs*7) predicted to result in loss of function via nonsense-mediated decay. Therefore, this criterion is applied at Very Strong strength because this truncating variant is expected to cause LoF in DNMT3A, a gene with established LOF disease mechanism.

PS1

PS1 (Not Applied)

According to standard ACMG guidelines, the rule for PS1 is: "Same amino acid change as a known pathogenic variant but different nucleotide change". The evidence for this variant shows: it results in a frameshift rather than an amino acid substitution matching a known pathogenic variant. Therefore, this criterion is not applied at Not Applied strength because the variant does not produce the same amino acid change as any known pathogenic variant.

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 information on de novo status or parental testing is available. Therefore, this criterion is not applied at Not Applied strength because de novo status cannot be evaluated without parental confirmation.

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 indicate that truncating mutations in DNMT3A lead to haploinsufficiency and disrupted DNA methylation patterns. Therefore, this criterion is applied at Strong strength because multiple well-established functional assays support a damaging effect of this truncating variant.

PS4

PS4 (Not Applied)

According to standard ACMG guidelines, the rule for PS4 is: "Prevalence in affected individuals significantly increased compared with controls". The evidence for this variant shows: no case-control or prevalence data are available. Therefore, this criterion is not applied at Not Applied strength because prevalence in affected individuals cannot be assessed.

PM1

PM1 (Not Applied)

According to standard ACMG guidelines, the rule for PM1 is: "Located in a mutational hot spot or well-established functional domain without benign variation". The evidence for this variant shows: no data indicating that c.2536 lies within a known mutational hotspot or critical domain. Therefore, this criterion is not applied at Not Applied strength.

PM2

PM2 (Moderate)

According to standard ACMG guidelines, the rule for PM2 is: "Absent from controls (or at extremely low frequency if recessive)". The evidence for this variant shows: it is absent from population databases including gnomAD. Therefore, this criterion is applied at Moderate strength because the variant is not observed in large control cohorts.

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: zygosity and phasing information are unavailable. Therefore, this criterion is not applied at Not Applied strength because trans configuration cannot be determined.

PM4

PM4 (Not Applied)

According to standard ACMG guidelines, the rule for PM4 is: "Protein length changes due to in-frame deletions/insertions or stop-loss variants". The evidence for this variant shows: it causes a frameshift and premature truncation rather than an in-frame change. Therefore, this criterion is not applied at Not Applied strength.

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". The evidence for this variant shows: it is not a missense variant. Therefore, this criterion is not applied at Not Applied 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: there is no data on de novo occurrence. Therefore, this criterion is not applied at Not Applied strength.

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: family segregation data are not provided. Therefore, this criterion is not applied at Not Applied strength.

PP2

PP2 (Not Applied)

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

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/gene product (e.g., conservation, splicing impact)". The evidence for this variant shows: computational tools including SpliceAI (max score 0.05) predict minimal splicing impact and no deleterious signals. Therefore, this criterion is not applied at Not Applied strength.

PP4

PP4 (Not Applied)

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

PP5

PP5 (Not Applied)

According to standard ACMG guidelines, the rule for PP5 is: "Reputable source reports variant as pathogenic, but without accessible evidence". The evidence for this variant shows: it is not reported in ClinVar or other reputable sources. Therefore, this criterion is not applied at Not Applied strength.

BA1

BA1 (Not Applied)

According to standard ACMG guidelines, the rule for BA1 is: "Allele frequency is too high for the disorder (based on population data)". The evidence for this variant shows: it is absent from population databases. Therefore, this criterion is not applied at Not Applied strength.

BS1

BS1 (Not Applied)

According to standard ACMG guidelines, the rule for BS1 is: "Allele frequency is greater than expected for the disorder". The evidence for this variant shows: it is absent from population databases. Therefore, this criterion is not applied at Not Applied strength.

BS2

BS2 (Not Applied)

According to standard ACMG guidelines, the rule for BS2 is: "Observed in healthy individuals with full penetrance expected at an early age". The evidence for this variant shows: there are no reports in healthy cohorts. Therefore, this criterion is not applied at Not Applied strength.

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 data support a damaging effect. Therefore, this criterion is not applied at Not Applied strength.

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: segregation information is unavailable. Therefore, this criterion is not applied at Not Applied strength.

BP1

BP1 (Not Applied)

According to standard ACMG guidelines, the rule for BP1 is: "Missense variant in a gene where only LoF causes disease". The evidence for this variant shows: it is a frameshift variant. Therefore, this criterion is not applied at Not Applied strength.

BP2

BP2 (Not Applied)

According to standard ACMG guidelines, the rule for BP2 is: "Observed in trans with a pathogenic variant for a dominantly inherited disorder or in cis with a pathogenic variant". The evidence for this variant shows: phasing data are not available. Therefore, this criterion is not applied at Not Applied strength.

BP3

BP3 (Not Applied)

According to standard ACMG guidelines, the rule for BP3 is: "In-frame deletions/insertions in a repetitive region without known function". The evidence for this variant shows: it is a frameshift deletion not located in a repetitive region. Therefore, this criterion is not applied at Not Applied strength.

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 (conservation, splicing, etc)". The evidence for this variant shows: SpliceAI predicts minimal splice impact (max score 0.05) and other in silico tools do not indicate deleterious effect. Therefore, this criterion is applied at Supporting strength because computational data suggest no significant splicing or functional 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 alternate molecular diagnoses are reported. Therefore, this criterion is not applied at Not Applied strength.

BP6

BP6 (Not Applied)

According to standard ACMG guidelines, the rule for BP6 is: "Reputable source reports variant as benign, but without accessible evidence". The evidence for this variant shows: no benign reports are available. Therefore, this criterion is not applied at Not Applied strength.

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 synonymous. Therefore, this criterion is not applied at Not Applied strength.