Genetic Information

Gene & Transcript Details

Gene
MAP2K1
Transcript
NM_002755.4 MANE Select
Total Exons
Reference Sequence
NC_000015.9
Alternative Transcripts
IDStatusDetails
NM_002755.4 MANE Select 2547 nt | 437–1618
NM_002755.3 RefSeq Select 2603 nt | 476–1657

Variant Details

HGVS Notation
NM_002755.4:c.158T>C
Protein Change
F53S
Location
Exon 2 (Exon 2 of )
2
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
Likely Pathogenic
1 publications
Publications List
PMID: 18413255

Variant summary: MAP2K1 c.158T>C (p.Phe53Ser) results in a non-conservative amino acid change in the encoded protein sequence. Four of five in-silico tools predict a damaging effect of the variant on protein function. The variant was absent in 251374 control chromosomes. c.158T>C has been reported in the literature in individuals affected with Cardiofaciocutaneous Syndrome (example, Rodrigues-Viciana_2006, Yoon_2007, Siegel_2011, Bessis_2019). These data indicate that the variant is likely to be associated with disease. At least one publication reports experimental evidence evaluating an impact on protein function. The most pronounced variant effect results in significant stimulation of ERK phosphorylation relative to the wild-type control (example, Rodriguez-Viciana_2008). One clinical diagnostic laboratory and an expert panel (ClinGen RASopathy Variant Curation Expert Panel) have submitted clinical-significance assessments for this variant to ClinVar after 2014 without evidence for independent evaluation. Both submitters classified the variant as pathogenic. Based on the evidence outlined above, the variant was classified as pathogenic.

Clinical Statement

"This variant has been reported in ClinVar as Pathogenic (2 clinical laboratories) and as Likely Pathogenic by ClinGen RASopathy Variant Curation Expert Panel expert panel."

COSMIC Somatic Evidence

Open
COSMIC ID
COSM9868057
Recurrence
2 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 MAP2K1.

Functional Studies & Therapeutic Relevance

Functional Summary

Error in OpenAI Consolidation. OncoKB: MAP2K1F53SMAP2K1F53SSomaticNCBI Gene:5604|Show additional gene information Variant OverviewMAP2K1, an intracellular kinase, is mutated at low frequencies in various cancer types including melanoma, colorectal and lung cancers.The MAP2K1 F53S mutation is likely oncogenic.Hide mutation effect description The MEK1 F53S mutation is located in the negative regulatory region of the protein and occurs as a result of a mutation in exon 2 of the MAP2K1 gene. Expression of the MEK1 F53S mutation in 293T and HEK293 cells demonstrated that it is activating as measured by increased downstream pathway activation associated with mutant MEK1 compared to wildtype MEK1 that is dependent on upstream activation by BRAF (PMID: 16439621, 17981815). JAX-CKB: MAP2K1 F53S lies within the negative regulatory region of the Map2k1 protein (PMID: 24241536). F53S demonstrates phosphorylation profile similar to wild-type Map2k1 (PMID: 29753091), and results in increased Erk phosphorylation in cell culture (PMID: 16439621, PMID: 12370306).

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
Mixed/VUS
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 11 bp
- Donor Loss (DL) 0.0 155 bp
+ Acceptor Gain (AG) 0.0 -141 bp
+ Donor Gain (DG) 0.0 19 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:
PS3

PS3 (Unknown (Pre-LLM))

From pre-LLM assessment (LLM Failed)

PM2

PM2 (Unknown (Pre-LLM))

From pre-LLM assessment (LLM Failed)

PP5

PP5 (Unknown (Pre-LLM))

From pre-LLM assessment (LLM Failed)

BP4

BP4 (Unknown (Pre-LLM))

From pre-LLM assessment (LLM Failed)