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Variant: NM_000138.5(FBN1):c.5747G>A (p.Cys1916Tyr)

CA016026

42391 (ClinVar)

Gene: FBN1
Condition: Marfan syndrome
Inheritance Mode: Autosomal dominant inheritance
UUID: c5663969-cd67-40de-80f4-b96e8399345c
Approved on: 2024-02-22
Published on: 2024-02-22

HGVS expressions

NM_000138.5:c.5747G>A
NM_000138.5(FBN1):c.5747G>A (p.Cys1916Tyr)
NC_000015.10:g.48446747C>T
CM000677.2:g.48446747C>T
NC_000015.9:g.48738944C>T
CM000677.1:g.48738944C>T
NC_000015.8:g.46526236C>T
NG_008805.2:g.204042G>A
ENST00000559133.6:c.5747G>A
ENST00000674301.2:c.5747G>A
ENST00000684448.1:n.4421G>A
ENST00000316623.10:c.5747G>A
ENST00000674301.1:c.746G>A
ENST00000316623.9:c.5747G>A
ENST00000537463.6:c.*1510G>A
ENST00000559133.5:c.1054G>A
NM_000138.4:c.5747G>A
More

Likely Pathogenic

Met criteria codes 4
PP3 PP2 PM2_Supporting PM1_Strong
Not Met criteria codes 1
PM5

Evidence Links 0

Expert Panel

Criteria Specification Information

Criteria Specifications for this VCEP
Evidence submitted by expert panel
FBN1 VCEP
The NM_00138 c.5747G>A, is a missense variant in FBN1predicted to cause a substitution of a cysteine by tyrosine at amino acid 1916 (p.Cys1916Tyr). This variant was found in a proband diagnosed with Marfan syndrome (PMID 24793577, PP4). This variant has been reported three times in ClinVar: once as pathogenic, once as likely pathogenic, and once as uncertain significance (Variation ID: 42391). This variant is not present in gnomAD(PM2_sup; https://gnomad.broadinstitute.org/ v2.1.1). This variant affects a cysteine residue in a calcium binding EGF domain. Cysteine residues are believed to be involved in the formation of disulfide bridges which are essential for the protein structure (PM1_strong). Computational prediction tools and conservation analysis suggest that this variant may impact the protein (REVEL: 0.967, PP3). The constraint z-score for missense variants affecting FBN1 is 5.06 (PP2). In summary, this variant meets criteria to be classified as likely pathogenic for Marfan syndrome based on the ACMG/AMP criteria applied, as specified by the ClinGen FBN1 VCEP: PM1_Strong, PM2_Sup, PP2, PP3, PP4.
Met criteria codes
PP3
REVEL: 0.967
PP2
The constraint z-score for missense variants affecting FBN1 is 5.06
PM2_Supporting
Absent in gnomAD
PM1_Strong
Cys-disrupting residue in cbEGF-like domain
Not Met criteria codes
PM5
No code specific comments provided, please refer to the summary above or general recommendations provided in the guideline
Curation History
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