NM_000900.5:c.87T>A

Variant summary

Our verdict is Pathogenic.
+11 Pathogenic
-7
-6
-1
0
+5
+6
+9
+10
B
LB
VUS
LP
P
The variant received 11 classification points (ACMG Germline Pathogenicity v2019). PVS1PM2PP5

The NM_000900.5(MGP):c.87T>A (p.Tyr29*) variant causes a stop gained change involving the alteration of a non-conserved nucleotide. The variant is predicted to lead to nonsense-mediated mRNA decay (NMD). The variant is absent from the gnomAD population database at sites with sufficient sequencing coverage. In-silico predictor (REVEL) classifies this variant as likely benign. Splicing prediction tools (SpliceAI) predict no significant impact on normal splicing. Variant has been reported in ClinVar as Pathogenic/Likely Pathogenic (no review stars).

Frequency

Genomes: not found (cov: 32)
Exomes 𝑓: 0.0 ( 0 hom. )
Failed GnomAD Quality Control

Consequence

MGP
NM_000900.5 stop_gained

Scores

3
1
3

Clinical Significance

Pathogenic no assertion criteria provided P:1

Conservation

PhyloP100: 1.16

Publications

0 publications found
Variant links:
Genes affected
MGP (HGNC:7060): (matrix Gla protein) This gene encodes a member of the osteocalcin/matrix Gla family of proteins. The encoded vitamin K-dependent protein is secreted by chondrocytes and vascular smooth muscle cells, and functions as a physiological inhibitor of ectopic tissue calcification. Carboxylation status of the encoded protein is associated with calcification of the vasculature in human patients with cardiovascular disease and calcification of the synovial membranes in osteoarthritis patients. Mutations in this gene cause Keutel syndrome in human patients, which is characterized by abnormal cartilage calcification, peripheral pulmonary stenosis and facial hypoplasia. [provided by RefSeq, Sep 2016]
MGP Gene-Disease associations (from GenCC):
  • Keutel syndrome
    Inheritance: AR Classification: DEFINITIVE, STRONG, SUPPORTIVE Submitted by: ClinGen, G2P, PanelApp Australia, Orphanet, Ambry Genetics, Labcorp Genetics (formerly Invitae)
  • skeletal dysplasia
    Inheritance: AD Classification: STRONG Submitted by: PanelApp Australia

Genome browser will be placed here

new If you want to explore the variant's impact on the transcript NM_000900.5, check out the Mutation Effect Viewer. This is especially useful for frameshift variants or if you want to visualize the effect of exon loss / intron retention.

Classification according to ACMG Germline Pathogenicity v2019

Classification was made for transcript:

Our verdict: Pathogenic. The variant received 11 points.

PVS1Null variant in LOF gene
Frameshift/stop-gained, NMD predicted, LoF disease mechanism — very strong (PVS1); Stop-gained (exon 2 of 4) at amino acid 29 of 103, predicted to trigger nonsense-mediated mRNA decay. Loss of function is a known disease mechanism for this gene.
PM2Absent/extremely low in population databases
Absent from gnomAD (AD/XL gene) — PM2; Absent from gnomAD at well-covered site (MOI: AD+AR) (threshold 0.0001) — PM2 moderate.
PP5Reputable source reports pathogenic
ClinVar 0-star pathogenic — supporting (PP5); ClinVar germline classification: Pathogenic/Likely Pathogenic, 0 star(s).

Variant Effect in Transcripts

Automated classification analysis was done for transcript: NM_000900.5. You can select a different transcript below to see updated classification assignments.

RefSeq Transcripts

MGP NM_000900.5
MANE Select
HGVSc / HGVSpc.87T>Ap.Tyr29*
Exon rankExon 2 of 4
stop_gained
Prot: NP_000891.2
MGP NM_001190839.3
HGVSc / HGVSpc.162T>Ap.Tyr54*
Exon rankExon 3 of 5
stop_gained
Prot: NP_001177768.1UniProt: P08493-2

Ensembl Transcripts

MGP ENST00000539261.6
TSL:1MANE Select
HGVSc / HGVSpc.87T>Ap.Tyr29*
Exon rankExon 2 of 4
stop_gained
Prot: ENSP00000445907.1UniProt: P08493-1
MGP ENST00000507170.2
TSL:1
HGVSc / HGVSpn.150T>A
Exon rankExon 2 of 2
non_coding_transcript_exon
Prot:
MGP ENST00000228938.5
TSL:3
HGVSc / HGVSpc.162T>Ap.Tyr54*
Exon rankExon 3 of 5
stop_gained
Prot: ENSP00000228938.5UniProt: P08493-2

Frequencies

Allele frequencies (AF), counts (AC/AN), homozygotes and coverage

Common (AF > 0.05 / Hom > 5)
Rare (AF ≤ 0.0001 / Hom ≤ 1)
Global population databases 3 sources
GnomAD3 genomes
Cov:32
GnomAD4 exome Warning
AF: 0.00
AC:0
Hom:0
AN:1308592
Cov:24
GnomAD4 genome
Cov:32
Showing 3 sources

ClinVar

ClinVar submissions
Significance:Pathogenic
Revision:no assertion criteria provided
View on ClinVar
Pathogenic
VUS
Benign
Condition
1
-
-
Keutel syndrome (1)

Computational Scores

AlphaGenome AVI
Pathogenic32
BayesDel_addAF
Pathogenic0.60D
BayesDel_noAF
Pathogenic0.55
CADD
Pathogenic35
DANN
Uncertain0.98
Eigen
Benign-0.050
Eigen_PC
Benign-0.25
FATHMM_MKL
Benign0.73D
GPN-Star LLR
N/A-6.2
GPN-Star score
N/A6.2
Mutation Taster
N/A12/188disease causing (ClinVar)
PhyloP100
Benign1.2
PromoterAI
N/A0.016Neutral
Showing 13 of 13 scores

Splicing Scores

Pangolin (max)
Benign0.020
SpliceAI score (max)
Benign0.010
Details are displayed if max score is > 0.2
Splicevardb
N/A2.0
Showing 3 of 3 scores

Find out detailed SpliceAI scores and Pangolin per-transcript scores at spliceailookup.broadinstitute.org

MaxEntScan Visualizer can be used to analyze the impact of this mutation on the neighboring sequence.

MaveDB functional evidence

MaveDB is a public repository of laboratory assays that test the effect of thousands of variants at once — treat this as supporting research data and interpret it with great caution; it is not a clinical classification.

No MaveDB functional evidence found for this variant.

Publications

Other links and lift over

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