4-113333195-C-T

Variant summary

Our verdict is Benign. Variant got -21 ACMG points: 0P and 21B. BP4_StrongBP6_Very_StrongBP7BS1BS2

The NM_001148.6(ANK2):​c.3366C>T​(p.Asn1122Asn) variant causes a synonymous change involving the alteration of a non-conserved nucleotide. The variant allele was found at a frequency of 0.000776 in 1,614,006 control chromosomes in the GnomAD database, including 10 homozygotes. In-silico tool predicts a benign outcome for this variant. Variant has been reported in ClinVar as Likely benign (★★).

Frequency

Genomes: 𝑓 0.0025 ( 1 hom., cov: 32)
Exomes 𝑓: 0.00060 ( 9 hom. )

Consequence

ANK2
NM_001148.6 synonymous

Scores

2

Clinical Significance

Benign/Likely benign criteria provided, multiple submitters, no conflicts B:11

Conservation

PhyloP100: -1.05
Variant links:
Genes affected
ANK2 (HGNC:493): (ankyrin 2) This gene encodes a member of the ankyrin family of proteins that link the integral membrane proteins to the underlying spectrin-actin cytoskeleton. Ankyrins play key roles in activities such as cell motility, activation, proliferation, contact and the maintenance of specialized membrane domains. Most ankyrins are typically composed of three structural domains: an amino-terminal domain containing multiple ankyrin repeats; a central region with a highly conserved spectrin binding domain; and a carboxy-terminal regulatory domain which is the least conserved and subject to variation. The protein encoded by this gene is required for targeting and stability of Na/Ca exchanger 1 in cardiomyocytes. Mutations in this gene cause long QT syndrome 4 and cardiac arrhythmia syndrome. Multiple transcript variants encoding different isoforms have been described. [provided by RefSeq, Dec 2011]

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ACMG classification

Classification made for transcript

Verdict is Benign. Variant got -21 ACMG points.

BP4
Computational evidence support a benign effect (BayesDel_noAF=-0.55).
BP6
Variant 4-113333195-C-T is Benign according to our data. Variant chr4-113333195-C-T is described in ClinVar as [Likely_benign]. Clinvar id is 238577.Status of the report is criteria_provided_multiple_submitters_no_conflicts, 2 stars. Variant chr4-113333195-C-T is described in Lovd as [Benign].
BP7
Synonymous conserved (PhyloP=-1.05 with no splicing effect.
BS1
Variant frequency is greater than expected in population afr. gnomad4 allele frequency = 0.00246 (375/152134) while in subpopulation AFR AF= 0.00737 (306/41496). AF 95% confidence interval is 0.00669. There are 1 homozygotes in gnomad4. There are 191 alleles in male gnomad4 subpopulation. Median coverage is 32. This position pass quality control queck.
BS2
High AC in GnomAd4 at 375 AD gene.

Transcripts

RefSeq

Gene Transcript HGVSc HGVSp Effect Exon rank MANE Protein UniProt
ANK2NM_001148.6 linkc.3366C>T p.Asn1122Asn synonymous_variant Exon 29 of 46 ENST00000357077.9 NP_001139.3 Q01484-4

Ensembl

Gene Transcript HGVSc HGVSp Effect Exon rank TSL MANE Protein Appris UniProt
ANK2ENST00000357077.9 linkc.3366C>T p.Asn1122Asn synonymous_variant Exon 29 of 46 1 NM_001148.6 ENSP00000349588.4 Q01484-4

Frequencies

GnomAD3 genomes
AF:
0.00244
AC:
371
AN:
152016
Hom.:
1
Cov.:
32
show subpopulations
Gnomad AFR
AF:
0.00735
Gnomad AMI
AF:
0.00
Gnomad AMR
AF:
0.00223
Gnomad ASJ
AF:
0.00
Gnomad EAS
AF:
0.000385
Gnomad SAS
AF:
0.00249
Gnomad FIN
AF:
0.00
Gnomad MID
AF:
0.00
Gnomad NFE
AF:
0.000265
Gnomad OTH
AF:
0.000478
GnomAD3 exomes
AF:
0.00132
AC:
333
AN:
251414
Hom.:
3
AF XY:
0.00116
AC XY:
157
AN XY:
135880
show subpopulations
Gnomad AFR exome
AF:
0.00917
Gnomad AMR exome
AF:
0.000607
Gnomad ASJ exome
AF:
0.000198
Gnomad EAS exome
AF:
0.0000544
Gnomad SAS exome
AF:
0.00402
Gnomad FIN exome
AF:
0.0000924
Gnomad NFE exome
AF:
0.000220
Gnomad OTH exome
AF:
0.00163
GnomAD4 exome
AF:
0.000601
AC:
878
AN:
1461872
Hom.:
9
Cov.:
31
AF XY:
0.000652
AC XY:
474
AN XY:
727238
show subpopulations
Gnomad4 AFR exome
AF:
0.00839
Gnomad4 AMR exome
AF:
0.000537
Gnomad4 ASJ exome
AF:
0.000115
Gnomad4 EAS exome
AF:
0.000101
Gnomad4 SAS exome
AF:
0.00395
Gnomad4 FIN exome
AF:
0.0000187
Gnomad4 NFE exome
AF:
0.000125
Gnomad4 OTH exome
AF:
0.00111
GnomAD4 genome
AF:
0.00246
AC:
375
AN:
152134
Hom.:
1
Cov.:
32
AF XY:
0.00257
AC XY:
191
AN XY:
74366
show subpopulations
Gnomad4 AFR
AF:
0.00737
Gnomad4 AMR
AF:
0.00223
Gnomad4 ASJ
AF:
0.00
Gnomad4 EAS
AF:
0.000386
Gnomad4 SAS
AF:
0.00291
Gnomad4 FIN
AF:
0.00
Gnomad4 NFE
AF:
0.000265
Gnomad4 OTH
AF:
0.000473
Alfa
AF:
0.00134
Hom.:
0
Bravo
AF:
0.00288
Asia WGS
AF:
0.00144
AC:
5
AN:
3478
EpiCase
AF:
0.000382
EpiControl
AF:
0.000415

ClinVar

Significance: Benign/Likely benign
Submissions summary: Benign:11
Revision: criteria provided, multiple submitters, no conflicts
LINK: link

Submissions by phenotype

not provided Benign:4
Dec 12, 2017
ARUP Laboratories, Molecular Genetics and Genomics, ARUP Laboratories
Significance: Benign
Review Status: criteria provided, single submitter
Collection Method: clinical testing

- -

Mar 03, 2015
GeneDx
Significance: Benign
Review Status: criteria provided, single submitter
Collection Method: clinical testing

- -

Apr 04, 2016
Women's Health and Genetics/Laboratory Corporation of America, LabCorp
Significance: Benign
Review Status: criteria provided, single submitter
Collection Method: clinical testing

Variant Summary: The variant of interest causes a synonymous change involving a non-conserved nucleotide with 5/5 in silico programs via Alamut predicting no significant effect on splicing, although these predictions have yet to be functionally assessed. The variant of interest was observed in the large, broad control population, ExAC, with an allele frequency of 169/121394 (1/718), which significantly exceeds the predicted maximum expected allele frequency for a pathogenic ANK2 variant of 1/100000. The variant of interest has not been reported in affected individuals via publications and/or reputable databases/clinical laboratories, to our knowledge. Therefore, taking into consideration the nature of this variant being a synonymous change and the high frequency in controls, it is classified as Benign. -

Sep 01, 2024
CeGaT Center for Human Genetics Tuebingen
Significance: Likely benign
Review Status: criteria provided, single submitter
Collection Method: clinical testing

ANK2: BP4, BP7 -

not specified Benign:3
-
Genome Diagnostics Laboratory, University Medical Center Utrecht
Significance: Benign
Review Status: no assertion criteria provided
Collection Method: clinical testing

- -

-
Clinical Genetics, Academic Medical Center
Significance: Benign
Review Status: no assertion criteria provided
Collection Method: clinical testing

- -

-
Joint Genome Diagnostic Labs from Nijmegen and Maastricht, Radboudumc and MUMC+
Significance: Benign
Review Status: no assertion criteria provided
Collection Method: clinical testing

- -

Cardiac arrhythmia, ankyrin-B-related Benign:2
Jan 13, 2018
Illumina Laboratory Services, Illumina
Significance: Likely benign
Review Status: criteria provided, single submitter
Collection Method: clinical testing

This variant was observed in the ICSL laboratory as part of a predisposition screen in an ostensibly healthy population. It had not been previously curated by ICSL or reported in the Human Gene Mutation Database (HGMD: prior to June 1st, 2018), and was therefore a candidate for classification through an automated scoring system. Utilizing variant allele frequency, disease prevalence and penetrance estimates, and inheritance mode, an automated score was calculated to assess if this variant is too frequent to cause the disease. Based on the score and internal cut-off values, a variant classified as likely benign is not then subjected to further curation. The score for this variant resulted in a classification of likely benign for this disease. -

Dec 05, 2021
Genome-Nilou Lab
Significance: Benign
Review Status: criteria provided, single submitter
Collection Method: clinical testing

- -

Long QT syndrome Benign:1
Dec 30, 2024
Labcorp Genetics (formerly Invitae), Labcorp
Significance: Benign
Review Status: criteria provided, single submitter
Collection Method: clinical testing

- -

Cardiovascular phenotype Benign:1
Jan 21, 2017
Ambry Genetics
Significance: Benign
Review Status: criteria provided, single submitter
Collection Method: clinical testing

This alteration is classified as benign based on a combination of the following: seen in unaffected individuals, population frequency, intact protein function, lack of segregation with disease, co-occurrence, RNA analysis, in silico models, amino acid conservation, lack of disease association in case-control studies, and/or the mechanism of disease or impacted region is inconsistent with a known cause of pathogenicity. -

Computational scores

Source: dbNSFP v4.3

Name
Calibrated prediction
Score
Prediction
BayesDel_noAF
Benign
-0.55
CADD
Benign
0.31
DANN
Benign
0.46

Splicing

Name
Calibrated prediction
Score
Prediction
SpliceAI score (max)
0.020
Details are displayed if max score is > 0.2

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

Publications

LitVar

Below is the list of publications found by LitVar. It may be empty.

Other links and lift over

dbSNP: rs114896457; hg19: chr4-114254351; API