11-65535999-A-C

Variant summary

Our verdict is Likely pathogenic. The variant received 6 ACMG points: 6P and 0B. PM2PM5PP3_Moderate

The NM_020680.4(SCYL1):​c.1433A>C​(p.Asp478Ala) variant causes a missense change. The variant was absent in control chromosomes in GnomAD project. In-silico tool predicts a pathogenic outcome for this variant. No clinical diagnostic laboratories have submitted clinical-significance assessments for this variant to ClinVar. Another variant affecting the same amino acid position, but resulting in a different missense (i.e. D478G) has been classified as Pathogenic.

Frequency

Genomes: not found (cov: 33)

Consequence

SCYL1
NM_020680.4 missense

Scores

8
8
1

Clinical Significance

Not reported in ClinVar

Conservation

PhyloP100: 6.40

Publications

0 publications found
Variant links:
Genes affected
SCYL1 (HGNC:14372): (SCY1 like pseudokinase 1) This gene encodes a transcriptional regulator belonging to the SCY1-like family of kinase-like proteins. The protein has a divergent N-terminal kinase domain that is thought to be catalytically inactive, and can bind specific DNA sequences through its C-terminal domain. It activates transcription of the telomerase reverse transcriptase and DNA polymerase beta genes. The protein has been localized to the nucleus, and also to the cytoplasm and centrosomes during mitosis. Multiple transcript variants encoding different isoforms have been found for this gene. [provided by RefSeq, Jul 2008]
SCYL1 Gene-Disease associations (from GenCC):
  • acute infantile liver failure-cerebellar ataxia-peripheral sensory motor neuropathy syndrome
    Inheritance: AR Classification: STRONG, MODERATE, SUPPORTIVE Submitted by: G2P, Labcorp Genetics (formerly Invitae), Ambry Genetics, PanelApp Australia, Orphanet

Genome browser will be placed here

ACMG classification

Classification was made for transcript

Our verdict: Likely_pathogenic. The variant received 6 ACMG points.

PM2
Very rare variant in population databases, with high coverage;
PM5
Other missense variant is known to change same aminoacid residue: Variant chr11-65535999-A-G is described in ClinVar as Pathogenic. ClinVar VariationId is 446291.Status of the report is no_assertion_criteria_provided, 0 stars.
PP3
MetaRNN computational evidence supports a deleterious effect, 0.924

Variant Effect in Transcripts

ACMG analysis was done for transcript: NM_020680.4. You can select a different transcript below to see updated ACMG assignments.

RefSeq Transcripts

Sel.
GeneTranscriptTagsHGVScHGVSpEffectExon RankProteinUniProt
SCYL1
NM_020680.4
MANE Select
c.1433A>Cp.Asp478Ala
missense
Exon 11 of 18NP_065731.3
SCYL1
NM_001425179.1
c.1433A>Cp.Asp478Ala
missense
Exon 11 of 18NP_001412108.1
SCYL1
NM_001425180.1
c.1430A>Cp.Asp477Ala
missense
Exon 11 of 18NP_001412109.1

Ensembl Transcripts

Sel.
GeneTranscriptTagsHGVScHGVSpEffectExon RankProteinUniProt
SCYL1
ENST00000270176.10
TSL:1 MANE Select
c.1433A>Cp.Asp478Ala
missense
Exon 11 of 18ENSP00000270176.5Q96KG9-1
SCYL1
ENST00000420247.6
TSL:1
c.1433A>Cp.Asp478Ala
missense
Exon 11 of 18ENSP00000408192.2Q96KG9-2
SCYL1
ENST00000524944.5
TSL:1
c.1433A>Cp.Asp478Ala
missense
Exon 11 of 17ENSP00000432175.1E9PS17

Frequencies

GnomAD3 genomes
Cov.:
33
GnomAD4 exome
Cov.:
32
GnomAD4 genome
Cov.:
33

ClinVar

Not reported in ClinVar

Computational scores

Source: dbNSFP v4.9

Name
Calibrated prediction
Score
Prediction
AlphaMissense
Pathogenic
1.0
BayesDel_addAF
Pathogenic
0.29
D
BayesDel_noAF
Pathogenic
0.18
CADD
Pathogenic
27
DANN
Uncertain
1.0
DEOGEN2
Uncertain
0.77
D
Eigen
Pathogenic
0.77
Eigen_PC
Pathogenic
0.70
FATHMM_MKL
Uncertain
0.87
D
LIST_S2
Benign
0.85
T
M_CAP
Uncertain
0.16
D
MetaRNN
Pathogenic
0.92
D
MetaSVM
Uncertain
-0.27
T
PhyloP100
6.4
PrimateAI
Uncertain
0.67
T
PROVEAN
Pathogenic
-7.1
D
REVEL
Pathogenic
0.70
Sift
Uncertain
0.0030
D
Sift4G
Uncertain
0.0050
D
Polyphen
1.0
D
Vest4
0.92
MutPred
0.74
Loss of helix (P = 0.028)
MVP
0.62
MPC
1.3
ClinPred
1.0
D
GERP RS
5.5
PromoterAI
-0.0080
Neutral
Varity_R
0.77
gMVP
0.84
Mutation Taster
=10/90
disease causing

Splicing

Name
Calibrated prediction
Score
Prediction
SpliceAI score (max)
0.010
Details are displayed if max score is > 0.2

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

Publications

Other links and lift over

dbSNP: rs1554969925; hg19: chr11-65303470; API