chr16-89553941-T-A

Variant summary

Our verdict is Likely pathogenic. The variant received 8 ACMG points: 8P and 0B. PM1PM2PM5PP3_Moderate

The NM_003119.4(SPG7):​c.2084T>A​(p.Leu695Gln) 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. L695V) has been classified as Uncertain significance.

Frequency

Genomes: not found (cov: 32)

Consequence

SPG7
NM_003119.4 missense

Scores

11
4

Clinical Significance

Not reported in ClinVar

Conservation

PhyloP100: 6.02

Publications

2 publications found
Variant links:
Genes affected
SPG7 (HGNC:11237): (SPG7 matrix AAA peptidase subunit, paraplegin) This gene encodes a mitochondrial metalloprotease protein that is a member of the AAA family. Members of this protein family share an ATPase domain and have roles in diverse cellular processes including membrane trafficking, intracellular motility, organelle biogenesis, protein folding, and proteolysis. Mutations in this gene cause autosomal recessive spastic paraplegia 7. Two transcript variants encoding distinct isoforms have been identified. [provided by RefSeq, Mar 2014]
SPG7 Gene-Disease associations (from GenCC):
  • hereditary spastic paraplegia 7
    Inheritance: AR, AD Classification: DEFINITIVE, STRONG, SUPPORTIVE Submitted by: G2P, Orphanet, PanelApp Australia, Laboratory for Molecular Medicine, Labcorp Genetics (formerly Invitae)
  • autosomal dominant optic atrophy
    Inheritance: AD Classification: STRONG Submitted by: PanelApp Australia
  • lateral sclerosis
    Inheritance: AD Classification: SUPPORTIVE Submitted by: Orphanet

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ACMG classification

Classification was made for transcript

Our verdict: Likely_pathogenic. The variant received 8 ACMG points.

PM1
In a hotspot region, there are 4 aminoacids with missense pathogenic changes in the window of +-8 aminoacids around while only 0 benign, 10 uncertain in NM_003119.4
PM2
Very rare variant in population databases, with high coverage;
PM5
Other missense variant is known to change same aminoacid residue: Variant chr16-89553941-T-C is described in ClinVar as Pathogenic/Likely_pathogenic. ClinVar VariationId is 219448.Status of the report is criteria_provided_multiple_submitters_no_conflicts, 2 stars.
PP3
MetaRNN computational evidence supports a deleterious effect, 0.888

Variant Effect in Transcripts

ACMG analysis was done for transcript: NM_003119.4. You can select a different transcript below to see updated ACMG assignments.

RefSeq Transcripts

Sel.
GeneTranscriptTagsHGVScHGVSpEffectExon RankProteinUniProt
SPG7
NM_003119.4
MANE Select
c.2084T>Ap.Leu695Gln
missense
Exon 15 of 17NP_003110.1Q9UQ90-1
SPG7
NM_001363850.1
c.2084T>Ap.Leu695Gln
missense
Exon 15 of 18NP_001350779.1A0A2R8Y3M4

Ensembl Transcripts

Sel.
GeneTranscriptTagsHGVScHGVSpEffectExon RankProteinUniProt
SPG7
ENST00000645818.2
MANE Select
c.2084T>Ap.Leu695Gln
missense
Exon 15 of 17ENSP00000495795.2Q9UQ90-1
SPG7
ENST00000268704.7
TSL:1
c.2063T>Ap.Leu688Gln
missense
Exon 15 of 17ENSP00000268704.3A0A2U3TZH1
SPG7
ENST00000918773.1
c.2174T>Ap.Leu725Gln
missense
Exon 15 of 17ENSP00000588832.1

Frequencies

GnomAD3 genomes
Cov.:
32
GnomAD4 exome
Cov.:
32
GnomAD4 genome
Cov.:
32
Alfa
AF:
0.00
Hom.:
0
Bravo
AF:
0.00000378

ClinVar

Not reported in ClinVar

Computational scores

Source: dbNSFP v4.9

Name
Calibrated prediction
Score
Prediction
AlphaMissense
Pathogenic
0.65
BayesDel_addAF
Pathogenic
0.41
D
BayesDel_noAF
Pathogenic
0.36
CADD
Pathogenic
28
DANN
Uncertain
0.99
DEOGEN2
Pathogenic
0.80
D
Eigen
Pathogenic
0.76
Eigen_PC
Pathogenic
0.72
FATHMM_MKL
Pathogenic
0.99
D
LIST_S2
Uncertain
0.93
D
M_CAP
Pathogenic
0.33
D
MetaRNN
Pathogenic
0.89
D
MetaSVM
Uncertain
0.61
D
MutationAssessor
Pathogenic
2.9
M
PhyloP100
6.0
PrimateAI
Uncertain
0.63
T
REVEL
Pathogenic
0.91
Polyphen
1.0
D
MutPred
0.66
Gain of disorder (P = 0.0236)
MVP
0.97
MPC
0.80
ClinPred
0.99
D
GERP RS
5.2
RBP_binding_hub_radar
0.92
RBP_regulation_power_radar
2.6
Varity_R
0.90
gMVP
0.84

Splicing

Name
Calibrated prediction
Score
Prediction
SpliceAI score (max)
0.0
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: rs864622094; hg19: chr16-89620349; API