chr22-26465579-A-C

Variant summary

Our verdict is Likely benign. The variant received -2 ACMG points: 2P and 4B. PM2BP4_Strong

The NM_022081.6(HPS4):​c.707-28T>G variant causes a intron change involving the alteration of a non-conserved nucleotide. The variant was absent in control chromosomes in GnomAD project. In-silico tool predicts a benign outcome for this variant. No clinical diagnostic laboratories have submitted clinical-significance assessments for this variant to ClinVar. There are indicators that this mutation may affect the branch point..

Frequency

Genomes: not found (cov: 31)
Exomes 𝑓: 0.0 ( 0 hom. )
Failed GnomAD Quality Control

Consequence

HPS4
NM_022081.6 intron

Scores

2

Clinical Significance

Not reported in ClinVar

Conservation

PhyloP100: 0.491

Publications

9 publications found
Variant links:
Genes affected
HPS4 (HGNC:15844): (HPS4 biogenesis of lysosomal organelles complex 3 subunit 2) This gene encodes a protein component of biogenesis of lysosome-related organelles complexes (BLOC). BLOC complexes are important for the formation of endosomal-lysosomal organelles such as melanosomes and platelet dense granules. Mutations in this gene result in subtype 4 of Hermansky-Pudlak syndrome, a form of albinism. Alternative splicing results in multiple transcript variants. [provided by RefSeq, Aug 2012]
HPS4 Gene-Disease associations (from GenCC):
  • Hermansky-Pudlak syndrome 4
    Inheritance: AR Classification: DEFINITIVE, STRONG, MODERATE Submitted by: Labcorp Genetics (formerly Invitae), ClinGen, G2P, Ambry Genetics
  • Hermansky-Pudlak syndrome with pulmonary fibrosis
    Inheritance: AR Classification: SUPPORTIVE Submitted by: Orphanet

Genome browser will be placed here

ACMG classification

Classification was made for transcript

Our verdict: Likely_benign. The variant received -2 ACMG points.

PM2
Very rare variant in population databases, with high coverage;
BP4
This position, referring to a specific DNA site, is a probable branch point but is likely benign (scored 3 / 10, using the threshold of <=3). The score ranges from 0 to 10, with values ≤3 considered benign and >5 classified as pathogenic. Computational evidence support a benign effect (BayesDel_noAF=-0.82).

Variant Effect in Transcripts

ACMG analysis was done for transcript: NM_022081.6. You can select a different transcript below to see updated ACMG assignments.

RefSeq Transcripts

Sel.
GeneTranscriptTagsHGVScHGVSpEffectExon RankProteinUniProt
HPS4
NM_022081.6
MANE Select
c.707-28T>G
intron
N/ANP_071364.4
HPS4
NM_001349900.2
c.761-28T>G
intron
N/ANP_001336829.1F1LLU8
HPS4
NM_001349901.1
c.761-28T>G
intron
N/ANP_001336830.1F1LLU8

Ensembl Transcripts

Sel.
GeneTranscriptTagsHGVScHGVSpEffectExon RankProteinUniProt
HPS4
ENST00000398145.7
TSL:1 MANE Select
c.707-28T>G
intron
N/AENSP00000381213.2Q9NQG7-1
HPS4
ENST00000402105.7
TSL:1
c.692-28T>G
intron
N/AENSP00000384185.3Q9NQG7-3
HPS4
ENST00000439453.5
TSL:1
n.*225-28T>G
intron
N/AENSP00000406764.1F8WC53

Frequencies

GnomAD3 genomes
Cov.:
31
GnomAD4 exome
Data not reliable, filtered out with message: AC0
AF:
0.00
AC:
0
AN:
1430964
Hom.:
0
Cov.:
24
AF XY:
0.00
AC XY:
0
AN XY:
714046
African (AFR)
AF:
0.00
AC:
0
AN:
32834
American (AMR)
AF:
0.00
AC:
0
AN:
44668
Ashkenazi Jewish (ASJ)
AF:
0.00
AC:
0
AN:
25948
East Asian (EAS)
AF:
0.00
AC:
0
AN:
39492
South Asian (SAS)
AF:
0.00
AC:
0
AN:
85598
European-Finnish (FIN)
AF:
0.00
AC:
0
AN:
52676
Middle Eastern (MID)
AF:
0.00
AC:
0
AN:
5686
European-Non Finnish (NFE)
AF:
0.00
AC:
0
AN:
1084642
Other (OTH)
AF:
0.00
AC:
0
AN:
59420
GnomAD4 genome
Cov.:
31

ClinVar

Not reported in ClinVar

Computational scores

Source: dbNSFP v4.9

Name
Calibrated prediction
Score
Prediction
BayesDel_noAF
Benign
-0.82
CADD
Benign
12
DANN
Benign
0.66
PhyloP100
0.49
BranchPoint Hunter
3.0

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: rs3213583; hg19: chr22-26861545; API