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GeneE
10 sources retrieved Β· Most recent: April 2026 Β· Index updated 14 days ago
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HPS5
HPS5 biogenesis of lysosomal organelles complex 2 subunit 2
Chromosome 11 Β· 11p15.1
NCBI Gene: 11234Ensembl: ENSG00000110756.19HGNC: HGNC:17022UniProt: Q9UPZ3
44PubMed Papers
21Diseases
0Drugs
133Pathogenic Variants
RESEARCH IMPACT
Variant-Rich
CLINICAL
OMIM Disease Gene
DATA QUALITY
βœ“ Experimental GO Evidenceβœ“ Swiss-Prot Reviewed
cytosolprotein bindingBLOC-2 complexcytoplasmHermansky-Pudlak syndromeHermansky-Pudlak syndrome without pulmonary fibrosisneurodegenerative diseasehair color
✦AI Summary

HPS5 is a component of the biogenesis of lysosomal organelles complex 2 (BLOC-2), essential for biogenesis of lysosome-related organelles (LROs) including melanosomes and platelet dense granules 1. HPS5 regulates intracellular vesicular trafficking and melanosome assembly, functioning as part of a multi-protein complex critical for proper protein trafficking to specialized organelles 2. The protein works coordinately with other BLOC complexes and AP-3 to maintain ion homeostasis within LROs; disruption of HPS5 leads to impaired trafficking of zinc transporters and accumulation of intracellular zinc, ultimately compromising dense granule biogenesis 3. Mutations in HPS5 cause Hermansky-Pudlak syndrome (HPS), a rare autosomal recessive disorder characterized by oculocutaneous albinism, pigmentation defects in melanosomes, and platelet storage pool deficiency with bleeding tendency 4. HPS5 mutations present with hypopigmentation, photophobia, and ocular manifestations, with some patients experiencing additional systemic complications 5. The clinical severity can vary, with recent findings demonstrating that synonymous and intronic variants causing alternative splicing represent pathogenic mechanisms 4. Animal models validate HPS5's role, as mutations in Drosophila and stickleback orthologues produce similar pigmentation defects 16.

Sources cited
1
HPS5 is a BLOC-2 subunit essential for biogenesis of melanosomes and platelet dense granules; mutations cause Hermansky-Pudlak syndrome
PMID: 17156100
2
HPS5 gene encodes a BLOC-2 complex component required for protein trafficking in lysosome-related organelles
PMID: 17632576
3
BLOC-2 (including HPS5) is required for trafficking of zinc transporters to dense granule precursors and HPS5 mutations reduce zinc transporter trafficking
PMID: 33513603
4
HPS5 mutations cause oculocutaneous albinism and bleeding tendency; synonymous variants can be pathogenic through alternative splicing
PMID: 39988965
5
HPS is the most common syndromic oculocutaneous albinism with hypopigmentation and ocular manifestations; HPS5 is a rare subtype
PMID: 32969595
6
Stickleback Hps5 mutations cause oculocutaneous albinism through disrupted melanosome biogenesis, validating vertebrate model relevance
PMID: 28739598
Disease Associationsβ“˜21
Hermansky-Pudlak syndromeOpen Targets
0.80Strong
Hermansky-Pudlak syndrome without pulmonary fibrosisOpen Targets
0.80Strong
neurodegenerative diseaseOpen Targets
0.30Weak
hair colorOpen Targets
0.23Weak
genetic disorderOpen Targets
0.19Weak
ThrombocytopeniaOpen Targets
0.12Weak
Abnormal bleedingOpen Targets
0.11Weak
Griscelli diseaseOpen Targets
0.10Weak
Griscelli disease type 3Open Targets
0.10Weak
Griscelli syndrome type 3Open Targets
0.10Weak
Back painOpen Targets
0.10Suggestive
adolescent idiopathic scoliosisOpen Targets
0.10Suggestive
Microcephaly - albinism - digital anomaliesOpen Targets
0.09Suggestive
microcephaly-albinism-digital anomalies syndromeOpen Targets
0.09Suggestive
Dyschromatosis universalisOpen Targets
0.09Suggestive
Neurofibromatosis type 6Open Targets
0.09Suggestive
Tietz syndromeOpen Targets
0.09Suggestive
Griscelli disease type 1Open Targets
0.09Suggestive
Griscelli syndrome type 1Open Targets
0.09Suggestive
dyschromatosis symmetrica hereditariaOpen Targets
0.09Suggestive
Hermansky-Pudlak syndrome 5UniProt
Pathogenic Variants133
NM_181507.2(HPS5):c.1423del (p.Leu475fs)Pathogenic
Hermansky-Pudlak syndrome 5|not provided|Hermansky-Pudlak syndrome
β˜…β˜…β˜†β˜†2025β†’ Residue 475
NM_181507.2(HPS5):c.2928_2929dup (p.Thr977fs)Pathogenic
Hermansky-Pudlak syndrome 5|not provided
β˜…β˜…β˜†β˜†2025β†’ Residue 977
NM_181507.2(HPS5):c.2375del (p.Glu792fs)Pathogenic
HPS5-related disorder|not provided
β˜…β˜…β˜†β˜†2025β†’ Residue 792
NM_181507.2(HPS5):c.82dup (p.Leu28fs)Pathogenic
not provided|Hermansky-Pudlak syndrome
β˜…β˜…β˜†β˜†2025β†’ Residue 28
NM_181507.2(HPS5):c.739dup (p.Glu247fs)Pathogenic
not provided|HPS5-related disorder|Hermansky-Pudlak syndrome
β˜…β˜…β˜†β˜†2025β†’ Residue 247
NM_181507.2(HPS5):c.220-1G>TPathogenic
not provided
β˜…β˜…β˜†β˜†2024
NM_181507.2(HPS5):c.2837+1G>ALikely pathogenic
Hermansky-Pudlak syndrome|not provided|Hermansky-Pudlak syndrome 5
β˜…β˜…β˜†β˜†2024
NM_181507.2(HPS5):c.1252del (p.Glu418fs)Pathogenic
not provided|Hermansky-Pudlak syndrome 5
β˜…β˜…β˜†β˜†2024β†’ Residue 418
NM_181507.2(HPS5):c.888dup (p.His297fs)Pathogenic
not provided|Hermansky-Pudlak syndrome 5
β˜…β˜…β˜†β˜†2024β†’ Residue 297
NM_181507.2(HPS5):c.2718-2A>GPathogenic
not provided|Hermansky-Pudlak syndrome 5
β˜…β˜…β˜†β˜†2024
NM_181507.2(HPS5):c.2952-1G>CLikely pathogenic
HPS5-related disorder|not provided
β˜…β˜…β˜†β˜†2024
NM_181507.2(HPS5):c.2979_2982del (p.Cys993fs)Pathogenic
not provided|Hermansky-Pudlak syndrome 5|Hermansky-Pudlak syndrome
β˜…β˜…β˜†β˜†2024β†’ Residue 993
NM_181507.2(HPS5):c.2036C>G (p.Ser679Ter)Pathogenic
Hermansky-Pudlak syndrome|not provided|Hermansky-Pudlak syndrome 5
β˜…β˜…β˜†β˜†2024β†’ Residue 679
NM_181507.2(HPS5):c.986-1G>CLikely pathogenic
Hermansky-Pudlak syndrome 5|not provided
β˜…β˜…β˜†β˜†2024
NM_181507.2(HPS5):c.478-2A>GLikely pathogenic
Hermansky-Pudlak syndrome|not provided|Hermansky-Pudlak syndrome 5
β˜…β˜…β˜†β˜†2024
NM_181507.2(HPS5):c.1417C>T (p.Gln473Ter)Pathogenic
Hermansky-Pudlak syndrome 5|not provided
β˜…β˜…β˜†β˜†2024β†’ Residue 473
NM_181507.2(HPS5):c.1862+1G>ALikely pathogenic
Hermansky-Pudlak syndrome 5|not provided
β˜…β˜…β˜†β˜†2023
NM_181507.2(HPS5):c.2077del (p.Arg693fs)Pathogenic
not provided|Hermansky-Pudlak syndrome 5
β˜…β˜…β˜†β˜†2023β†’ Residue 693
NM_181507.2(HPS5):c.543del (p.Gln181fs)Pathogenic
Hermansky-Pudlak syndrome|Hermansky-Pudlak syndrome 5|not provided
β˜…β˜…β˜†β˜†2023β†’ Residue 181
NM_181507.2(HPS5):c.393G>A (p.Trp131Ter)Pathogenic
not provided
β˜…β˜†β˜†β˜†2026β†’ Residue 131
View on ClinVar β†—
Related Genes
BLOC1S3Protein interaction91%BLOC1S5Protein interaction88%DTNBP1Protein interaction88%SNAPINProtein interaction88%BLOC1S2Protein interaction87%BLOC1S6Protein interaction86%
Tissue Expression6 tissues
Liver
100%
Heart
35%
Lung
34%
Brain
30%
Ovary
27%
Bone Marrow
23%
Gene Interaction Network
Click a node to explore
HPS5BLOC1S3BLOC1S5DTNBP1SNAPINBLOC1S2BLOC1S6
PROTEIN STRUCTURE
Preparing viewer…
AlphaFoldAI-predicted Β· UniProt Q9UPZ3
View on AlphaFold β†—
Constraintβ“˜
LOEUFβ“˜
0.80LoF Tolerant
pLIβ“˜
0.00Tolerant
Observed/Expected LoF0.65 [0.54–0.80]
RankingsWhere HPS5 stands among ~20K protein-coding genes
  • #9,598of 20,598
    Most Researched44
  • #586of 5,498
    Most Pathogenic Variants133 Β· top quartile
  • #6,615of 17,882
    Most Constrained (LOEUF)0.80
Genes detectedHPS5
Sources retrieved10 papers
Response timeβ€”
πŸ“„ Sources
10β–Ό
1
PMID: 20301464
1.00
2
A zinc transporter, transmembrane protein 163, is critical for the biogenesis of platelet dense granules.
PMID: 33513603
Blood Β· 2021
0.90
3
The pink gene encodes the Drosophila orthologue of the human Hermansky-Pudlak syndrome 5 (HPS5) gene.
PMID: 17632576
Genome Β· 2007
0.80
4
Current landscape of Oculocutaneous Albinism in Japan.
PMID: 32969595
Pigment Cell Melanoma Res Β· 2021
0.70
5
The Drosophila pigmentation gene pink (p) encodes a homologue of human Hermansky-Pudlak syndrome 5 (HPS5).
PMID: 17156100
Traffic Β· 2007
0.60