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GeneE
10 sources retrieved Β· Most recent: April 2026 Β· Index updated 14 days ago
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PIGV
phosphatidylinositol glycan anchor biosynthesis class V
Chromosome 1 Β· 1p36.11
NCBI Gene: 55650Ensembl: ENSG00000060642.12HGNC: HGNC:26031UniProt: A0A6I8PU65
19PubMed Papers
21Diseases
0Drugs
9Pathogenic Variants
CLINICAL
OMIM Disease Gene
DATA QUALITY
βœ“ Experimental GO Evidenceβœ“ Swiss-Prot Reviewed
alpha-1,6-mannosyltransferase activitydol-P-Man:Man(1)GlcN-acyl-PI alpha-1,6-mannosyltransferase activityGPI anchor biosynthetic processendoplasmic reticulum membranehyperphosphatasia-intellectual disability syndromeDisorder of glycosphingolipid and glycosylphosphatidylinositol anchor glycosylationgenetic disorderElevated circulating alkaline phosphatase concentration
✦AI Summary

PIGV encodes an alpha-1,6-mannosyltransferase that catalyzes a critical step in glycosylphosphatidylinositol (GPI) anchor biosynthesis 1. The enzyme transfers the second mannose via an alpha-1,6 bond from dolichol-phosphate-mannose to an intermediate substrate, participating in the seventh step of GPI-anchor biosynthesis. GPI-anchored proteins are essential for maintaining epithelial integrity during development, as demonstrated in C. elegans where PIGV reduction caused epithelial breaches, internal cysts, and surface ruptures during morphogenesis 1. These defects could be rescued by strengthening membrane-cortex connections, indicating GPI-anchored proteins are crucial for withstanding developmental stresses. PIGV mutations cause hyperphosphatasia with impaired intellectual development syndrome 1 (HPMRS1), characterized by intellectual disability, distinctive facial features, brachytelephalangy, and elevated serum alkaline phosphatase 23. The clinical spectrum also includes anorectal malformations, Hirschsprung disease, and renal anomalies 2. The most common mutation (c.1022C>A) accounts for approximately 80% of cases 24. Additionally, ER stress can downregulate PIGV through the ATF6-miR449c-5p pathway, contributing to cancer progression by disrupting GPI anchor assembly 5.

Sources cited
1
PIGV encodes an alpha-1,6-mannosyltransferase involved in GPI anchor biosynthesis and is essential for epithelial integrity during development
PMID: 25807459
2
PIGV mutations cause HPMRS1 with characteristic features including intellectual disability, facial dysmorphism, and the c.1022C>A mutation being most common
PMID: 24129430
3
PIGV mutations are associated with hyperphosphatasia-mental retardation syndrome with expanded clinical spectrum including anorectal malformations
PMID: 21739589
4
The c.1022C>A mutation is found in Polish patients with HPMRS1 presenting diverse neurological and behavioral symptoms
PMID: 37372388
5
ER stress downregulates PIGV through ATF6-miR449c-5p pathway, affecting GPI anchor assembly and contributing to cancer progression
PMID: 37390992
Disease Associationsβ“˜21
hyperphosphatasia-intellectual disability syndromeOpen Targets
0.69Moderate
Disorder of glycosphingolipid and glycosylphosphatidylinositol anchor glycosylationOpen Targets
0.50Moderate
genetic disorderOpen Targets
0.45Moderate
Elevated circulating alkaline phosphatase concentrationOpen Targets
0.26Weak
metabolic syndromeOpen Targets
0.07Suggestive
response to combination chemotherapyOpen Targets
0.07Suggestive
hair colorOpen Targets
0.06Suggestive
HeterotaxiaOpen Targets
0.06Suggestive
genito-palato-cardiac syndromeOpen Targets
0.06Suggestive
physical activityOpen Targets
0.06Suggestive
congenital unilateral hypoplasia of depressor anguli orisOpen Targets
0.05Suggestive
Microcephaly - cardiac defect - lung malsegmentationOpen Targets
0.04Suggestive
microcephaly-cardiac defect-lung malsegmentation syndromeOpen Targets
0.04Suggestive
22q11.2 deletion syndromeOpen Targets
0.04Suggestive
heterotaxy, visceral, 12, autosomalOpen Targets
0.04Suggestive
congenital heart defects, multiple types, 6Open Targets
0.04Suggestive
Congenitally uncorrected transposition of the great arteriesOpen Targets
0.04Suggestive
congenital heart defects, multiple types, 4Open Targets
0.04Suggestive
congenital heart diseaseOpen Targets
0.04Suggestive
congenital heart defects, multiple types, 7Open Targets
0.04Suggestive
Hyperphosphatasia with impaired intellectual development syndrome 1UniProt
Pathogenic Variants9
NM_017837.4(PIGV):c.467G>A (p.Cys156Tyr)Pathogenic
Hyperphosphatasia with intellectual disability syndrome 1|not provided
β˜…β˜…β˜†β˜†2026β†’ Residue 156
NM_017837.4(PIGV):c.1022C>A (p.Ala341Glu)Pathogenic
Hyperphosphatasia with intellectual disability syndrome 1|Inborn genetic diseases|not provided|Hyperphosphatasia-intellectual disability syndrome|PIGV-related disorder
β˜…β˜…β˜†β˜†2025β†’ Residue 341
NM_017837.4(PIGV):c.607C>T (p.Arg203Cys)Pathogenic
not provided|Hyperphosphatasia with intellectual disability syndrome 1
β˜…β˜…β˜†β˜†2025β†’ Residue 203
NM_017837.4(PIGV):c.1253C>A (p.Ala418Asp)Likely pathogenic
Inborn genetic diseases|not provided
β˜…β˜…β˜†β˜†2025β†’ Residue 418
NM_017837.4(PIGV):c.781dup (p.Thr261fs)Likely pathogenic
not provided
β˜…β˜†β˜†β˜†2018β†’ Residue 261
NM_017837.4(PIGV):c.55C>T (p.Arg19Cys)Likely pathogenic
Hyperphosphatasia with intellectual disability syndrome 1
β˜…β˜†β˜†β˜†2017β†’ Residue 19
NM_017837.4(PIGV):c.1022C>T (p.Ala341Val)Pathogenic
Hyperphosphatasia with intellectual disability syndrome 1
β˜†β˜†β˜†β˜†2010β†’ Residue 341
NM_017837.4(PIGV):c.766C>A (p.Gln256Lys)Pathogenic
Hyperphosphatasia with intellectual disability syndrome 1
β˜†β˜†β˜†β˜†2010β†’ Residue 256
NM_017837.4(PIGV):c.1154A>C (p.His385Pro)Pathogenic
Hyperphosphatasia with intellectual disability syndrome 1
β˜†β˜†β˜†β˜†2010β†’ Residue 385
View on ClinVar β†—
Related Genes
PIGFShared pathway100%PIGXShared pathway100%PGAP4Shared pathway100%CWH43Shared pathway100%PIGYProtein interaction100%ALG3Protein interaction99%
Tissue Expression6 tissues
Liver
100%
Ovary
39%
Bone Marrow
32%
Lung
30%
Brain
24%
Heart
23%
Gene Interaction Network
Click a node to explore
PIGVPIGFPIGXPGAP4CWH43PIGYALG3
PROTEIN STRUCTURE
Preparing viewer…
AlphaFoldAI-predicted Β· UniProt Q9NUD9
View on AlphaFold β†—
Constraintβ“˜
LOEUFβ“˜
0.90LoF Tolerant
pLIβ“˜
0.00Tolerant
Observed/Expected LoF0.65 [0.48–0.90]
RankingsWhere PIGV stands among ~20K protein-coding genes
  • #14,492of 20,598
    Most Researched19
  • #3,001of 5,498
    Most Pathogenic Variants9
  • #8,172of 17,882
    Most Constrained (LOEUF)0.90
Genes detectedPIGV
Sources retrieved10 papers
Response timeβ€”
πŸ“„ Sources
10β–Ό
1
Metabolic Cardiomyopathies and Cardiac Defects in Inherited Disorders of Carbohydrate Metabolism: A Systematic Review.
PMID: 37239976
Int J Mol Sci Β· 2023
1.00
2
Characteristics of Neuroimaging and Behavioural Phenotype in Polish Patients with PIGV-CDG-An Observational Study and Literature Review.
PMID: 37372388
Genes (Basel) Β· 2023
0.90
3
Alternative splicing in ovarian cancer.
PMID: 39425166
Cell Commun Signal Β· 2024
0.80
4
Delineation of PIGV mutation spectrum and associated phenotypes in hyperphosphatasia with mental retardation syndrome.
PMID: 24129430
Eur J Hum Genet Β· 2014
0.70
5
Glycosyl phosphatidylinositol anchor biosynthesis is essential for maintaining epithelial integrity during Caenorhabditis elegans embryogenesis.
PMID: 25807459
PLoS Genet Β· 2015
0.60