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GeneE
10 sources retrieved Β· Most recent: April 2026 Β· Index updated 14 days ago
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HGSNAT
heparan-alpha-glucosaminide N-acetyltransferase
Chromosome 8 Β· 8p11.21-p11.1
NCBI Gene: 138050Ensembl: ENSG00000165102.16HGNC: HGNC:26527UniProt: Q68CP4
38PubMed Papers
22Diseases
0Drugs
188Pathogenic Variants
RESEARCH IMPACT
Variant-Rich
CLINICAL
OMIM Disease Gene
DATA QUALITY
βœ“ Experimental GO Evidenceβœ“ Swiss-Prot Reviewed
protein complex oligomerizationacyltransferase activitylysosomal transportlysosomal membranemucopolysaccharidosis type 3Cretinitis pigmentosa 73mucopolysaccharidosis type 3Retinal dystrophy
✦AI Summary

HGSNAT (heparan-alpha-glucosaminide N-acetyltransferase) is a lysosomal membrane acetyltransferase that catalyzes a unique transmembrane acetylation reaction essential for heparan sulfate degradation 1. The enzyme acetylates the non-reducing terminal alpha-glucosamine residue of intralysosomal heparan sulfate, converting it into a substrate for luminal alpha-N-acetyl glucosaminidase 2. This involves transporting the acetyl group from cytosolic acetyl-CoA across the lysosomal membrane via a central pore within a dimeric enzyme structure, with catalysis mediated by a histidine-aspartic acid dyad 1. HGSNAT deficiency causes mucopolysaccharidosis type IIIC (Sanfilippo syndrome C), a severe autosomal recessive lysosomal storage disease characterized by heparan sulfate accumulation 2. Clinical presentation includes progressive neurological deterioration with onset typically around 4 years of age, featuring speech deterioration, mental decline, and hyperactivity, with relatively mild somatic symptoms 3. Additionally, biallelic HGSNAT variants can cause adult-onset nonsyndromic retinitis pigmentosa with pericentral retinal degeneration and reduced photoreceptor function, distinct from syndromic MPS IIIC 4. Current treatment options remain limited, though enzyme replacement therapy and gene therapy show promise 5. Cardiac complications have been documented in carbohydrate-linked lysosomal storage diseases including HGSNAT deficiency 6.

Sources cited
1
HGSNAT structure, transmembrane acetylation mechanism, histidine-aspartic acid catalytic dyad, and pore structure for acetyl-CoA transport
PMID: 38918376
2
HGSNAT enzyme function in acetylating terminal glucosamine residues of heparan sulfate and MPS IIIC pathophysiology
PMID: 19479962
3
Clinical presentation of MPS IIIC with onset symptoms, diagnosis delay, and genetic variants in HGSNAT
PMID: 37014526
4
HGSNAT variants associated with adult-onset nonsyndromic retinitis pigmentosa with pericentral retinal degeneration
PMID: 32770643
5
MPS III clinical features, diagnosis methods, and treatment approaches including enzyme replacement and gene therapy
PMID: 25851924
6
HGSNAT deficiency included in carbohydrate-linked lysosomal storage diseases with cardiac complications
PMID: 37239976
Disease Associationsβ“˜22
mucopolysaccharidosis type 3COpen Targets
0.85Strong
retinitis pigmentosa 73Open Targets
0.79Strong
mucopolysaccharidosis type 3Open Targets
0.74Strong
Retinal dystrophyOpen Targets
0.55Moderate
retinitis pigmentosaOpen Targets
0.51Moderate
neurodegenerative diseaseOpen Targets
0.47Moderate
genetic disorderOpen Targets
0.47Moderate
inherited retinal dystrophyOpen Targets
0.37Weak
synovial plica syndromeOpen Targets
0.27Weak
Intellectual disabilityOpen Targets
0.11Weak
ovarian dysfunctionOpen Targets
0.06Suggestive
McLeod neuroacanthocytosis syndromeOpen Targets
0.03Suggestive
cholesteryl ester storage diseaseOpen Targets
0.03Suggestive
lysosomal acid lipase deficiencyOpen Targets
0.03Suggestive
retinopathyOpen Targets
0.03Suggestive
retinitis pigmentosa 1Open Targets
0.03Suggestive
retinal degenerationOpen Targets
0.01Suggestive
Neurodevelopmental disorderOpen Targets
0.01Suggestive
metachromatic leukodystrophyOpen Targets
0.01Suggestive
hyperinsulinemic hypoglycemia, familial, 4Open Targets
0.01Suggestive
Mucopolysaccharidosis 3CUniProt
Retinitis pigmentosa 73UniProt
Pathogenic Variants188
NM_152419.3(HGSNAT):c.743G>C (p.Gly248Ala)Likely pathogenic
Retinitis pigmentosa 73|Retinitis pigmentosa 73;Mucopolysaccharidosis, MPS-III-C|Mucopolysaccharidosis, MPS-III-C
β˜…β˜…β˜†β˜†2026β†’ Residue 248
NM_152419.3(HGSNAT):c.1348del (p.Asp450fs)Pathogenic
Mucopolysaccharidosis, MPS-III-C;Retinitis pigmentosa 73|Mucopolysaccharidosis, MPS-III-C|not provided
β˜…β˜…β˜†β˜†2026β†’ Residue 450
NM_152419.3(HGSNAT):c.739del (p.Arg247fs)Pathogenic
not provided|Mucopolysaccharidosis, MPS-III-C|Retinitis pigmentosa 73;Mucopolysaccharidosis, MPS-III-C|Sanfilippo syndrome
β˜…β˜…β˜†β˜†2026β†’ Residue 247
NM_152419.3(HGSNAT):c.1516C>T (p.Arg506Ter)Pathogenic
Mucopolysaccharidosis, MPS-III-C|Retinitis pigmentosa 73;Mucopolysaccharidosis, MPS-III-C|Sanfilippo syndrome|not provided|Retinal dystrophy|Retinitis pigmentosa 73
β˜…β˜…β˜†β˜†2026β†’ Residue 506
NM_152419.3(HGSNAT):c.852-2A>CPathogenic
not provided|Mucopolysaccharidosis, MPS-III-C|Retinitis pigmentosa 73;Mucopolysaccharidosis, MPS-III-C
β˜…β˜…β˜†β˜†2026
NM_152419.3(HGSNAT):c.1270G>A (p.Gly424Ser)Likely pathogenic
Mucopolysaccharidosis, MPS-III-C|Retinitis pigmentosa 73;Mucopolysaccharidosis, MPS-III-C|Sanfilippo syndrome
β˜…β˜…β˜†β˜†2026β†’ Residue 424
NM_152419.3(HGSNAT):c.493+1G>APathogenic
Mucopolysaccharidosis, MPS-III-C|Mucopolysaccharidosis, MPS-III-C;Retinitis pigmentosa 73|Sanfilippo syndrome|Inborn genetic diseases|not provided
β˜…β˜…β˜†β˜†2025
NM_152419.3(HGSNAT):c.1031G>A (p.Arg344His)Pathogenic
Mucopolysaccharidosis, MPS-III-C|Retinitis pigmentosa 73;Mucopolysaccharidosis, MPS-III-C|not provided
β˜…β˜…β˜†β˜†2025β†’ Residue 344
NM_152419.3(HGSNAT):c.1250+1G>APathogenic
not provided|Mucopolysaccharidosis, MPS-III-C|Mucopolysaccharidosis, MPS-III-C;Retinitis pigmentosa 73|Retinal dystrophy|Sanfilippo syndrome|Retinitis pigmentosa 73|Acute myeloid leukemia
β˜…β˜…β˜†β˜†2025
NM_152419.3(HGSNAT):c.234+1G>APathogenic
Mucopolysaccharidosis, MPS-III-C|not provided|Mucopolysaccharidosis, MPS-III-C;Retinitis pigmentosa 73|Retinal dystrophy|Sanfilippo syndrome
β˜…β˜…β˜†β˜†2025
NM_152419.3(HGSNAT):c.1464+1G>APathogenic
not provided|Mucopolysaccharidosis, MPS-III-C|Retinitis pigmentosa 73;Mucopolysaccharidosis, MPS-III-C
β˜…β˜…β˜†β˜†2025
NM_152419.3(HGSNAT):c.372-2A>GPathogenic
Mucopolysaccharidosis, MPS-III-C|Sanfilippo syndrome|Mucopolysaccharidosis, MPS-III-C;Retinitis pigmentosa 73|not provided
β˜…β˜…β˜†β˜†2025
NM_152419.3(HGSNAT):c.1466C>A (p.Ala489Glu)Pathogenic
Mucopolysaccharidosis, MPS-III-C|Retinitis pigmentosa 73;Mucopolysaccharidosis, MPS-III-C
β˜…β˜…β˜†β˜†2025β†’ Residue 489
NM_152419.3(HGSNAT):c.1622C>T (p.Ser541Leu)Pathogenic
Retinitis pigmentosa 73;Mucopolysaccharidosis, MPS-III-C|Retinal dystrophy|Mucopolysaccharidosis, MPS-III-C|Sanfilippo syndrome|Retinitis pigmentosa 73|not provided
β˜…β˜…β˜†β˜†2025β†’ Residue 541
NM_152419.3(HGSNAT):c.852-1G>APathogenic
Mucopolysaccharidosis, MPS-III-C|Retinitis pigmentosa 73;Mucopolysaccharidosis, MPS-III-C
β˜…β˜…β˜†β˜†2025
NM_152419.3(HGSNAT):c.1700G>A (p.Trp567Ter)Pathogenic
Retinitis pigmentosa 73;Mucopolysaccharidosis, MPS-III-C|Mucopolysaccharidosis, MPS-III-C
β˜…β˜…β˜†β˜†2025β†’ Residue 567
NM_152419.3(HGSNAT):c.1170del (p.Trp390fs)Pathogenic
not provided|Retinitis pigmentosa 73;Mucopolysaccharidosis, MPS-III-C|Mucopolysaccharidosis, MPS-III-C
β˜…β˜…β˜†β˜†2025β†’ Residue 390
NM_152419.3(HGSNAT):c.1411G>A (p.Glu471Lys)Pathogenic
not provided|Sanfilippo syndrome|Mucopolysaccharidosis, MPS-III-C|Retinitis pigmentosa 73;Mucopolysaccharidosis, MPS-III-C|Retinitis pigmentosa 73
β˜…β˜…β˜†β˜†2025β†’ Residue 471
NM_152419.3(HGSNAT):c.1782_1815del (p.Phe595fs)Pathogenic
Mucopolysaccharidosis, MPS-III-C|Mucopolysaccharidosis, MPS-III-C;Retinitis pigmentosa 73
β˜…β˜…β˜†β˜†2025β†’ Residue 595
NM_152419.3(HGSNAT):c.1271G>T (p.Gly424Val)Pathogenic
not provided|Retinitis pigmentosa 73;Mucopolysaccharidosis, MPS-III-C
β˜…β˜…β˜†β˜†2025β†’ Residue 424
View on ClinVar β†—
Related Genes
ESCO1Protein interaction80%NAGLUProtein interaction77%ESCO2Protein interaction72%SGSHProtein interaction65%GNSProtein interaction62%WDCPShared pathway33%
Tissue Expression6 tissues
Ovary
100%
Bone Marrow
73%
Lung
72%
Liver
62%
Heart
58%
Brain
39%
Gene Interaction Network
Click a node to explore
HGSNATESCO1NAGLUESCO2SGSHGNSWDCP
PROTEIN STRUCTURE
Preparing viewer…
PDB8JL3 Β· 2.59 Γ… Β· EM
View on RCSB β†—
Constraintβ“˜
LOEUFβ“˜
1.00LoF Tolerant
pLIβ“˜
0.00Tolerant
Observed/Expected LoF0.76 [0.58–1.00]
RankingsWhere HGSNAT stands among ~20K protein-coding genes
  • #10,493of 20,598
    Most Researched38
  • #375of 5,498
    Most Pathogenic Variants188 Β· top 10%
  • #9,659of 17,882
    Most Constrained (LOEUF)1.00
Genes detectedHGSNAT
Sources retrieved10 papers
Response timeβ€”
πŸ“„ Sources
10β–Ό
1
PMID: 20301590
1.00
2
Metabolic Cardiomyopathies and Cardiac Defects in Inherited Disorders of Carbohydrate Metabolism: A Systematic Review.
PMID: 37239976
Int J Mol Sci Β· 2023
0.90
3
Structural and mechanistic insights into a lysosomal membrane enzyme HGSNAT involved in Sanfilippo syndrome.
PMID: 38918376
Nat Commun Β· 2024
0.80
4
Mucopolysaccharidosis type IIIC in chinese mainland: clinical and molecular characteristics of ten patients and report of six novel variants in the HGSNAT gene.
PMID: 37014526
Metab Brain Dis Β· 2023
0.70
5
Sanfilippo syndrome type C: mutation spectrum in the heparan sulfate acetyl-CoA: alpha-glucosaminide N-acetyltransferase (HGSNAT) gene.
PMID: 19479962
Hum Mutat Β· 2009
0.60