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GeneE
10 sources retrieved Β· Most recent: April 2026 Β· Index updated 14 days ago
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IDUA
alpha-L-iduronidase
Chromosome 4 Β· 4p16.3
NCBI Gene: 3425Ensembl: ENSG00000127415.14HGNC: HGNC:5391UniProt: P35475
98PubMed Papers
23Diseases
0Drugs
414Pathogenic Variants
RESEARCH IMPACT
Variant-Rich
CLINICAL
OMIM Disease Gene
DATA QUALITY
βœ“ Experimental GO Evidenceβœ“ Swiss-Prot Reviewed
heparin proteoglycan catabolic processL-iduronidase activitydermatan sulfate proteoglycan catabolic processextracellular exosomeScheie syndromeHurler syndromeHurler-Scheie syndromemucopolysaccharidosis type 1
✦AI Summary

IDUA encodes alpha-L-iduronidase, a lysosomal hydrolase essential for glycosaminoglycan (GAG) catabolism, specifically degrading dermatan sulfate and heparan sulfate 1. The enzyme functions in the lysosomal lumen to catalyze disaccharide metabolism and proteoglycan breakdown. IDUA deficiency causes mucopolysaccharidosis type I (MPS I), a lysosomal storage disorder with three phenotypic variants: severe Hurler disease with CNS involvement, intermediate Hurler/Scheie disease, and mild Scheie disease without cognitive impairment 1. Disease severity correlates poorly with genotype, though homozygous nonsense mutations typically produce Hurler phenotype 1. Over 200 pathogenic variants exist, with p.Trp402Ter, p.Gln70Ter, and p.Pro533Arg being geographically common 2. MPS I presents with progressive skeletal dysostosis multiplex, airway obstruction, corneal clouding, joint contractures, cardiac valve disease, and hepatosplenomegaly 1. IDUA deficiency also causes cardiac complications including cardiomyopathy and arrhythmias 3. Treatment options include enzyme replacement therapy (laronidase), which improves GAG excretion and respiratory function, and hematopoietic stem cell transplantation, which can stabilize cognitive decline when performed early 1. Emerging gene therapy approaches using lentiviral vectors to deliver functional IDUA in autologous hematopoietic stem cells show promising results, achieving supraphysiologic enzyme activity and metabolic correction in both peripheral tissues and CNS 4.

Sources cited
1
IDUA is a lysosomal hydrolase whose deficiency causes MPS I with three phenotypic variants; clinical manifestations including skeletal, cardiac, respiratory, and neurological features; current treatments with HSCT and ERT
PMID: 25345091
2
Worldwide distribution and frequencies of common IDUA pathogenic variants including p.Trp402Ter, p.Gln70Ter, and p.Pro533Arg
PMID: 29393969
3
IDUA-related MPS I is a carbohydrate-linked lysosomal storage disease presenting with cardiac manifestations
PMID: 37239976
4
Gene therapy approach using lentiviral vectors to deliver IDUA in autologous hematopoietic stem cells achieves supraphysiologic enzyme activity and metabolic correction in peripheral tissues and CNS
PMID: 34788506
⚠Limited data available β€” This gene has 4 indexed publications. Summary and analysis may be incomplete.
Disease Associationsβ“˜23
Scheie syndromeOpen Targets
0.85Strong
Hurler syndromeOpen Targets
0.85Strong
Hurler-Scheie syndromeOpen Targets
0.85Strong
mucopolysaccharidosis type 1Open Targets
0.81Strong
mucopolysaccharidosisOpen Targets
0.70Strong
genetic disorderOpen Targets
0.51Moderate
nephrolithiasis susceptibility caused by SLC26A1Open Targets
0.50Moderate
bone fractureOpen Targets
0.48Moderate
calcium oxalate urolithiasisOpen Targets
0.46Moderate
nephrolithiasis, calcium oxalateOpen Targets
0.46Moderate
Interstitial pneumonitisOpen Targets
0.43Moderate
Alzheimer diseaseOpen Targets
0.41Moderate
hypersulfaturiaOpen Targets
0.41Moderate
Parkinson diseaseOpen Targets
0.37Weak
upper extremity fractureOpen Targets
0.36Weak
mucopolysaccharidosis type 4AOpen Targets
0.35Weak
type 1 diabetes mellitusOpen Targets
0.34Weak
radius fractureOpen Targets
0.31Weak
atrial fibrillationOpen Targets
0.30Weak
ulna fractureOpen Targets
0.29Weak
Mucopolysaccharidosis 1HUniProt
Mucopolysaccharidosis 1H/SUniProt
Mucopolysaccharidosis 1SUniProt
Pathogenic Variants414
NM_000203.5(IDUA):c.882dup (p.Ile295fs)Pathogenic
Mucopolysaccharidosis type 1
β˜…β˜…β˜…β˜†2026β†’ Residue 295
NM_000203.5(IDUA):c.1A>C (p.Met1Leu)Pathogenic
Hurler syndrome|Mucopolysaccharidosis type 1
β˜…β˜…β˜…β˜†2026β†’ Residue 1
NM_000203.5(IDUA):c.1249_1275del (p.Thr417_His425del)Likely pathogenic
Hurler syndrome|Mucopolysaccharidosis type 1
β˜…β˜…β˜…β˜†2026β†’ Residue 417
NM_000203.5(IDUA):c.3G>A (p.Met1Ile)Pathogenic
Hurler syndrome|Mucopolysaccharidosis type 1
β˜…β˜…β˜…β˜†2026β†’ Residue 1
NM_000203.5(IDUA):c.1591del (p.Arg531fs)Pathogenic
Hurler syndrome|Mucopolysaccharidosis type 1
β˜…β˜…β˜…β˜†2026β†’ Residue 531
NM_000203.5(IDUA):c.568_581del (p.Asn190fs)Pathogenic
Mucopolysaccharidosis type 1|not provided
β˜…β˜…β˜…β˜†2026β†’ Residue 190
NM_000203.5(IDUA):c.1727+4C>TPathogenic
Hurler syndrome|Mucopolysaccharidosis type 1
β˜…β˜…β˜…β˜†2026
NM_000203.5(IDUA):c.187C>T (p.Gln63Ter)Pathogenic
Mucopolysaccharidosis type 1|Hurler syndrome
β˜…β˜…β˜…β˜†2026β†’ Residue 63
NM_000203.5(IDUA):c.1641dup (p.Pro548fs)Likely pathogenic
Mucopolysaccharidosis type 1
β˜…β˜…β˜…β˜†2026β†’ Residue 548
NM_000203.5(IDUA):c.1599del (p.Ser534fs)Likely pathogenic
Mucopolysaccharidosis type 1
β˜…β˜…β˜…β˜†2026β†’ Residue 534
NM_000203.5(IDUA):c.1727+1G>APathogenic
Mucopolysaccharidosis type 1
β˜…β˜…β˜…β˜†2026
NM_000203.5(IDUA):c.530T>C (p.Phe177Ser)Likely pathogenic
Mucopolysaccharidosis type 1
β˜…β˜…β˜…β˜†2025β†’ Residue 177
NM_000203.5(IDUA):c.1883G>C (p.Arg628Pro)Likely pathogenic
Hurler syndrome|not provided|Mucopolysaccharidosis type 1|Mucopolysaccharidosis, MPS-IV-A
β˜…β˜…β˜…β˜†2025β†’ Residue 628
NM_000203.5(IDUA):c.1045G>C (p.Asp349His)Likely pathogenic
Mucopolysaccharidosis, MPS-II|Mucopolysaccharidosis type 1|IDUA-related disorder
β˜…β˜…β˜…β˜†2025β†’ Residue 349
NM_000203.5(IDUA):c.820G>T (p.Glu274Ter)Pathogenic
Hurler syndrome|Mucopolysaccharidosis type 1
β˜…β˜…β˜…β˜†2025β†’ Residue 274
NM_000203.5(IDUA):c.1044C>A (p.Asn348Lys)Likely pathogenic
Mucopolysaccharidosis type 1
β˜…β˜…β˜…β˜†2025β†’ Residue 348
NM_000203.5(IDUA):c.972+2T>CPathogenic
Mucopolysaccharidosis type 1
β˜…β˜…β˜…β˜†2025
NM_000203.5(IDUA):c.349del (p.Tyr117fs)Pathogenic
Mucopolysaccharidosis type 1
β˜…β˜…β˜…β˜†2025β†’ Residue 117
NM_000203.5(IDUA):c.943G>T (p.Asp315Tyr)Likely pathogenic
Mucopolysaccharidosis type 1
β˜…β˜…β˜…β˜†2025β†’ Residue 315
NM_000203.5(IDUA):c.1861C>T (p.Arg621Ter)Pathogenic
Hurler syndrome|Mucopolysaccharidosis type 1|not provided|Hurler syndrome;Mucopolysaccharidosis, MPS-I-H/S;Mucopolysaccharidosis, MPS-I-S
β˜…β˜…β˜…β˜†2025β†’ Residue 621
View on ClinVar β†—
Related Genes
HYAL1Protein interaction95%SPAM1Protein interaction94%HYAL3Protein interaction92%HYAL2Protein interaction92%SIProtein interaction84%MGAMProtein interaction84%
Tissue Expression6 tissues
Ovary
100%
Liver
86%
Lung
75%
Bone Marrow
68%
Heart
16%
Brain
4%
Gene Interaction Network
Click a node to explore
IDUAHYAL1SPAM1HYAL3HYAL2SIMGAM
PROTEIN STRUCTURE
Preparing viewer…
PDB6I6R Β· 2.02 Γ… Β· X-ray
View on RCSB β†—
Constraintβ“˜
LOEUFβ“˜
1.34LoF Tolerant
pLIβ“˜
0.00Tolerant
Observed/Expected LoF1.09 [0.89–1.34]
RankingsWhere IDUA stands among ~20K protein-coding genes
  • #4,887of 20,598
    Most Researched98 Β· top quartile
  • #133of 5,498
    Most Pathogenic Variants414 Β· top 5%
  • #14,067of 17,882
    Most Constrained (LOEUF)1.34
Genes detectedIDUA
Sources retrieved10 papers
Response timeβ€”
πŸ“„ Sources
10β–Ό
1
Novel vectors and approaches for gene therapy in liver diseases.
PMID: 34159305
JHEP Rep Β· 2021
1.00
2
Hematopoietic Stem- and Progenitor-Cell Gene Therapy for Hurler Syndrome.
PMID: 34788506
N Engl J Med Β· 2021
0.90
3
Molecular characterization of 355 mucopolysaccharidosis patients reveals 104 novel mutations.
PMID: 22976768
J Inherit Metab Dis Β· 2013
0.80
4
Metabolic Cardiomyopathies and Cardiac Defects in Inherited Disorders of Carbohydrate Metabolism: A Systematic Review.
PMID: 37239976
Int J Mol Sci Β· 2023
0.70
5
Identification of incompletely penetrant variants and interallelic interactions in autosomal recessive disorders by a population-genetic approach.
PMID: 34405919
Hum Mutat Β· 2021
0.60