mucolipidosis type III, alpha/beta

disease
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Also known as ML 3ML 3 AML 3 alpha/betaML III alpha/betaML3MLIIImucolipidosis 3mucolipidosis IIImucolipidosis type 3 alpha/betamucolipidosis type 3Amucolipidosis type IIIPseudo Hurler Polydystrophypseudo-Hurler polydystrophy

Summary

mucolipidosis type III, alpha/beta (MONDO:0018931) is a disease caused by GNPTAB (GenCC Definitive), with 1 cohort gene and 1 clinical trial.

At a glance

  • Prevalence: 1-5 / 10 000 (Europe) [Orphanet-validated]
  • Causal gene: GNPTAB (GenCC Definitive)
  • Cohort genes: 1
  • ClinVar variants: 1,432
  • Phenotypes (HPO): 36
  • Clinical trials: 1

Clinical features

Epidemiology

Prevalence records

6 prevalence record(s), Orphanet:

TypeClassValueGeographyValidation
Annual incidence1-9 / 1 000 0000.985EuropeValidated
Point prevalence1-5 / 10 00029.55EuropeValidated
Annual incidence<1 / 1 000 0000.08NetherlandsValidated
Annual incidence1-9 / 1 000 0001.68PortugalValidated
Point prevalence1-9 / 100 0002.4NetherlandsValidated
Point prevalence1-5 / 10 00050.4PortugalValidated

Signs & symptoms

Clinical features (HPO)

36 HPO clinical features (Orphanet curated; top 36 by frequency):

HPO IDTermFrequency
HP:0000269Prominent occiputVery frequent (80-99%)
HP:0000505Visual impairmentVery frequent (80-99%)
HP:0001387Joint stiffnessVery frequent (80-99%)
HP:0003264Deficiency of N-acetylglucosamine-1-phosphotransferaseVery frequent (80-99%)
HP:0003272Abnormality of the hip boneVery frequent (80-99%)
HP:0003312Abnormal form of the vertebral bodiesVery frequent (80-99%)
HP:0004322Short statureVery frequent (80-99%)
HP:0004493Craniofacial hyperostosisVery frequent (80-99%)
HP:0008818Large iliac wingsVery frequent (80-99%)
HP:0008821Hypoplastic inferior iliaVery frequent (80-99%)
HP:0025261Stiff fingerVery frequent (80-99%)
HP:0002857Genu valgumFrequent (30-79%)
HP:0003307HyperlordosisFrequent (30-79%)
HP:0007957Corneal opacityFrequent (30-79%)
HP:0000280Coarse facial featuresFrequent (30-79%)
HP:0000938OsteopeniaFrequent (30-79%)
HP:0000943Dysostosis multiplexFrequent (30-79%)
HP:0001385Hip dysplasiaFrequent (30-79%)
HP:0001634Mitral valve prolapseFrequent (30-79%)
HP:0002650ScoliosisFrequent (30-79%)
HP:0002758OsteoarthritisFrequent (30-79%)
HP:0009837Bullet-shaped distal phalanges of the handFrequent (30-79%)
HP:0012185Constrictive median neuropathyFrequent (30-79%)
HP:0012532Chronic painFrequent (30-79%)
HP:0030680Abnormal cardiovascular system morphologyFrequent (30-79%)
HP:0034337Claw hand deformityFrequent (30-79%)
HP:0100543Cognitive impairmentFrequent (30-79%)
HP:0000885Broad ribsOccasional (5-29%)
HP:0001072Thickened skinOccasional (5-29%)
HP:0001653Mitral regurgitationOccasional (5-29%)
HP:0001659Aortic regurgitationOccasional (5-29%)
HP:0002091Restrictive ventilatory defectOccasional (5-29%)
HP:0002176Spinal cord compressionOccasional (5-29%)
HP:0002515Waddling gaitOccasional (5-29%)
HP:0004349Reduced bone mineral densityOccasional (5-29%)
HP:0012378FatigueOccasional (5-29%)

Identifiers

Disease identifiers

FieldValue
Canonical namemucolipidosis type III, alpha/beta
Mondo IDMONDO:0018931
OMIM252600
Orphanet423461, 577
DOIDDOID:0080071
SNOMED CT65764006
UMLSC0033788
MedGen10988
GARD0017704
NORD1624
Is cancer (heuristic)no

Also known as: ML 3 · ML 3 A · ML 3 alpha/beta · ML III alpha/beta · ML3 · MLIII · mucolipidosis 3 · mucolipidosis III · mucolipidosis type 3 alpha/beta · mucolipidosis type 3A · mucolipidosis type III · Pseudo Hurler Polydystrophy · pseudo-Hurler polydystrophy

Data availability: 1,432 ClinVar variants · 4 GenCC gene-disease records.

Disease family

Classification path: disease › human disease › disease by developmental or physiological process › metabolic diseasedevelopmental anomaly of metabolic originmucolipidosis › familial mucolipidosis › mucolipidosis type III, alpha/beta

Related subtypes (4): GNPTG-mucolipidosis, mucolipidosis type IV, sialidosis type 2, sialidosis type 1

Genetics & variants

GWAS landscape

No GWAS associations recorded — common-variant (GWAS) studies don’t cover this disease (typical for Mendelian / rare diseases). See the curated gene cohort and Mendelian overlap below.

Variant details and genetic-evidence tiers

ClinVar germline variants

600 retrieved; paginated sample, class counts are floors:

353 likely benign, 100 uncertain significance, 61 pathogenic, 49 likely pathogenic, 15 pathogenic/likely pathogenic, 12 conflicting classifications of pathogenicity, 5 benign, 5 benign/likely benign

ClinVarVariant (HGVS)GeneClassificationReview
1012255NM_024312.5(GNPTAB):c.2980_2983del (p.Ala994fs)GNPTABPathogeniccriteria provided, single submitter
1069206NM_024312.5(GNPTAB):c.118-1G>AGNPTABPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
1071081NM_024312.5(GNPTAB):c.1491del (p.Gly498fs)GNPTABPathogeniccriteria provided, single submitter
1072768NM_024312.5(GNPTAB):c.2657_2660del (p.Asp886fs)GNPTABPathogeniccriteria provided, single submitter
1072782NM_024312.5(GNPTAB):c.2423del (p.Leu808fs)GNPTABPathogeniccriteria provided, single submitter
1072926NM_024312.5(GNPTAB):c.2868_2869del (p.Met957fs)GNPTABPathogeniccriteria provided, multiple submitters, no conflicts
1072933NM_024312.5(GNPTAB):c.3345del (p.Met1116fs)GNPTABPathogeniccriteria provided, single submitter
1073529NM_024312.5(GNPTAB):c.3300_3301del (p.Lys1100fs)GNPTABPathogeniccriteria provided, single submitter
1074624NM_024312.5(GNPTAB):c.1906del (p.Arg636fs)GNPTABPathogeniccriteria provided, single submitter
1075110NM_024312.5(GNPTAB):c.2737_2738del (p.Gln913fs)GNPTABPathogeniccriteria provided, single submitter
1075885NM_024312.5(GNPTAB):c.178_179del (p.Ile60fs)GNPTABPathogeniccriteria provided, single submitter
1362626NM_024312.5(GNPTAB):c.269del (p.Leu90fs)GNPTABPathogeniccriteria provided, single submitter
1364183NM_024312.5(GNPTAB):c.3100del (p.Ala1034fs)GNPTABPathogeniccriteria provided, single submitter
1382090NM_024312.5(GNPTAB):c.2675del (p.Leu892fs)GNPTABPathogeniccriteria provided, single submitter
1385807NM_024312.5(GNPTAB):c.2334_2338del (p.Ser778fs)GNPTABPathogeniccriteria provided, single submitter
1388742NM_024312.5(GNPTAB):c.3034C>T (p.Gln1012Ter)GNPTABPathogeniccriteria provided, single submitter
1393484NM_024312.5(GNPTAB):c.29_32dup (p.Thr12fs)GNPTABPathogeniccriteria provided, single submitter
1396985NM_024312.5(GNPTAB):c.1186G>T (p.Glu396Ter)GNPTABPathogeniccriteria provided, single submitter
1398708NM_024312.5(GNPTAB):c.280C>T (p.Gln94Ter)GNPTABPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
1399558NM_024312.5(GNPTAB):c.2654_2655del (p.Thr885fs)GNPTABPathogeniccriteria provided, single submitter
1416350NM_024312.5(GNPTAB):c.88_89del (p.Thr30fs)GNPTABPathogeniccriteria provided, single submitter
1424471NM_024312.5(GNPTAB):c.2303_2306del (p.Lys768fs)GNPTABPathogeniccriteria provided, single submitter
1429731NM_024312.5(GNPTAB):c.1936dup (p.Gln646fs)GNPTABPathogeniccriteria provided, single submitter
1431816NM_024312.5(GNPTAB):c.1503_1521dup (p.Gly508delinsCysLeuLeuLeuTer)GNPTABPathogeniccriteria provided, single submitter
1437885NM_024312.5(GNPTAB):c.1122dup (p.Arg375fs)GNPTABPathogeniccriteria provided, single submitter
1445251NM_024312.5(GNPTAB):c.1292T>A (p.Leu431Ter)GNPTABPathogeniccriteria provided, single submitter
1451810NM_024312.5(GNPTAB):c.1499del (p.Asn500fs)GNPTABPathogeniccriteria provided, single submitter
1451897NM_024312.5(GNPTAB):c.2923del (p.Glu975fs)GNPTABPathogeniccriteria provided, single submitter
1452514NM_024312.5(GNPTAB):c.2098dup (p.Ile700fs)GNPTABPathogeniccriteria provided, single submitter
1452854NM_024312.5(GNPTAB):c.1871del (p.Glu624fs)GNPTABPathogeniccriteria provided, single submitter

Genes & proteins

Mendelian disease overlap and somatic drivers

GenCC: 10 · Orphanet: 2 · OMIM-shared: 0 · Dual-evidence (GWAS+Mendelian): 0

GenCC gene–disease validity (cohort genes)

the Disease column is the GenCC-asserted condition — a cohort gene’s strongest validity may be for a related predisposition syndrome.

GeneClassificationInheritanceDiseaseRecords
GNPTABDefinitiveAutosomal recessivemucolipidosis type II10

Orphanet rare-disease linkage (cohort genes)

GeneOrphanet IDRare disease
GNPTABOrphanet:423461Mucolipidosis type III alpha/beta
GNPTABOrphanet:576Mucolipidosis type II

Cohort genes → proteins

1 cohort genes, 1 distinct canonical proteins.

Evidence partition

SubsetGenes
multi_evidence1

Cohort genes (full)

SymbolHGNCEnsemblUniProtNameEvidence
GNPTABHGNC:29670ENSG00000111670Q3T906N-acetylglucosamine-1-phosphotransferase subunits alpha/betagencc,clinvar

Cohort function summary

Lead sentence per gene, UniProt-curated.

SymbolProtein nameFunction (lead sentence)
GNPTABN-acetylglucosamine-1-phosphotransferase subunits alpha/betaCatalyzes the formation of mannose 6-phosphate (M6P) markers on high mannose type oligosaccharides in the Golgi apparatus.

Protein-family classification

Druggable: 1 · Difficult: 0 · Unknown: 0 · Druggable fraction: 1.0

Family distribution

Cohort families vs a genome-wide background (hypergeometric, BH-FDR; fold = observed/expected). Counts kept; sorted by enrichment, so the catch-all Other/Unknown bucket no longer leads.

FamilyGenesFoldFDR
Enzyme (other)112.0×0.083

Per-gene assignment

SymbolFamilyDruggable?ECInterPro (top 3)
GNPTABEnzyme (other)yes2.7.8.17Notch_dom, EF_hand_dom, DMAP1-bd

Expression context

Cohort genes with no expression data: 0.

1 cohort gene are a single-cell marker in ≥1 SCXA experiment.

Breadth distribution (Bgee present_calls)

BucketGenes
narrow (1-5 tissues)0
moderate (6-20)0
broad (>20)1
unknown0

Top tissues across cohort

TissueCohort genes
endothelial cell1
sural nerve1
tibia1

Per-gene tissue summary (top 30)

SymbolBgee breadthFANTOM5 breadthSCXATop tissues
GNPTAB290ubiquitousmarkertibia, endothelial cell, sural nerve

Protein interactions among cohort

Intra-cohort edges: 0.

Hub genes (top 10 by interactor count)

SymbolInteractor count
GNPTAB1,518

Structural data

PDB: 1 · AlphaFold-only: 0 · No structure: 0

Cohort genes with PDB structures (top 30)

SymbolUniProtPDB entries
GNPTABQ3T9065

Function

Pathway analysis

Distinct Reactome pathways touched by cohort: 0. Enrichment computed across 1 evidence-associated genes (0 with Reactome annotation).

GO biological processes by enrichment

Over-representation of cohort genes vs the genome-wide background (hypergeometric test, Benjamini-Hochberg FDR; fold = observed/expected over 1 annotated cohort genes). Counts and members are kept as ground-truth; sorted by enrichment.

GO termCohort genesFoldFDRSample cohort genes
N-glycan processing to lysosome18426.0×5e-04GNPTAB
secretion of lysosomal enzymes13370.4×6e-04GNPTAB
carbohydrate phosphorylation12106.5×6e-04GNPTAB
lysosome organization1306.4×0.003GNPTAB

Therapeutics

Drug target analysis

Approved (phase 4): 0 · Phase ≥3: 0 · Phased (≥1): 0 · Undrugged: 1

Druggability breadth: 0 of 1 evidence-associated genes (0%) have a ChEMBL target (buckets above are over the deeply-mined display cohort).

Top cohort targets by molecule count

SymbolMoleculesMax phase
GNPTAB00

Bioactivity and enzyme data

Enzyme cohort genes (≥1 EC): 1.

Cohort enzymes (BRENDA EC)

SymbolEC numbersNames
GNPTAB2.7.8.17UDP-N-acetylglucosamine-lysosomal-enzyme N-acetylglucosaminephosphotransferase

Pharmacogenomics

Cohort genes with a PharmGKB record: 1; with CPIC/DPWG dosing guidelines: 0.

No cohort gene has a CPIC/DPWG genotype-guided dosing guideline (PharmGKB).

Chemical tractability of cohort targets

0 approved/phased compounds have measured bioactivity against a cohort gene (and aren’t yet in disease-level trials). This is a research / tractability signal, NOT a therapeutic recommendation — a bioactivity row often reflects off-target or screening binding (e.g. promiscuous kinase inhibitors against a cohort kinase), implying no disease mechanism.

Druggability pyramid

Cohort genes binned by druggability tier (high → low):

TierDefinitionGenesSymbols
AApproved (phase 4 drug)0
BPhased (≥1) drug, not yet approved0
CDruggable family + PDB, no drug1GNPTAB
DDruggable family + AlphaFold only, no drug0
EDifficult family or no structure, no drug0

Undrugged target profiles

1 cohort genes are undrugged. Ranked by ‘starting-point quality’ (assay depth + drugged-partner adjacency).

SymbolChEMBL assaysDrugged partners (top 3)
GNPTAB0

Clinical trials & evidence

Clinical trials

Clinical trials: 1.

Phase distribution (across all retrieved trials)

PhaseTrials
Not specified1

Top trials by phase / activity

NCTPhaseStatusTitle
NCT01891422Not specifiedCOMPLETEDLongitudinal Studies of the Glycoproteinoses