GNPTG-mucolipidosis

disease
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Also known as ML 3 gammaML III gammamucolipidosis type 3 gammamucolipidosis type III gamma

Summary

GNPTG-mucolipidosis (MONDO:0009652) is a disease caused by GNPTG (GenCC Definitive), with 2 cohort genes.

At a glance

  • Prevalence: Unknown (Worldwide) [Orphanet-validated]
  • Causal gene: GNPTG (GenCC Definitive)
  • Cohort genes: 2
  • ClinVar variants: 288

Clinical features

No curated clinical features (Orphanet) for this disease.

Identifiers

Disease identifiers

FieldValue
Canonical nameGNPTG-mucolipidosis
Mondo IDMONDO:0009652
MeSHC565367
OMIM252605
Orphanet423470
DOIDDOID:0080678
NCITC129978
UMLSC1854896
MedGen340743
GARD0017705
Is cancer (heuristic)no

Also known as: GNPTG-mucolipidosis · ML 3 gamma · ML III gamma · mucolipidosis type 3 gamma · mucolipidosis type III gamma

Data availability: 288 ClinVar variants · 6 GenCC gene-disease records · 2 cell lines.

Disease family

Classification path: disease › human disease › disease by developmental or physiological process › metabolic diseasedevelopmental anomaly of metabolic originmucolipidosis › familial mucolipidosis › GNPTG-mucolipidosis

Related subtypes (4): mucolipidosis type IV, sialidosis type 2, mucolipidosis type III, alpha/beta, 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

288 retrieved; paginated sample, class counts are floors:

113 uncertain significance, 45 likely pathogenic, 42 likely benign, 31 conflicting classifications of pathogenicity, 26 pathogenic, 19 pathogenic/likely pathogenic, 5 benign/likely benign, 4 not provided, 3 benign

ClinVarVariant (HGVS)GeneClassificationReview
1069744NM_032520.5(GNPTG):c.739A>T (p.Lys247Ter)GNPTGPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
1072459NM_032520.5(GNPTG):c.750_753del (p.Lys250fs)GNPTGPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
1342959NM_032520.5(GNPTG):c.190_193dup (p.Phe65fs)GNPTGPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
1354910NM_032520.5(GNPTG):c.203C>A (p.Ser68Ter)GNPTGPathogeniccriteria provided, multiple submitters, no conflicts
1450957NM_032520.5(GNPTG):c.607C>T (p.Gln203Ter)GNPTGPathogeniccriteria provided, multiple submitters, no conflicts
1526169NM_032520.5(GNPTG):c.638_639del (p.Leu212_Phe213insTer)GNPTGPathogeniccriteria provided, multiple submitters, no conflicts
1685858NM_032520.5(GNPTG):c.735C>A (p.Cys245Ter)GNPTGPathogeniccriteria provided, single submitter
2080282NM_032520.5(GNPTG):c.108del (p.Phe36fs)GNPTGPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
2119911NM_032520.5(GNPTG):c.701del (p.Pro234fs)GNPTGPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
21713NM_032520.5(GNPTG):c.196C>T (p.Arg66Ter)GNPTGPathogeniccriteria provided, multiple submitters, no conflicts
21714NM_032520.5(GNPTG):c.318-1G>CGNPTGPathogeniccriteria provided, single submitter
21715NM_032520.5(GNPTG):c.344ACA[1] (p.Asn116del)GNPTGPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
21719NM_032520.5(GNPTG):c.609+28_610-16delGNPTGPathogenicno assertion criteria provided
21720NM_032520.5(GNPTG):c.610-1G>TGNPTGPathogenicno assertion criteria provided
21721NM_032520.5(GNPTG):c.610-2A>GGNPTGPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
21723NM_032520.5(GNPTG):c.639del (p.Phe213fs)GNPTGPathogenicno assertion criteria provided
2444155NM_032520.5(GNPTG):c.610-1G>AGNPTGPathogeniccriteria provided, multiple submitters, no conflicts
2731676NM_032520.5(GNPTG):c.384_385dup (p.Cys129fs)GNPTGPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
2795NM_032520.4(GNPTG):c.611delGGNPTGPathogenicno assertion criteria provided
2796NM_032520.5(GNPTG):c.640_667del (p.Glu214fs)GNPTGPathogenicno assertion criteria provided
2797NM_032520.5(GNPTG):c.333G>A (p.Trp111Ter)GNPTGPathogeniccriteria provided, multiple submitters, no conflicts
2799NM_032520.5(GNPTG):c.316G>A (p.Gly106Ser)GNPTGPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
3004658NM_032520.5(GNPTG):c.461_465del (p.Pro154fs)GNPTGPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
30685NM_032520.5(GNPTG):c.527-10G>AGNPTGPathogenicno assertion criteria provided
3578452NM_032520.5(GNPTG):c.514dup (p.His172fs)GNPTGPathogeniccriteria provided, multiple submitters, no conflicts
39080NM_032520.5(GNPTG):c.445del (p.Ala149fs)GNPTGPathogenicno assertion criteria provided
437454NM_032520.4(GNPTG):c.499dup (p.Leu167Profs)GNPTGPathogeniccriteria provided, multiple submitters, no conflicts
453175NM_032520.5(GNPTG):c.318-1G>AGNPTGPathogeniccriteria provided, multiple submitters, no conflicts
4807182NM_032520.5(GNPTG):c.559C>T (p.Gln187Ter)GNPTGPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
4816330NM_032520.5(GNPTG):c.13del (p.Leu5fs)GNPTGPathogeniccriteria provided, single submitter

Genes & proteins

Mendelian disease overlap and somatic drivers

GenCC: 6 · Orphanet: 1 · 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
GNPTGDefinitiveAutosomal recessiveGNPTG-mucolipidosis6

Orphanet rare-disease linkage (cohort genes)

GeneOrphanet IDRare disease
GNPTGOrphanet:423470Mucolipidosis type III gamma

Cohort genes → proteins

2 cohort genes, 2 distinct canonical proteins.

Evidence partition

SubsetGenes
multi_evidence2

Cohort genes (full)

SymbolHGNCEnsemblUniProtNameEvidence
GNPTGHGNC:23026ENSG00000090581Q9UJJ9N-acetylglucosamine-1-phosphotransferase subunit gammagencc,clinvar
UNKLHGNC:14184ENSG00000059145Q9H9P5Putative E3 ubiquitin-protein ligase UNKLclinvar

Cohort function summary

Lead sentence per gene, UniProt-curated.

SymbolProtein nameFunction (lead sentence)
GNPTGN-acetylglucosamine-1-phosphotransferase subunit gammaNon-catalytic subunit of the N-acetylglucosamine-1-phosphotransferase complex, an enzyme that catalyzes the formation of mannose 6-phosphate (M6P) markers on high mannose type oligosaccharides in the Golgi apparatus.
UNKLPutative E3 ubiquitin-protein ligase UNKLMay participate in a protein complex showing an E3 ligase activity regulated by RAC1.

Protein-family classification

Druggable: 1 · Difficult: 1 · Unknown: 0 · Druggable fraction: 0.5

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)16.0×0.228
Transcription factor14.1×0.228

Per-gene assignment

SymbolFamilyDruggable?ECInterPro (top 3)
GNPTGEnzyme (other)yes2.7.8.17Man6P_isomerase_rcpt-bd_dom_sf, DMAP1-bd, OS9-like_dom
UNKLTranscription factornoZnf_CCCH, Znf_RING, Znf_RING/FYVE/PHD

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)2
unknown0

Top tissues across cohort

TissueCohort genes
left adrenal gland1
right adrenal gland1
right adrenal gland cortex1
oocyte1
secondary oocyte1
sural nerve1

Per-gene tissue summary (top 30)

SymbolBgee breadthFANTOM5 breadthSCXATop tissues
GNPTG255ubiquitousmarkerright adrenal gland cortex, right adrenal gland, left adrenal gland
UNKL259ubiquitousyessecondary oocyte, oocyte, sural nerve

Protein interactions among cohort

Intra-cohort edges: 0.

Hub genes (top 10 by interactor count)

SymbolInteractor count
GNPTG1,199
UNKL675

Structural data

PDB: 0 · AlphaFold-only: 2 · No structure: 0

AlphaFold-only cohort genes (top 30 by pLDDT)

SymbolUniProtpLDDT
GNPTGQ9UJJ980.63
UNKLQ9H9P567.86

Function

Pathway analysis

Distinct Reactome pathways touched by cohort: 1. Enrichment computed across 2 evidence-associated genes (1 with Reactome annotation).

Pathways 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.

PathwayCohort genesFoldFDRSample cohort genes
Antigen processing: Ubiquitination & Proteasome degradation137.2×0.027UNKL

GO biological processes by enrichment

Over-representation of cohort genes vs the genome-wide background (hypergeometric test, Benjamini-Hochberg FDR; fold = observed/expected over 2 annotated cohort genes). Counts and members are kept as ground-truth; sorted by enrichment.

GO termCohort genesFoldFDRSample cohort genes
N-glycan processing to lysosome14213.0×7e-04GNPTG
carbohydrate phosphorylation11053.2×0.001GNPTG
protein ubiquitination120.7×0.048UNKL

Therapeutics

Drug target analysis

Approved (phase 4): 0 · Phase ≥3: 0 · Phased (≥1): 0 · Undrugged: 2

Druggability breadth: 0 of 2 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
GNPTG00
UNKL00

Bioactivity and enzyme data

Enzyme cohort genes (≥1 EC): 1.

Cohort enzymes (BRENDA EC)

SymbolEC numbersNames
GNPTG2.7.8.17UDP-N-acetylglucosamine-lysosomal-enzyme N-acetylglucosaminephosphotransferase

Pharmacogenomics

Cohort genes with a PharmGKB record: 2; 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 drug0
DDruggable family + AlphaFold only, no drug1GNPTG
EDifficult family or no structure, no drug1UNKL

Undrugged target profiles

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

SymbolChEMBL assaysDrugged partners (top 3)
GNPTG0
UNKL0

Clinical trials & evidence

Clinical trials

Clinical trials: 0.