neuropathy, hereditary motor and sensory, type VIc, with optic atrophy

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Also known as Charcot-Marie-Tooth Disease, Type 6CCMT 6CCMT6CHMSN 6CHMSN6C

Summary

neuropathy, hereditary motor and sensory, type VIc, with optic atrophy (MONDO:0032792) is a disease caused by PDXK (GenCC Strong), with 1 cohort gene and 1 clinical trial.

At a glance

  • Causal gene: PDXK (GenCC Strong)
  • Cohort genes: 1
  • ClinVar variants: 9
  • Clinical trials: 1

Clinical features

No curated clinical features (Orphanet) for this disease.

Identifiers

Disease identifiers

FieldValue
Canonical nameneuropathy, hereditary motor and sensory, type VIc, with optic atrophy
Mondo IDMONDO:0032792
OMIM618511
UMLSC5193137
MedGen1680245
GARD0025745
Is cancer (heuristic)no

Also known as: Charcot-Marie-Tooth Disease, Type 6C · CMT 6C · CMT6C · HMSN 6C · HMSN6C · neuropathy, hereditary motor and sensory, type VIc, with optic atrophy

Data availability: 9 ClinVar variants · 2 GenCC gene-disease records.

Disease family

Classification path: disease › human disease › disease by body system or component › nervous system disorderperipheral nervous system disorderperipheral neuropathyhereditary peripheral neuropathyhereditary motor and sensory neuropathyhereditary motor and sensory neuropathy type 6neuropathy, hereditary motor and sensory, type VIc, with optic atrophy

Related subtypes (2): neuropathy, hereditary motor and sensory, type 6A, neuropathy, hereditary motor and sensory, type 6B

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

9 retrieved; paginated sample, class counts are floors:

3 benign, 2 uncertain significance, 2 pathogenic, 1 likely benign, 1 likely pathogenic

ClinVarVariant (HGVS)GeneClassificationReview
636264NM_003681.5(PDXK):c.682G>A (p.Ala228Thr)PDXKPathogeniccriteria provided, multiple submitters, no conflicts
636265NM_003681.5(PDXK):c.659G>A (p.Arg220Gln)PDXKPathogenicno assertion criteria provided
812519NM_003681.5(PDXK):c.225T>A (p.Asn75Lys)PDXKLikely pathogeniccriteria provided, single submitter
2348515NM_003681.5(PDXK):c.842G>A (p.Gly281Glu)PDXKUncertain significancecriteria provided, multiple submitters, no conflicts
2434643NM_003681.5(PDXK):c.509G>A (p.Arg170Gln)PDXKUncertain significancecriteria provided, single submitter
1013193NM_003681.5(PDXK):c.374C>T (p.Ser125Leu)PDXKLikely benigncriteria provided, multiple submitters, no conflicts
1267789NM_003681.5(PDXK):c.*14T>CPDXKBenigncriteria provided, multiple submitters, no conflicts
1268466NM_003681.5(PDXK):c.387G>A (p.Pro129=)PDXKBenigncriteria provided, multiple submitters, no conflicts
1327004NM_003681.5(PDXK):c.331+26T>GPDXKBenigncriteria provided, multiple submitters, no conflicts

Genes & proteins

Mendelian disease overlap and somatic drivers

GenCC: 2 · Orphanet: 0 · 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
PDXKStrongAutosomal recessiveneuropathy, hereditary motor and sensory, type VIc, with optic atrophy2

Cohort genes → proteins

1 cohort genes, 1 distinct canonical proteins.

Evidence partition

SubsetGenes
multi_evidence1

Cohort genes (full)

SymbolHGNCEnsemblUniProtNameEvidence
PDXKHGNC:8819ENSG00000160209O00764Pyridoxal kinasegencc,clinvar

Cohort function summary

Lead sentence per gene, UniProt-curated.

SymbolProtein nameFunction (lead sentence)
PDXKPyridoxal kinaseCatalyzes the phosphorylation of the dietary vitamin B6 vitamers pyridoxal (PL), pyridoxine (PN) and pyridoxamine (PM) to form pyridoxal 5’-phosphate (PLP), pyridoxine 5’-phosphate (PNP) and pyridoxamine 5’-phosphate (PMP), respectively.

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
Kinase127.7×0.036

Per-gene assignment

SymbolFamilyDruggable?ECInterPro (top 3)
PDXKKinaseyes2.7.1.35PyrdxlKinase, PM/HMP-P_kinase-1, Ribokinase-like

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
adult organism1
left testis1
right testis1

Per-gene tissue summary (top 30)

SymbolBgee breadthFANTOM5 breadthSCXATop tissues
PDXK295ubiquitousmarkerleft testis, right testis, adult organism

Protein interactions among cohort

Intra-cohort edges: 0.

Hub genes (top 10 by interactor count)

SymbolInteractor count
PDXK1,899

Structural data

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

Cohort genes with PDB structures (top 30)

SymbolUniProtPDB entries
PDXKO0076410

Function

Pathway analysis

Distinct Reactome pathways touched by cohort: 2. Enrichment computed across 1 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
Vitamin B6 activation to pyridoxal phosphate13806.7×5e-04PDXK
Neutrophil degranulation123.1×0.043PDXK

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
pyridoxal 5’-phosphate salvage116852.0×9e-05PDXK
pyridoxal metabolic process116852.0×9e-05PDXK
pyridoxamine metabolic process18426.0×1e-04PDXK

Therapeutics

Drug target analysis

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

Druggability breadth: 1 of 1 evidence-associated genes (100%) have a ChEMBL target (buckets above are over the deeply-mined display cohort).

Genes with an approved drug

The molecule shown is one approved compound that hits the gene — not necessarily a drug of choice or one indicated for this disease.

SymbolExample approved molecule
PDXKSUNITINIB

Top cohort targets by molecule count

SymbolMoleculesMax phase
PDXK64

Drugs targeting cohort genes (top 30)

MoleculeMax phaseTargets in cohort
SUNITINIB4PDXK
VOLASERTIB3PDXK
LUCITANIB2PDXK
TG100-1152PDXK
FGFR INHIBITOR DEBIO 13472PDXK
BI-25362PDXK

Bioactivity and enzyme data

Enzyme cohort genes (≥1 EC): 1.

Cohort genes with ChEMBL bioactivity (full, sorted by assay count)

SymbolAssaysType breakdown
PDXK21Binding:20, ADMET:1

Cohort enzymes (BRENDA EC)

SymbolEC numbersNames
PDXK2.7.1.35pyridoxal kinase

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

6 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.

CompoundMax phaseCohort target (bioactivity)
SUNITINIB4PDXK
VOLASERTIB3PDXK
LUCITANIB2PDXK
TG100-1152PDXK
FGFR INHIBITOR DEBIO 13472PDXK
BI-25362PDXK

Druggability pyramid

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

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

Undrugged target profiles

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

Clinical trials & evidence

Clinical trials

Clinical trials: 1.

Phase distribution (across all retrieved trials)

PhaseTrials
Not specified1

Top trials by phase / activity

NCTPhaseStatusTitle
NCT05687474Not specifiedCOMPLETEDBaby Detect : Genomic Newborn Screening