Achromatopsia 6

disease
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Also known as ACHM6RCD3Aretinal cone dystrophy 3Aretinal cone dystrophy type 3A

Summary

Achromatopsia 6 (MONDO:0012398) is a disease caused by PDE6H (GenCC Strong), with 1 cohort gene.

At a glance

  • Causal gene: PDE6H (GenCC Strong)
  • Cohort genes: 1
  • ClinVar variants: 18

Clinical features

No curated clinical features (Orphanet) for this disease.

Identifiers

Disease identifiers

FieldValue
Canonical nameachromatopsia 6
Mondo IDMONDO:0012398
MeSHC566483
OMIM610024
DOIDDOID:0081025
GARD0010648
Is cancer (heuristic)no

Also known as: ACHM6 · RCD3A · retinal cone dystrophy 3A · retinal cone dystrophy type 3A

Data availability: 18 ClinVar variants · 4 GenCC gene-disease records.

Disease family

Classification path: disease › human disease › disease by body system or component › nervous system disorderperceptual disordersvision disordercolor vision disorderachromatopsiaachromatopsia 6

Related subtypes (5): achromatopsia 2, achromatopsia 3, blue cone monochromacy, achromatopsia 4, achromatopsia 7

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

18 retrieved; paginated sample, class counts are floors:

10 uncertain significance, 5 benign, 2 conflicting classifications of pathogenicity, 1 benign/likely benign

ClinVarVariant (HGVS)GeneClassificationReview
37245NM_006205.3(PDE6H):c.35C>G (p.Ser12Ter)PDE6HConflicting classifications of pathogenicitycriteria provided, conflicting classifications
881091NM_006205.3(PDE6H):c.232G>T (p.Ala78Ser)PDE6HConflicting classifications of pathogenicitycriteria provided, conflicting classifications
1098417NM_006205.3(PDE6H):c.237G>C (p.Gln79His)PDE6HUncertain significancecriteria provided, multiple submitters, no conflicts
307783NM_006205.3(PDE6H):c.*47G>CPDE6HUncertain significancecriteria provided, single submitter
307784NM_006205.3(PDE6H):c.*71C>TPDE6HUncertain significancecriteria provided, single submitter
307785NM_006205.3(PDE6H):c.*77C>GPDE6HUncertain significancecriteria provided, single submitter
307786NM_006205.3(PDE6H):c.*134G>APDE6HUncertain significancecriteria provided, single submitter
307789NM_006205.3(PDE6H):c.*358T>GPDE6HUncertain significancecriteria provided, single submitter
307790NM_006205.3(PDE6H):c.*369A>GPDE6HUncertain significancecriteria provided, single submitter
3780109NM_006205.3(PDE6H):c.135-2A>GPDE6HUncertain significancecriteria provided, single submitter
883445NM_006205.3(PDE6H):c.-42C>TPDE6HUncertain significancecriteria provided, single submitter
883446NM_006205.3(PDE6H):c.59G>A (p.Arg20His)PDE6HUncertain significancecriteria provided, multiple submitters, no conflicts
307780NM_006205.3(PDE6H):c.-59G>CPDE6HBenigncriteria provided, multiple submitters, no conflicts
307781NM_006205.3(PDE6H):c.-29G>CPDE6HBenign/Likely benigncriteria provided, multiple submitters, no conflicts
307782NM_006205.3(PDE6H):c.195A>G (p.Pro65=)PDE6HBenigncriteria provided, multiple submitters, no conflicts
307787NM_006205.3(PDE6H):c.*301A>GPDE6HBenigncriteria provided, multiple submitters, no conflicts
307788NM_006205.3(PDE6H):c.*319T>CPDE6HBenigncriteria provided, multiple submitters, no conflicts
883444NM_006205.3(PDE6H):c.-73C>APDE6HBenigncriteria 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
PDE6HStrongAutosomal recessiveachromatopsia 66

Orphanet rare-disease linkage (cohort genes)

GeneOrphanet IDRare disease
PDE6HOrphanet:49382Achromatopsia

Cohort genes → proteins

1 cohort genes, 1 distinct canonical proteins.

Evidence partition

SubsetGenes
multi_evidence1

Cohort genes (full)

SymbolHGNCEnsemblUniProtNameEvidence
PDE6HHGNC:8790ENSG00000139053Q13956Retinal cone rhodopsin-sensitive cGMP 3’,5’-cyclic phosphodiesterase subunit gammagencc,clinvar

Cohort function summary

Lead sentence per gene, UniProt-curated.

SymbolProtein nameFunction (lead sentence)
PDE6HRetinal cone rhodopsin-sensitive cGMP 3’,5’-cyclic phosphodiesterase subunit gammaParticipates in processes of transmission and amplification of the visual signal. cGMP-PDEs are the effector molecules in G-protein-mediated phototransduction in vertebrate rods and cones.

Protein-family classification

Druggable: 0 · Difficult: 0 · Unknown: 1 · Druggable fraction: 0.0

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
Other/Unknown11.8×0.558

Per-gene assignment

SymbolFamilyDruggable?ECInterPro (top 3)
PDE6HOther/UnknownnoPDE6_gamma, PDE6_gamma_sf

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
male germ line stem cell (sensu Vertebrata) in testis1
monocyte1
mononuclear cell1

Per-gene tissue summary (top 30)

SymbolBgee breadthFANTOM5 breadthSCXATop tissues
PDE6H83tissue_specificmarkermale germ line stem cell (sensu Vertebrata) in testis, monocyte, mononuclear cell

Protein interactions among cohort

Intra-cohort edges: 0.

Hub genes (top 10 by interactor count)

SymbolInteractor count
PDE6H875

Structural data

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

AlphaFold-only cohort genes (top 30 by pLDDT)

SymbolUniProtpLDDT
PDE6HQ1395667.61

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
positive regulation of G protein-coupled receptor signaling pathway11053.2×0.004PDE6H
positive regulation of epidermal growth factor receptor signaling pathway1495.6×0.004PDE6H
positive regulation of MAPK cascade180.6×0.013PDE6H
visual perception179.5×0.013PDE6H

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
PDE6HVARDENAFIL

Top cohort targets by molecule count

SymbolMoleculesMax phase
PDE6H64

Drugs targeting cohort genes (top 30)

MoleculeMax phaseTargets in cohort
VARDENAFIL4PDE6H
SILDENAFIL4PDE6H
TADALAFIL4PDE6H
DIPYRIDAMOLE4PDE6H
ZAPRINAST2PDE6H
TBA-73712PDE6H

Bioactivity and enzyme data

Enzyme cohort genes (≥1 EC): 0.

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

SymbolAssaysType breakdown
PDE6H51Binding:49, ADMET:2

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)
VARDENAFIL4PDE6H
SILDENAFIL4PDE6H
TADALAFIL4PDE6H
DIPYRIDAMOLE4PDE6H
ZAPRINAST2PDE6H
TBA-73712PDE6H

Druggability pyramid

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

TierDefinitionGenesSymbols
AApproved (phase 4 drug)1PDE6H
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: 0.