megalencephalic leukoencephalopathy with subcortical cysts 2A

disease
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Also known as megalencephalic leukoencephalopathy with subcortical cysts type 2AMLC2A

Summary

megalencephalic leukoencephalopathy with subcortical cysts 2A (MONDO:0013490) is a disease caused by HEPACAM (GenCC Strong), with 1 cohort gene.

At a glance

  • Causal gene: HEPACAM (GenCC Strong)
  • Cohort genes: 1
  • ClinVar variants: 27

Clinical features

No curated clinical features (Orphanet) for this disease.

Identifiers

Disease identifiers

FieldValue
Canonical namemegalencephalic leukoencephalopathy with subcortical cysts 2A
Mondo IDMONDO:0013490
OMIM613925
DOIDDOID:0080318
UMLSC3151355
MedGen462705
GARD0015728
Is cancer (heuristic)no

Also known as: megalencephalic leukoencephalopathy with subcortical cysts 2A · megalencephalic leukoencephalopathy with subcortical cysts type 2A · MLC2A

Data availability: 27 ClinVar variants · 3 GenCC gene-disease records.

Disease family

Classification path: disease › human disease › disease by body system or component › nervous system disordercentral nervous system disorderbrain disorderleukoencephalopathy, megalencephalicmegalencephalic leukoencephalopathy with subcortical cystsmegalencephalic leukoencephalopathy with subcortical cysts 2A

Related subtypes (2): megalencephalic leukoencephalopathy with subcortical cysts 2B, remitting, with or without intellectual disability, megalencephalic leukoencephalopathy with subcortical cysts 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

27 retrieved; paginated sample, class counts are floors:

9 uncertain significance, 5 conflicting classifications of pathogenicity, 4 pathogenic, 3 likely pathogenic, 2 benign, 2 pathogenic/likely pathogenic, 1 likely benign, 1 benign/likely benign

ClinVarVariant (HGVS)GeneClassificationReview
30914NM_152722.5(HEPACAM):c.587C>A (p.Ser196Tyr)HEPACAMPathogeniccriteria provided, single submitter
30915NM_152722.5(HEPACAM):c.789G>A (p.Trp263Ter)HEPACAMPathogenicno assertion criteria provided
30916NM_152722.5(HEPACAM):c.275G>A (p.Arg92Gln)HEPACAMPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
30917NM_152722.5(HEPACAM):c.631G>A (p.Asp211Asn)HEPACAMPathogenicno assertion criteria provided
30919NM_152722.5(HEPACAM):c.265G>A (p.Gly89Ser)HEPACAMPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
30921NM_152722.5(HEPACAM):c.274C>T (p.Arg92Trp)HEPACAMPathogeniccriteria provided, multiple submitters, no conflicts
30918NM_152722.5(HEPACAM):c.292C>T (p.Arg98Cys)HEPACAMLikely pathogeniccriteria provided, single submitter
3233388NM_152722.5(HEPACAM):c.949-2A>GHEPACAMLikely pathogeniccriteria provided, single submitter
433199NM_152722.5(HEPACAM):c.442C>T (p.Pro148Ser)HEPACAMLikely pathogenicno assertion criteria provided
1031210NM_152722.5(HEPACAM):c.259G>A (p.Val87Ile)HEPACAMConflicting classifications of pathogenicitycriteria provided, conflicting classifications
1218905NM_152722.5(HEPACAM):c.382G>A (p.Asp128Asn)HEPACAMConflicting classifications of pathogenicitycriteria provided, conflicting classifications
1367723NM_152722.5(HEPACAM):c.96G>C (p.Glu32Asp)HEPACAMConflicting classifications of pathogenicitycriteria provided, conflicting classifications
393149NM_152722.5(HEPACAM):c.1198G>A (p.Val400Met)HEPACAMConflicting classifications of pathogenicitycriteria provided, conflicting classifications
931058NM_152722.5(HEPACAM):c.139G>A (p.Val47Met)HEPACAMConflicting classifications of pathogenicitycriteria provided, conflicting classifications
1031211NM_152722.5(HEPACAM):c.665C>T (p.Pro222Leu)HEPACAMUncertain significancecriteria provided, single submitter
1031682NM_152722.5(HEPACAM):c.922A>G (p.Met308Val)HEPACAMUncertain significancecriteria provided, multiple submitters, no conflicts
1187211NM_152722.5(HEPACAM):c.217C>T (p.Arg73Trp)HEPACAMUncertain significancecriteria provided, multiple submitters, no conflicts
1203684NM_152722.5(HEPACAM):c.1169_1180del (p.Ser390_Arg393del)HEPACAMUncertain significancecriteria provided, multiple submitters, no conflicts
1339869NM_152722.5(HEPACAM):c.616C>T (p.Arg206Cys)HEPACAMUncertain significancecriteria provided, multiple submitters, no conflicts
1510664NM_152722.5(HEPACAM):c.484G>A (p.Glu162Lys)HEPACAMUncertain significancecriteria provided, multiple submitters, no conflicts
3362692NM_152722.5(HEPACAM):c.1238A>T (p.Glu413Val)HEPACAMUncertain significancecriteria provided, single submitter
435410NM_152722.5(HEPACAM):c.614C>T (p.Thr205Ile)HEPACAMUncertain significancecriteria provided, multiple submitters, no conflicts
638399NM_152722.5(HEPACAM):c.128T>A (p.Ile43Asn)HEPACAMUncertain significancecriteria provided, multiple submitters, no conflicts
1582595NM_152722.5(HEPACAM):c.1062C>T (p.Pro354=)HEPACAMLikely benigncriteria provided, multiple submitters, no conflicts
262680NM_152722.5(HEPACAM):c.618C>T (p.Arg206=)HEPACAMBenigncriteria provided, multiple submitters, no conflicts
262681NM_152722.5(HEPACAM):c.652A>G (p.Met218Val)HEPACAMBenigncriteria provided, multiple submitters, no conflicts
303335NM_152722.5(HEPACAM):c.258G>A (p.Glu86=)HEPACAMBenign/Likely benigncriteria provided, multiple submitters, no conflicts

Genes & proteins

Mendelian disease overlap and somatic drivers

GenCC: 8 · 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
HEPACAMStrongAutosomal recessivemegalencephalic leukoencephalopathy with subcortical cysts 2A8

Orphanet rare-disease linkage (cohort genes)

GeneOrphanet IDRare disease
HEPACAMOrphanet:210548Macrocephaly-intellectual disability-autism syndrome
HEPACAMOrphanet:2478Megalencephalic leukoencephalopathy with subcortical cysts

Cohort genes → proteins

1 cohort genes, 1 distinct canonical proteins.

Evidence partition

SubsetGenes
multi_evidence1

Cohort genes (full)

SymbolHGNCEnsemblUniProtNameEvidence
HEPACAMHGNC:26361ENSG00000165478Q14CZ8Hepatic and glial cell adhesion moleculegencc,clinvar

Cohort function summary

Lead sentence per gene, UniProt-curated.

SymbolProtein nameFunction (lead sentence)
HEPACAMHepatic and glial cell adhesion moleculeInvolved in regulating cell motility and cell-matrix interactions.

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
Antibody/Immunoglobulin129.2×0.034

Per-gene assignment

SymbolFamilyDruggable?ECInterPro (top 3)
HEPACAMAntibody/ImmunoglobulinyesIg_sub2, Ig_sub, Ig-like_dom

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
lateral globus pallidus1
medial globus pallidus1
substantia nigra pars reticulata1

Per-gene tissue summary (top 30)

SymbolBgee breadthFANTOM5 breadthSCXATop tissues
HEPACAM167broadmarkerlateral globus pallidus, substantia nigra pars reticulata, medial globus pallidus

Protein interactions among cohort

Intra-cohort edges: 0.

Hub genes (top 10 by interactor count)

SymbolInteractor count
HEPACAM661

Structural data

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

Cohort genes with PDB structures (top 30)

SymbolUniProtPDB entries
HEPACAMQ14CZ81

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
protein localization to cell-cell junction11872.4×0.002HEPACAM
regulation of cell cycle174.6×0.027HEPACAM
immune response147.1×0.027HEPACAM
cell adhesion137.5×0.027HEPACAM

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
HEPACAM00

Bioactivity and enzyme data

Enzyme cohort genes (≥1 EC): 0.

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 drug1HEPACAM
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)
HEPACAM0

Clinical trials & evidence

Clinical trials

Clinical trials: 0.