Weill-Marchesani 4 syndrome, recessive

disease
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Also known as 15q26.3 microdeletion syndromeichthyosis-short stature-brachydactyly-microspherophakia syndromeWeill-Marchesani syndrome 4Weill-Marchesani-like syndromeWMS4

Summary

Weill-Marchesani 4 syndrome, recessive (MONDO:0013176) is a disease caused by ADAMTS17 (GenCC Definitive), with 1 cohort gene.

At a glance

  • Prevalence: <1 / 1 000 000 (Worldwide) [Orphanet-validated]
  • Causal gene: ADAMTS17 (GenCC Definitive)
  • Cohort genes: 1
  • ClinVar variants: 361

Clinical features

Epidemiology

Prevalence records

2 prevalence record(s), Orphanet:

TypeClassValueGeographyValidation
Cases/families7WorldwideValidated
Point prevalence<1 / 1 000 000WorldwideValidated

Identifiers

Disease identifiers

FieldValue
Canonical nameWeill-Marchesani 4 syndrome, recessive
Mondo IDMONDO:0013176
MeSHC567710
OMIM613195
Orphanet363992
DOIDDOID:0061276
UMLSC2750787
MedGen416383
GARD0017579
Is cancer (heuristic)no

Also known as: 15q26.3 microdeletion syndrome · ichthyosis-short stature-brachydactyly-microspherophakia syndrome · Weill-Marchesani syndrome 4 · Weill-Marchesani-like syndrome · WMS4

Data availability: 361 ClinVar variants · 6 GenCC gene-disease records.

Disease family

Classification path: disease › human disease › disease by etiologic mechanism › disease of genetic or genomic mechanism › hereditary disease › autosomal genetic disease › Weill-Marchesani syndromeWeill-Marchesani 4 syndrome, recessive

Related subtypes (3): Weill-Marchesani syndrome 1, Weill-Marchesani syndrome 2, dominant, Weill-Marchesani syndrome 3

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

361 retrieved; paginated sample, class counts are floors:

165 uncertain significance, 91 benign, 65 conflicting classifications of pathogenicity, 20 likely benign, 6 benign/likely benign, 5 likely pathogenic, 5 pathogenic, 4 pathogenic/likely pathogenic

ClinVarVariant (HGVS)GeneClassificationReview
1687282NM_139057.4(ADAMTS17):c.662_665del (p.Glu221fs)ADAMTS17Pathogeniccriteria provided, single submitter
2570579NM_139057.4(ADAMTS17):c.2560C>T (p.Arg854Ter)ADAMTS17Pathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
3065118NM_139057.4(ADAMTS17):c.197_206del (p.Arg66fs)ADAMTS17Pathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
3153NM_139057.4(ADAMTS17):c.2456-1dupADAMTS17Pathogenicno assertion criteria provided
3154NM_139057.4(ADAMTS17):c.760C>T (p.Gln254Ter)ADAMTS17Pathogenicno assertion criteria provided
4761867NM_139057.4(ADAMTS17):c.1297C>T (p.Arg433Ter)ADAMTS17Pathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
492940NM_139057.4(ADAMTS17):c.652del (p.Asp218fs)ADAMTS17Pathogeniccriteria provided, single submitter
492941NM_139057.4(ADAMTS17):c.873+1G>TADAMTS17Pathogenicno assertion criteria provided
3155NM_139057.4(ADAMTS17):c.1721+1G>ALOC130058037Pathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
1210284NM_139057.4(ADAMTS17):c.2254A>G (p.Ile752Val)ADAMTS17Likely pathogenicno assertion criteria provided
1210285NM_139057.4(ADAMTS17):c.2984G>A (p.Arg995Gln)ADAMTS17Likely pathogenicno assertion criteria provided
2570576NM_139057.4(ADAMTS17):c.1239G>C (p.Met413Ile)ADAMTS17Likely pathogeniccriteria provided, single submitter
2570577NM_139057.4(ADAMTS17):c.1630G>T (p.Gly544Ter)ADAMTS17Likely pathogeniccriteria provided, single submitter
2570578NC_000015.10:g.(99976222_99993049)_(99997590_100048857)delLOC113939942Likely pathogeniccriteria provided, single submitter
1442566NM_139057.4(ADAMTS17):c.1873G>T (p.Ala625Ser)ADAMTS17Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
1528321NM_139057.4(ADAMTS17):c.1924_1929dup (p.Lys642_Glu643dup)ADAMTS17Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
315247NM_139057.4(ADAMTS17):c.3285G>A (p.Ser1095=)ADAMTS17Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
315250NM_139057.4(ADAMTS17):c.3165G>C (p.Thr1055=)ADAMTS17Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
315254NM_139057.4(ADAMTS17):c.3127+12C>TADAMTS17Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
315256NM_139057.4(ADAMTS17):c.3068G>A (p.Cys1023Tyr)ADAMTS17Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
315259NM_139057.4(ADAMTS17):c.2883G>A (p.Pro961=)ADAMTS17Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
315263NM_139057.4(ADAMTS17):c.2592-9G>AADAMTS17Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
315264NM_139057.4(ADAMTS17):c.2592-10C>TADAMTS17Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
315268NM_139057.4(ADAMTS17):c.2364G>A (p.Ala788=)ADAMTS17Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
315271NM_139057.4(ADAMTS17):c.2109G>A (p.Lys703=)ADAMTS17Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
315272NM_139057.4(ADAMTS17):c.2067C>T (p.Cys689=)ADAMTS17Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
315273NM_139057.4(ADAMTS17):c.2040C>T (p.Ile680=)ADAMTS17Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
315274NM_139057.4(ADAMTS17):c.2004C>T (p.His668=)ADAMTS17Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
315275NM_139057.4(ADAMTS17):c.1981T>C (p.Tyr661His)ADAMTS17Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
315276NM_139057.4(ADAMTS17):c.1971C>G (p.Pro657=)ADAMTS17Conflicting classifications of pathogenicitycriteria provided, conflicting classifications

Genes & proteins

Mendelian disease overlap and somatic drivers

GenCC: 6 · 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
ADAMTS17DefinitiveAutosomal recessiveWeill-Marchesani 4 syndrome, recessive6

Orphanet rare-disease linkage (cohort genes)

GeneOrphanet IDRare disease
ADAMTS17Orphanet:3449Weill-Marchesani syndrome
ADAMTS17Orphanet:363992Ichthyosis-short stature-brachydactyly-microspherophakia syndrome

Cohort genes → proteins

1 cohort genes, 1 distinct canonical proteins.

Evidence partition

SubsetGenes
multi_evidence1

Cohort genes (full)

SymbolHGNCEnsemblUniProtNameEvidence
ADAMTS17HGNC:17109ENSG00000140470Q8TE56A disintegrin and metalloproteinase with thrombospondin motifs 17gencc,clinvar

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
Protease136.6×0.027

Per-gene assignment

SymbolFamilyDruggable?ECInterPro (top 3)
ADAMTS17ProteaseyesTSP1_rpt, Peptidase_M12B, Peptidase_M12B_N

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
primordial germ cell in gonad1
right lobe of liver1
thymus1

Per-gene tissue summary (top 30)

SymbolBgee breadthFANTOM5 breadthSCXATop tissues
ADAMTS17177broadmarkerthymus, primordial germ cell in gonad, right lobe of liver

Protein interactions among cohort

Intra-cohort edges: 0.

Hub genes (top 10 by interactor count)

SymbolInteractor count
ADAMTS17817

Structural data

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

AlphaFold-only cohort genes (top 30 by pLDDT)

SymbolUniProtpLDDT
ADAMTS17Q8TE5670.06

Function

Pathway analysis

Distinct Reactome pathways touched by cohort: 9. 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
Defective B3GALTL causes PpS1308.6×0.014ADAMTS17
O-glycosylation of TSR domain-containing proteins1300.5×0.014ADAMTS17
Diseases associated with O-glycosylation of proteins1215.5×0.014ADAMTS17
O-linked glycosylation1144.6×0.014ADAMTS17
Diseases of glycosylation1131.3×0.014ADAMTS17
Diseases of metabolism180.4×0.019ADAMTS17
Post-translational protein modification119.2×0.067ADAMTS17
Disease113.1×0.081ADAMTS17
Metabolism of proteins112.4×0.081ADAMTS17

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
extracellular matrix organization1122.1×0.016ADAMTS17
proteolysis134.2×0.029ADAMTS17

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
ADAMTS1700

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 drug0
DDruggable family + AlphaFold only, no drug1ADAMTS17
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)
ADAMTS170

Clinical trials & evidence

Clinical trials

Clinical trials: 0.