Geleophysic dysplasia

disease
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Also known as geleophysic dwarfismgeleophysic dwarfism syndrome

Summary

Geleophysic dysplasia (MONDO:0000127) is a disease with 4 cohort genes. The dominant Reactome pathway is Elastic fibre formation (3 cohort genes).

At a glance

  • Prevalence: <1 / 1 000 000 (Worldwide) [Orphanet-validated]
  • Cohort genes: 4
  • ClinVar variants: 120
  • Phenotypes (HPO): 47

Clinical features

Epidemiology

Prevalence records

2 prevalence record(s), Orphanet:

TypeClassValueGeographyValidation
Cases/families27WorldwideValidated
Point prevalence<1 / 1 000 000WorldwideValidated

Signs & symptoms

Clinical features (HPO)

47 HPO clinical features (Orphanet curated; top 47 by frequency):

HPO IDTermFrequency
HP:0004322Short statureVery frequent (80-99%)
HP:0000219Thin upper lip vermilionFrequent (30-79%)
HP:0000311Round faceFrequent (30-79%)
HP:0000343Long philtrumFrequent (30-79%)
HP:0000405Conductive hearing impairmentFrequent (30-79%)
HP:0000463Anteverted naresFrequent (30-79%)
HP:0001072Thickened skinFrequent (30-79%)
HP:0001156BrachydactylyFrequent (30-79%)
HP:0001376Limitation of joint mobilityFrequent (30-79%)
HP:0001511Intrauterine growth retardationFrequent (30-79%)
HP:0001561PolyhydramniosFrequent (30-79%)
HP:0001773Short footFrequent (30-79%)
HP:0002099AsthmaFrequent (30-79%)
HP:0002750Delayed skeletal maturationFrequent (30-79%)
HP:0003026Short long boneFrequent (30-79%)
HP:0003196Short noseFrequent (30-79%)
HP:0004279Short palmFrequent (30-79%)
HP:0004890Elevated pulmonary artery pressureFrequent (30-79%)
HP:0006536Airway obstructionFrequent (30-79%)
HP:0011428Short fetal femur lengthFrequent (30-79%)
HP:0034281Phalangeal cone-shaped epiphysesFrequent (30-79%)
HP:0034350Valvular pulmonary stenosisFrequent (30-79%)
HP:0034392Joint contractureFrequent (30-79%)
HP:0040261Increased size of nasopharyngeal adenoidsFrequent (30-79%)
HP:0410018Recurrent ear infectionsFrequent (30-79%)
HP:0000316HypertelorismOccasional (5-29%)
HP:0000483AstigmatismOccasional (5-29%)
HP:0000540HypermetropiaOccasional (5-29%)
HP:0000545MyopiaOccasional (5-29%)
HP:0001385Hip dysplasiaOccasional (5-29%)
HP:0001601LaryngomalaciaOccasional (5-29%)
HP:0001602Laryngeal stenosisOccasional (5-29%)
HP:0001650Aortic valve stenosisOccasional (5-29%)
HP:0001653Mitral regurgitationOccasional (5-29%)
HP:0001718Mitral stenosisOccasional (5-29%)
HP:0002091Restrictive ventilatory defectOccasional (5-29%)
HP:0002093Respiratory insufficiencyOccasional (5-29%)
HP:0002240HepatomegalyOccasional (5-29%)
HP:0002777Tracheal stenosisOccasional (5-29%)
HP:0002779TracheomalaciaOccasional (5-29%)
HP:0002870Obstructive sleep apneaOccasional (5-29%)
HP:0006530Abnormal pulmonary interstitial morphologyOccasional (5-29%)
HP:0006695Atrioventricular canal defectOccasional (5-29%)
HP:0012185Constrictive median neuropathyOccasional (5-29%)
HP:0030051Tip-toe gaitOccasional (5-29%)
HP:0000501GlaucomaVery rare (<1-4%)
HP:0000821HypothyroidismVery rare (<1-4%)

Identifiers

Disease identifiers

FieldValue
Canonical namegeleophysic dysplasia
Mondo IDMONDO:0000127
OMIM231050
Orphanet2623
DOIDDOID:0111724
ICD-11518828851
SNOMED CT28557005
UMLSC3489726
MedGen483679
GARD0002449
MedDRA10063361
Is cancer (heuristic)no

Also known as: geleophysic dwarfism · geleophysic dwarfism syndrome

Data availability: 120 ClinVar variants · 2 GenCC gene-disease records · 1 cell line.

Disease family

An umbrella term covering 3 Mondo subtypes.

Classification path: disease › human disease › disease by etiologic mechanism › disease of genetic or genomic mechanism › hereditary diseaseskeletal dysplasiaacromelic dysplasiageleophysic dysplasia

Related subtypes (17): Acromicric dysplasia, pseudohypoparathyroidism type 1A, Angel-shaped phalango-epiphyseal dysplasia, Myhre syndrome, Leri pleonosteosis, peripheral dysostosis, short-rib thoracic dysplasia 9 with or without polydactyly, terminal osseous dysplasia-pigmentary defects syndrome, intellectual disability-balding-patella luxation-acromicria syndrome, acrocapitofemoral dysplasia, pseudohypoparathyroidism type 1C, pseudopseudohypoparathyroidism, short stature-brachydactyly-obesity-global developmental delay syndrome, trichorhinophalangeal syndrome, Weill-Marchesani syndrome, craniofacial conodysplasia, acrodysostosis

Subtypes (3): geleophysic dysplasia 1, geleophysic dysplasia 2, geleophysic dysplasia 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

120 retrieved; paginated sample, class counts are floors:

59 conflicting classifications of pathogenicity, 35 benign/likely benign, 22 benign, 2 uncertain significance, 1 likely benign, 1 pathogenic/likely pathogenic

ClinVarVariant (HGVS)GeneClassificationReview
430150NM_000138.5(FBN1):c.5183C>T (p.Ala1728Val)FBN1Pathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
695NM_014694.4(ADAMTSL2):c.340G>A (p.Glu114Lys)ADAMTSL2Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
137300NM_000138.5(FBN1):c.-176A>TFBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
137306NM_000138.5(FBN1):c.3590-8T>CFBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
161237NM_000138.5(FBN1):c.7846A>G (p.Ile2616Val)FBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
161244NM_000138.5(FBN1):c.1027G>A (p.Gly343Arg)FBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
16445NM_000138.5(FBN1):c.8176C>T (p.Arg2726Trp)FBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
178035NM_000138.5(FBN1):c.247+10T>CFBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
199954NM_000138.5(FBN1):c.8185A>C (p.Lys2729Gln)FBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
199960NM_000138.5(FBN1):c.902G>T (p.Gly301Val)FBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
228686NM_000138.5(FBN1):c.6314-15G>AFBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
237081NM_000138.5(FBN1):c.1884C>T (p.Cys628=)FBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
237089NM_000138.5(FBN1):c.3936C>T (p.Ser1312=)FBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
237105NM_000138.5(FBN1):c.783T>C (p.Asn261=)FBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
237106NM_000138.5(FBN1):c.8149G>A (p.Glu2717Lys)FBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
263431NM_000138.5(FBN1):c.7056C>T (p.Ser2352=)FBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
263632NM_000138.5(FBN1):c.3089A>G (p.Asn1030Ser)FBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
264529NM_000138.5(FBN1):c.2094G>T (p.Pro698=)FBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
281682NM_000138.5(FBN1):c.4998C>T (p.Thr1666=)FBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
316306NM_000138.5(FBN1):c.*2395G>AFBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
316328NM_000138.5(FBN1):c.*1580G>AFBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
316331NM_000138.5(FBN1):c.*1484C>TFBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
316337NM_000138.5(FBN1):c.*1325T>CFBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
316338NM_000138.5(FBN1):c.*1298C>GFBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
316340NM_000138.5(FBN1):c.*1245C>TFBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
316345NM_000138.5(FBN1):c.*967C>TFBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
316350NM_000138.5(FBN1):c.*764G>AFBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
316360NM_000138.5(FBN1):c.8310C>T (p.His2770=)FBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
316362NM_000138.5(FBN1):c.8202C>T (p.Asn2734=)FBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
316364NM_000138.5(FBN1):c.7661G>A (p.Arg2554Gln)FBN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications

Genes & proteins

Mendelian disease overlap and somatic drivers

GenCC: 64 · Orphanet: 19 · 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
FBN1StrongAutosomal dominantAcromicric dysplasia24
LTBP2StrongAutosomal dominantgeleophysic dysplasia 320
LTBP3StrongAutosomal dominantgeleophysic dysplasia 320

Orphanet rare-disease linkage (cohort genes)

GeneOrphanet IDRare disease
FBN1Orphanet:1885Isolated ectopia lentis
FBN1Orphanet:2084Glaucoma-ectopia lentis-microspherophakia-stiff joints-short stature syndrome
FBN1Orphanet:2462Shprintzen-Goldberg syndrome
FBN1Orphanet:2623Geleophysic dysplasia
FBN1Orphanet:2833Stiff skin syndrome
FBN1Orphanet:284963Marfan syndrome type 1
FBN1Orphanet:284979Neonatal Marfan syndrome
FBN1Orphanet:300382Progeroid and marfanoid aspect-lipodystrophy syndrome
FBN1Orphanet:3449Weill-Marchesani syndrome
FBN1Orphanet:91387Familial thoracic aortic aneurysm and aortic dissection
FBN1Orphanet:969Acromicric dysplasia
LTBP2Orphanet:238763Glaucoma secondary to spherophakia/ectopia lentis and megalocornea
LTBP2Orphanet:3449Weill-Marchesani syndrome
LTBP2Orphanet:98976Congenital glaucoma
LTBP3Orphanet:2623Geleophysic dysplasia
LTBP3Orphanet:2899Brachyolmia-amelogenesis imperfecta syndrome
LTBP3Orphanet:969Acromicric dysplasia
ADAMTSL2Orphanet:1901Dermatosparaxis Ehlers-Danlos syndrome
ADAMTSL2Orphanet:2623Geleophysic dysplasia

Cohort genes → proteins

4 cohort genes, 4 distinct canonical proteins.

Evidence partition

SubsetGenes
multi_evidence4

Cohort genes (full)

SymbolHGNCEnsemblUniProtNameEvidence
FBN1HGNC:3603ENSG00000166147P35555Fibrillin-1gencc,clinvar
LTBP2HGNC:6715ENSG00000119681Q14767Latent-transforming growth factor beta-binding protein 2gencc,clinvar
LTBP3HGNC:6716ENSG00000168056Q9NS15Latent-transforming growth factor beta-binding protein 3gencc,clinvar
ADAMTSL2HGNC:14631ENSG00000197859Q86TH1ADAMTS-like protein 2clinvar

Cohort function summary

Lead sentence per gene, UniProt-curated.

SymbolProtein nameFunction (lead sentence)
FBN1Fibrillin-1Structural component of the 10-12 nm diameter microfibrils of the extracellular matrix, which conveys both structural and regulatory properties to load-bearing connective tissues.
LTBP2Latent-transforming growth factor beta-binding protein 2May play an integral structural role in elastic-fiber architectural organization and/or assembly.
LTBP3Latent-transforming growth factor beta-binding protein 3Key regulator of transforming growth factor beta (TGFB1, TGFB2 and TGFB3) that controls TGF-beta activation by maintaining it in a latent state during storage in extracellular space.

Protein-family classification

Druggable: 0 · Difficult: 0 · Unknown: 4 · 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/Unknown41.8×0.097

Per-gene assignment

SymbolFamilyDruggable?ECInterPro (top 3)
FBN1Other/UnknownnoEGF-type_Asp/Asn_hydroxyl_site, EGF, EGF-like_Ca-bd_dom
LTBP2Other/UnknownnoEGF-type_Asp/Asn_hydroxyl_site, EGF, EGF-like_Ca-bd_dom
LTBP3Other/UnknownnoEGF-type_Asp/Asn_hydroxyl_site, EGF, EGF-like_Ca-bd_dom
ADAMTSL2Other/UnknownnoTSP1_rpt, ADAMTS_spacer1, PLAC

Expression context

Cohort genes with no expression data: 0.

4 cohort genes 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)4
unknown0

Top tissues across cohort

TissueCohort genes
ascending aorta2
descending thoracic aorta2
thoracic aorta2
decidua1
skin of hip1
synovial joint1
cardiac atrium1
metanephros cortex1
right atrium auricular region1

Per-gene tissue summary (top 30)

SymbolBgee breadthFANTOM5 breadthSCXATop tissues
FBN1275ubiquitousmarkersynovial joint, skin of hip, decidua
LTBP2276ubiquitousmarkerdescending thoracic aorta, thoracic aorta, ascending aorta
LTBP3279broadmarkerdescending thoracic aorta, thoracic aorta, ascending aorta
ADAMTSL2159broadmarkerright atrium auricular region, metanephros cortex, cardiac atrium

Protein interactions among cohort

Intra-cohort edges: 4.

Hub genes (top 10 by interactor count)

SymbolInteractor count
FBN13,640
LTBP22,658
LTBP32,339
ADAMTSL2600

Intra-cohort edges

ABSources
ADAMTSL2FBN1string_interaction
ADAMTSL2LTBP3string_interaction
FBN1LTBP2string_interaction
FBN1LTBP3string_interaction

Structural data

PDB: 1 · AlphaFold-only: 3 · No structure: 0

Cohort genes with PDB structures (top 30)

SymbolUniProtPDB entries
FBN1P3555511

AlphaFold-only cohort genes (top 30 by pLDDT)

SymbolUniProtpLDDT
ADAMTSL2Q86TH167.10
LTBP3Q9NS1564.21
LTBP2Q1476758.33

Function

Pathway analysis

Distinct Reactome pathways touched by cohort: 20. Enrichment computed across 4 evidence-associated genes (4 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 4 annotated cohort genes). Counts and members are kept as ground-truth; sorted by enrichment.

PathwayCohort genesFoldFDRSample cohort genes
Elastic fibre formation3251.9×8e-07FBN1, LTBP2, LTBP3
TGF-beta receptor signaling activates SMADs3244.7×8e-07FBN1, LTBP2, LTBP3
Molecules associated with elastic fibres3231.5×8e-07FBN1, LTBP2, LTBP3
Signaling by TGF-beta Receptor Complex2100.2×7e-04LTBP2, LTBP3
Signaling by TGFB family members257.7×0.002LTBP2, LTBP3
Extracellular matrix organization231.6×0.005LTBP2, LTBP3
Defective B3GALTL causes PpS177.2×0.033ADAMTSL2
O-glycosylation of TSR domain-containing proteins175.1×0.033ADAMTSL2
Diseases associated with O-glycosylation of proteins153.9×0.041ADAMTSL2
O-linked glycosylation136.1×0.050ADAMTSL2
Integrin cell surface interactions133.6×0.050FBN1
Diseases of glycosylation132.8×0.050ADAMTSL2
Degradation of the extracellular matrix129.4×0.052FBN1
Post-translational protein phosphorylation125.0×0.056FBN1
Regulation of Insulin-like Growth Factor (IGF) transport and uptake by Insulin-like Growth Factor Binding Proteins (IGFBPs)121.6×0.060FBN1
Diseases of metabolism120.1×0.060ADAMTSL2
Signal Transduction25.1×0.060LTBP2, LTBP3
Post-translational protein modification14.8×0.214ADAMTSL2
Disease13.3×0.286ADAMTSL2
Metabolism of proteins13.1×0.286ADAMTSL2

GO biological processes by enrichment

Over-representation of cohort genes vs the genome-wide background (hypergeometric test, Benjamini-Hochberg FDR; fold = observed/expected over 4 annotated cohort genes). Counts and members are kept as ground-truth; sorted by enrichment.

GO termCohort genesFoldFDRSample cohort genes
lobar bronchus epithelium development14213.0×0.004ADAMTSL2
transforming growth factor beta receptor signaling pathway279.5×0.004LTBP2, LTBP3
post-embryonic eye morphogenesis11404.3×0.006FBN1
obsolete sequestering of BMP in extracellular matrix11053.2×0.006FBN1
obsolete sequestering of TGFbeta in extracellular matrix11053.2×0.006FBN1
negative regulation of osteoclast development1842.6×0.007FBN1
positive regulation of mesenchymal stem cell differentiation1601.9×0.007LTBP3
lung saccule development1526.6×0.007LTBP3
positive regulation of mesenchymal stem cell proliferation1526.6×0.007LTBP3
embryonic eye morphogenesis1383.0×0.009FBN1
cellular response to insulin-like growth factor stimulus1324.1×0.010FBN1
supramolecular fiber organization1263.3×0.010LTBP2
positive regulation of bone resorption1247.8×0.010LTBP3
negative regulation of bone mineralization1234.1×0.010LTBP3
bone remodeling1234.1×0.010LTBP3
negative regulation of chondrocyte differentiation1168.5×0.012LTBP3
cell adhesion mediated by integrin1168.5×0.012FBN1
bone morphogenesis1150.5×0.013LTBP3
negative regulation of osteoclast differentiation1135.9×0.013FBN1
metanephros development1127.7×0.013FBN1
protein targeting191.6×0.017LTBP2
camera-type eye development189.6×0.017FBN1
lung alveolus development187.8×0.017FBN1
chondrocyte differentiation175.2×0.019LTBP3
cellular response to transforming growth factor beta stimulus169.1×0.019FBN1
bone mineralization168.0×0.019LTBP3
protein secretion165.8×0.019LTBP2
glucose metabolic process163.8×0.019FBN1
negative regulation of transforming growth factor beta receptor signaling pathway143.4×0.027ADAMTSL2
glucose homeostasis132.7×0.034FBN1

Therapeutics

Drug target analysis

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

Druggability breadth: 0 of 4 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
FBN100
LTBP200
LTBP300
ADAMTSL200

Bioactivity and enzyme data

Enzyme cohort genes (≥1 EC): 0.

Pharmacogenomics

Cohort genes with a PharmGKB record: 4; 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 drug0
EDifficult family or no structure, no drug4FBN1, LTBP2, LTBP3, ADAMTSL2

Undrugged target profiles

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

SymbolChEMBL assaysDrugged partners (top 3)
FBN10
LTBP20
LTBP30
ADAMTSL20

Clinical trials & evidence

Clinical trials

Clinical trials: 0.