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Spondyloepimetaphyseal dysplasia, Isidor type

MedGen UID:
1676518
Concept ID:
C5190629
Disease or Syndrome
Synonyms: Spondyloepimetaphyseal dysplasia Isidor type; spondyloepimetaphyseal dysplasia, Isidor type
SNOMED CT: Spondyloepimetaphyseal dysplasia Isidor type (782821004)
 
Monarch Initiative: MONDO:0018254
Orphanet: ORPHA370015

Definition

A rare primary bone dysplasia disorder with characteristics of normal birth length with early postnatal growth deficiency resulting in severe disproportionate short stature (with short trunk and limbs), severe genu varum, flexion contractures in the hips and lumbar hyperlordosis. Radiological findings reveal platyspondyly with central indentation of vertebral endplates, progressive and severe epimetaphyseal abnormalities that primarily affect the lower limbs and include very small, irregular proximal femoral and knee epiphyses, severe coxa vara, delayed ossification of proximal femoral epiphyses and irregular distal femoral and proximal tibial metaphyses. [from SNOMEDCT_US]

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • CROGVSpondyloepimetaphyseal dysplasia, Isidor type

Recent clinical studies

Diagnosis

Isidor B, Geffroy L, de Courtivron B, Le Caignec C, Thiel CT, Mortier G, Cormier-Daire V, David A, Toutain A
Am J Med Genet A 2013 Oct;161A(10):2645-51. Epub 2013 Aug 16 doi: 10.1002/ajmg.a.36132. PMID: 23956136

Prognosis

Isidor B, Geffroy L, de Courtivron B, Le Caignec C, Thiel CT, Mortier G, Cormier-Daire V, David A, Toutain A
Am J Med Genet A 2013 Oct;161A(10):2645-51. Epub 2013 Aug 16 doi: 10.1002/ajmg.a.36132. PMID: 23956136

Clinical prediction guides

Le Caignec C, Ory B, Lamoureux F, O'Donohue MF, Orgebin E, Lindenbaum P, Téletchéa S, Saby M, Hurst A, Nelson K, Gilbert SR, Wilnai Y, Zeitlin L, Segev E, Tesfaye R, Nizon M, Cogne B, Bezieau S, Geoffroy L, Hamel A, Mayrargue E, de Courtivron B, Decock-Giraudaud A, Charrier C, Pichon O, Retière C, Redon R, Pepler A, McWalter K, Da Costa L, Toutain A, Gleizes PE, Baud'huin M, Isidor B
Am J Hum Genet 2019 Nov 7;105(5):1040-1047. Epub 2019 Oct 17 doi: 10.1016/j.ajhg.2019.09.024. PMID: 31630789Free PMC Article
Isidor B, Geffroy L, de Courtivron B, Le Caignec C, Thiel CT, Mortier G, Cormier-Daire V, David A, Toutain A
Am J Med Genet A 2013 Oct;161A(10):2645-51. Epub 2013 Aug 16 doi: 10.1002/ajmg.a.36132. PMID: 23956136

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