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Omodysplasia

MedGen UID:
1388973
Concept ID:
C4510897
Disease or Syndrome
Synonym: omodysplasia
SNOMED CT: Omodysplasia (725164008)
Modes of inheritance:
Autosomal recessive inheritance
MedGen UID:
141025
Concept ID:
C0441748
Intellectual Product
Source: Orphanet
A mode of inheritance that is observed for traits related to a gene encoded on one of the autosomes (i.e., the human chromosomes 1-22) in which a trait manifests in individuals with two pathogenic alleles, either homozygotes (two copies of the same mutant allele) or compound heterozygotes (whereby each copy of a gene has a distinct mutant allele).
Autosomal dominant inheritance
MedGen UID:
141047
Concept ID:
C0443147
Intellectual Product
Source: Orphanet
A mode of inheritance that is observed for traits related to a gene encoded on one of the autosomes (i.e., the human chromosomes 1-22) in which a trait manifests in heterozygotes. In the context of medical genetics, an autosomal dominant disorder is caused when a single copy of the mutant allele is present. Males and females are affected equally, and can both transmit the disorder with a risk of 50% for each child of inheriting the mutant allele.
 
Monarch Initiative: MONDO:0017136
OMIM® Phenotypic series: PS258315
Orphanet: ORPHA2733

Definition

Omodysplasia is a rare skeletal dysplasia characterised by severe limb shortening and facial dysmorphism. Two types of omodysplasia have been described: an autosomal recessive or generalised form (also referred to as micromelic dysplasia with dislocation of radius) marked by severe micromelic dwarfism with predominantly rhizomelic shortening of both the upper and lower limbs, and an autosomal dominant form in which stature is normal and shortening is limited to the upper limbs. In total, less than 40 cases of omodysplasia have been described in the literature so far, with the majority of reported cases concerning the autosomal recessive form of the disease. The aetiology remains unknown but a paternally inherited paracentric inversion of 15q13 to q21.3 has been detected in one family. [from SNOMEDCT_US]

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • Omodysplasia

Professional guidelines

PubMed

Borochowitz Z, Sabo E, Misselevitch I, Boss JH
Am J Med Genet 1998 Mar 19;76(3):238-44. doi: 10.1002/(sici)1096-8628(19980319)76:3<238::aid-ajmg7>3.0.co;2-m. PMID: 9508243

Recent clinical studies

Etiology

Elçioglu NH, Gustavson KH, Wilkie AO, Yüksel-Apak M, Spranger JW
Pediatr Radiol 2004 Jan;34(1):75-82. Epub 2003 Oct 18 doi: 10.1007/s00247-003-1064-9. PMID: 14566439

Diagnosis

Nagasaki K, Nishimura G, Kikuchi T, Nyuzuki H, Sasaki S, Ogawa Y, Saitoh A
Am J Med Genet A 2018 Mar;176(3):739-742. Epub 2018 Jan 31 doi: 10.1002/ajmg.a.38623. PMID: 29383834
Saal HM, Prows CA, Guerreiro I, Donlin M, Knudson L, Sund KL, Chang CF, Brugmann SA, Stottmann RW
Hum Mol Genet 2015 Jun 15;24(12):3399-409. Epub 2015 Mar 10 doi: 10.1093/hmg/ddv088. PMID: 25759469Free PMC Article
Gordon BL, Champaigne NL, Rogers RC, Frias JL, Leroy JG
Am J Med Genet A 2014 May;164A(5):1234-8. Epub 2014 Jan 23 doi: 10.1002/ajmg.a.36408. PMID: 24458798
Tan TY, McGillivray G, Kornman L, Fink AM, Superti-Furga A, Bonafé L, Francis DI, Savarirayan R
Am J Med Genet A 2005 Jun 15;135(3):324-7. doi: 10.1002/ajmg.a.30754. PMID: 15887278
Venditti CP, Farmer J, Russell KL, Friedrich CA, Alter C, Canning D, Whitaker L, Mennuti MT, Driscoll DA, Zackai EH
Am J Med Genet 2002 Aug 1;111(2):169-77. doi: 10.1002/ajmg.10555. PMID: 12210345

Prognosis

Kemp JP, Morris JA, Medina-Gomez C, Forgetta V, Warrington NM, Youlten SE, Zheng J, Gregson CL, Grundberg E, Trajanoska K, Logan JG, Pollard AS, Sparkes PC, Ghirardello EJ, Allen R, Leitch VD, Butterfield NC, Komla-Ebri D, Adoum AT, Curry KF, White JK, Kussy F, Greenlaw KM, Xu C, Harvey NC, Cooper C, Adams DJ, Greenwood CMT, Maurano MT, Kaptoge S, Rivadeneira F, Tobias JH, Croucher PI, Ackert-Bicknell CL, Bassett JHD, Williams GR, Richards JB, Evans DM
Nat Genet 2017 Oct;49(10):1468-1475. Epub 2017 Sep 4 doi: 10.1038/ng.3949. PMID: 28869591Free PMC Article
Campos-Xavier AB, Martinet D, Bateman J, Belluoccio D, Rowley L, Tan TY, Baxová A, Gustavson KH, Borochowitz ZU, Innes AM, Unger S, Beckmann JS, Mittaz L, Ballhausen D, Superti-Furga A, Savarirayan R, Bonafé L
Am J Hum Genet 2009 Jun;84(6):760-70. Epub 2009 May 28 doi: 10.1016/j.ajhg.2009.05.002. PMID: 19481194Free PMC Article
al Gazali LI, Abou al-Asaad F
Clin Dysmorphol 1995 Jan;4(1):52-6. PMID: 7735505

Clinical prediction guides

Zhu X, Xu M, Leu NA, Morrisey EE, Millar SE
Dis Model Mech 2023 Mar 1;16(3) Epub 2023 Mar 24 doi: 10.1242/dmm.049876. PMID: 36867021Free PMC Article
Capurro M, Izumikawa T, Suarez P, Shi W, Cydzik M, Kaneiwa T, Gariepy J, Bonafe L, Filmus J
J Cell Biol 2017 Sep 4;216(9):2911-2926. Epub 2017 Jul 10 doi: 10.1083/jcb.201605119. PMID: 28696225Free PMC Article
Gordon BL, Champaigne NL, Rogers RC, Frias JL, Leroy JG
Am J Med Genet A 2014 May;164A(5):1234-8. Epub 2014 Jan 23 doi: 10.1002/ajmg.a.36408. PMID: 24458798
Campos-Xavier AB, Martinet D, Bateman J, Belluoccio D, Rowley L, Tan TY, Baxová A, Gustavson KH, Borochowitz ZU, Innes AM, Unger S, Beckmann JS, Mittaz L, Ballhausen D, Superti-Furga A, Savarirayan R, Bonafé L
Am J Hum Genet 2009 Jun;84(6):760-70. Epub 2009 May 28 doi: 10.1016/j.ajhg.2009.05.002. PMID: 19481194Free PMC Article
Venditti CP, Farmer J, Russell KL, Friedrich CA, Alter C, Canning D, Whitaker L, Mennuti MT, Driscoll DA, Zackai EH
Am J Med Genet 2002 Aug 1;111(2):169-77. doi: 10.1002/ajmg.10555. PMID: 12210345

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