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Microcephalic osteodysplastic primordial dwarfism types I and III

MedGen UID:
1380769
Concept ID:
C4319565
Congenital Abnormality
Synonyms: brachymelic primordial dwarfism; cephaloskeletal dysplasia; low-birth-weight dwarfism with skeletal dysplasia; Microcephalic osteodysplastic primordial dwarfism Taybi Linder type; microcephalic osteodysplastic primordial dwarfism type 1; microcephalic osteodysplastic primordial dwarfism types 1 and 3; microcephalic osteodysplastic primordial dwarfism types I and III; Microcephalic osteodysplastic primordial dwarfism, Taybi-Linder type; microcephalic osteodysplastic primordial dwarfism, Taybi-Linder type; MOPD 1; MOPD types I and III; osteodysplastic primordial dwarfism type I; Primordial microcephalic dwarfism Crachami type; Primordial microcephalic dwarfism, Crachami type; primordial microcephalic dwarfism, Crachami type; Taybi Linder syndrome; Taybi-Linder syndrome
SNOMED CT: Microcephalic osteodysplastic primordial dwarfism types I and III (725461009); Microcephalic osteodysplastic primordial dwarfism Taybi Linder type (725461009); Primordial microcephalic dwarfism Crachami type (725461009); Taybi Linder syndrome (725461009)
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).
 
Monarch Initiative: MONDO:0016994
Orphanet: ORPHA2636

Definition

Disease with characteristics of intrauterine and postnatal growth retardation, microcephaly, facial dysmorphism, skeletal dysplasia, low-birth weight and brain anomalies. Although microcephalic osteodysplastic primordial dwarfism (MOPD) types 1 and 3 were originally described as two separate entities on the basis of radiological criteria (notably small differences in pelvic and long bone structure), later reports confirmed that the two forms represent different modes of expression of the same syndrome. Although the causative gene remains unknown, homozygosity mapping has allowed identification of a candidate gene region on chromosome 2q (2q14.2-q14.3). Histological studies suggest that MOPD types 1 and 3 result from a basic defect in cell proliferation and tissue differentiation. Transmitted as autosomal recessive trait. [from SNOMEDCT_US]

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • CROGVMicrocephalic osteodysplastic primordial dwarfism types I and III

Recent clinical studies

Etiology

Akalın A, Şimşek-Kiper PÖ, Taşkıran EZ, Karaosmanoğlu B, Utine GE, Boduroğlu K
Am J Med Genet A 2023 Apr;191(4):1119-1127. Epub 2023 Jan 11 doi: 10.1002/ajmg.a.63120. PMID: 36630262
Khetarpal P, Das S, Panigrahi I, Munshi A
Mol Genet Genomics 2016 Feb;291(1):1-15. Epub 2015 Sep 1 doi: 10.1007/s00438-015-1110-y. PMID: 26323792
Juric-Sekhar G, Kapur RP, Glass IA, Murray ML, Parnell SE, Hevner RF
Acta Neuropathol 2011 Apr;121(4):545-54. Epub 2010 Sep 21 doi: 10.1007/s00401-010-0748-0. PMID: 20857301Free PMC Article
Fukuzawa R, Sato S, Sullivan MJ, Nishimura G, Hasegawa T, Matsuo N
Am J Med Genet 2002 Nov 15;113(1):93-6. doi: 10.1002/ajmg.10716. PMID: 12400072
Sigaudy S, Toutain A, Moncla A, Fredouille C, Bourlière B, Ayme S, Philip N
Am J Med Genet 1998 Oct 30;80(1):16-24. PMID: 9800907

Diagnosis

Khetarpal P, Das S, Panigrahi I, Munshi A
Mol Genet Genomics 2016 Feb;291(1):1-15. Epub 2015 Sep 1 doi: 10.1007/s00438-015-1110-y. PMID: 26323792
Galasso C, Lo-Castro A, Lalli C, Cerminara C, Curatolo P
Pediatr Neurol 2008 Jun;38(6):435-8. doi: 10.1016/j.pediatrneurol.2008.02.011. PMID: 18486828
Nadjari M, Fasouliotis SJ, Ariel I, Raas-Rothschild A, Bar-Ziv J, Elchalal U
Prenat Diagn 2000 Aug;20(8):666-9. doi: 10.1002/1097-0223(200008)20:8<666::aid-pd887>3.0.co;2-l. PMID: 10951479

Clinical prediction guides

Juric-Sekhar G, Kapur RP, Glass IA, Murray ML, Parnell SE, Hevner RF
Acta Neuropathol 2011 Apr;121(4):545-54. Epub 2010 Sep 21 doi: 10.1007/s00401-010-0748-0. PMID: 20857301Free PMC Article
Meinecke P, Passarge E
J Med Genet 1991 Nov;28(11):795-800. doi: 10.1136/jmg.28.11.795. PMID: 1770539Free PMC Article

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