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Mesomelic dysplasia, Kantaputra type(MMDK)

MedGen UID:
331880
Concept ID:
C1835009
Disease or Syndrome
Synonyms: Mesomelic dysplasia Thai type; Mesomelic dysplasia with ankle carpal and tarsal synostosis
SNOMED CT: Mesomelic dysplasia Kantaputra type (719397009); Kantaputra mesomelic dysplasia (719397009); Mesomelic dysplasia Thai type (719397009)
Modes of inheritance:
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:0007977
OMIM®: 156232
Orphanet: ORPHA1836

Definition

Kantaputra mesomelic dysplasia (MMDK) is a rare autosomal dominant skeletal disease characterized by symmetric marked shortening of the upper and lower limbs. The ulnae are very short and the radii are bowed. The distal humerus has a dumbbell shape, whereas the hands are relatively normal but show progressive flexion contractures of the proximal interphalangeal joints. Carpal and tarsal synostoses are observed in some individuals. In the lower limbs, the feet are fixed in plantar flexion with the sole facing backward, causing 'ballerina-like standing.' The prominent distal fibula (ventral aspect) is considered to be the signature finding of the syndrome. The calcaneus is small or missing, and a small fibula and talus as well as fibulocalcaneal synostosis are characteristic features. The tibial bony knot articulates with the proximal end of the fibula (summary by Kantaputra et al., 2010). Mesomelia and synostosis are also cardinal features of the mesomelia-synostoses syndrome (600383). See 613681 for discussion of the chromosome 2q31.1 duplication syndrome, which shows cytogenetic and phenotypic overlap with MMDK. [from OMIM]

Clinical features

From HPO
Mesomelia
MedGen UID:
107808
Concept ID:
C0549306
Congenital Abnormality
Shortening of the middle parts of the limbs (forearm and lower leg) in relation to the upper and terminal segments.
Radial bowing
MedGen UID:
347136
Concept ID:
C1859399
Anatomical Abnormality
A bending or abnormal curvature of the radius.
Tarsal synostosis
MedGen UID:
539393
Concept ID:
C0265654
Congenital Abnormality
Synostosis (bony fusion) involving one or more bones of the tarsus (calcaneus, talus, cuboid, navicular, cuneiiform bones).
Carpal synostosis
MedGen UID:
98468
Concept ID:
C0431863
Congenital Abnormality
Synostosis (bony fusion) involving one or more bones of the carpus (scaphoid, lunate, triquetrum, trapezium, trapezoid, capitate, hamate, pisiform).

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • CROGVMesomelic dysplasia, Kantaputra type
Follow this link to review classifications for Mesomelic dysplasia, Kantaputra type in Orphanet.

Recent clinical studies

Etiology

Siwicka KA, Kitoh H, Nishiyama M, Ishiguro N
J Pediatr Orthop B 2008 Sep;17(5):271-6. doi: 10.1097/BPB.0b013e32830cc3c8. PMID: 19471182
Kantaputra PN
Am J Med Genet A 2004 Jul 1;128A(1):1-5. doi: 10.1002/ajmg.a.20642. PMID: 15211646

Diagnosis

Lim BC, Min BJ, Park WY, Oh SK, Woo MJ, Choi JS, Kim KJ, Hwang YS, Chae JH
J Child Neurol 2014 Feb;29(2):260-4. Epub 2013 Mar 1 doi: 10.1177/0883073813478659. PMID: 23456534
Siwicka KA, Kitoh H, Nishiyama M, Ishiguro N
J Pediatr Orthop B 2008 Sep;17(5):271-6. doi: 10.1097/BPB.0b013e32830cc3c8. PMID: 19471182
Kwee ML, van de Sluijs JA, van Vugt JM, Wijnaendts LC, Gille JJ
Am J Med Genet A 2004 Aug 1;128A(4):404-9. doi: 10.1002/ajmg.a.30120. PMID: 15264287

Prognosis

Siwicka KA, Kitoh H, Nishiyama M, Ishiguro N
J Pediatr Orthop B 2008 Sep;17(5):271-6. doi: 10.1097/BPB.0b013e32830cc3c8. PMID: 19471182
Kantaputra PN
Am J Med Genet A 2004 Jul 1;128A(1):1-5. doi: 10.1002/ajmg.a.20642. PMID: 15211646

Clinical prediction guides

Fujimoto M, Kantaputra PN, Ikegawa S, Fukushima Y, Sonta S, Matsuo M, Ishida T, Matsumoto T, Kondo S, Tomita H, Deng HX, D'urso M, Rinaldi MM, Ventruto V, Takagi T, Nakamura Y, Niikawa N
J Hum Genet 1998;43(1):32-6. doi: 10.1007/s100380050033. PMID: 9609995

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