Literature DB >> 24966961

A novel small deletion mutation in RUNX2 gene in one Chinese family with cleidocranial dysplasia.

Ting Chen1, Jin Hou1, Ling-Ling Hu2, Jie Gao1, Bu-Ling Wu1.   

Abstract

Cleidocranial dysplasia (CCD) is a skeletal dysplasia with autosomal-dominant inheritance. The runt related transcription factor 2 (RUNX2) gene is the only gene in which mutations are known to cause CCD. We report identification of a novel small deletions mutation in the RUNX2 gene in a Chinese family with CCD. A 29-year-old female was diagnosed as proband of CCD based on the clinical findings, which show delayed closure of the fontanels, hypoplastic or aplastic clavicles and dental anomalies. Similar dental and skeletal symptoms were also observed in the other three affected individuals. We prepared genomic DNA from all four affected individuals, unaffected individual from her family members, as well as 100 unrelated healthy controls. PCR was conducted using the above genomic DNA as template and the RUNX2 gene-specific primers. The PCR product was subjected to direct sequencing and the sequence was compared to that of RUNX2 gene within the NCBI database. We detected a small deletion CCTA from nucleotide 635 to nucleotide 638 in exon 3 of RUNX2 gene of the proband. This will lead to the introduction of a translational stop codon at codon 220, resulting in a truncated RUNX2 protein, and therefore within the runt domain of the RUNX2 protein. We detected the same mutation in the the other three affected individuals, and did not detect any mutation in the unaffected family members or the 100 unrelated healthy controls, demonstrating that this is a novel missense mutation in RUNX2 gene and therefore, contributes to the molecular diagnosis of CCD.

Entities:  

Keywords:  Cleidocranial dysplasia; RUNX2 gene; mutation

Mesh:

Substances:

Year:  2014        PMID: 24966961      PMCID: PMC4069909     

Source DB:  PubMed          Journal:  Int J Clin Exp Pathol        ISSN: 1936-2625


  19 in total

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2.  CBFA1 mutation analysis and functional correlation with phenotypic variability in cleidocranial dysplasia.

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4.  Four novel RUNX2 mutations including a splice donor site result in the cleidocranial dysplasia phenotype.

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Journal:  J Cell Physiol       Date:  2006-04       Impact factor: 6.384

5.  Dysregulation of chondrogenesis in human cleidocranial dysplasia.

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6.  Functional analysis of a novel RUNX2 missense mutation found in a family with cleidocranial dysplasia.

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7.  Mutational analysis of RUNX2 gene in Chinese patients with cleidocranial dysplasia.

Authors:  Chenying Zhang; Shuguo Zheng; Yixiang Wang; Yuming Zhao; Junxia Zhu; Lihong Ge
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8.  Genomic organization, expression of the human CBFA1 gene, and evidence for an alternative splicing event affecting protein function.

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9.  A novel in-frame deletion of the RUNX2 gene causes a classic form of cleidocranial dysplasia.

Authors:  Masaki Matsushita; Hiroshi Kitoh; Hiroshi Kaneko; Kenichi Mishima; Yasutomo Itoh; Yoshihito Tokita; Naoki Ishiguro
Journal:  J Bone Miner Metab       Date:  2013-04-05       Impact factor: 2.626

10.  Cleidocranial dysplasia syndrome: clinical characteristics and mutation study of a Chinese family.

Authors:  Shengguo Wang; Shu Zhang; Yanmin Wang; Yangxi Chen; Li Zhou
Journal:  Int J Clin Exp Med       Date:  2013-10-25
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  2 in total

1.  Novel mutation of RUNX2 gene in a patient with cleidocranial dysplasia.

Authors:  Ya-Wun Guo; Chih-Yang Chiu; Chien-Lin Liu; Tjin-Shing Jap; Liang-Yu Lin
Journal:  Int J Clin Exp Pathol       Date:  2015-01-01

Review 2.  Delayed Eruption of Permanent Dentition and Maxillary Contraction in Patients with Cleidocranial Dysplasia: Review and Report of a Family.

Authors:  A Impellizzeri; G Midulla; U Romeo; C La Monaca; E Barbato; G Galluccio
Journal:  Int J Dent       Date:  2018-07-04
  2 in total

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