Literature DB >> 28027977

Cleidocranial dysplasia: Clinical, endocrinologic and molecular findings in 15 patients from 11 families.

Firdevs Dinçsoy Bir1, Nuriye Dinçkan2, Yeliz Güven3, Firdevs Baş4, Umut Altunoğlu5, Senem S Kuvvetli6, Şükran Poyrazoğlu4, Güven Toksoy5, Hülya Kayserili7, Z Oya Uyguner5.   

Abstract

Cleidocranial dysplasia (CCD) is an autosomal dominant disorder characterized by skeletal anomalies such as delayed closure of the cranial sutures, underdeveloped or absent clavicles, multiple dental abnormalities, short stature and osteoporosis. RUNX2, encoding Runt DNA-binding domain protein important in osteoblast differentiation, is the only known gene related to the disease and identified as responsible in 70% of the cases. Our clinical evaluations revealed that short stature present at a rate of 28.6%, osteoporosis at a rate of 57.1% and osteopenia at 21.4%. In this study, RUNX2 sequencing revealed nine different variations in 11 families, eight being pathogenic of which one was novel gross insertion (c.1271_1272ins20) and one other being predicted benign in frame gross deletion (c.241_258del).
Copyright © 2016 Elsevier Masson SAS. All rights reserved.

Entities:  

Keywords:  Cleidocranial dysplasia; Dental abnormalities; Osteoporosis; RUNX2; Underdeveloped clavicles

Mesh:

Substances:

Year:  2016        PMID: 28027977     DOI: 10.1016/j.ejmg.2016.12.007

Source DB:  PubMed          Journal:  Eur J Med Genet        ISSN: 1769-7212            Impact factor:   2.708


  11 in total

1.  Surgical Management and Evaluation of the Craniofacial Growth and Morphology in Cleidocranial Dysplasia.

Authors:  Stephen L Greene; Chung How Kau; Somsak Sittitavornwong; Kathlyn Powell; Noel K Childers; Mary MacDougall; Ejvis Lamani
Journal:  J Craniofac Surg       Date:  2018-06       Impact factor: 1.046

2.  Syringic acid, a phenolic acid, promotes osteoblast differentiation by stimulation of Runx2 expression and targeting of Smad7 by miR-21 in mouse mesenchymal stem cells.

Authors:  B Arumugam; K Balagangadharan; N Selvamurugan
Journal:  J Cell Commun Signal       Date:  2018-01-19       Impact factor: 5.782

Review 3.  Demographic, clinical, and radiological characteristics of cleidocranial dysplasia: A systematic review of cases reported in south America.

Authors:  Eder Cano-Pérez; Claudio Gómez-Alegría; Fredy Pomares Herrera; Doris Gómez-Camargo; Dacia Malambo-García
Journal:  Ann Med Surg (Lond)       Date:  2022-04-10

4.  Cleidocranial dysplasia.

Authors:  Stepan Kutilek; Roman Machytka; Petr Munzar
Journal:  Sudan J Paediatr       Date:  2019

5.  A novel gene mutation of Runx2 in cleidocranial dysplasia.

Authors:  You-Jian Peng; Qiao-Yun Chen; Dong-Jie Fu; Zhi-Ming Liu; Tian-Tian Mao; Jun Li; Wen-Ting She
Journal:  J Huazhong Univ Sci Technolog Med Sci       Date:  2017-10-20

6.  Analysis of novel RUNX2 mutations in Chinese patients with cleidocranial dysplasia.

Authors:  Xianli Zhang; Yang Liu; Xiaozhe Wang; Xiangyu Sun; Chenying Zhang; Shuguo Zheng
Journal:  PLoS One       Date:  2017-07-24       Impact factor: 3.240

7.  Cleidocranial dysplasia syndrome with epilepsy: a case report.

Authors:  Yimei Ma; Fumin Zhao; Dan Yu
Journal:  BMC Pediatr       Date:  2019-04-08       Impact factor: 2.125

8.  A Case of Cleidocranial Dysplasia with a Novel Mutation and Growth Velocity Gain with Growth Hormone Treatment

Authors:  Emine Çamtosun; Ayşehan Akıncı; Emine Demiral; İbrahim Tekedereli; Ahmet Sığırcı
Journal:  J Clin Res Pediatr Endocrinol       Date:  2018-11-23

9.  A Novel 90-kbp Deletion of RUNX2 Associated with Cleidocranial Dysplasia.

Authors:  Yanli Zhang; Xiaohong Duan
Journal:  Genes (Basel)       Date:  2022-06-23       Impact factor: 4.141

10.  Cleidocranial dysplasia with growth hormone deficiency: a case report.

Authors:  Nozomi Takaki; Jun Mori; Satoshi Matsuo; Toshio Osamura; Toshimi Michigami
Journal:  BMC Pediatr       Date:  2020-01-16       Impact factor: 2.125

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