| Literature DB >> 28027977 |
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).Entities:
Keywords: Cleidocranial dysplasia; Dental abnormalities; Osteoporosis; RUNX2; Underdeveloped clavicles
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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