Literature DB >> 23220435

A new phenotypic variant in cleidocranial dysplasia (CCD) associated with mutation c.391C>T of the RUNX2 gene.

Michele Callea1, Fabiana Fattori, Izzet Yavuz, Enrico Bertini.   

Abstract

The RUNX2 gene is a physiological regulatory gene implicated in the development of cleidocranial dysplasia (CCD). A 13-month-old child presented with clinical features of CCD. At the age of 3 years the diagnosis was corroborated by clinical genetic assessment and DNA analysis, revealing a missense mutation p.R131C (c.391C>T) in RUNX2. At the age of 8 years the child was found to have a unique dental phenotype, represented by lack of supernumerary teeth and congenital absence of one tooth. A simple therapeutic approach was adopted, consisting of interceptive orthodontic treatment. The presence of this specific missense mutation in RUNX2, associated with the lack of typical supernumerary teeth may suggest a phenotype-genotype association.

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Year:  2012        PMID: 23220435      PMCID: PMC4542990          DOI: 10.1136/bcr-12-2011-5422

Source DB:  PubMed          Journal:  BMJ Case Rep        ISSN: 1757-790X


  12 in total

1.  Cleidocranial dysplasia: oral features and genetic analysis of 11 patients.

Authors:  A Bufalino; L M R Paranaíba; A F Gouvêa; L A Gueiros; H Martelli-Júnior; J J Junior; M A Lopes; E Graner; O P De Almeida; P A Vargas; R D Coletta
Journal:  Oral Dis       Date:  2011-10-24       Impact factor: 3.511

2.  Deletions of the RUNX2 gene are present in about 10% of individuals with cleidocranial dysplasia.

Authors:  Claus E Ott; Gundula Leschik; Fabienne Trotier; Louise Brueton; Han G Brunner; Wim Brussel; Encarna Guillen-Navarro; Claudia Haase; Juergen Kohlhase; Dieter Kotzot; Andrew Lane; Min Ae Lee-Kirsch; Susanne Morlot; Marleen E H Simon; Elisabeth Steichen-Gersdorf; David H Tegay; Hartmut Peters; Stefan Mundlos; Eva Klopocki
Journal:  Hum Mutat       Date:  2010-08       Impact factor: 4.878

3.  Osf2/Cbfa1: a transcriptional activator of osteoblast differentiation.

Authors:  P Ducy; R Zhang; V Geoffroy; A L Ridall; G Karsenty
Journal:  Cell       Date:  1997-05-30       Impact factor: 41.582

Review 4.  Cleidocranial dysplasia: clinical and molecular genetics.

Authors:  S Mundlos
Journal:  J Med Genet       Date:  1999-03       Impact factor: 6.318

Review 5.  The genetic basis of tooth development and dental defects.

Authors:  Irma Thesleff
Journal:  Am J Med Genet A       Date:  2006-12-01       Impact factor: 2.802

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

Authors:  Hyo-Jin Kim; Soon-Hyeun Nam; Hyun-Jung Kim; Hyo-Sang Park; Hyun-Mo Ryoo; Shin-Yoon Kim; Tae-Joon Cho; Seung-Gon Kim; Suk-Chul Bae; In-San Kim; Janet L Stein; Andre J van Wijnen; Gary S Stein; Jane B Lian; Je-Yong Choi
Journal:  J Cell Physiol       Date:  2006-04       Impact factor: 6.384

7.  Runx2 (Cbfa1) inhibits Shh signaling in the lower but not upper molars of mouse embryos and prevents the budding of putative successional teeth.

Authors:  X-P Wang; T Aberg; M J James; D Levanon; Y Groner; I Thesleff
Journal:  J Dent Res       Date:  2005-02       Impact factor: 6.116

8.  Microduplications upstream of MSX2 are associated with a phenocopy of cleidocranial dysplasia.

Authors:  Claus Eric Ott; Hendrikje Hein; Silke Lohan; Jeannette Hoogeboom; Nicola Foulds; Johannes Grünhagen; Sigmar Stricker; Pablo Villavicencio-Lorini; Eva Klopocki; Stefan Mundlos
Journal:  J Med Genet       Date:  2012-06-20       Impact factor: 6.318

9.  Development of the dentition in cleidocranial dysplasia.

Authors:  B L Jensen; S Kreiborg
Journal:  J Oral Pathol Med       Date:  1990-02       Impact factor: 4.253

10.  Two domains unique to osteoblast-specific transcription factor Osf2/Cbfa1 contribute to its transactivation function and its inability to heterodimerize with Cbfbeta.

Authors:  K Thirunavukkarasu; M Mahajan; K W McLarren; S Stifani; G Karsenty
Journal:  Mol Cell Biol       Date:  1998-07       Impact factor: 4.272

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  6 in total

Review 1.  Identification of a novel mutation of RUNX2 in a family with supernumerary teeth and craniofacial dysplasia by whole-exome sequencing: A case report and literature review.

Authors:  Dan Ma; Xuxia Wang; Jun Guo; Jun Zhang; Tao Cai
Journal:  Medicine (Baltimore)       Date:  2018-08       Impact factor: 1.889

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

3.  Anti-USAG-1 therapy for tooth regeneration through enhanced BMP signaling.

Authors:  A Murashima-Suginami; H Kiso; Y Tokita; E Mihara; Y Nambu; R Uozumi; Y Tabata; K Bessho; J Takagi; M Sugai; K Takahashi
Journal:  Sci Adv       Date:  2021-02-12       Impact factor: 14.136

4.  Abnormal eruption of teeth in relation to FGFR1 heterozygote mutation: a rare case of osteoglophonic dysplasia with 4-year follow-up.

Authors:  Yuchun Zou; Hanyu Lin; Weijia Chen; Lin Chang; Senxin Cai; You-Guang Lu; Linyu Xu
Journal:  BMC Oral Health       Date:  2022-02-11       Impact factor: 2.757

5.  Local application of Usag-1 siRNA can promote tooth regeneration in Runx2-deficient mice.

Authors:  Sayaka Mishima; Katsu Takahashi; Honoka Kiso; Akiko Murashima-Suginami; Yoshihito Tokita; Jun-Ichiro Jo; Ryuji Uozumi; Yukiko Nambu; Boyen Huang; Hidemitsu Harada; Toshihisa Komori; Manabu Sugai; Yasuhiko Tabata; Kazuhisa Bessho
Journal:  Sci Rep       Date:  2021-07-01       Impact factor: 4.379

6.  Antagonistic Functions of USAG-1 and RUNX2 during Tooth Development.

Authors:  Yumiko Togo; Katsu Takahashi; Kazuyuki Saito; Honoka Kiso; Hiroko Tsukamoto; Boyen Huang; Motoko Yanagita; Manabu Sugai; Hidemitsu Harada; Toshihisa Komori; Akira Shimizu; Mary MacDougall; Kazuhisa Bessho
Journal:  PLoS One       Date:  2016-08-12       Impact factor: 3.240

  6 in total

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