Literature DB >> 28371282

MED13L haploinsufficiency syndrome: A de novo frameshift and recurrent intragenic deletions due to parental mosaicism.

Toshiyuki Yamamoto1,2, Keiko Shimojima1,2, Yumiko Ondo2, Shuichi Shimakawa3, Nobuhiko Okamoto4.   

Abstract

MED13L haploinsufficiency syndrome is a clinical condition manifesting intellectual disability and developmental delay in association with various complications including congenital heart defects and dysmorphic features. Most of the previously reported patients showed de novo loss-of-function mutations in MED13L. Additional three patients with MED13L haploinsufficiency syndrome were identified here in association with rare complications. One patient had a de novo deletion (c.257delT) and T2-weighted high intensity in the occipital white matter on magnetic resonance imaging. Two siblings exhibited an intragenic deletion involving exons 3-14, which led to an in-frame deletion in MED13L. The deletion was inherited from their carrier mother who possessed low frequency mosaicism. The older sister of the siblings showed craniosynostosis; this condition has never been reported in patients with MED13L haploinsufficiency syndrome. Dysmorphic features were observed in these patients; however, most of the findings were nonspecific. Further information would be necessary to understand this clinical condition better.
© 2017 Wiley Periodicals, Inc.

Entities:  

Keywords:  MED13L haploinsufficiency syndrome; craniosynostosis; intellectual disability; loss-of-function; mosaic

Mesh:

Substances:

Year:  2017        PMID: 28371282     DOI: 10.1002/ajmg.a.38168

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  7 in total

Review 1.  Report of a de novo c.2605C > T (p.Pro869Ser) change in the MED13L gene and review of the literature for MED13L-related intellectual disability.

Authors:  Zhi Yi; Ying Zhang; Zhenfeng Song; Hong Pan; Chengqing Yang; Fei Li; Jiao Xue; Zhenghai Qu
Journal:  Ital J Pediatr       Date:  2020-07-09       Impact factor: 2.638

2.  Analysis of Polymorphisms in the Mediator Complex Subunit 13-like (Med13L) Gene in the Context of Immune Function and Development of Experimental Arthritis.

Authors:  Samra Sardar; Katrine Kanne; Åsa Andersson
Journal:  Arch Immunol Ther Exp (Warsz)       Date:  2018-06-27       Impact factor: 4.291

3.  MED13L-related intellectual disability due to paternal germinal mosaicism.

Authors:  Beáta Bessenyei; István Balogh; Attila Mokánszki; Anikó Ujfalusi; Rolph Pfundt; Katalin Szakszon
Journal:  Cold Spring Harb Mol Case Stud       Date:  2022-01-10

4.  A complex molecular switch directs stress-induced cyclin C nuclear release through SCFGrr1-mediated degradation of Med13.

Authors:  David C Stieg; Stephen D Willis; Vidyaramanan Ganesan; Kai Li Ong; Joseph Scuorzo; Mia Song; Julianne Grose; Randy Strich; Katrina F Cooper
Journal:  Mol Biol Cell       Date:  2017-12-06       Impact factor: 4.138

5.  Early-Onset Diabetes Mellitus in a Patient With a Chromosome 13q34qter Microdeletion Including IRS2.

Authors:  Naru Babaya; Shinsuke Noso; Yoshihisa Hiromine; Hiroyuki Ito; Yasunori Taketomo; Toshiyuki Yamamoto; Yumiko Kawabata; Hiroshi Ikegami
Journal:  J Endocr Soc       Date:  2018-09-11

Review 6.  Potential roles of mediator Complex Subunit 13 in Cardiac Diseases.

Authors:  Wenqian Zhou; He Cai; Jia Li; He Xu; Xiang Wang; Hongbo Men; Yang Zheng; Lu Cai
Journal:  Int J Biol Sci       Date:  2021-01-01       Impact factor: 6.580

7.  A Japanese patient with a 2p25.3 terminal deletion presented with early-onset obesity, intellectual disability and diabetes mellitus: A case report.

Authors:  Taka-Aki Sakaue; Yoshinari Obata; Yuya Fujishima; Junji Kozawa; Michio Otsuki; Toshiyuki Yamamoto; Norikazu Maeda; Hitoshi Nishizawa; Iichiro Shimomura
Journal:  J Diabetes Investig       Date:  2021-08-31       Impact factor: 4.232

  7 in total

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