Literature DB >> 12467752

Frequent low penetrance mutations in the Lamin A/C gene, causing Emery Dreifuss muscular dystrophy.

Michal Vytopil1, Enzo Ricci, Antonio Dello Russo, Frank Hanisch, Stephan Neudecker, Stephan Zierz, Roberta Ricotti, Laurence Demay, Pascale Richard, Manfred Wehnert, Gisèle Bonne, Luciano Merlini, Daniela Toniolo.   

Abstract

Emery Dreifuss muscular dystrophy is a genetically heterogeneous disorder characterized by the clinical triad of early onset contractures, progressive muscular wasting and weakness with humeroperoneal distribution and cardiac conduction defects. Mutations in the Lamin A/C (LMNA) gene are responsible for the autosomal dominant and the autosomal recessive forms. Familiar and sporadic patients carrying mutations in the LMNA gene show high variability in the clinical symptomatology and age of onset. In this report, we describe four families harboring missense mutations in the LMNA gene and we show that the effect of mutations ranges from silent to fully penetrant. We suggest that incomplete penetrance of dominant mutations in the LMNA gene is a common feature and we emphasize the significance of mutational analysis in relatives of sporadic cases of laminopathies, as asymptomatic carriers face high risk of sudden cardiac death.

Entities:  

Mesh:

Substances:

Year:  2002        PMID: 12467752     DOI: 10.1016/s0960-8966(02)00178-5

Source DB:  PubMed          Journal:  Neuromuscul Disord        ISSN: 0960-8966            Impact factor:   4.296


  11 in total

Review 1.  Mouse models of the laminopathies.

Authors:  Colin L Stewart; Serguei Kozlov; Loren G Fong; Stephen G Young
Journal:  Exp Cell Res       Date:  2007-03-31       Impact factor: 3.905

Review 2.  Molecular genetics of autosomal-recessive axonal Charcot-Marie-Tooth neuropathies.

Authors:  Rafaëlle Bernard; Annachiara De Sandre-Giovannoli; Valérie Delague; Nicolas Lévy
Journal:  Neuromolecular Med       Date:  2006       Impact factor: 3.843

Review 3.  Where genotype is not predictive of phenotype: towards an understanding of the molecular basis of reduced penetrance in human inherited disease.

Authors:  David N Cooper; Michael Krawczak; Constantin Polychronakos; Chris Tyler-Smith; Hildegard Kehrer-Sawatzki
Journal:  Hum Genet       Date:  2013-07-03       Impact factor: 4.132

4.  Mislocalization to the nuclear envelope: an effect of the dystonia-causing torsinA mutation.

Authors:  Rose E Goodchild; William T Dauer
Journal:  Proc Natl Acad Sci U S A       Date:  2004-01-07       Impact factor: 11.205

5.  A laminopathic mutation disrupting lamin filament assembly causes disease-like phenotypes in Caenorhabditis elegans.

Authors:  Erin M Bank; Kfir Ben-Harush; Naama Wiesel-Motiuk; Rachel Barkan; Naomi Feinstein; Oren Lotan; Ohad Medalia; Yosef Gruenbaum
Journal:  Mol Biol Cell       Date:  2011-06-08       Impact factor: 4.138

6.  Titin Truncating Variants in Dilated Cardiomyopathy - Prevalence and Genotype-Phenotype Correlations.

Authors:  Maria Franaszczyk; Przemyslaw Chmielewski; Grazyna Truszkowska; Piotr Stawinski; Ewa Michalak; Malgorzata Rydzanicz; Malgorzata Sobieszczanska-Malek; Agnieszka Pollak; Justyna Szczygieł; Joanna Kosinska; Adam Parulski; Tomasz Stoklosa; Agnieszka Tarnowska; Marcin M Machnicki; Bogna Foss-Nieradko; Malgorzata Szperl; Agnieszka Sioma; Mariusz Kusmierczyk; Jacek Grzybowski; Tomasz Zielinski; Rafal Ploski; Zofia T Bilinska
Journal:  PLoS One       Date:  2017-01-03       Impact factor: 3.240

Review 7.  Cellular and Animal Models of Striated Muscle Laminopathies.

Authors:  Hannah A Nicolas; Marie-Andrée Akimenko; Frédérique Tesson
Journal:  Cells       Date:  2019-03-29       Impact factor: 6.600

8.  Characterization of lamin mutation phenotypes in Drosophila and comparison to human laminopathies.

Authors:  Andrés Muñoz-Alarcón; Maja Pavlovic; Jasmine Wismar; Bertram Schmitt; Maria Eriksson; Per Kylsten; Mitchell S Dushay
Journal:  PLoS One       Date:  2007-06-13       Impact factor: 3.240

9.  Mutations in LMNA modulate the lamin A--Nesprin-2 interaction and cause LINC complex alterations.

Authors:  Liu Yang; Martina Munck; Karthic Swaminathan; Larisa E Kapinos; Angelika A Noegel; Sascha Neumann
Journal:  PLoS One       Date:  2013-08-20       Impact factor: 3.240

10.  Novel insights into the disease etiology of laminopathies.

Authors:  Chin Yee Ho; Diana E Jaalouk; Jan Lammerding
Journal:  Rare Dis       Date:  2013-11-06
View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.