Literature DB >> 17845137

Therapy of Marfan syndrome.

Daniel P Judge1, Harry C Dietz.   

Abstract

Marfan syndrome is a common inherited disorder of connective tissue caused by deficiency of the matrix protein fibrillin-1. Effective surgical therapy for the most life-threatening manifestation, aortic root aneurysm, has led to a nearly normal lifespan for affected individuals who are appropriately recognized and treated. Traditional medical therapies, such as beta-adrenergic receptor blockade, are used to slow pathologic aortic growth and decrease the risk of aortic dissection by decreasing hemodynamic stress. New insights regarding the pathogenesis of Marfan syndrome have developed from investigation of murine models of this disorder. Fibrillin-1 deficiency is associated with excess signaling by transforming growth factor beta (TGFbeta). TGFbeta antagonists have shown great success in improving or preventing several manifestations of Marfan syndrome in these mice, including aortic aneurysm. These results highlight the potential for development of targeted therapies based on discovery of disease genes and interrogation of pathogenesis in murine models.

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Year:  2008        PMID: 17845137     DOI: 10.1146/annurev.med.59.103106.103801

Source DB:  PubMed          Journal:  Annu Rev Med        ISSN: 0066-4219            Impact factor:   13.739


  19 in total

Review 1.  The pathogenesis of aortopathy in Marfan syndrome and related diseases.

Authors:  Jeffrey A Jones; John S Ikonomidis
Journal:  Curr Cardiol Rep       Date:  2010-03       Impact factor: 2.931

2.  Long form of latent TGF-β binding protein 1 (Ltbp1L) regulates cardiac valve development.

Authors:  Vesna Todorovic; Erin Finnegan; Laina Freyer; Lior Zilberberg; Mitsuhiko Ota; Daniel B Rifkin
Journal:  Dev Dyn       Date:  2011-01       Impact factor: 3.780

3.  High incidence and severity of periodontitis in patients with Marfan syndrome in Japan.

Authors:  Jun-Ichi Suzuki; Yasushi Imai; Mieko Aoki; Daishi Fujita; Norio Aoyama; Yuko Tada; Hiroshi Akazawa; Yuichi Izumi; Mitsuaki Isobe; Issei Komuro; Ryozo Nagai; Yasunobu Hirata
Journal:  Heart Vessels       Date:  2013-11-08       Impact factor: 2.037

4.  ADAMTSL6β protein rescues fibrillin-1 microfibril disorder in a Marfan syndrome mouse model through the promotion of fibrillin-1 assembly.

Authors:  Masahiro Saito; Misaki Kurokawa; Masahito Oda; Masamitsu Oshima; Ko Tsutsui; Kazutaka Kosaka; Kazuhisa Nakao; Miho Ogawa; Ri-ichiroh Manabe; Naoto Suda; Ganburged Ganjargal; Yasunobu Hada; Toshihide Noguchi; Toshio Teranaka; Kiyotoshi Sekiguchi; Toshiyuki Yoneda; Takashi Tsuji
Journal:  J Biol Chem       Date:  2011-08-31       Impact factor: 5.157

5.  Molecular mechanisms of inherited thoracic aortic disease - from gene variant to surgical aneurysm.

Authors:  Elizabeth Robertson; Candice Dilworth; Yaxin Lu; Brett Hambly; Richmond Jeremy
Journal:  Biophys Rev       Date:  2014-12-06

6.  A new mouse model for marfan syndrome presents phenotypic variability associated with the genetic background and overall levels of Fbn1 expression.

Authors:  Bruno L Lima; Enrico J C Santos; Gustavo R Fernandes; Christian Merkel; Marco R B Mello; Juliana P A Gomes; Marina Soukoyan; Alexandre Kerkis; Silvia M G Massironi; José A Visintin; Lygia V Pereira
Journal:  PLoS One       Date:  2010-11-30       Impact factor: 3.240

7.  The importance of genetic testing in the clinical management of patients with Marfan syndrome and related disorders.

Authors:  Mine Arslan-Kirchner; Yskert von Kodolitsch; Jörg Schmidtke
Journal:  Dtsch Arztebl Int       Date:  2008-07-04       Impact factor: 5.594

8.  Aortic Dimensions, Biophysical Properties, and Plasma Biomarkers in Children and Adults with Marfan or Loeys-Dietz Syndrome.

Authors:  Jason Z Cui; Kevin C Harris; Koen Raedschelders; Zsuzsanna Hollander; James E Potts; Astrid De Souza; Marla Kiess; Bruce M McManus; Pascal Bernatchez; Leslie A Raffin; Heidi Paine; Cornelis van Breemen; George G S Sandor; Mitra Esfandiarei
Journal:  CJC Open       Date:  2020-12-28

9.  Multi-scale biomechanical remodeling in aging and genetic mutant murine mitral valve leaflets: insights into Marfan syndrome.

Authors:  Russell A Gould; Ravi Sinha; Hamza Aziz; Rosanne Rouf; Harry C Dietz; Daniel P Judge; Jonathan Butcher
Journal:  PLoS One       Date:  2012-09-11       Impact factor: 3.240

10.  Informative STR Markers for Marfan Syndrome in Birjand, Iran.

Authors:  Ezzat Dadkhah; Masood Ziaee; Mohammad Hossein Davari; Toba Kazemi; Mohammad Reza Abbaszadegan
Journal:  Iran J Basic Med Sci       Date:  2012-09       Impact factor: 2.699

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