Literature DB >> 33475959

Network Analysis Reveals Proteins Associated with Aortic Dilatation in Mucopolysaccharidoses.

Thiago Corrêa1,2, Bruno César Feltes3, Esteban Alberto Gonzalez1,2, Guilherme Baldo1,2, Ursula Matte4,5.   

Abstract

Mucopolysaccharidoses are caused by a deficiency of enzymes involved in the degradation of glycosaminoglycans. Heart diseases are a significant cause of morbidity and mortality in MPS patients, even in conditions in which enzyme replacement therapy is available. In this sense, cardiovascular manifestations, such as heart hypertrophy, cardiac function reduction, increased left ventricular chamber, and aortic dilatation, are among the most frequent. However, the downstream events which influence the heart dilatation process are unclear. Here, we employed systems biology tools together with transcriptomic data to investigate new elements that may be involved in aortic dilatation in Mucopolysaccharidoses syndrome. We identified candidate genes involved in biological processes related to inflammatory responses, deposition of collagen, and lipid accumulation in the cardiovascular system that may be involved in aortic dilatation in the Mucopolysaccharidoses I and VII. Furthermore, we investigated the molecular mechanisms of losartan treatment in Mucopolysaccharidoses I mice to underscore how this drug acts to prevent aortic dilation. Our data indicate that the association between the TGF-b signaling pathway, Fos, and Col1a1 proteins can play an essential role in aortic dilation's pathophysiology and its subsequent improvement by losartan treatment.

Entities:  

Keywords:  Aortic dilatation; Col1a1; Fos; MPS I; MPS VII; TGF-b

Year:  2021        PMID: 33475959     DOI: 10.1007/s12539-020-00406-3

Source DB:  PubMed          Journal:  Interdiscip Sci        ISSN: 1867-1462            Impact factor:   2.233


  63 in total

1.  Mucopolysaccharidosis I under enzyme replacement therapy with laronidase--a mortality case with autopsy report.

Authors:  H-Y Lin; S-P Lin; C-K Chuang; M-R Chen; B-F Chen; J E Wraith
Journal:  J Inherit Metab Dis       Date:  2005       Impact factor: 4.982

2.  Introduction to Brazilian Guidelines to Diagnosis, Treatment,and Monitoring for Gaucher Disease, Fabry Disease, Mucopolysaccharidosis I, and Pompe Disease.

Authors:  Ana Maria Martins
Journal:  J Pediatr       Date:  2009-10       Impact factor: 4.406

3.  Aortic root dilatation in patients with mucopolysaccharidoses and the impact of enzyme replacement therapy.

Authors:  Fabiano de Oliveira Poswar; Carolina Fischinger Moura de Souza; Roberto Giugliani; Guilherme Baldo
Journal:  Heart Vessels       Date:  2018-08-22       Impact factor: 2.037

4.  Molecular characterization of 355 mucopolysaccharidosis patients reveals 104 novel mutations.

Authors:  Laura M Pollard; Julie R Jones; Tim C Wood
Journal:  J Inherit Metab Dis       Date:  2012-09-14       Impact factor: 4.982

Review 5.  Clinical presentation and diagnosis of mucopolysaccharidoses.

Authors:  Molly Stapleton; Nivethitha Arunkumar; Francyne Kubaski; Robert W Mason; Orii Tadao; Shunji Tomatsu
Journal:  Mol Genet Metab       Date:  2018-01-31       Impact factor: 4.797

6.  A complete deficiency of Hyaluronoglucosaminidase 1 (HYAL1) presenting as familial juvenile idiopathic arthritis.

Authors:  Lisa Imundo; Charles A Leduc; Saurav Guha; Marc Brown; Giorgio Perino; Lara Gushulak; Barbara Triggs-Raine; Wendy K Chung
Journal:  J Inherit Metab Dis       Date:  2011-05-11       Impact factor: 4.982

Review 7.  Critical review of current MPS guidelines and management.

Authors:  Molly Stapleton; Hiroo Hoshina; Kazuki Sawamoto; Francyne Kubaski; Robert W Mason; William G Mackenzie; Mary Theroux; Hironori Kobayashi; Seiji Yamaguchi; Yasuyuki Suzuki; Toshiyuki Fukao; Orii Tadao; Hiroyuki Ida; Shunji Tomatsu
Journal:  Mol Genet Metab       Date:  2018-07-07       Impact factor: 4.797

8.  Identification and characterization of 13 new mutations in mucopolysaccharidosis type I patients.

Authors:  Ursula Matte; Gouri Yogalingam; Doug Brooks; Sandra Leistner; Ida Schwartz; Luciane Lima; Denise Y Norato; Jaime M Brum; Clare Beesley; Bryan Winchester; Roberto Giugliani; John J Hopwood
Journal:  Mol Genet Metab       Date:  2003-01       Impact factor: 4.797

9.  Identification and molecular characterization of alpha-L-iduronidase mutations present in mucopolysaccharidosis type I patients undergoing enzyme replacement therapy.

Authors:  G Yogalingam; X-H Guo; V J Muller; D A Brooks; P R Clements; E D Kakkis; J J Hopwood
Journal:  Hum Mutat       Date:  2004-09       Impact factor: 4.878

10.  Mucopolysacccharidoses: From understanding to treatment, a century of discoveries.

Authors:  Roberto Giugliani
Journal:  Genet Mol Biol       Date:  2012-12-18       Impact factor: 1.771

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