Literature DB >> 12694340

Fabry disease: diagnosis and treatment.

Frank Breunig1, Frank Weidemann, Meinrad Beer, Andreas Eggert, Vera Krane, Matthias Spindler, Jörn Sandstede, Jörg Strotmann, Christoph Wanner.   

Abstract

Fabry disease is an X-linked lysosomal storage disorder that results from a deficiency of the enzyme alpha-galactosidase A (alpha-Gal A). The lack of alpha-Gal A causes an intracellular accumulation of glycosphingolipids, mainly globotriaosyceramide (GL3). Affected organs include, among others, the vascular endothelium, heart, brain, and kidneys, leading to end-stage renal disease (ESRD). Since Fabry disease cannot be cured at present, clinical management is symptomatic. Enzyme replacement therapy (ERT) with recombinant alpha-Gal A has been introduced as a new therapeutic option for the treatment of Fabry patients. Short-term (one year) clinical studies have positively correlated ERT with improvement of clinical symptoms and microvascular endothelial cell clearance. Treatment outcome concerning severe organ manifestations such as proteinuria and renal function impairment, left ventricular hypertrophy, and heart failure in the long run has yet to be shown. In our studies we used sensitive and noninvasive techniques such as ultrasound-based strain rate imaging and magnetic resonance imaging (MRI), combined with MR-spectroscopy (MR-S), for the quantification of functional abnormalities at an early stage of the disease and during long-term follow-up. Future issues should determine the appropriate timing to start therapy and how children and heterozygous females should be managed. Given the diagnostic and therapeutic potential today, it is of importance to identify patients at an early stage and to start therapeutic intervention before progression of organ damage is inevitable.

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Year:  2003        PMID: 12694340     DOI: 10.1046/j.1523-1755.63.s84.5.x

Source DB:  PubMed          Journal:  Kidney Int Suppl        ISSN: 0098-6577            Impact factor:   10.545


  13 in total

1.  A survey of the pain experienced by males and females with Fabry disease.

Authors:  Andrea L Gibas; Regan Klatt; Jack Johnson; Joe T R Clarke; Joel Katz
Journal:  Pain Res Manag       Date:  2006       Impact factor: 3.037

Review 2.  Progressive hearing loss in Fabry's disease: a case report.

Authors:  Florian M Barras; Raphaël Maire
Journal:  Eur Arch Otorhinolaryngol       Date:  2006-03-07       Impact factor: 2.503

3.  Cross-sectional baseline analysis of electrocardiography in a large cohort of patients with untreated Fabry disease.

Authors:  Markus Niemann; Tanja Hartmann; Mehdi Namdar; Frank Breunig; Meinrad Beer; Wolfram Machann; Sebastian Herrmann; Georg Ertl; Christoph Wanner; Frank Weidemann
Journal:  J Inherit Metab Dis       Date:  2012-10-11       Impact factor: 4.982

Review 4.  Agalsidase Beta: a review of its use in the management of Fabry disease.

Authors:  Gillian M Keating; Dene Simpson
Journal:  Drugs       Date:  2007       Impact factor: 9.546

Review 5.  Understanding the gastrointestinal manifestations of Fabry disease: promoting prompt diagnosis.

Authors:  Claire Zar-Kessler; Amel Karaa; Katherine Bustin Sims; Virginia Clarke; Braden Kuo
Journal:  Therap Adv Gastroenterol       Date:  2016-04-15       Impact factor: 4.409

6.  Disease rarity, carrier status, and gender: a triple disadvantage for women with Fabry disease.

Authors:  Andrea L Gibas; Regan Klatt; Jack Johnson; Joe T R Clarke; Joel Katz
Journal:  J Genet Couns       Date:  2008-10-16       Impact factor: 2.537

Review 7.  Fabry disease in the era of enzyme replacement therapy: a renal perspective.

Authors:  Monique E Cho; Jeffrey B Kopp
Journal:  Pediatr Nephrol       Date:  2004-04-03       Impact factor: 3.714

8.  [Juvenile ischemic brain infarction. Clinical aspects, etiology spectrum, diagnosis and therapy].

Authors:  D G Nabavi; A Allroggen; E B Ringelstein
Journal:  Nervenarzt       Date:  2004-02       Impact factor: 1.214

9.  Electrical Changes in Resting, Exercise, and Holter Electrocardiography in Fabry Cardiomyopathy.

Authors:  Johannes Krämer; Peter Nordbeck; Stefan Störk; Christian Ritter; Georg Ertl; Christoph Wanner; Frank Weidemann
Journal:  JIMD Rep       Date:  2015-10-27

10.  Imaging Mass Spectrometry Reveals Acyl-Chain- and Region-Specific Sphingolipid Metabolism in the Kidneys of Sphingomyelin Synthase 2-Deficient Mice.

Authors:  Masayuki Sugimoto; Masato Wakabayashi; Yoichi Shimizu; Takeshi Yoshioka; Kenichi Higashino; Yoshito Numata; Tomohiko Okuda; Songji Zhao; Shota Sakai; Yasuyuki Igarashi; Yuji Kuge
Journal:  PLoS One       Date:  2016-03-24       Impact factor: 3.240

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