Literature DB >> 12799787

[Fabry's disease: new therapeutic options for this lysosomal storage disorder].

A J Grau1, M Schwaninger, H H Goebel, M Beck.   

Abstract

Fabry's disease is an x-linked, recessive, lysosomal storage disorder that results from deficient alpha-galactosidase A activity with pathological sphingolipid deposition mainly in endothelium, smooth muscle cells, kidneys, central and peripheral nervous system, and myocardium. Clinical manifestation mostly occurs during childhood and adolescence with severe pain attacks or chronic pain mainly in hands and feet, hypohydrosis, and skin lesions (angiokeratoma). In more advanced disease stages, renal and cerebrovascular complications develop with proteinuria and later renal failure and cerebral ischemia caused by cerebral microangiopathy, dilatative arteriopathy, or cardiac embolism. Heterozygote female carriers are severely affected more often than was previously considered. The diagnosis is based on the detection of deficient alpha-galactosidase A activity in leukocytes, fibroblasts, or tissue biopsies. Two randomised placebo-controlled studies showed that enzyme replacement is effective by demonstrating either reduced pain or reduced tissue sphingolipid deposition. Early diagnosis of Fabry's disease is important in view of these new causal therapeutic options.

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Year:  2003        PMID: 12799787     DOI: 10.1007/s00115-003-1513-6

Source DB:  PubMed          Journal:  Nervenarzt        ISSN: 0028-2804            Impact factor:   1.214


  28 in total

1.  Anderson-Fabry disease: clinical manifestations and impact of disease in a cohort of 60 obligate carrier females.

Authors:  K D MacDermot; A Holmes; A H Miners
Journal:  J Med Genet       Date:  2001-11       Impact factor: 6.318

Review 2.  Neuropathic pain in Anderson-Fabry disease: pathology and therapeutic options.

Authors:  J MacDermot; K D MacDermot
Journal:  Eur J Pharmacol       Date:  2001-10-19       Impact factor: 4.432

3.  Regional cerebral hyperperfusion and nitric oxide pathway dysregulation in Fabry disease: reversal by enzyme replacement therapy.

Authors:  D F Moore; L T Scott; M T Gladwin; G Altarescu; C Kaneski; K Suzuki; M Pease-Fye; R Ferri; R O Brady; P Herscovitch; R Schiffmann
Journal:  Circulation       Date:  2001-09-25       Impact factor: 29.690

4.  Enhanced endothelium-dependent vasodilation in Fabry disease.

Authors:  G Altarescu; D F Moore; R Pursley; U Campia; S Goldstein; M Bryant; J A Panza; R Schiffmann
Journal:  Stroke       Date:  2001-07       Impact factor: 7.914

5.  Safety and efficacy of recombinant human alpha-galactosidase A replacement therapy in Fabry's disease.

Authors:  C M Eng; N Guffon; W R Wilcox; D P Germain; P Lee; S Waldek; L Caplan; G E Linthorst; R J Desnick
Journal:  N Engl J Med       Date:  2001-07-05       Impact factor: 91.245

6.  Fabry disease: impaired autonomic function.

Authors:  W J Cable; E H Kolodny; R D Adams
Journal:  Neurology       Date:  1982-05       Impact factor: 9.910

Review 7.  Fabry disease: recent advances in enzyme replacement therapy.

Authors:  Dominique P Germain
Journal:  Expert Opin Investig Drugs       Date:  2002-10       Impact factor: 6.206

8.  Diffuse central neuronal involvement in Fabry disease: a proton MRS imaging study.

Authors:  G Tedeschi; S Bonavita; T K Banerjee; A Virta; R Schiffmann
Journal:  Neurology       Date:  1999-05-12       Impact factor: 9.910

9.  Enzyme replacement therapy in Fabry disease: a randomized controlled trial.

Authors:  R Schiffmann; J B Kopp; H A Austin; S Sabnis; D F Moore; T Weibel; J E Balow; R O Brady
Journal:  JAMA       Date:  2001-06-06       Impact factor: 56.272

10.  A phase 1/2 clinical trial of enzyme replacement in fabry disease: pharmacokinetic, substrate clearance, and safety studies.

Authors:  C M Eng; M Banikazemi; R E Gordon; M Goldman; R Phelps; L Kim; A Gass; J Winston; S Dikman; J T Fallon; S Brodie; C B Stacy; D Mehta; R Parsons; K Norton; M O'Callaghan; R J Desnick
Journal:  Am J Hum Genet       Date:  2001-02-01       Impact factor: 11.025

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  2 in total

1.  Availability of and access to orphan drugs: an international comparison of pharmaceutical treatments for pulmonary arterial hypertension, Fabry disease, hereditary angioedema and chronic myeloid leukaemia.

Authors:  Carl Rudolf Blankart; Tom Stargardt; Jonas Schreyögg
Journal:  Pharmacoeconomics       Date:  2011-01       Impact factor: 4.981

2.  The microstructure of cornea verticillata in Fabry disease and amiodarone-induced keratopathy: a confocal laser-scanning microscopy study.

Authors:  Karen Falke; Armin Büttner; Michael Schittkowski; Oliver Stachs; Robert Kraak; Andrey Zhivov; Arndt Rolfs; Rudolf Guthoff
Journal:  Graefes Arch Clin Exp Ophthalmol       Date:  2008-10-18       Impact factor: 3.117

  2 in total

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