Literature DB >> 27555236

Fabry Disease: A Disorder of Childhood Onset.

Raphael Schiffmann1, Markus Ries2.   

Abstract

BACKGROUND: Fabry disease, an X-linked disorder of glycosphingolipids, markedly increases the risk of systemic vasculopathy, ischemic stroke, small-fiber peripheral neuropathy, cardiac dysfunction, and chronic kidney disease.
METHODS: We performed an extensive PubMed search on the topic of Fabry disease and drew from our cumulative 43 years of experience.
RESULTS: Most of these complications are nonspecific in nature and clinically indistinguishable from similar abnormalities that occur in the context of more common disorders in the general population. This disease is caused by variants of the GLA gene, and its incidence may have been underestimated. However, one must also guard against overdiagnosis of Fabry disease and unjustified enzyme replacement therapy, because some of the gene variants are benign. Specific therapy for Fabry disease has been developed in the last few years, but its clinical effect has been modest. Novel therapeutic agents are being developed. Standard "nonspecific" medical and surgical therapy is necessary and effective in slowing deterioration or compensating for organ failure in patients with Fabry disease.
CONCLUSIONS: Fabry disease is a treatable and modifiable genetic risk factor for a myriad of clinical organ complications. Fabry disease may be frequently overlooked but on occasion overdiagnosed.
Copyright © 2016 Elsevier Inc. All rights reserved.

Entities:  

Keywords:  X-linked; angiokeratoma; genetic disease; heart disease; sphingolipids; stroke

Mesh:

Year:  2016        PMID: 27555236     DOI: 10.1016/j.pediatrneurol.2016.07.001

Source DB:  PubMed          Journal:  Pediatr Neurol        ISSN: 0887-8994            Impact factor:   3.372


  9 in total

Review 1.  Enzyme replacement therapy and beyond-in memoriam Roscoe O. Brady, M.D. (1923-2016).

Authors:  Markus Ries
Journal:  J Inherit Metab Dis       Date:  2017-03-17       Impact factor: 4.982

2.  Recommendations for the inclusion of Fabry disease as a rare febrile condition in existing algorithms for fever of unknown origin.

Authors:  Raffaele Manna; Roberto Cauda; Sandro Feriozzi; Giovanni Gambaro; Antonio Gasbarrini; Didier Lacombe; Avi Livneh; Alberto Martini; Huri Ozdogan; Antonio Pisani; Eleonora Riccio; Elena Verrecchia; Lorenzo Dagna
Journal:  Intern Emerg Med       Date:  2017-07-19       Impact factor: 3.397

Review 3.  Induced pluripotent stem cell models of lysosomal storage disorders.

Authors:  Daniel K Borger; Benjamin McMahon; Tamanna Roshan Lal; Jenny Serra-Vinardell; Elma Aflaki; Ellen Sidransky
Journal:  Dis Model Mech       Date:  2017-06-01       Impact factor: 5.758

4.  Different renal phenotypes in related adult males with Fabry disease with the same classic genotype.

Authors:  Renzo Mignani; Mariarita Moschella; Giovanna Cenacchi; Ilaria Donati; Marta Flachi; Daniela Grimaldi; Davide Cerretani; Paola De Giovanni; Marcello Montevecchi; Angelo Rigotti; Alessandro Ravasio
Journal:  Mol Genet Genomic Med       Date:  2017-05-08       Impact factor: 2.183

5.  Glucosylceramide synthase inhibition with lucerastat lowers globotriaosylceramide and lysosome staining in cultured fibroblasts from Fabry patients with different mutation types.

Authors:  R W D Welford; A Mühlemann; M Garzotti; V Rickert; P M A Groenen; O Morand; N Üçeyler; M R Probst
Journal:  Hum Mol Genet       Date:  2018-10-01       Impact factor: 6.150

6.  Longitudinal study on ocular manifestations in a cohort of patients with Fabry disease.

Authors:  Langis Michaud
Journal:  PLoS One       Date:  2019-06-27       Impact factor: 3.240

7.  Efficacy and safety of migalastat in a Japanese population: a subgroup analysis of the ATTRACT study.

Authors:  Ichiei Narita; Toya Ohashi; Norio Sakai; Takashi Hamazaki; Nina Skuban; Jeffrey P Castelli; Hjalmar Lagast; Jay A Barth
Journal:  Clin Exp Nephrol       Date:  2019-12-30       Impact factor: 2.801

8.  Recurrent fever of unknown origin: An overlooked symptom of Fabry disease.

Authors:  Yi Luo; Di Wu; Min Shen
Journal:  Mol Genet Genomic Med       Date:  2020-08-14       Impact factor: 2.183

9.  The effect of the glucosylceramide synthase inhibitor lucerastat on cardiac repolarization: results from a thorough QT study in healthy subjects.

Authors:  Markus S Mueller; Patricia N Sidharta; Christine Voors-Pette; Borje Darpo; Hongqi Xue; Jasper Dingemanse
Journal:  Orphanet J Rare Dis       Date:  2020-10-27       Impact factor: 4.123

  9 in total

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