Control of proteinuria with increased doses of agalsidase alfa in a patient with Fabry disease with atypical genotype-phenotype expression

Nefrologia. 2015 Nov-Dec;35(6):578-81. doi: 10.1016/j.nefro.2015.08.001. Epub 2015 Sep 15.

Abstract

Fabry disease is a rare X-linked lysosomal storage disorder of glycosphingolipids, caused by the partial or complete deficiency of the lysosomal enzyme alpha-galactosidase A (a-Gal A). The missense mutation pN215S usually causes a milder form of the disease with isolated cardiac involvement. We report a case of a male Fabry patient with the pN215S mutation and a generalized disease. He suffered a relapse in proteinuria which responded to increased doses of the administered recombinant enzyme. Individualization of enzyme replacement therapy must be considered in selected cases characterized by clinical deterioration.

Keywords: Enfermedad de Fabry; Enzyme replacement therapy; Fabry disease; Mutación pN215S; Mutation pN215S; Proteinuria; Terapia sustitutiva enzimática.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Disease Progression
  • Dose-Response Relationship, Drug
  • Fabry Disease / complications*
  • Fabry Disease / genetics
  • Genetic Association Studies
  • Heart Ventricles / pathology
  • Humans
  • Hypertrophy, Left Ventricular / etiology
  • Hypertrophy, Left Ventricular / pathology
  • Isoenzymes / administration & dosage
  • Isoenzymes / therapeutic use
  • Male
  • Mutation, Missense*
  • Organ Size / drug effects
  • Proteinuria / drug therapy*
  • Proteinuria / etiology
  • Recombinant Proteins / administration & dosage
  • Recombinant Proteins / therapeutic use
  • Recurrence
  • alpha-Galactosidase / administration & dosage
  • alpha-Galactosidase / genetics*
  • alpha-Galactosidase / therapeutic use

Substances

  • Isoenzymes
  • Recombinant Proteins
  • agalsidase alfa
  • alpha-Galactosidase