Literature DB >> 23980562

Intrafamilial phenotypic variability in four families with Anderson-Fabry disease.

M Rigoldi1, D Concolino, A Morrone, F Pieruzzi, R Ravaglia, F Furlan, F Santus, P Strisciuglio, G Torti, R Parini.   

Abstract

We analysed the clinical history of 16 hemizygous males affected by Anderson-Fabry Disease, from four families, to verify their intrafamilial phenotypic variability. Seven male patients, ranging from 26 to 61 years of age, died, whereas nine (age range 23-55) are alive. Eleven patients have undergone enzyme replacement therapy (ERT) for a period of 5-10 years. We have found a wide range of intrafamilial phenotypic variability in these families, both in terms of target-organs and severity of the disease. Overall, our findings confirm previous data from the literature showing a high degree of intrafamilial phenotypic variability in patients carrying the same mutation. Furthermore, our results underscore the difficulty in giving accurate prognostic information to patients during genetic counselling, both in terms of rate of disease progression and involvement of different organs, when such prognosis is solely based on the patient's family history.
© 2013 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.

Entities:  

Keywords:  Anderson-Fabry disease; Fabry; GLA gene; agalsidase alpha; agalsidase beta; alpha-galactosidase deficiency; genetic counselling; intrafamilial variability; phenotypic heterogeneity

Mesh:

Year:  2013        PMID: 23980562     DOI: 10.1111/cge.12261

Source DB:  PubMed          Journal:  Clin Genet        ISSN: 0009-9163            Impact factor:   4.438


  30 in total

1.  A classic variant of Fabry disease in a family with the M296I late-onset variant.

Authors:  Shuma Hirashio; Reiko Kagawa; Go Tajima; Takao Masaki
Journal:  CEN Case Rep       Date:  2020-09-09

2.  High variability of Fabry disease manifestations in an extended Italian family.

Authors:  Giuseppe Cammarata; Pasquale Fatuzzo; Margherita Stefania Rodolico; Paolo Colomba; Luigi Sicurella; Francesco Iemolo; Carmela Zizzo; Riccardo Alessandro; Caterina Bartolotta; Giovanni Duro; Ines Monte
Journal:  Biomed Res Int       Date:  2015-04-22       Impact factor: 3.411

3.  Taming molecular flexibility to tackle rare diseases.

Authors:  Maria Vittoria Cubellis; Marc Baaden; Giuseppina Andreotti
Journal:  Biochimie       Date:  2015-04-02       Impact factor: 4.079

4.  p.R301X Mutation and Variable Phenotypic Appearance of Fabry Disease.

Authors:  Ruya Ozelsancak; Bulent Uyar
Journal:  Am J Case Rep       Date:  2016-05-09

5.  Urinary Podocyte Loss Is Increased in Patients with Fabry Disease and Correlates with Clinical Severity of Fabry Nephropathy.

Authors:  Brent Fall; C Ronald Scott; Michael Mauer; Stuart Shankland; Jeffrey Pippin; Jonathan A Jefferson; Eric Wallace; David Warnock; Behzad Najafian
Journal:  PLoS One       Date:  2016-12-16       Impact factor: 3.240

Review 6.  The Large Phenotypic Spectrum of Fabry Disease Requires Graduated Diagnosis and Personalized Therapy: A Meta-Analysis Can Help to Differentiate Missense Mutations.

Authors:  Valentina Citro; Marco Cammisa; Ludovica Liguori; Chiara Cimmaruta; Jan Lukas; Maria Vittoria Cubellis; Giuseppina Andreotti
Journal:  Int J Mol Sci       Date:  2016-12-01       Impact factor: 5.923

7.  Genetic variants associated with Fabry disease progression despite enzyme replacement therapy.

Authors:  Francesca Scionti; Maria Teresa Di Martino; Simona Sestito; Angela Nicoletti; Francesca Falvo; Katia Roppa; Mariamena Arbitrio; Pietro Hiram Guzzi; Giuseppe Agapito; Antonio Pisani; Eleonora Riccio; Daniela Concolino; Licia Pensabene
Journal:  Oncotarget       Date:  2017-11-18

8.  Home infusion program with enzyme replacement therapy for Fabry disease: The experience of a large Italian collaborative group.

Authors:  D Concolino; L Amico; M D Cappellini; E Cassinerio; M Conti; M A Donati; F Falvo; A Fiumara; M Maccarone; R Manna; A Matucci; M B Musumeci; A Nicoletti; R Nisticò; F Papadia; R Parini; D Peluso; L Pensabene; A Pisani; G Pistone; M Rigoldi; I Romani; M Tenuta; G Torti; M Veroux; E Zachara
Journal:  Mol Genet Metab Rep       Date:  2017-06-22

9.  Gastrointestinal Symptoms of Patients with Fabry Disease.

Authors:  Licia Pensabene; Simona Sestito; Angela Nicoletti; Francesca Graziano; Pietro Strisciuglio; Daniela Concolino
Journal:  Gastroenterol Res Pract       Date:  2015-12-31       Impact factor: 2.260

10.  Skin globotriaosylceramide 3 deposits are specific to Fabry disease with classical mutations and associated with small fibre neuropathy.

Authors:  Rocco Liguori; Alex Incensi; Silvia de Pasqua; Renzo Mignani; Enrico Fileccia; Marisa Santostefano; Elena Biagini; Claudio Rapezzi; Silvia Palmieri; Ilaria Romani; Walter Borsini; Alessandro Burlina; Roberto Bombardi; Marco Caprini; Patrizia Avoni; Vincenzo Donadio
Journal:  PLoS One       Date:  2017-07-03       Impact factor: 3.240

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