Literature DB >> 28596458

Prevalence of Fabry Disease and Outcomes in Young Canadian Patients With Cryptogenic Ischemic Cerebrovascular Events.

Sylvain Lanthier1, Gustavo Saposnik2, Gerald Lebovic2, Karen Pope2, Daniel Selchen2, David F Moore2.   

Abstract

BACKGROUND AND
PURPOSE: Previous studies reported Fabry disease in 0% to 4% of young patients with cryptogenic ischemic stroke (IS). We sought to determine the prevalence of Fabry and outcomes among young Canadians with cryptogenic IS or transient ischemic attack (TIA).
METHODS: We prospectively enrolled individuals aged 18 to 55 with IS or speech or motor TIA, and no cause identified despite predetermined investigation. α-galactosidase-A gene was sequenced for Fabry diagnosis. National Institutes of Health Stroke Scale score was measured at presentation to quantify stroke severity. Modified Rankin Scale determined functional outcomes ≤7 days after presentation and 6 months later.
RESULTS: We enrolled 365 patients with IS and 32 with TIA. α-galactosidase-A sequencing identified a single carrier of a genetic variant of unknown significance (p.R118C) and no well-recognized pathogenic variants. Mean National Institutes of Health Stroke Scale score was 3.1. Proportion of patients with modified Rankin Scale of 0 to 2 was 70.7% at ≤7 days and 87.4% at 6 months. National Institutes of Health Stroke Scale score at presentation and diabetes mellitus predicted 6-month modified Rankin Scale. Thirteen patients experienced 5 recurrent IS and 9 TIA during follow-up. No patient died. Most patients (98.7%) returned home. Among previous workers, 43% had residual working limitations.
CONCLUSIONS: In this Canadian cohort of patients with cryptogenic IS or TIA, the prevalence of Fabry was 0.3% if p.R118C variant is considered as pathogenic. This suggests that more cost-effective methods should be applied for diagnosis of Fabry rather than systematic genetic screening in this population. Overall, cryptogenic IS in young adults is associated with favorable outcomes.
© 2017 American Heart Association, Inc.

Entities:  

Keywords:  Fabry disease; outcome; prevalence; speech

Mesh:

Year:  2017        PMID: 28596458     DOI: 10.1161/STROKEAHA.116.016083

Source DB:  PubMed          Journal:  Stroke        ISSN: 0039-2499            Impact factor:   7.914


  9 in total

1.  Stroke in young adults: Five new things.

Authors:  Nirav Bhatt; Amer M Malik; Seemant Chaturvedi
Journal:  Neurol Clin Pract       Date:  2018-12

2.  Screening for Fabry Disease in Patients With Juvenile Systemic Lupus Erythematosus.

Authors:  Ertugrul Kiykim; Sezgin Şahİn; Tanyel ZubarioĞlu; Kenan Barut; Amra Adrovic; Mehmet Şerif Cansever; Ayşe Çiğdem AktuĞlu Zeybek; Özgür KasapÇopur
Journal:  Arch Rheumatol       Date:  2020-02-07       Impact factor: 1.472

Review 3.  Diagnosis and Screening of Patients with Fabry Disease.

Authors:  Irfan Vardarli; Christoph Rischpler; Ken Herrmann; Frank Weidemann
Journal:  Ther Clin Risk Manag       Date:  2020-06-22       Impact factor: 2.423

Review 4.  Renal Manifestations of Fabry Disease: A Narrative Review.

Authors:  Cassiano Augusto Braga Silva; José A Moura-Neto; Marlene Antônia Dos Reis; Osvaldo Merege Vieira Neto; Fellype Carvalho Barreto
Journal:  Can J Kidney Health Dis       Date:  2021-01-19

Review 5.  Prevalence of Fabry Disease in Patients With Cryptogenic Strokes: A Systematic Review.

Authors:  Juan Fernando Ortiz; Jashank Parwani; Paul W Millhouse; Ahmed Eissa-Garcés; Gashaw Hassen; Victor D Cuenca; Ivan Mateo Alzamora; Mahika Khurana; Domenica Herrera-Bucheli; Abbas Altamimi; Adam Atoot; Wilson Cueva
Journal:  Cureus       Date:  2021-11-08

Review 6.  Expert opinion on the recognition, diagnosis and management of children and adults with Fabry disease: a multidisciplinary Turkey perspective.

Authors:  Fatih Ezgu; Erkan Alpsoy; Zerrin Bicik Bahcebasi; Ozgur Kasapcopur; Melis Palamar; Huseyin Onay; Binnaz Handan Ozdemir; Mehmet Akif Topcuoglu; Omac Tufekcioglu
Journal:  Orphanet J Rare Dis       Date:  2022-03-02       Impact factor: 4.123

7.  Effectiveness of plasma lyso-Gb3 as a biomarker for selecting high-risk patients with Fabry disease from multispecialty clinics for genetic analysis.

Authors:  Hiroki Maruyama; Kaori Miyata; Mariko Mikame; Atsumi Taguchi; Chu Guili; Masaru Shimura; Kei Murayama; Takeshi Inoue; Saori Yamamoto; Koichiro Sugimura; Koichi Tamita; Toshihiro Kawasaki; Jun Kajihara; Akifumi Onishi; Hitoshi Sugiyama; Teiko Sakai; Ichijiro Murata; Takamasa Oda; Shigeru Toyoda; Kenichiro Hanawa; Takeo Fujimura; Shigehisa Ura; Mimiko Matsumura; Hideki Takano; Satoshi Yamashita; Gaku Matsukura; Ryushi Tazawa; Tsuyoshi Shiga; Mio Ebato; Hiroshi Satoh; Satoshi Ishii
Journal:  Genet Med       Date:  2018-03-15       Impact factor: 8.822

8.  How common are single gene mutations as a cause for lacunar stroke? A targeted gene panel study.

Authors:  Rhea Y Y Tan; Matthew Traylor; Karyn Megy; Daniel Duarte; Sri V V Deevi; Olga Shamardina; Rutendo P Mapeta; Willem H Ouwehand; Stefan Gräf; Kate Downes; Hugh S Markus
Journal:  Neurology       Date:  2019-11-12       Impact factor: 9.910

9.  Plasma Globotriaosylsphingosine and α-Galactosidase A Activity as a Combined Screening Biomarker for Fabry Disease in a Large Japanese Cohort.

Authors:  Hiroki Maruyama; Atsumi Taguchi; Mariko Mikame; Atsushi Izawa; Naoki Morito; Kazufumi Izaki; Toshiyuki Seto; Akifumi Onishi; Hitoshi Sugiyama; Norio Sakai; Kenji Yamabe; Yukio Yokoyama; Satoshi Yamashita; Hiroshi Satoh; Shigeru Toyoda; Michihiro Hosojima; Yumi Ito; Ryushi Tazawa; Satoshi Ishii
Journal:  Curr Issues Mol Biol       Date:  2021-06-19       Impact factor: 2.976

  9 in total

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