Literature DB >> 32189245

Clinical course and pathological findings of two late-onset Fabry hemizygous patients including mulberry cell counts after enzyme replacement therapy.

Homare Shimohata1, Marina Yamashita2, Kentaro Ohgi2, Hiroshi Maruyama2, Mamiko Takayasu2, Kouichi Hirayama2, Masaki Kobayashi2.   

Abstract

Fabry disease is an X-linked inherited lysosomal storage disorder caused by a deficiency of α-galactosidase A activity, resulting in the intracellular accumulation of globotriaosylceramide and related glycosphingolipids. The phenotypes of Fabry disease in both males and females are grouped into two categories: the classical type and the late-onset type. The classical type shows general symptoms including angiokeratoma(s), acroparesthesia, hypohidrosis, corneal opacity, and gastrointestinal symptoms from an early age. The late-onset type shows cardiac or renal (or both) symptoms from a late age. We present herein the clinical course and pathological findings of two late-onset hemizygous Fabry patients after the initiation of enzyme replacement therapy (ERT), along with their mulberry cell counts during treatment. One patient's case was a renal-variant type without general symptoms; he showed stable renal function and mild proteinuria but little histological improvement with no change in the mulberry cell count during ERT. The other patient had a cardiac-variant type with renal pathological abnormality. He achieved a mild improvement of renal pathological findings, and his mulberry cell count gradually decreased during the treatment. These findings indicate that monitoring the mulberry cell count might help assess the efficacy of ERT, as a renal pathology tool.

Entities:  

Keywords:  Fabry nephropathy; Late-onset type; Mulberry cell; Podocyte; Renal biopsy

Year:  2020        PMID: 32189245      PMCID: PMC7320132          DOI: 10.1007/s13730-020-00463-z

Source DB:  PubMed          Journal:  CEN Case Rep        ISSN: 2192-4449


  16 in total

1.  Fabry's disease discovered with chance urinary mulberry cells: a case report.

Authors:  Takashi Nakamichi; Mariko Miyazaki; Keisuke Nakayama; Mitsuhiro Sato; Naoki Akiu; Toshinobu Sato; Hiroshi Sato; Sadayoshi Ito
Journal:  CEN Case Rep       Date:  2012-10-31

2.  Long-Term Dose-Dependent Agalsidase Effects on Kidney Histology in Fabry Disease.

Authors:  Rannveig Skrunes; Camilla Tøndel; Sabine Leh; Kristin Kampevold Larsen; Gunnar Houge; Einar Skulstad Davidsen; Carla Hollak; André B P van Kuilenburg; Frédéric M Vaz; Einar Svarstad
Journal:  Clin J Am Soc Nephrol       Date:  2017-06-16       Impact factor: 8.237

3.  Agalsidase benefits renal histology in young patients with Fabry disease.

Authors:  Camilla Tøndel; Leif Bostad; Kristin Kampevold Larsen; Asle Hirth; Bjørn Egil Vikse; Gunnar Houge; Einar Svarstad
Journal:  J Am Soc Nephrol       Date:  2013-01       Impact factor: 10.121

4.  Urinary mulberry cells and mulberry bodies are useful tool to detect late-onset Fabry disease.

Authors:  Homare Shimohata; Hiroshi Maruyama; Yasunori Miyamoto; Mamiko Takayasu; Kouichi Hirayama; Masaki Kobayashi
Journal:  CEN Case Rep       Date:  2017-06-07

5.  Plasma lyso-Gb3: a biomarker for monitoring fabry patients during enzyme replacement therapy.

Authors:  Hitoshi Sakuraba; Tadayasu Togawa; Takahiro Tsukimura; Hiroshi Kato
Journal:  Clin Exp Nephrol       Date:  2017-12-29       Impact factor: 2.801

6.  Scoring system for renal pathology in Fabry disease: report of the International Study Group of Fabry Nephropathy (ISGFN).

Authors:  Agnes B Fogo; Leif Bostad; Einar Svarstad; William J Cook; Solange Moll; Federic Barbey; Laurette Geldenhuys; Michael West; Dusan Ferluga; Bojan Vujkovac; Alexander J Howie; Aine Burns; Roy Reeve; Stephen Waldek; Laure-Hélène Noël; Jean-Pierre Grünfeld; Carmen Valbuena; João Paulo Oliveira; Justus Müller; Frank Breunig; Xiao Zhang; David G Warnock
Journal:  Nephrol Dial Transplant       Date:  2009-10-15       Impact factor: 5.992

7.  Fabry's disease: alpha-galactosidase deficiency.

Authors:  J A Kint
Journal:  Science       Date:  1970-02-27       Impact factor: 47.728

8.  A Renal Variant of Fabry Disease Diagnosed by the Presence of Urinary Mulberry Cells.

Authors:  Homare Shimohata; Yujiro Ogawa; Hiroshi Maruyama; Kouichi Hirayama; Masaki Kobayashi
Journal:  Intern Med       Date:  2016-12-01       Impact factor: 1.271

9.  A Cardiac Variant of Fabry Disease Diagnosed with Chance Urinary Mulberry Cells.

Authors:  Rina Onishi; Koshiro Kanaoka; Junichi Sugiura; Motoko Tokunaga; Yasuhiro Takemoto; Kenji Onoue; Yuta Yamamoto; Manabu Horii; Yoshihiko Saito
Journal:  Intern Med       Date:  2018-07-06       Impact factor: 1.271

10.  Antiproteinuric therapy and Fabry nephropathy: factors associated with preserved kidney function during agalsidase-beta therapy.

Authors:  David G Warnock; Christie P Thomas; Bojan Vujkovac; Ruth C Campbell; Joel Charrow; Dawn A Laney; Leslie L Jackson; William R Wilcox; Christoph Wanner
Journal:  J Med Genet       Date:  2015-10-21       Impact factor: 6.318

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