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J Nephropathol. 2015;4(3): 91-96.
doi: 10.12860/jnp.2015.17
PMID: 26312237
PMCID: PMC4544560
Scopus ID: 84936942286
  Abstract View: 3669
  PDF Download: 1356

Case Series

Clinical and biochemical investigation of male patients exhibiting membranous cytoplasmic bodies in biopsied kidney tissues; a pitfall in diagnosis of Fabry disease

Hitoshi Sakuraba 1*, Takahiro Tsukimura 2, Toshie Tanaka 1, Tadayasu Togawa 2, Naoki Takahashi 3, Daisuke Mikami 3, Sachiko Wakai 4, Yasuhiro Akai 5

1 Department of Clinical Genetics, Meiji Pharmaceutical University, Kiyose, Tokyo, Japan
2 Departmentof Functional Bioanalysis, Meiji Pharmaceutical University, Kiyose, Tokyo, Japan
3 Division of Nephrology, Department of General Medicine, Faculty of Medical Sciences, University of Fukui, Eiheiji, Fukui, Japan
4 Division of Nephrology, Department of Internal Medicine, Ohkubo Hospital, Tokyo Metropolitan Health and Medical Treatment Corporation, Shinjuku, Tokyo, Japan
5 The 1st Department of Internal Medicine, Nara Medical University, Kashihara, Nara, Japan
*Corresponding Author: *Corresponding author: Prof. Hitoshi Sakuraba, Department of Clinical Genetics, Meiji Pharmaceutical University, Noshio, Kiyose, Tokyo, Japan. , Email: sakuraba@my-pharm.ac.jp

Abstract

Background: The existence of membranous cytoplasmic bodies in biopsied kidney tissues is one of the important findings when considering Fabry disease as the first choice diagnosis. However, there are possible acquired lysosomal diseases associated with pharmacological toxicity, although less attention has been paid to them.

Case Presentation: We experienced 3 male patients presenting with proteinuria and specific pathological changes strongly suggesting Fabry disease. We sought detailed clinical and biochemical information to avoid a wrong diagnosis. The patients were examined clinically and pathologically, and plasma α-galactosidase A (GLA) activity and the globotriaosylsphingosine (lyso-Gb3) concentrations were measured. Electron microscopic examination revealed numerous membranous inclusion bodies in podocytes, and biochemical analysis revealed normal GLA activity and a normal lyso-Gb3 level in plasma, showing that they did not have Fabry disease. They suffered from hyperlipidemia, myeloma, or lupus nephritis. They had received pitavastatin calcium, clarithromycin, loxoprofen and/or prednisolone, and there was no medication history of cationic amphiphilic drugs.

Conclusions: In this case series, the etiology of the inclusions was not clarified. However, these cases indicate that careful attention should be paid on diagnosis of patients exhibiting inclusion bodies in kidney cells, and it is important to confirm their past and present illnesses, and medication history as well as to measure the GLA activity and lyso-Gb3 level.


Implication for health policy/practice/research/medical education:

Three male patients are described in this case series, presenting with proteinuria and membranous cytoplasmic bodies (MCBs) in podocytes in kidney biopsies. MCBs in kidney cells are known to be the most important pathological finding for Fabry disease. But the results of assaying of α-galactosidase A (GLA) activity and measurement of the globotriaosylsphingosine concentration in plasma revealed that our patients did not have Fabry disease. Although the etiology of the MCBs has not been clarified yet, we should pay attention on diagnosis in cases exhibiting MCBs in kidney cells, and biochemical confirmation is necessary to avoid a pitfall in diagnosis of Fabry disease.

Please cite this paper as: Sakuraba H, Tsukimura T, Tanaka T, Togawa T, Takahashi N, Mikami D, et al. Clinical and biochemical investigation of male patients exhibiting membranous cytoplasmic bodies in biopsied kidney tissues; a pitfall in diagnosis of Fabry disease. J Nephropathol. 2015;4(3):91-96. DOI: 10.12860/jnp.2015.17

 

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Revision: 28 Jan 2015
ePublished: 01 Jul 2015
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