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Kidney Podocyte Zebra Bodies after Lung Transplantation for Lymphangioleiomyomatosis

A 55-year-old woman showed progressive renal dysfunction after unilateral deceased-donor lung transplantation for lymphangioleiomyomatosis. A kidney biopsy showed a striped pattern of interstitial fibrosis, suggesting calcineurin inhibitor toxicity, and zebra body accumulation predominantly in the p...

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Autores principales: Ishide, Takashi, Nishi, Hiroshi, Miyano, Shinako, Hirakawa, Yosuke, Honda, Kenjiro, Abe, Hiroyuki, Sato, Masaaki, Nangaku, Masaomi
Formato: Online Artículo Texto
Lenguaje:English
Publicado: The Japanese Society of Internal Medicine 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10372279/
https://www.ncbi.nlm.nih.gov/pubmed/36351586
http://dx.doi.org/10.2169/internalmedicine.0882-22
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author Ishide, Takashi
Nishi, Hiroshi
Miyano, Shinako
Hirakawa, Yosuke
Honda, Kenjiro
Abe, Hiroyuki
Sato, Masaaki
Nangaku, Masaomi
author_facet Ishide, Takashi
Nishi, Hiroshi
Miyano, Shinako
Hirakawa, Yosuke
Honda, Kenjiro
Abe, Hiroyuki
Sato, Masaaki
Nangaku, Masaomi
author_sort Ishide, Takashi
collection PubMed
description A 55-year-old woman showed progressive renal dysfunction after unilateral deceased-donor lung transplantation for lymphangioleiomyomatosis. A kidney biopsy showed a striped pattern of interstitial fibrosis, suggesting calcineurin inhibitor toxicity, and zebra body accumulation predominantly in the podocytes, characteristics of Fabry disease. Nevertheless, she had no extra-renal symptoms of the disease, and gene testing identified no known pathogenic variant or exon deletion. Our case report and literature review suggest that this atypical lysosomal inclusion may be phospholipidosis induced by sertraline. Potential underlying etiologies linking zebra body deposits may be not only hereditary but also drug-induced phospholipidosis.
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spelling pubmed-103722792023-07-28 Kidney Podocyte Zebra Bodies after Lung Transplantation for Lymphangioleiomyomatosis Ishide, Takashi Nishi, Hiroshi Miyano, Shinako Hirakawa, Yosuke Honda, Kenjiro Abe, Hiroyuki Sato, Masaaki Nangaku, Masaomi Intern Med Case Report A 55-year-old woman showed progressive renal dysfunction after unilateral deceased-donor lung transplantation for lymphangioleiomyomatosis. A kidney biopsy showed a striped pattern of interstitial fibrosis, suggesting calcineurin inhibitor toxicity, and zebra body accumulation predominantly in the podocytes, characteristics of Fabry disease. Nevertheless, she had no extra-renal symptoms of the disease, and gene testing identified no known pathogenic variant or exon deletion. Our case report and literature review suggest that this atypical lysosomal inclusion may be phospholipidosis induced by sertraline. Potential underlying etiologies linking zebra body deposits may be not only hereditary but also drug-induced phospholipidosis. The Japanese Society of Internal Medicine 2022-11-09 2023-07-01 /pmc/articles/PMC10372279/ /pubmed/36351586 http://dx.doi.org/10.2169/internalmedicine.0882-22 Text en Copyright © 2023 by The Japanese Society of Internal Medicine https://creativecommons.org/licenses/by-nc-nd/4.0/The Internal Medicine is an Open Access journal distributed under the Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License. To view the details of this license, please visit (https://creativecommons.org/licenses/by-nc-nd/4.0/).
spellingShingle Case Report
Ishide, Takashi
Nishi, Hiroshi
Miyano, Shinako
Hirakawa, Yosuke
Honda, Kenjiro
Abe, Hiroyuki
Sato, Masaaki
Nangaku, Masaomi
Kidney Podocyte Zebra Bodies after Lung Transplantation for Lymphangioleiomyomatosis
title Kidney Podocyte Zebra Bodies after Lung Transplantation for Lymphangioleiomyomatosis
title_full Kidney Podocyte Zebra Bodies after Lung Transplantation for Lymphangioleiomyomatosis
title_fullStr Kidney Podocyte Zebra Bodies after Lung Transplantation for Lymphangioleiomyomatosis
title_full_unstemmed Kidney Podocyte Zebra Bodies after Lung Transplantation for Lymphangioleiomyomatosis
title_short Kidney Podocyte Zebra Bodies after Lung Transplantation for Lymphangioleiomyomatosis
title_sort kidney podocyte zebra bodies after lung transplantation for lymphangioleiomyomatosis
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10372279/
https://www.ncbi.nlm.nih.gov/pubmed/36351586
http://dx.doi.org/10.2169/internalmedicine.0882-22
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