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Cell Transplantation Combined with Recombinant Collagen Peptides for the Treatment of Fabry Disease

Fabry disease is caused by a decrease in or loss of the activity of alpha-galactosidase, which causes its substrates globotriaosylceramide (Gb3) and globotriaosylsphingosine (lyso-Gb3) to accumulate in cells throughout the body. This accumulation results in progressive kidney injury due to glomerulo...

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Autores principales: Kami, Daisuke, Yamanami, Masashi, Tsukimura, Takahiro, Maeda, Hideki, Togawa, Tadayasu, Sakuraba, Hitoshi, Gojo, Satoshi
Formato: Online Artículo Texto
Lenguaje:English
Publicado: SAGE Publications 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7873760/
https://www.ncbi.nlm.nih.gov/pubmed/33300391
http://dx.doi.org/10.1177/0963689720976362
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author Kami, Daisuke
Yamanami, Masashi
Tsukimura, Takahiro
Maeda, Hideki
Togawa, Tadayasu
Sakuraba, Hitoshi
Gojo, Satoshi
author_facet Kami, Daisuke
Yamanami, Masashi
Tsukimura, Takahiro
Maeda, Hideki
Togawa, Tadayasu
Sakuraba, Hitoshi
Gojo, Satoshi
author_sort Kami, Daisuke
collection PubMed
description Fabry disease is caused by a decrease in or loss of the activity of alpha-galactosidase, which causes its substrates globotriaosylceramide (Gb3) and globotriaosylsphingosine (lyso-Gb3) to accumulate in cells throughout the body. This accumulation results in progressive kidney injury due to glomerulosclerosis and in heart failure due to hypertrophy. Enzyme replacement therapy (ERT) has been used as the standard therapy for Fabry disease, but it causes a significant financial burden, and regular administration is inconvenient for patients. Because of the short half-life of alpha-galactosidase in vivo, therapeutic methods that can supplement or replace ERT are expected to involve continuous release of alpha-galactosidase, even at low doses. Cell transplantation therapy is one of these methods; however, its use has been hindered by the short-term survival of transplanted cells. CellSaic technology, which utilizes cell spheroids that form after cells are seeded simultaneously with a recombinant collagen peptide scaffold called a μ-piece, has been used to improve cell survival upon implantation. In this study, syngeneic murine embryonic fibroblasts were used to generate CellSaic that were transplanted into Fabry mice. These spheroids survived for 28 days in the renal subcapsular space with forming blood vessels. These results indicate CellSaic technology could be a platform to promote cellular graft survival and may facilitate the development of cell transplantation methods for lysosomal diseases.
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spelling pubmed-78737602021-02-19 Cell Transplantation Combined with Recombinant Collagen Peptides for the Treatment of Fabry Disease Kami, Daisuke Yamanami, Masashi Tsukimura, Takahiro Maeda, Hideki Togawa, Tadayasu Sakuraba, Hitoshi Gojo, Satoshi Cell Transplant Original Article Fabry disease is caused by a decrease in or loss of the activity of alpha-galactosidase, which causes its substrates globotriaosylceramide (Gb3) and globotriaosylsphingosine (lyso-Gb3) to accumulate in cells throughout the body. This accumulation results in progressive kidney injury due to glomerulosclerosis and in heart failure due to hypertrophy. Enzyme replacement therapy (ERT) has been used as the standard therapy for Fabry disease, but it causes a significant financial burden, and regular administration is inconvenient for patients. Because of the short half-life of alpha-galactosidase in vivo, therapeutic methods that can supplement or replace ERT are expected to involve continuous release of alpha-galactosidase, even at low doses. Cell transplantation therapy is one of these methods; however, its use has been hindered by the short-term survival of transplanted cells. CellSaic technology, which utilizes cell spheroids that form after cells are seeded simultaneously with a recombinant collagen peptide scaffold called a μ-piece, has been used to improve cell survival upon implantation. In this study, syngeneic murine embryonic fibroblasts were used to generate CellSaic that were transplanted into Fabry mice. These spheroids survived for 28 days in the renal subcapsular space with forming blood vessels. These results indicate CellSaic technology could be a platform to promote cellular graft survival and may facilitate the development of cell transplantation methods for lysosomal diseases. SAGE Publications 2020-12-10 /pmc/articles/PMC7873760/ /pubmed/33300391 http://dx.doi.org/10.1177/0963689720976362 Text en © The Author(s) 2020 https://creativecommons.org/licenses/by-nc/4.0/ This article is distributed under the terms of the Creative Commons Attribution-NonCommercial 4.0 License (https://creativecommons.org/licenses/by-nc/4.0/) which permits non-commercial use, reproduction and distribution of the work without further permission provided the original work is attributed as specified on the SAGE and Open Access pages (https://us.sagepub.com/en-us/nam/open-access-at-sage).
spellingShingle Original Article
Kami, Daisuke
Yamanami, Masashi
Tsukimura, Takahiro
Maeda, Hideki
Togawa, Tadayasu
Sakuraba, Hitoshi
Gojo, Satoshi
Cell Transplantation Combined with Recombinant Collagen Peptides for the Treatment of Fabry Disease
title Cell Transplantation Combined with Recombinant Collagen Peptides for the Treatment of Fabry Disease
title_full Cell Transplantation Combined with Recombinant Collagen Peptides for the Treatment of Fabry Disease
title_fullStr Cell Transplantation Combined with Recombinant Collagen Peptides for the Treatment of Fabry Disease
title_full_unstemmed Cell Transplantation Combined with Recombinant Collagen Peptides for the Treatment of Fabry Disease
title_short Cell Transplantation Combined with Recombinant Collagen Peptides for the Treatment of Fabry Disease
title_sort cell transplantation combined with recombinant collagen peptides for the treatment of fabry disease
topic Original Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7873760/
https://www.ncbi.nlm.nih.gov/pubmed/33300391
http://dx.doi.org/10.1177/0963689720976362
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