TY - JOUR
T1 - Cell Transplantation Combined with Recombinant Collagen Peptides for the Treatment of Fabry Disease
AU - Kami, Daisuke
AU - Yamanami, Masashi
AU - Tsukimura, Takahiro
AU - Maeda, Hideki
AU - Togawa, Tadayasu
AU - Sakuraba, Hitoshi
AU - Gojo, Satoshi
N1 - Publisher Copyright:
© The Author(s) 2020.
PY - 2020
Y1 - 2020
N2 - 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.
AB - 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.
KW - CellSaic
KW - Fabry disease
KW - alpha-galactosidase (GLA in humans and Gla in mice)
KW - cell transplantation
KW - globotriaosylceramide (Gb3)
KW - globotriaosylsphingosine (lyso-Gb3)
KW - spheroid
UR - https://www.scopus.com/pages/publications/85097917043
U2 - 10.1177/0963689720976362
DO - 10.1177/0963689720976362
M3 - Article
C2 - 33300391
AN - SCOPUS:85097917043
SN - 0963-6897
VL - 29
JO - Cell Transplantation
JF - Cell Transplantation
ER -