Journal article icon

Journal article

Generation and characterization of two iPSC lines derived from subjects with free sialic acid storage disorder (FSASD)

Abstract:
Free sialic acid storage disorder (FSASD) is a rare, autosomal recessive, neurodegenerative disorder caused by biallelic mutations in SLC17A5, encoding the lysosomal transmembrane sialic acid exporter, SLC17A5. Defects in SLC17A5 lead to lysosomal accumulation of free sialic acid and other acid hexoses. The clinical spectrum of FSASD ranges from mild (Salla disease) to severe infantile forms. The pathobiology underlying FSASD remains elusive. In this study, two induced pluripotent stem cell (iPSC) lines were generated from a mild and an intermediate FSASD patient and characterized to provide much-needed additional models for basic and translational studies.
Publication status:
Published
Peer review status:
Peer reviewed

Actions


Access Document


Publisher copy:
10.1016/j.scr.2024.103600

Authors


More by this author
Institution:
University of Oxford
Division:
MSD
Department:
NDM
Sub department:
Target Discovery Institute
Oxford college:
Wolfson College
Role:
Author
ORCID:
0000-0001-6333-4049


More from this funder
Funder identifier:
https://ror.org/00baak391


Publisher:
Elsevier
Journal:
Stem Cell Research More from this journal
Volume:
81
Article number:
103600
Publication date:
2024-10-22
Acceptance date:
2024-10-20
DOI:
EISSN:
1876-7753
ISSN:
1873-5061
Pmid:
39461116


Language:
English
Keywords:
Pubs id:
2054309
Local pid:
pubs:2054309
Deposit date:
2024-12-02

Terms of use



Views and Downloads






If you are the owner of this record, you can report an update to it here: Report update to this record

TO TOP