fd0f00ad36872b8725540232b0ee3740 ijmolsci_a2020v21n24p9621.pdf 4ebb68c487f06fad2246a4764111c6664a13b994 ijmolsci_a2020v21n24p9621.pdf 9bec9ddd3a3d6b19cbbb69465ba73c176207b84fe926a5b0f946a3158a6d79c4 ijmolsci_a2020v21n24p9621.pdf Title: Preclinical Research in Glycogen Storage Diseases: A Comprehensive Review of Current Animal Models Subject: GSD are a group of disorders characterized by a defect in gene expression of specific enzymes involved in glycogen breakdown or synthesis, commonly resulting in the accumulation of glycogen in various tissues (primarily the liver and skeletal muscle). Several different GSD animal models have been found to naturally present spontaneous mutations and others have been developed and characterized in order to further understand the physiopathology of these diseases and as a useful tool to evaluate potential therapeutic strategies. In the present work we have reviewed a total of 42 different animal models of GSD, including 26 genetically modified mouse models, 15 naturally occurring models (encompassing quails, cats, dogs, sheep, cattle and horses), and one genetically modified zebrafish model. To our knowledge, this is the most complete list of GSD animal models ever reviewed. Importantly, when all these animal models are analyzed together, we can observe some common traits, as well as model specific differences, that would be overlooked if each model was only studied in the context of a given GSD. Keywords: glycogen storage diseases; animal models; therapy Author: Aitana Almodóvar-Payá, Mónica Villarreal-Salazar, Noemí de Luna, Gisela Nogales-Gadea, Alberto Real-Martínez, Antoni L. Andreu, Miguel Angel Martín, Joaquin Arenas, Alejandro Lucia, John Vissing, Thomas Krag and Tomàs Pinós Creator: LaTeX with hyperref Producer: pdfTeX-1.40.21 CreationDate: Mon Dec 21 02:37:50 2020 CET ModDate: Mon Dec 21 02:47:31 2020 CET Custom Metadata: no Metadata Stream: no Tagged: no UserProperties: no Suspects: no Form: none JavaScript: no Pages: 48 Encrypted: no Page size: 595.276 x 841.89 pts (A4) Page rot: 0 File size: 698614 bytes Optimized: no PDF version: 1.7 name type encoding emb sub uni object ID ------------------------------------ ----------------- ---------------- --- --- --- --------- FYMAKE+Pxsy Type 1 Builtin yes yes no 10 0 XEFGQJ+VnURWPalladioL Type 1 Custom yes yes no 14 0 RWBFJP+URWPalladioL-Ital Type 1 Custom yes yes no 19 0 OSNIXR+URWPalladioL-Bold Type 1 Custom yes yes no 24 0 JXVPXA+URWPalladioL-Roma Type 1 Custom yes yes no 28 0 QLMOVM+Rpxr Type 1 Builtin yes yes no 32 0 HSNKGA+VnURWPalladioL-Bold Type 1 Custom yes yes no 36 0 QTLELQ+Pxbsy Type 1 Builtin yes yes no 40 0 IRWHZX+TimesNewRomanPSMT Type 1C WinAnsi yes yes no 48 0 CKUPDE+TimesNewRomanPS-BoldItalicMT Type 1C WinAnsi yes yes no 51 0 MCBCCI+SourceSansRoman-Regular Type 1C Custom yes yes no 73 0 MCBCCM+SourceSansRoman.613wght Type 1C Custom yes yes no 77 0 MCBCCO+SourceSansRoman-Bold Type 1C Custom yes yes no 81 0 VVSYYB+EURM10 Type 1 Builtin yes yes no 91 0 WOZBHT+Rpxb Type 1 Builtin yes yes no 95 0 GANEOA+Rpxi Type 1 Builtin yes yes no 99 0 MPUDFL+PazoMath Type 1 Builtin yes yes no 106 0 BFKLTO+URWPalladioL-BoldItal Type 1 Custom yes yes no 122 0 BGEWMG+Rpxbi Type 1 Builtin yes yes no 126 0 VSHNAE+Rpcxr Type 1 Builtin yes yes no 161 0 PDXFNG+PazoMath-Bold Type 1 Builtin yes yes no 171 0 UZLZOY+Rpxmi Type 1 Builtin yes yes no 216 0 Jhove (Rel. 1.28.0, 2023-05-18) Date: 2024-10-02 04:23:26 CEST RepresentationInformation: ijmolsci_a2020v21n24p9621.pdf ReportingModule: PDF-hul, Rel. 1.12.4 (2023-03-16) LastModified: 2023-03-31 11:40:09 CEST Size: 698614 Format: PDF Version: 1.7 Status: Well-Formed and valid SignatureMatches: PDF-hul MIMEtype: application/pdf PDFMetadata: Objects: 1327 FreeObjects: 1 IncrementalUpdates: 0 DocumentCatalog: PageLayout: SinglePage PageMode: UseNone Outlines: Item: Title: Animal Models in Research Destination: section.1 Item: Title: Glycogen Storage Diseases Destination: section.2 Children: Item: Title: Introduction Destination: subsection.2.1 Item: Title: Animal Models Destination: subsection.2.2 Item: Title: Types of GSD Destination: subsection.2.3 Children: Item: Title: GSD Type 0 (GSD-0; Aglycogenosis) Destination: subsubsection.2.3.1 Item: Title: GSD Type I (GSD-I; von Gierke Disease) Destination: subsubsection.2.3.2 Item: Title: GSD Type II (GSD-II; Pompe Disease) Destination: subsubsection.2.3.3 Item: Title: GSD Type III (GSD-III; Cori Disease) Destination: subsubsection.2.3.4 Item: Title: GSD Type IV (GSD-IV; Andersen Disease) Destination: subsubsection.2.3.5 Item: Title: GSD Type V (GSD-V; McArdle Disease) Destination: subsubsection.2.3.6 Item: Title: GSD Type VI (GSD-VI; Hers Disease) Destination: subsubsection.2.3.7 Item: Title: GSD Type VII (GSD-VII; Tarui Disease) Destination: subsubsection.2.3.8 Item: Title: GSD Type XV (GSD-XV) Destination: subsubsection.2.3.9 Item: Title: Critical Discussion Destination: section.3 Item: Title: Conclusions Destination: section.4 Item: Title: References Destination: Item.3 Info: Title: Preclinical Research in Glycogen Storage Diseases: A Comprehensive Review of Current Animal Models Author: Aitana Almodóvar-Payá, Mónica Villarreal-Salazar, Noemí de Luna, Gisela Nogales-Gadea, Alberto Real-Martínez, Antoni L. Andreu, Miguel Angel Martín, Joaquin Arenas, Alejandro Lucia, John Vissing, Thomas Krag and Tomàs Pinós Subject: GSD are a group of disorders characterized by a defect in gene expression of specific enzymes involved in glycogen breakdown or synthesis, commonly resulting in the accumulation of glycogen in various tissues (primarily the liver and skeletal muscle). Several different GSD animal models have been found to naturally present spontaneous mutations and others have been developed and characterized in order to further understand the physiopathology of these diseases and as a useful tool to evaluate potential therapeutic strategies. In the present work we have reviewed a total of 42 different animal models of GSD, including 26 genetically modified mouse models, 15 naturally occurring models (encompassing quails, cats, dogs, sheep, cattle and horses), and one genetically modified zebrafish model. To our knowledge, this is the most complete list of GSD animal models ever reviewed. Importantly, when all these animal models are analyzed together, we can observe some common traits, as well as model specific differences, that would be overlooked if each model was only studied in the context of a given GSD. 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