Distribution of mono-, di- and trisialo gangliosides in the brain of Actinopterygian fishes

Viljetić, Barbara; Labak, Irena; Majić, Senka; Štambuk, Anamaria; Heffer-Lauc, Marija

doi:10.1016/j.bbagen.2011.12.010

Cited by 12 publications

(8 citation statements)

References 25 publications

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“…This mirrors previous findings that ganglioside GM1 accumulates as secondary storage material in NPC (Lloyd-Evans et al 2008 ) and confirms ganglioside GM1 storage as a phenotype in the npc1 morpholino model. We, and others, have confirmed the presence of gangliosides in zebrafish larvae by HPTLC (Supplemental Figure 2c) (Saslowsky et al 2010 ; Viljetić et al 2012 ; Chisada et al 2009 ). The presence of substantial cholesterol and ganglioside GM1 storage (Fig.…”

Section: Resultssupporting

confidence: 63%

“…Cholesterol and glycosphingolipids, gangliosides GM1 and GM2, form the primary storage material in the LSDs Niemann–Pick type C (NPC) and GM1/GM2 gangliosidosis, respectively, but have also been found as secondary storage material in many other LSDs (Lloyd-Evans and Platt 2010 ; Walkley and Vanier 2009 ). Considering the widespread presence of cholesterol and gangliosides as secondary storage in LSDs, it is fortunate that well-characterised probes to image these lipids exist (Viljetić et al 2012 ; Chisada et al 2009 ). These include filipin, a blue fluorescent polyene antibiotic that selectively binds cholesterol (Schroeder et al 1971 ; Kruth and Vaughan 1980 ), and cholera toxin subunit B (CtxB), produced by Vibrio cholerae bacteria and that selectively binds ganglioside GM1, which is present in zebrafish, as is ganglioside GM2 (Holmgren et al 1973 ; Saslowsky et al 2010 ; Boutry et al 2018 ).…”

Section: Introductionmentioning

confidence: 99%

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Visualisation of cholesterol and ganglioside GM1 in zebrafish models of Niemann–Pick type C disease and Smith–Lemli–Opitz syndrome using light sheet microscopy

Cook

Bladen

Smith

et al. 2020

Histochem Cell Biol

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Lysosomal storage diseases are the most common cause of neurodegeneration in children. They are characterised at the cellular level by the accumulation of storage material within lysosomes. There are very limited therapeutic options, and the search for novel therapies has been hampered as few good small animal models are available. Here, we describe the use of light sheet microscopy to assess lipid storage in drug and morpholino induced zebrafish models of two diseases of cholesterol homeostasis with lysosomal dysfunction: First, Niemann–Pick type C disease (NPC), caused by mutations in the lysosomal transmembrane protein NPC1, characterised by intralysosomal accumulation of cholesterol and several other lipids. Second, Smith–Lemli–Opitz syndrome (SLOS), caused by mutations in 7-dehydrocholesterol reductase, which catalyses the last step of cholesterol biosynthesis and is characterised by intralysosomal accumulation of dietary cholesterol. This is the first description of a zebrafish SLOS model. We find that zebrafish accurately model lysosomal storage and disease-specific phenotypes in both diseases. Increased cholesterol and ganglioside GM1 were observed in sections taken from NPC model fish, and decreased cholesterol in SLOS model fish, but these are of limited value as resolution is poor, and accurate anatomical comparisons difficult. Using light sheet microscopy, we were able to observe lipid changes in much greater detail and identified an unexpected accumulation of ganglioside GM1 in SLOS model fish. Our data demonstrate, for the first time in zebrafish, the immense potential that light sheet microscopy has in aiding the resolution of studies involving lysosomal and lipid disorders.

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Section: Resultssupporting

confidence: 63%

Section: Introductionmentioning

confidence: 99%

Visualisation of cholesterol and ganglioside GM1 in zebrafish models of Niemann–Pick type C disease and Smith–Lemli–Opitz syndrome using light sheet microscopy

Cook

Bladen

Smith

et al. 2020

Histochem Cell Biol

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“…82–86 In adult zebrafish brain, c-series gangliosides comprise up to 70% of the total gangliosides. 87 Accordingly, damaged zebrafish brains have high regeneration capacity, and the brain is rich in A2B5 + cells. 88 In proliferating NSCs of mammals, GT3-synthase (GT3S or ST-III), an enzyme for the synthesis for c-series gangliosides (A2B5 antigens), is highly expressed.…”

Section: A2b5 In Progenitor Cellsmentioning

confidence: 99%

Gangliosides in Nerve Cell Specification

Itokazu

Wang

2018

Progress in Molecular Biology and Translational Science

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The central nervous system is generated from progenitor cells that are recognized as neural stem cells (NSCs). NSCs are defined as undifferentiated neural cells that are characterized by the capacity for self-renewal and multipotency. Throughout neural development, NSCs undergo proliferation, migration, and cellular differentiation, and dynamic changes are observed in the composition of carbohydrate-rich molecules, including gangliosides. Gangliosides are sialic acid-containing glycosphingolipids with essential and multifaceted functions in brain development and NSC maintenance, which reflects the complexity of brain development. Our group has pioneered research on the importance of gangliosides for growth factor receptor signaling and epigenetic regulation of ganglioside biosynthesis in NSCs. We found that GD3 is the predominant ganglioside species in NSCs (>80%) and modulates NSC proliferation by interacting with epidermal growth factor receptor signaling. In postnatal brain, GD3 is required for long-term maintenance of NSCs. Deficiency in GD3 leads to developmental and behavioral deficits, such as depression. The synthesis of GD3 is switched to the synthesis of complex, brain-type gangliosides, namely, GM1, GD1a, GD1b, and GT1b, resulting in terminal differentiation and loss of “stemness” of NSCs. In this process, GM1 is augmented by a novel GM1-modulated epigenetic gene regulation mechanism of glycosyltransferases at a later differentiation stage. Consequently, our research suggests that stage-specific gangliosides play specific roles in maintaining NSC activities and in cell fate determination.

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“…The same neurons have been shown to express NgR [31] . A study by Viljetic et al showed that neurons in the granule cell layer of the zebrafish cerebellum express the gangliosides GD1a and GT1b, while Purkinje neurons do Katarina Vajn, et al Axonal regeneration after SCI in zebrafish and mammals 405 not [32] . it is also worth mentioning that Schwann cells, which weakly or do not express Nogo-A and MAG, proliferate and remyelinate zebrafish CNS axons after SCI [4] .…”

Section: Magmentioning

confidence: 99%

Axonal regeneration after spinal cord injury in zebrafish and mammals: differences, similarities, translation

et al. 2013

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Spinal cord injury (SCI) in mammals results in functional deficits that are mostly permanent due in part to the inability of severed axons to regenerate. Several types of growth-inhibitory molecules expressed at the injury site contribute to this regeneration failure. The responses of axons to these inhibitors vary greatly within and between organisms, reflecting axons' characteristic intrinsic propensity for regeneration. In the zebrafish (Danio rerio) many but not all axons exhibit successful regeneration after SCI. This review presents and compares the intrinsic and extrinsic determinants of axonal regeneration in the injured spinal cord in mammals and zebrafish. A better understanding of the molecules and molecular pathways underlying the remarkable individualism among neurons in mature zebrafish may support the development of therapies for SCI and their translation to the clinic.

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Distribution of mono-, di- and trisialo gangliosides in the brain of Actinopterygian fishes

Cited by 12 publications

References 25 publications

Visualisation of cholesterol and ganglioside GM1 in zebrafish models of Niemann–Pick type C disease and Smith–Lemli–Opitz syndrome using light sheet microscopy

Visualisation of cholesterol and ganglioside GM1 in zebrafish models of Niemann–Pick type C disease and Smith–Lemli–Opitz syndrome using light sheet microscopy

Gangliosides in Nerve Cell Specification

Axonal regeneration after spinal cord injury in zebrafish and mammals: differences, similarities, translation

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