Bis(monoacylglycero)phosphate, a peculiar phospholipid to control the fate of cholesterol: Implications in pathology

Hullin-Matsuda, Françoise; Luquain-Costaz, Céline; Bouvier, J B; Delton-Vandenbroucke, Isabelle

doi:10.1016/j.plefa.2009.09.006

Cited by 80 publications

(108 citation statements)

References 134 publications

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“…Because BMP is localized to endosomal/lysosomal membranes ( 57 ), a lysosomal expansion from stored gangliosides could simply increase the amount of a lipid localized in these compartments. However, BMP does not increase proportionally with lysosomal size, and it is not stored as a secondary material in all LSDs ( 56 ). Hence, lysosomal expansion might not be the only mechanism for BMP elevation ( 20,56 ).…”

Section: Discussionmentioning

confidence: 96%

“…However, BMP does not increase proportionally with lysosomal size, and it is not stored as a secondary material in all LSDs ( 56 ). Hence, lysosomal expansion might not be the only mechanism for BMP elevation ( 20,56 ).…”

Section: Discussionmentioning

confidence: 96%

“…We found that BMP was stored as a secondary material in the brains of GM1 and GM2 gangliosidoses. An explanation for the secondary storage of BMP in gangliosidosis brain has not been established ( 56 ). Because BMP is localized to endosomal/lysosomal membranes ( 57 ), a lysosomal expansion from stored gangliosides could simply increase the amount of a lipid localized in these compartments.…”

Section: Discussionmentioning

confidence: 99%

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Bis(monoacylglycero)phosphate: a secondary storage lipid in the gangliosidoses

Akgoc

Sena‐Esteves

Martin

et al. 2015

Journal of Lipid Research

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Bis(monoacylglycero)phosphate (BMP) is a negatively charged glycerophospholipid with an unusual sn-1;sn-1\u27 structural configuration. BMP is primarily enriched in endosomal/lysosomal membranes. BMP is thought to play a role in glycosphingolipid degradation and cholesterol transport. Elevated BMP levels have been found in many lysosomal storage diseases (LSDs), suggesting an association with lysosomal storage material. The gangliosidoses are a group of neurodegenerative LSDs involving the accumulation of either GM1 or GM2 gangliosides resulting from inherited deficiencies in beta-galactosidase or beta-hexosaminidase, respectively. Little information is available on BMP levels in gangliosidosis brain tissue. Our results showed that the content of BMP in brain was significantly greater in humans and in animals (mice, cats, American black bears) with either GM1 or GM2 ganglioside storage diseases, than in brains of normal subjects. The storage of BMP and ganglioside GM2 in brain were reduced similarly following adeno-associated viral-mediated gene therapy in Sandhoff disease mice. We also found that C22:6, C18:0, and C18:1 were the predominant BMP fatty acid species in gangliosidosis brains. The results show that BMP accumulates as a secondary storage material in the brain of a broad range of mammals with gangliosidoses

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Section: Discussionmentioning

confidence: 96%

Section: Discussionmentioning

confidence: 96%

Section: Discussionmentioning

confidence: 99%

See 1 more Smart Citation

Bis(monoacylglycero)phosphate: a secondary storage lipid in the gangliosidoses

Akgoc

Sena‐Esteves

Martin

et al. 2015

Journal of Lipid Research

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show abstract

“…Bouvier et al (38) found that the C 22:6 /C 22:6 species of BMP was highly sensitive to oxidative stress, showing a significant decrease in macrophages after exposure to oxidative stresses, although many studies demonstrated the dramatic increase of BMP levels in the tissue of animals and humans with the lysosomal disorder, such as Niemann-Pick C disease, certain druginduced lipidosis, and anti-phospholipid syndrome (39,40). We carried out LC/MS analysis to measure the BMP levels, as compared with our current quantitative BMP measurement.…”

Section: Journal Of Biological Chemistry 27439mentioning

confidence: 99%

Heat Shock Protein 70.1 (Hsp70.1) Affects Neuronal Cell Fate by Regulating Lysosomal Acid Sphingomyelinase

Zhu

Yoshimoto

Yamashima

2014

Journal of Biological Chemistry

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Background:The role of Hsp70.1 in neurodegeneration remains unknown. Results: We demonstrate differential profiles in activation of -calpain, bis(monoacylglycero)phosphate (BMP) levels, Hsp70.1-BMP interaction, and acid sphingomyelinase (ASM) activity between motor cortex and CA1 after ischemia/reperfusion. Conclusion: Hsp70.1-mediated ASM activity affects neuronal cell fate by regulating lysosomal membrane stability. Significance: This finding provides new insights into the mechanism of neuronal death.

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“…These changes in BMP composition are likely to alter the organization of the lysosomal membrane domains and interfere with lysosomal function. A role for BMP in lysosomal diseases is indicative, but the pathological significance is not known ( 14 ).…”

Section: Lipid Extractions and Mass Spectrometric Quantifi Cationmentioning

confidence: 99%

Selective reduction of bis(monoacylglycero)phosphate ameliorates the storage burden in a THP-1 macrophage model of Gaucher disease

Hein

Duplock

Fuller

2013

Journal of Lipid Research

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esterifi ed with fatty acids. The stereochemical confi guration of BMP differs as each glycerol moiety is esterifi ed through only the sn 1 position to the phosphate and contains a single fatty acid ( 1 ). Evidence suggests that BMP is synthesized from its structural isomer, phosphatidylglycerol ( 2 ). The fatty acid from the sn 2 position is removed with phospholipase A2 to produce lysophosphatidylglycerol, which then undergoes a transacylation reaction on the sn 2 position of the glycerol head group ( 3, 4 ). The subsequent steps, which must remove the fatty acid from the sn 1 position, rearrange the phosphoryl ester from the sn 3 to the sn 1 position and esterify the sn 2 position to produce the fi nal product, BMP, have not yet been adequately described. The synthesis of BMP is believed to occur in the lysosomal network, as BMP is highly enriched in the internal membranes of lysosomes ( 5 ).The negative charge of BMP, coupled with its proposed cone-shaped structure, enables it to participate in the organization of the membranes of the lysosomal network, contributing to its multivesicular/multilamellar morphology ( 6, 7 ). These properties of BMP also facilitate lysosomal lipid degradation, which takes place on the surface of the inner membranes of lysosomes. At the acidic pH of the lysosome, BMP contributes its negative charge to enhance adherence of the polycationic enzymes and activator proteins, aiding in lipid hydrolysis ( 8 ). Together, the high BMP and low cholesterol content of the internal lysosomal membrane ensures the selective degradation of lipids destined for lysosomal degradation without affecting the limiting membrane of the lysosome ( 9 ). Bis(monoacylglycero)phosphate (BMP) belongs to the group of glycerophospholipids that consist of a glycerol-3-phosphate backbone whose sn 1 and sn 2 positions are

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Bis(monoacylglycero)phosphate, a peculiar phospholipid to control the fate of cholesterol: Implications in pathology

Cited by 80 publications

References 134 publications

Bis(monoacylglycero)phosphate: a secondary storage lipid in the gangliosidoses

Bis(monoacylglycero)phosphate: a secondary storage lipid in the gangliosidoses

Heat Shock Protein 70.1 (Hsp70.1) Affects Neuronal Cell Fate by Regulating Lysosomal Acid Sphingomyelinase

Selective reduction of bis(monoacylglycero)phosphate ameliorates the storage burden in a THP-1 macrophage model of Gaucher disease

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