Spastic paraplegia-linked phospholipase PAPLA1 is necessary for development, reproduction, and energy metabolism in Drosophila

Gáliková, Martina; Klepsatel, Peter; Münch, Judith; Kühnlein, Ronald P.

doi:10.1038/srep46516

Cited by 12 publications

(10 citation statements)

References 55 publications

(124 reference statements)

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“…Drosophila PAPLA1 is an ortholog of Manduca TGL and has been shown to possess TAG hydrolase activity in vitro (Arrese et al 2006). PAPLA1 mutant animals show a complex spectrum of phenotypes including impaired fecundity and fertility, decreased photoreceptor sensitivity, and metabolic abnormalities (Kunduri et al 2014;Gáliková et al 2017). However, TAG storage and mobilization are comparable in PAPLA1 mutants and wild-type animals (Gáliková et al 2017), which makes the hypothesis of in vivo relevance of PAPLA1 in Drosophila TAG mobilization controversial.…”

Section: Regulation Of Lipolysismentioning

confidence: 99%

Triacylglycerol Metabolism in Drosophila melanogaster

2018

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Triacylglycerol (TAG) is the most important caloric source with respect to energy homeostasis in animals. In addition to its evolutionarily conserved importance as an energy source, TAG turnover is crucial to the metabolism of structural and signaling lipids. These neutral lipids are also key players in development and disease. Here, we review the metabolism of TAG in the Drosophila model system. Recently, the fruit fly has attracted renewed attention in research due to the unique experimental approaches it affords in studying the tissue-autonomous and interorgan regulation of lipid metabolism in vivo. Following an overview of the systemic control of fly body fat stores, we will cover lipid anabolic, enzymatic, and regulatory processes, which begin with the dietary lipid breakdown and de novo lipogenesis that results in lipid droplet storage. Next, we focus on lipolytic processes, which mobilize storage TAG to make it metabolically accessible as either an energy source or as a building block for biosynthesis of other lipid classes. Since the buildup and breakdown of fat involves various organs, we highlight avenues of lipid transport, which are at the heart of functional integration of organismic lipid metabolism. Finally, we draw attention to some "missing links" in basic neutral lipid metabolism and conclude with a perspective on how fly research can be exploited to study functional metabolic roles of diverse lipids.

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Section: Regulation Of Lipolysismentioning

confidence: 99%

Triacylglycerol Metabolism in Drosophila melanogaster

2018

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“…Fat and glycogen content were determined using standard protocols based on the colourimetric assays [68]. Both types of energy reserves were measured from the same starting homogenates, prepared as described in detail in [69]. Lipids were quantified using the Triglycerides kit (Randox, TR1697).…”

Section: Fat and Glycogen Quantificationmentioning

confidence: 99%

The influence of developmental diet on reproduction and metabolism in Drosophila

et al. 2020

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Background: The adaptive significance of phenotypic changes elicited by environmental conditions experienced early in life has long attracted attention in evolutionary biology. In this study, we used Drosophila melanogaster to test whether the developmental diet produces phenotypes better adapted to cope with similar nutritional conditions later in life. To discriminate among competing hypotheses on the underlying nature of developmental plasticity, we employed a full factorial design with several developmental and adult diets. Specifically, we examined the effects of early-and late-life diets (by varying their yeast and sugar contents) on reproductive fitness and on the amount of energy reserves (fat and glycogen) in two wild-caught populations. Results: We found that individuals that had developed on either low-yeast or high-sugar diet showed decreased reproductive performance regardless of their adult nutritional environment. The lower reproductive fitness might be caused by smaller body size and reduced ovariole number. Overall, these results are consistent with the silver spoon concept, which posits that development in a suboptimal environment negatively affects fitness-associated traits. On the other hand, the higher amount of energy reserves (fat) in individuals that had developed in a suboptimal environment might represent either an adaptive response or a side-effect of compensatory feeding. Conclusion: Our findings suggest that the observed differences in the adult physiology induced by early-life diet likely result from inevitable and general effects of nutrition on the development of reproductive and metabolic organs, rather than from adaptive mechanisms.

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“…Additional diseases with neuromuscular involvement that are also associated with glycogen cacostasis include NPC1, HSP, ALS and Zellweger spectrum disorder (ZSD). For instance, reductions in GAA expression and glycogen storage manifest as secondary metabolic abnormalities in NPC1 (Rauniyar et al, 2015 ), and phospholipase PAPLA1, associated with HSP, regulates the expression of several regulators of carbohydrate metabolism and glycogen storage (Galiková et al, 2017 ). Glycogen levels are elevated in the spinal cords of sALS patients, and interestingly, although acidic GAA activity is not altered, neutral GAA activity is up regulated, thus suggesting that glycogen storage occurs outside of the lysosome during the course of the disease.…”

Section: Pompe Disease Alpha-glucosidase and Glycogenmentioning

confidence: 99%

Lipid Involvement in Neurodegenerative Diseases of the Motor System: Insights from Lysosomal Storage Diseases

Dodge

2017

Front. Mol. Neurosci.

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Lysosomal storage diseases (LSDs) are a heterogeneous group of rare inherited metabolic diseases that are frequently triggered by the accumulation of lipids inside organelles of the endosomal-autophagic-lysosomal system (EALS). There is now a growing realization that disrupted lysosomal homeostasis (i.e., lysosomal cacostasis) also contributes to more common neurodegenerative disorders such as Parkinson disease (PD). Lipid deposition within the EALS may also participate in the pathogenesis of some additional neurodegenerative diseases of the motor system. Here, I will highlight the lipid abnormalities and clinical manifestations that are common to LSDs and several diseases of the motor system, including amyotrophic lateral sclerosis (ALS), atypical forms of spinal muscular atrophy, Charcot–Marie–Tooth disease (CMT), hereditary spastic paraplegia (HSP), multiple system atrophy (MSA), PD and spinocerebellar ataxia (SCA). Elucidating the underlying basis of intracellular lipid mislocalization as well as its consequences in each of these disorders will likely provide innovative targets for therapeutic research.

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Spastic paraplegia-linked phospholipase PAPLA1 is necessary for development, reproduction, and energy metabolism in Drosophila

Cited by 12 publications

References 55 publications

Triacylglycerol Metabolism in Drosophila melanogaster

Triacylglycerol Metabolism in Drosophila melanogaster

The influence of developmental diet on reproduction and metabolism in Drosophila

Lipid Involvement in Neurodegenerative Diseases of the Motor System: Insights from Lysosomal Storage Diseases

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