Reference Report

Reference
Citation	Wightman, P.J., Jackson, G.R., Dipple, K.M. (2012). Glycerol hypersensitivity in a Drosophila model for glycerol kinase deficiency is affected by mutations in eye pigmentation genes. PLoS ONE 7(3): e31779. (Export to RIS)
FlyBase ID	FBrf0217823
Publication Type	Research paper
PubMed ID	22427807
PubMed Abstract	Glycerol kinase plays a critical role in metabolism by converting glycerol to glycerol 3-phosphate in an ATP dependent reaction. In humans, glycerol kinase deficiency results in a wide range of phenotypic variability; patients can have severe metabolic and CNS abnormalities, while others possess hyperglycerolemia and glyceroluria with no other apparent phenotype. In an effort to help understand the pathogenic mechanisms underlying the phenotypic variation, we have created a Drosophila model for glycerol kinase deficiency by RNAi targeting of dGyk (CG18374) and dGK (CG7995). As expected, RNAi flies have reduced glycerol kinase RNA expression, reduced phosphorylation activity and elevated glycerol levels. Further investigation revealed these flies to be hypersensitive to fly food supplemented with glycerol. Due to the hygroscopic nature of glycerol, we predict glycerol hypersensitivity is a result of greater susceptibility to desiccation, suggesting glycerol kinase to play an important role in desiccation resistance in insects. To evaluate a role for genetic modifier loci in determining severity of the glycerol hypersensitivity observed in knockdown flies, we performed a preliminary screen of lethal transposon insertion mutant flies using a glycerol hypersensitive survivorship assay. We demonstrate that this type of screen can identify both enhancer and suppressor genetic loci of glycerol hypersensitivity. Furthermore, we found that the glycerol hypersensitivity phenotype can be enhanced or suppressed by null mutations in eye pigmentation genes. Taken together, our data suggest proteins encoded by eye pigmentation genes play an important role in desiccation resistance and that eye pigmentation genes are strong modifiers of the glycerol hypersensitive phenotype identified in our Drosophila model for glycerol kinase deficiency.
DOI	10.1371/journal.pone.0031779
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Language of Publication	English
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Publication Type	Journal
Abbreviation	PLoS ONE
Title	PLoS ONE
Publication Year	2006-
ISBN/ISSN	1932-6203
Data from Reference
Alleles (23)
	bw¹ bw¹⁶ bw¹⁹ g¹ Gk^c06596 Gk^{dsRNA.Scer\UAS} Gk^EY01412 Gk^f05001 Gk^Scer\UAS.Ex Gyk^DG05111 Gyk^{dsRNA.Scer\UAS} Gyk^e00237 Gyk^EY21867 Gyk^Scer\UAS.Ex ry¹ Scer\GAL4^c564 Scer\GAL4^elav-C155 Scer\GAL4^GMR.PS Scer\GAL4^how-24B Scer\GAL4^αTub84B.PL st¹ v¹ y¹
Constructs (6)
	P{GAL4-GMR.S} P{tubP-GAL4} P{UAS-Gk.Ex} P{UAS-Gk.IR} P{UAS-Gyk.Ex} P{UAS-Gyk.IR}
Genes (10)
	Atpα bw g Gk Gyk ry Scer\GAL4 st v y
Insertions (6)
	P{GawB}c564 P{GawB}elav^C155 P{GawB}how^24B PBac{PB}Gk^c06596 PBac{RB}Gyk^e00237 PBac{WH}Gk^f05001
Natural transposons (1)
	P-element