Developmental control of inositol phosphate biosynthesis is altered in the brain of both curly and phenotypically normal straight tail mutant mice

Alebous, Hana Dawood; Cartee, Robert T.; Vaccari, David A.; Wright, O'Neil; Ahmed, A; Hood, Ronald D.; Johnson, Margaret Dean

doi:10.1002/bdra.20608

Cited by 4 publications

(4 citation statements)

References 35 publications

(32 reference statements)

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“…They observed significant behavioral differences, with the CT strain being hyperactive, hyperreactive, and memory deficient. These results correlate well with our finding that there is a significantly higher level of inositol, overall, in the ct-CT cerebellum when compared to levels of inositol in the cerebellum of CBA and st-CT mice [18]. Given the number of published reports documenting the crucial roles of inositol in the CNS and findings that suggest inositol supplementation reduces neural tube defects (NTDs) [17], it was vital to ask questions concerning the spatial control of de novo inositol phosphate biosynthesis in the developing mammalian brain [18].…”

Section: Introductionsupporting

confidence: 92%

“…These results correlate well with our finding that there is a significantly higher level of inositol, overall, in the ct-CT cerebellum when compared to levels of inositol in the cerebellum of CBA and st-CT mice [18]. Given the number of published reports documenting the crucial roles of inositol in the CNS and findings that suggest inositol supplementation reduces neural tube defects (NTDs) [17], it was vital to ask questions concerning the spatial control of de novo inositol phosphate biosynthesis in the developing mammalian brain [18]. Microscopic studies presented here extend biochemical results, which revealed that there is a significantly lower level of inositol, overall, in the st-CT cerebellum when compared to levels of inositol in the cerebellum of CBA and ct-CT mice [18].…”

Section: Introductionsupporting

confidence: 92%

“…Given the number of published reports documenting the crucial roles of inositol in the CNS and findings that suggest inositol supplementation reduces neural tube defects (NTDs) [17], it was vital to ask questions concerning the spatial control of de novo inositol phosphate biosynthesis in the developing mammalian brain [18]. Microscopic studies presented here extend biochemical results, which revealed that there is a significantly lower level of inositol, overall, in the st-CT cerebellum when compared to levels of inositol in the cerebellum of CBA and ct-CT mice [18]. No significiant difference in levels of inositol was detected in the cerebrum of CBA, st-CT, and st-CT.…”

Section: Introductionmentioning

confidence: 99%

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Expression of 1L-myo-Inositol -1-Phosphate Synthase (EC 5.5.1.4) is Deregulated in the Cerebellum of Curly Tail Mutant Mice

Alebous

Steele

Johnson

2020

JAMMR

Self Cite

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Previous research, defining spatial control of inositol phosphate biosynthesis in the developing brain of CBA (normal) and CT [curly tail (ct-CT) and straight tail (st-CT)] mutant mice implicated a role for 1l-myo-inositol 1-phosphate synthase (MIP) in normal functioning of the central nervous system. Biochemical research indicated that MIP enzymatic activity, conversion of glucose 6-phosphate into inositol phosphate, is highest in the cerebellum of ct-CT and lowest in st-CT, when compared to that of CBA mice. Here, we utilized microscopic and biochemical investigations to analyze and extend previous findings of MIP expression in the cerebellum. Results of this research indicated that MIP expression correlates, well, with its enzymatic activity in the cerebellum of CBA and CT mutant mice. Statistical analyses of fluorescent micrographs detected a significant difference in fluorescence intensity between MIP from ct-CT, st-CT, and CBA mice. These data support vital links between inositol phosphate biosynthesis, MIP expression, and normal functioning of the cerebellum. Moreover, published data, identifying significant behavioral differences in the CT mutant, as well as data linking motor and non-motor cerebellar functions to abnormal levels of inositol, support the conclusion that aspects of normal cerebellar functions require temporal and spatial control of inositol phosphate biosynthesis, MIP expression.

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Section: Introductionsupporting

confidence: 92%

Section: Introductionsupporting

confidence: 92%

Section: Introductionmentioning

confidence: 99%

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Expression of 1L-myo-Inositol -1-Phosphate Synthase (EC 5.5.1.4) is Deregulated in the Cerebellum of Curly Tail Mutant Mice

Alebous

Steele

Johnson

2020

JAMMR

Self Cite

View full text Add to dashboard Cite

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“…Inositol supplementation is known to reduce sterility, enhance early embryonic development, and reduce the incidence of neural tube defects in mammals (Ting et al 2003; Alebous et al 2009; Colazingari et al 2014). Together this suggests that MIPS may play a role in reproduction and development.…”

Section: Resultsmentioning

confidence: 99%

Disruption ofINOS, a Gene Encodingmyo-Inositol Phosphate Synthase, Causes Male Sterility inDrosophila melanogaster

Jackson

Flores

Eldon

et al. 2018

G3 Genes|Genomes|Genetics

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Inositol is a precursor for the phospholipid membrane component phosphatidylinositol (PI), involved in signal transduction pathways, endoplasmic reticulum stress, and osmoregulation. Alterations of inositol metabolism have been implicated in human reproductive issues, the therapeutic effects of drugs used to treat epilepsy and bipolar disorder, spinal cord defects, and diseases including diabetes and Alzheimer’s. The sole known inositol synthetic enzyme is myo-inositol synthase (MIPS), and the homolog in Drosophilia melanogaster is encoded by the Inos gene. Three identical deletion strains (inosΔDF/CyO) were constructed, confirmed by PCR and sequencing, and homozygotes (inosΔDF/inosΔDF) were shown to lack the transcript encoding the MIPS enzyme. Without inositol, homozygous inosΔDF deletion fertilized eggs develop only to the first-instar larval stage. When transferred as pupae to food without inositol, however, inosΔDF homozygotes die significantly sooner than wild-type flies. Even with dietary inositol the homozygous inosΔDF males are sterile. An inos allele, with a P-element inserted into the first intron, fails to complement this male sterile phenotype. An additional copy of the Inos gene inserted into another chromosome rescues all the phenotypes. These genetic and phenotypic analyses establish D. melanogaster as an excellent model organism in which to examine the role of inositol synthesis in development and reproduction.

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Comprehensive hippocampal metabolite responses to PM2.5 in young mice

Xia

et al. 2018

Ecotoxicology and Environmental Safety

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Developmental control of inositol phosphate biosynthesis is altered in the brain of both curly and phenotypically normal straight tail mutant mice

Abstract: These findings implicate a role for MIP in the maturation of the CNS and evoke a hypothesis regarding the regulation of inositol phosphate biosynthesis in brain development.

Cited by 4 publications

References 35 publications

Expression of 1L-myo-Inositol -1-Phosphate Synthase (EC 5.5.1.4) is Deregulated in the Cerebellum of Curly Tail Mutant Mice

Expression of 1L-myo-Inositol -1-Phosphate Synthase (EC 5.5.1.4) is Deregulated in the Cerebellum of Curly Tail Mutant Mice

Disruption ofINOS, a Gene Encodingmyo-Inositol Phosphate Synthase, Causes Male Sterility inDrosophila melanogaster

Comprehensive hippocampal metabolite responses to PM2.5 in young mice

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