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. 2010;50(1):365-72.
doi: 10.1016/j.advenzreg.2009.10.017. Epub 2009 Nov 13.

Expression of inositol 1,3,4-trisphosphate 5/6-kinase (ITPK1) and its role in neural tube defects

Philip W Majerus  1 , David B Wilson, Chunfen Zhang, Peter J Nicholas, Monita P Wilson
Affiliations

Expression of inositol 1,3,4-trisphosphate 5/6-kinase (ITPK1) and its role in neural tube defects

Philip W Majerus et al. Adv Enzyme Regul. 2010.
No abstract available

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Figures

Fig. 1
Fig. 1
Activities of ITPK1. (A) Phosphorylation of Ins(1,3,4)P3 and isomerization of IP4 isomers and (B) reversible phosphorylation of Ins(3,4,5,6)P4.
Fig. 2
Fig. 2
The pathway for the synthesis of the higher phosphorylated forms of inositol in mammalian cells.
Fig. 3
Fig. 3
RT-PCR of Itpk1 (upper panel) and L19 (lower panel) from 2 animals each heterozygous for the gene trap allele (A,B), homozygous for the gene trap allele (C,D), or wild type (E,F). The ribosomal protein L19 is used as a loading control.
Fig. 4
Fig. 4
Expression of Itpk1 in mouse tissues. Tissue sections from mice containing the Itpk1-βgal gene trap allele were subjected to Xgal staining. (A) Whole mount of bisected kidney showing expression in vasculature within the renal cortex (c) but not medulla (me). (B,C) Expression in vascular smooth muscle of renal arterioles (a), including those adjacent to glomeruli (g). (D) Expression in an intercostal arteriole (a) between a rib (r) and skeletal muscle (sk). (E) Expression in smooth muscle cells of the vasa vasorum. (F) Expression in arterioles (a), serosal cells (s) and visceral smooth muscle (m) of the small intestine. (G,H) Expression in visceral smooth muscle (m) of the esophagus (e) and small intestine (si). (I) Weak expression in duodenal crypts. (J) Whole mount showing expression in vas deferens (vd) but not testis (t). (K) Expression in brain stem neurons (n). (L) Expression in pia mater (pm) of the brain.
Fig. 5
Fig. 5
NTDs in Itpk1 hypomorphic mouse embryos. (A) Exencephaly in an E12.5 Itpk1 hypomorph. The inset shows an H&E stained section through the open NTD (*). (B) Exencephaly in an E12.5 Itpk1 hypomorph. The inset shows a dorsal view of the open NTD (arrowhead). (C) Severe exencephaly (arrowheads) in an E11.5 Itpk1 hypomorph. (D) Splayed hindlimbs (hl) in conjunction with a caudal NTD in an E12.5 Itpk1 hypomorph. The inset shows a higher magnification of the boxed region. Note the open and kinked neural tube (arrowheads).
Fig. 6
Fig. 6
Measurement of myo-inositol. (A). Time course for inositol dehydrogenase reaction. (B). Concentration-dependence of inositol dehydrogenase.
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