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The first part of our results demonstrate that enterocytic apoptosis is in part

responsible for triggering intestinal barrier dysfunction upon I/R challenge, and glucose

uptake mediated by SGLT1 attenuated the loss of intestinal barrier function partly via

anti-apoptotic PI3K/Akt signaling (Figure. 11).

The histological findings suggest that mesenteric I/R triggers epithelial apoptosis

that accompanies villous disintegration and decreased crypt cell proliferation. Changes

in intestinal permeability in I/R rats were demonstrated by the transmural HRP flux rate

ex vivo and by the lumen-to-blood passage of enterally-administered FD4 and

gadodiamide in vivo. The increase of epithelial permeability paralleled the augmentation

of BT in I/R intestines, indicating gut barrier damage. These pathological changes were

attenuated by enteral instillation of ZVAD, providing direct evidence that epithelial

apoptosis is in part responsible for triggering intestinal barrier damage and bacterial

influx. This study is thus the first to demonstrate that modification of epithelial caspase

activity reduces intestinal permeability and BT in animals. We also showed that

pretreatment with ZVAD diminished the increase of MPO activity and the production of

TNFα and MIP-1α in ischemic intestines, suggesting that epithelial apoptosis and

barrier dysfunction preceded mucosal inflammation.

The intestinal permeability was evaluated using ex vivo and in vivo assays. A

twofold increase of the transmural HRP flux was observed in the intestinal tissues in I/R

rats compared with sham controls. However, the increased HRP flux was only evident at

30-60 and 60-90 minutes after the addition of the probe to Ussing chambers; the

delayed observation and the extracorporeal, oxygenated setting may produce artificial

results of the extent of I/R injury. In light of these technical limitations, we also

evaluated gut permeability in vivo by measuring the luminal-to-blood passage of

enterally administered FD4 or gadodiamide during the reperfusion period. The plasma

level of FD4 was four times higher in I/R rats than in sham controls, indicating

compromised intestinal barrier function after ischemic challenge. A novel MRI-based

assay developed previously from our laboratory was used to detect the real-time

permeability changes in vivo. This more sensitive assay showed a five- to tenfold

increase of SNR in liver and kidney in ischemic rats at the early phase (<15 minutes) of

reperfusion, supporting the hypothesis that it is an acute break of the epithelial barrier

that leads to augmented portal drainage of luminal probes. The kidney SNR peaked at

15 minutes post-reperfusion and declined afterwards, which was likely due to the high

concentration of gadodiamide in the renal medulla and calyces leading to a greater T2

shortening effect than T1 shortening effect. These results are in agreement with others

showing increased gut permeability upon I/R challenge (Sun et al., 1998; Khanna et al.,

2001). In comparison to the HRP flux assay and the fluorescence intensity test, this

MRI-based method is fast and sensitive, and allows us to rapidly detect changes in gut

permeability and provides a means for further studies of targeted pharmacological

intervention.

Physiological cytoprotective mechanisms contributing to resistance against

apoptosis include upregulation of glucose transporters (e.g. SGLT1, GLUT1, and

GLUT4) and maintenance of high intracellular glucose concentration (Sun et al., 1994;

Hall et al., 2001; Russo et al., 2004; Yu et al., 2005; Yu et al., 2006; Wofford et al., 2008;

Yu et al., 2008). The phenomenon of glucose protection has been documented in a

number of cell types, including neurons, leukocytes, myocardiocytes, and vascular

smooth muscle cells (Sun et al., 1994; Hall et al., 2001; Russo et al., 2004; Wofford et

al., 2008). Previous in vitro studies utilizing human intestinal epithelial Caco-2 cells

have also demonstrated that SGLT1 glucose uptake inhibits cell apoptosis and barrier

impairment caused by bacterial and parasitic products (Yu et al., 2005; Yu et al., 2006;

Yu et al., 2008). Based on our finding that epithelial apoptosis is partly responsible for

intestinal barrier dysfunction upon I/R challenge, we sought to investigate the use of

glucose supplementation to correct excessive cell death and to improve gut barrier

integrity. In I/R rats, glucose uptake mediated by the phloridzin-sensitive SGLT1

protected the intestinal epithelium from apoptosis and attenuated the increase in

permeability and BT, leading to diminished inflammatory responses. Taken together

with previous studies (Yu et al., 2005; Yu et al., 2006; Yu et al., 2008), our finding

suggests that SGLT1-mediated cytoprotection may operate under a number of metabolic

and microbial stress conditions.

The signal transduction pathways responsible for promoting cell survival were also

investigated. Membrane translocation and increased phosphorylation of Akt in villous

epithelial cells were paralleled by enhanced Akt kinase activity in the gut mucosa

following glucose uptake by SGLT1. This finding is consistent with previous studies

showing that Akt is a downstream step of a signaling pathway induced by Na+-glucose

cotransport (Zhao et al., 2004; Shiue et al., 2005), which triggered the activation of

Na+-H+ exchanger (NHE). Our pharmacological blockade studies with LY294002 and

wortmannin confirmed that PI3K/Akt activation is involved in the glucose-mediated

protection against epithelial apoptosis, mucosal pathology and gut barrier damage

induced by I/R. Previous reports documented that PI3K/Akt signaling inhibits cell

apoptosis by phosphorylating Bad, either directly or indirectly via mTOR (Chang et al.,

2003; LoPiccolo et al., 2008). A number of other downstream targets of Akt, such as

IκBα/NFκBα, FoxO1/3a and GSK3, are also involved in promoting cell survival and

cell cycle progression (Liang et al., 2003; Urbich et al., 2005; Dan et al., 2008; Bai et al.,

2009). We showed here that SGLT1 glucose uptake induced the phosphorylation of Akt

and downstream targets such as mTOR, Bad and FoxO1/3a in intestinal mucosa. These

results provide an explanation of the anti-apoptotic effects of enteral glucose, which

may contribute to cytoprotective mechanisms in intestinal I/R. It is noteworthy that

recent evidence points to an unanticipated role of Bad in linking pathways of glucose

metabolism and cell apoptosis (Danial et al., 2003; Danial, 2008). Danial et al.

demonstrated that Bad resides in a mitochondrial complex with glucokinase and

participates in mitochondrial respiration in response to glucose (Danial et al., 2003;

Danial, 2008). The phosphorylation status of Bad is associated with glucokinase activity;

glucose deprivation results in dephosphorylation of Bad and Bad-dependent cell death

(Danial et al., 2003; Danial, 2008). These findings highlight the role that mitochondrial

Bad plays in coordinating glucose metabolism and the apoptotic machinery. Whether

the phosphorylation status of mitochondrial Bad affects the mode of glucose

metabolism in ischemic intestines requires further investigation.

Recent reports indicated that the IκBα/NFκB pathway is involved in anti-apoptotic

events in intestinal epithelial cells against various pathogenic stimuli (Potoka et al.,

2000; Egan et al., 2004; Nenci et al., 2007). However, our results demonstrated that

instillation of enteric glucose decreased the levels of phosphorylated IκBα in ischemic

guts. Despite links between Akt and IκBα/NFκB pathways in the mechanism of cell

survival (Chang et al., 2003; Dan et al., 2008; Bai et al., 2009), the IκBα/NFκB pathway

is not involved in the glucose-mediated anti-apoptotic signaling in intestinal epithelium.

The upregulation of phospho-IκBα may be partly responsible for proinflammatory

cytokine production after ischemic challenge. Overall, we demonstrated that SGLT1

glucose uptake in intestinal epithelial cells activated anti-apoptotic pathways that

involved PI3K/Akt signaling and increased phosphorylation of mTOR, Bad and

FoxO1/3a.

Accumulating data supports the notion that SGLT1 orchestrates a number of

fundamental cellular processes besides its canonical absorptive function. Previous

reports showed that activation of SGLT1 induced the recruitment of GLUT2 to the

brush border membrane for diffusive glucose transport via a PKCβ-dependent pathway

(Kellett et al., 2000; Kellett, 2001). Others documented that cotransport of Na+ with

glucose triggered the activation of Akt and phosphorylation of cytoskeleton-associated

ezrin, leading to recruitment of NHE to apical membrane that facilitated absorption of

sodium and hydrogen in intestines (Zhao et al., 2004; Shiue et al., 2005). A recent study

by Palazzo et al. demonstrated that activation of SGLT1 suppressed bacterial

LPS-induced NFκB signaling in intestinal epithelial cells, and suggested that SGLT1

plays a novel immunomodulatory role (Palazzo et al., 2008). Moreover, oral ingestion

but not intraperitoneal administration of glucose attenuated proinflammatory cytokine

production and protected endotoxemic mice from lethal septic shock (Palazzo et al.,

2008). Clinically, oral rehydration therapy that targets SGLT1 to drive passive diffusion

of water is a widely-used supportive therapy for diarrheal patients. Early enteral

nutrition (EN) is advocated for patients with multiple pathologies for its known benefit

in lowering the risk of septic complications compared to parenteral supplementation.

One area that has gained much attention in nutrition therapy nowadays is preoperative

oral carbohydrate loading (CHO) (Fearon et al., 2005; Martindale et al., 2006). This

novel concept involves giving patients an isotonic carbohydrate solution at midnight the

day before surgery and 2-3 hours preoperatively, in contrast to the general routine of

overnight fasting before surgery. Patients given CHO showed reduced postoperative

insulin resistance and shorter hospital stay after colorectal resection (Soop et al., 2004).

The advantage of early EN and CHO may also involve SGLT1-mediated nutritive and

non-nutritive functions.

The second part of our study demonstrates a novel mechanism through which

glycolytic pyruvate confers resistance to RIP-dependent necroptosis in hypoxic

colorectal carcinoma via mitochondrial superoxide scavenging. Despite long-standing

observation of cell necrosis in the hypoxic core of colorectal tumors, there is an

apparent lack of knowledge on its molecular mechanisms. In our study, we

demonstrated that oxygen and glucose deprivation induced RIP-1/3 signaling and

morphological necrotic features, i.e. rupture of plasma membrane, 8 to 24 hours after

the onset of challenge in colorectal carcinoma cells. Recent studies from other

laboratories have also showed necroptosis in intestinal epithelial cells in models of

chronic intestinal inflammation in gene-deficient mice (Gunther et al., 2011; Welz et al.,

2011). Although there is no data on the timing of necrotic death caused by hypoxic

stress in normal human epithelial cells, rapid villous necrosis in jejunum and colon was

found in rats after 60 minutes of hemorrhagic shock or 40 minutes of mesenteric

ischemia (Chang et al., 2005; Higa et al., 2007), showing that normal cells are more

sensitive to oxygen and nutrient deprivation compared to cancer cells. It is noteworthy

that normoxic controls in glucose-free media displayed no sign of necroptosis,

indicating that glucose deprivation alone did not trigger necrotic death.

Mitochondrial dysfunctions and free radical generation have also been implicated

in the necrotic process caused by cytotoxic agents.(Goossens et al., 1995; Temkin et al.,

2006; Zhang et al., 2009). The transient mitochondrial hyperpolarization observed in

our study may reflect a temporal reversal of electron transport chain ATP synthase

activity due to a decline in intracellular oxygen, leading to adverse proton pumping

against the electrochemical gradient to intermembranous spaces. Moreover, the final

mitochondrial potential collapse correlated well with organelle swelling at later time

points (16-24 hrs) of hypoxia. Interestingly, we identified high levels of mitochondrial

superoxide production prior to plasma membrane disintegration in hypoxic colonic

carcinoma cells. This seemingly paradoxical situation of ROS emission in hypoxia has

also been previously documented in cardiomyocytes after infarction (Becker et al., 1999;

Camara et al., 2007). The generation of mitochondrial ROS has been suggested to be

caused by electron leak to oxygen in the respiratory chain complexes whereby low

levels of oxygen remain inside cells at the early stage of hypoxic challenge (Turrens,

2003). By using antioxidants, we demonstrated that mitochondria-derived superoxide

plays a critical role in hypoxia-induced necrotic mechanisms and is upstream of RIP

signals.

We further showed that GLUT-dependent glucose uptake and glycolytic

metabolism inhibits hypoxia-induced RIP signaling and necrotic features in colorectal

cancer cells. Several studies have implicated that glycolytically generated ATP may

restore energy supply in cancer cells and act as a key factor for glucose-mediated death

resistance against hypoxia and genotoxic agents (Xu et al., 2005; Zhou et al., 2012).

Recent data in activated macrophages and B lymphocytes have demonstrated that

glycolytic ATP promotes cell survival via maintenance of transmembrane potential of

defective mitochondria that are unable to generate ATP (Dufort et al., 2007; Garedew et

al., 2010). Here, we focused on the role of glycolytic pyruvate, and examined if

cell-permeable derivative may lead to anti-necrotic effects. We demonstrated that

hypoxia-induced necroptosis (i.e. LDH leakage, RIP activation, and morphological

damage) was reverted by pyruvate, however, the resistance was uncoupled with

restoration of cellular energy and mitochondrial potential. Another line of evidence

showed that glucose addition reduced the mitochondrial ROS levels but did not alter the

redox enzyme activity. The aforementioned data indicate that a non-enzymatic free

radical scavenging mechanism, such as those reported with pyruvate, may contribute to

death resistance. Indeed, our results showed that supplementation of pyruvate

significantly reduced the mitochondrial superoxide levels in hypoxic cells in an

ATP-independent manner. It is noteworthy that glucose-mediated recovery of

mitochondrial transmembrane potential in hypoxic cells is not dependent on pyruvate

but may rely on ATP. Taken together, glycolytic metabolism may promote hypoxic cell

survival by a two-fold mechanism including scavenging of mitochondrial superoxide by

pyruvate and maintenance of mitochondrial potential by ATP.

We confirmed that presence of glucose during hypoxic challenge did not ablate

oxygen deprivation stress as evidenced by the nuclear translocation of HIF1α. The

HIF1-targeted upregulation of GLUTs led to enhanced glucose uptake and glycolytic

metabolism, serving as a positive cycle to promote cell survival. Normal human small

intestinal epithelial cells express SGLT1 on apical membrane and GLUT2 on basolateral

membrane for luminal-to-serosal flux of dietary glucose absorption (Yu et al., 2005; Yu

et al., 2008). In contrast to normal colonocytes, human colorectal carcinoma specimens

abnormally display SGLT1 and GLUT isoforms 1-4 on the apical membrane or

cytoplasm of tumor cells (Godoy et al., 2006; Guo et al., 2011; de Wit et al., 2012).

Previous studies have indicated that transcriptional regulation of GLUT1 is mediated by

HIF1 binding to a nucleotide sequence corresponding to a hypoxia-responsive element

in promoter regions of GLUT1 gene, following oxygen deprivation (Ouiddir et al., 1999;

Hayashi et al., 2004).

Other than glucose transporters (GLUT1 and GLUT3), a large number of genes

encoding for glycolytic enzymes, angiogenic growth factors, cell survival and

proliferative signals, and prolyl hydroxylases (PHD) are also upregulated by HIF1

(Denko, 2008; Chiacchiera et al., 2009; Marin-Hernandez et al., 2009). In our study, a

transient drop of GLUTs expression was observed after 16 hrs but recovered after 24 hrs

of hypoxia. The temporary reduction of GLUTs expression may be due to the regulatory

loop of HIF1α/PHD. Under normoxic conditions, PHD-mediated hydroxylation of

proline residues on HIF1α permits its ubiquitination followed by proteosomal

degradation. Hypoxia sensing downregulates PHD activity and thus, stabilizes HIF1α

levels, leading to its nuclear translocation and downstream transcription of target genes

(Jaakkola et al., 2001; Kondo et al., 2006). Paradoxically, PHDs are themselves target

genes of HIF1 transcription, indicating a negative feedback mechanism (Marxsen et al.,

2004; Stiehl et al., 2006). In addition, HIF1α degrades within 8-12 hrs without de novo

synthesis (Millonig et al., 2009). Therefore, we speculate that the fluctuation of GLUTs

expression after the onset of hypoxia may reflect the HIF1α/PHD loop. Experiments to

address the temporal relationship, cellular distribution, and pathophysiological

significance of various isoforms of glucose transporters by HIF1 in colonic carcinoma

are currently under progress.

Angiogenic pathways and mediators have been extensively studied as potential

anti-cancer drugs for several decades. In some cases, patients fail to respond to

antiangiogenic agents or simply develop drug resistance. Tumors may display invasive

metastatic transition in response to the increasing hypoxic microenvironment by cancer

treatment (Zeng et al., 2010; Rapisarda et al., 2012). Here, our data show a critical role

of glycolytic pyruvate in death resistance and underscore site-specific glucose

transporters and metabolic pathways as potential candidates for the development of

novel cancer killing strategies. The resistance to antiangiogesis or hypoxic stress may be

overcome by combinational strategies with glucose- or pyruvate-targeted therapy. In

summary, the second part of the studies demonstrated glycolytic metabolism inhibits

hypoxia-induced RIP-dependentnecroptosis in colonic cancer cells by suppression of

mitochondrial ROS (Figure 24).

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