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1. CHAPTER 1 INTRODUCTION

1.4 Research summary…

Previous studies emphasized the toxin effect of TDH mainly located in the

intestine of gastrointestinal tract.(15) However; the relationships between TDH and

liver had not been reported or analyzed. TDH might not only be absorbed by intestine

but also probably caused secondary injury to the liver via venous return of portal

system (Figure 4). In this study, we aim to analyze the hepatotoxicity of TDH and G.

hollisae (toxin and infection model) by in vitro (Figure 5) and in vitro (Figure 6)

analyses and provide an evidence to report the acute injury and recover stages of liver

in living animals by 18F-FDG PET (positron emission tomography)/CT (computer

tomography) scan.

Figure 4 TDH might not only be absorbed by intestine but also probably caused secondary injury to the liver via venous return of portal system.

Seafood G.

hollisae

TDH Intestine Portal

vein Liver

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Figure 5 The parameters in analyzing the in vitro hepatotoxicity of TDH from G.

hollisae. Mouse and human liver cell served as an in vitro model.

Figure 6 The parameters in analyzing the in vivo hepatotoxicity of TDH from G.

hollisae and G. hollisae. BALB/c served as an in vivo model.

Morphological

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Chapter 2 Materials and Methods

2.1 Bacterial strains and materials

The G. hollisae strain ATCC 33564 was obtained in a freeze-dried form from

the Culture Collection and Research Center (Hsin-Chu, Taiwan). Phenyl Sepharose 6

Fast Flow and protein molecular weight standards were purchased from GE

Healthcare (Piscataway, NJ). The protein assay kits were obtained from Bio-Rad

(Hercules, CA). Protein purification chemicals were obtained from Calbiochem (La

Jolla, CA).

2.2 Molecular cloning, protein expression and purification, and characterization of G. hollisae recombinant thermostable direct hemolysin (Gh-rTDH)

The G. hollisae tdh gene was obtained via PCR using G. hollisae genomic DNA

as the template and two primers, YKW-hol-TDH-N1

(5’-ATGAAATACAGACATCT-3’) and YKW-hol-TDH-C1

(5’-TTATTGTTGAGATTCAC-3’). PCR reaction was carried out under the following

conditions: denaturation at 94 °C for 5 min followed by 35 cycles of denaturation at

94 °C for 15 s, annealing at 58 °C for 1 min, and extension at 72 °C for 1 min

followed by a final extension at 72 °C for 10 min. The amplified DNA fragment was

cloned into pCR2.1-TOPO (Invitrogen, Carlsbad, CA) vector in order to construct the

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recombinant plasmid pCR2.1®-TOPO-Gh-tdh. The recombinant plasmid was

sequenced using an ABI PRISM® 3100 Genetic analyzer according to the

manufacturer’s protocol (Applied Biosystems, Foster City, CA).

The pCR2.1-TOPO-Gh-tdh plasmid harboring the tdh gene was transformed

into Escherichia coli BL21(DE3)(pLysS) cells (Promega, Madison, WI) for

recombinant protein production and purification. In parallel, the pCR2.1-TOPO

plasmid was used as a negative control. Colonies were inoculated into Luria-Bertani

broth supplemented 50 g/mL kanamycin and grown for 16 hours at 37 °C. The cells

were harvested by centrifugation at 6,000 x g for 30 min, and then resuspended in 40

mL of 20 mM Tris-HCl (pH 7) buffer. The mixture was sonicated, and the cell debris

was removed by centrifugation at 12,000 x g for 30 min at 4 °C. Purification method

was according to a previously described method.25 Overall, the supernatant containing

produced Gh-rTDH protein was loaded on a Phenyl-Sepharose 6 Fast Flow column

pre-equilibrated with 20 mM Tris-HCl (pH 7) and eluted with a linear 0 to 50%

ethylene glycol gradient. Fractions exhibiting hemolysis were pooled, dialyzed, and

added with NaCl to a 200 mM concentration. The active sample was applied to a

Phenyl-Sepharose 6 Fast Flow column with 20 mM Tris-HCl (pH 7) and then eluted

with 4 void volumes of a step gradient consisting of 200, 100, 50 mM NaCl and 20

mM Tris-HCl (pH 7) buffers. Finally, the protein (Gh-rTDH) was eluted by a 20 mM

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Tris-HCl buffer and the Gh-rTDH was dialyzed against 0.1M PBS buffer (Na2HPO4,

NaH2PO4, NaCl, pH 7.1) at 4°C overnight for cell and animal experiments. The

molecular cloning, protein expression, and purification of Gh-rTDH were performed

according to previous reports (17, 24). The effect of endotoxin has been excluded

before the experiment started. In this study, the endotoxin contamination had been

excluded during protein preparation by the method of anion-exchange

chromatography using diethylaminoethane (DEAE) chromatographic matrices.(25, 26)

Other prevention strategies including staff education (the use of aseptic technique,

understanding the nature of contamination, and good housekeeping), and sterility tests

were routinely performed before the study started.

2.3 Protein electrophoresis, detection and confirmed by MALDI-TOF/TOF mass spectrometry

For sodium dodecylacrylamide gel electrophoresis (SDS-PAGE), the protein

sample was mixed with 5 x sample treatment buffer (125 mM Tris-HCl, pH 6.8, 2%

SDS, 10% glycerol, 5%-merceptoethanol, and 0.05% bromophenol blue), and heated

at 100 °C for 10 min. Electrophoresis was performed according to the manufacturer’s

instructions. After electrophoresis, the gel was soaked in Coomassie Blue R 250

staining solution for 30 min, then the gel was destained with the destaining solution I

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(40% methanol, 7% acetic acid) and II (5% methanol, 7% acetic acid) until the stained

band was distinct against a clear background. The protein identities of SDS-PAGE

bands corresponding to Gh-rTDH were confirmed by MALDI-TOF/TOF mass

spectrometry.

2.4 Analyzed the in vitro hepatotoxicity of Gh-rTDH

2.41 Cytoviability and morphological examination of Gh-rTDH treated human liver

cell and FL83B cells

FL83B (BCRC 60325) and primary human non-cancer cell (kindly provided by

the Liver Transplantation Center of one medical center in central Taiwan; IRB number:

120305) were cultured for use in these studies. Following attachment, the cells were

treated with Gh-rTDH at a concentration of 1 μg/ml for 24 hours at 37 °C; the treating

dose was determined according to the initial results of the IC50 determination (1 μg/ml,

obtained from MTT assay). Images of the experimental group, cellular morphology

were recorded microscopically at 4 time points (before Gh-rTDH exposure and after

exposure to Gh-rTDH for 8, 16, and 24 hours). In addition, cells treated with PBS

(mixed with culture medium) were served as control group, they were also observed

at the same time points with the experimental group. All experimental or control

groups under the same conditions. Hygromycin was not used in this study.

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2.42 Cytoviability assay

Cytoviability of human liver cell and FL83B cells were measured by the MTT

assay using 4 treatment durations (12, 16, 24 and 48 hours). In the MTT assay, cells

were treated with PBS as control groups and treated with Gh-rTDH at different

concentrations (10to 10-8 μg/ml mixed with culture medium and administered in a total volume of 250 μl). For control group, the same concentration of vehicle was

added to the culture medium. After culture for different treating durations (12hours,

16hours, 24hours and 48hours), cells were incubated with MTT for another 4 hours at

37°C. Overall, the medium was removed and DMSO was added into each well. The

absorbance of the samples was measured at 570 nm using a microtiter plate reader. All

experiments were performed independently for five times

2.43 Confocal microscopy

Confocal microscopy was used to investigate the locations where Gh-rTDH

invaded in liver cells. Gh-rTDH was conjugated with fluorescein isothiocyanat (FITC)

(emission 488nm, green) as Gh-rTDH-FITC and the reactions were performed using

the FluoReporter FITC Protein Labeling Kit (molecular probes) according to the

manufacturer’s protocol. FL83B cells were seeded in 8-well chamber slide (1×104

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cells/well) and incubated in the culture medium to attach. After cells were attached,

they were treated with 10 μg/ml of Gh-rTDH-FITC mixed with culture medium for 20

and 40 min in darkness. Subsequently, the cells were washed 3 times using PBS

(SIGMA) buffer and they were also stained with propidium iodide (PI) (SIGMA)

(emission 650 nm, red) with working solution 5mg/ml in PBS for 5 min in darkness.

Finally, the cells were washed 3 times using PBS buffer and observed at 26 °C by

confocal microscopy (Olympus FV300).

2.44 TUNEL assay

TUNEL assay was performed for analyzing the reason of cell death. FL83B cells were respectively administrated with 1 μg/ml of Gh-rTDH for 24 hours and PBS

(control group) and the result of TUNEL assay according to the manufacturer’s

protocol (ApoAlert® DNA Fragmentation Assay Kit) and were observed by confocal

microscopy (Olympus FV300).

2.5 Analyzed the in vivo hepatotoxicity of Gh-rTDH 2.51 BALB/c served as an in vivo model

A total of 114 female mice aged 6 weeks were obtained from the National

Laboratory Animal Center of Taiwan and were used to analyze in vivo hepatotoxicity.

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All mice were fed normal diets. This study was carried out in strict accordance with

the recommendations in the Guide for the Care and Use of Laboratory Animals of the

National Institutes of Health. The protocol was approved by the Committee on the

Ethics of Animal Experiments of the National Chiao Tung University (Permit

Number: 01001008). All surgery was performed under sodium pentobarbital

anesthesia, and all efforts were made to minimize suffering.

2.52 Withdraw blood for analyzing the liver functions (n=25)

Twenty-five mice were divided into 5 groups (n = 5 for each group). One group

served as a control group and was administered PBS; the other 4 groups were

administered different dosages of Gh-rTDH (0.1, 1, 10, and 100 μg) as a single

treatment. The dosage that might initiate organ injury in animals has never been

reported (information on natural infection in humans is also lacking). Therefore, the

treatment dosages were carefully determined and modified according to the initial

results of the IC50 determination (1 μg/ml, obtained from the MTT assay described above). All mice were treated using the same volume (200 μl), the same treatment

time (10:00 am) and via gastric tubes without volume loss (i.e., vomiting). One

hundred microliters of whole blood was withdrawn from the orbital vascular plexus of

each mouse using a capillary tube and no analgesics. There were 8 experimental

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sampling times: before treatment with PBS or Gh-rTDH and 4, 8, 16, 32, 64, 128 and

256 hours after treatment with PBS or Gh-rTDH. The blood samples were analyzed

for the continuation of liver function as assessed by glutamic-oxaloacetic

transaminase (GOT), glutamic-pyruvic transaminase (GPT), total/direct/indirect

bilirubin, albumin and globulin)(Reagents Beckman Coulter®). One-way ANOVA

analysis was used to analyze the significant differences between each treatments/time

point. All analyses were performed with the SPSS statistical package for Windows

(Version 15.0, SPSS Inc., Chicago, IL).

2.53 Withdraw blood for analyzing the cardiotoxicity and nephrotoxicity (n=20)

Twenty mice were divided into 4 groups (each n=5). One of the 4 groups was

served as control group which were fed with PBS and the other 3 groups were

respectively fed with Gh-rTDH in dosages of 1 μg, 10 μg and 100 μg in single

administration via gastric tubes (each mouse was fed at AM10:00 with total volumes

of 200 μl). Each mouse was also respectively withdrawn 100 μl of whole blood at the

5 different time points: (1) before feeding with PBS or Gh-rTDH; (2) after feeding

with PBS or Gh-rTDH for 4, 16, 64, and 256 hours. Their blood samples were

analyzed for nephrotoxicity by detecting the level of creatinine (Assay kit: Creatinine

Reagent, Beckman Coulter®; Supply: Beckman Coulter Synchron CX7 Analyzer) and

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for cardiotoxicity by detecting the levels of CK-MB (Assay kit: CK-MB Reagent

Pack, Beckman Coulter®; Supply: Beckman Coulter Synchron CX7 Analyzer) and

Troponin I (Assay kit: ADVIA Centaur TnI-Ultra Readypack®; Supply: Bayer ADVIA

Centaur). Above all, blood samples were diluted appropriately for enough volume to

be detected by analyzers and were operated according to the manufacturer’s protocol.

One-way ANOVA analysis was also used to analyze the significant differences

between each treatments/time point.

2.54 Liver biopsy (n=9)

Nine mice were divided into 3 groups, which were treated with PBS, 10 μg of

Gh-rTDH or 100 μg of Gh-rTDH (n = 3 in each group) in a single administration via a

gastric tube. All mice had their livers biopsied after 8 hours of treatment. Samples

were prepared with H&E staining from tissue harvested at the time of animal

sacrifice.

2.55 PET/CT scan (n=60)

In this study, the 18F-FDG (2-fluoro-2-deoxy-D-glucose) PET /CT scan was

used to take images in detection the liver cells metabolism in living animals after

exposure of Gh-rTDH and their trends were recorded (GE Medical System, Discovery

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ST). 18F-FDG PET/CT imaging provides precise fusion of molecular PET images

with high-quality anatomical CT images. Technical parameters used for CT portion of

PET/CT are designed as follow: CT scan type with helical full of 0.5 second, a

detector row configuration of 16x1.25mm, an interval space of 2.5 mm, the slice

thickness of 1.2 mm, pitchof 1.75:1 (high quality mode), a speed of 17.5mm per

rotation, scan FOV of large, voltage of 120 kVp and current of 200 mA. Technical

parameters used for PET portion of 18F-FDG PET/CT are designed as follow 10 min

in each bed, the FOV chosen for imaging reconstruction is 20 cm and PET resolution

is 4.5 mm FWHM. The reconstructive parameters are type 3D iteration.

Sixty mice were divided into 4 major groups and each group (n=15) was respectively

fed with PBS, 1 μg, 10 μg and 100 μg of Gh-rTDH in single administration via gastric

tubes. Among each group, mice were further grouped to receive 18F-FDG PET/CT

scan in different time points including the 8th (n=5), 72th (n=5) and 168th (n=5) hours

after feeding with Gh-rTDH. In the study, 0.07mCi 18F-FDG for each mouse was

given by tail vein injection before taking the image (Figure 7A). After injection the

18F-FDG, images taking were performed one hour later with appropriate general

anesthesia (Isoflurane) (Figure 7B-D). In our study, each mouse did not be proposed

to receive 18F-FDG PET/CT scan in every time points to follow up because of

recurrent general anesthesia in short time might cause severe hepatotoxicity and could

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influence the results of this study. In the 18F-FDG PET/CT images, the 18F-FDG

uptake value was calculated using region of interest (ROI). In each mouse, the ROIs

of liver and muscle were recorded for semi-quantification which was proposed to be

the ratios of liver/muscle 18F-FDG uptake level.

Figure 7 18F-FDG and Isoflurane were treated to each mouse before scan started. (A)

18F-FDG for each mouse was given by tail vein injection before taking the images. (B)

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Isoflurane was used to perform general anesthesia. (C) Mice were respectively lay

down on the box and (D) received 18F-FDG PET/CT scan.

2.6 Infection models –In vivo hepatotoxicity of the G. hollisae strain, Escherichia coli containing the recombinant Gh-tdh gene (E. coli-TOPO-tdh), and the E.

coli-TOPO strain in BALB/c mice (n=126).

An animal infection model was set up to demonstrate the hepatotoxicity of

bacterial infection. The G. hollisae strain (wild type), E. coli-TOPO-tdh, and E.

coli-TOPO strains were cultured. Seventy-five mice were divided into three major

groups (n=25 for each group) and infected with bacteria via oral administration. Two

groups were infected with G. hollisae and E. coli-TOPO-tdh to demonstrate their

hepatotoxicity; the third group was infected with E. coli-TOPO to serve as a control

group. For each major group, five subgroups were established (n=5 for each group)

according to their treatment dosage (107, 108, 109, 1010 and 1011 organisms/ml and

treated with the same volumes. One hundred microliters of whole blood was

withdrawn at 8 different time points: before treatment with bacteria and 4, 8, 16, 32,

64, 128 and 256 hours after treatment with bacteria. Blood samples were analyzed for

continued liver function (GOT, GPT, total bilirubin, albumin and globulin). In

addition, 6 mice were treated with 1011 organisms/ml of G. hollisae, E. coli-TOPO-tdh,

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and E. coli-TOPO (n=2 for each group). For these animals, liver biopsies and H&E

staining (200X) were performed 8 hours after bacterial treatment. Finally, 54 mice

were treated with G. hollisae, E. coli-TOPO-tdh and E. coli-TOPO (n=18 for each

group) with a single administration. Among each group, mice were sub-grouped for

treatment with bacteria at the concentrations of 107, 109 and 1011 organisms/ml (n=6

for each group). In each concentration group, mice received a PET/CT scan at 8, 72

and 168 hours (n=2 for each group) after bacterial treatment.

2.7 Analyzed the in vivo and in vitro hepatotoxicity of fiber from Gh-rTDH (n=34)

In this study, fiber form of Gh-rTDH was prepared by heat treatment at 60 °C,

and the aggregates were collected by centrifugation. The method of preparing fiber

form of Gh-rTDH was according to a previously described method.(16) The

productions of fiber form of Gh-rTDH were confirmed by testing their hemolytic

ability. FL83B cells were treating with fiber form of Gh-rTDH and morphological

examination and cytoviability assay were performed (procedures and conditions were

uniform with previous description in the method section of 2.4). Moreover, mice

(n=25) were also fed with fiber form of Gh-rTDH and their liver functions including

the levels of GOT, GPT, total bilirubin, albumin and globulin were recorded. Liver

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biopsy and pathological images were taken in mice (n=8) for further analyzing the

hepatotoxicity of mice (procedures and conditions were uniform with previous

description in the method section of 2.5).

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Chapter 3 Results

3.1 Identification of the Gh-rTDH purified from G. hollisae

Electrophoresis of the homogeneous protein revealed a molecular mass of ~ 22

kDa as determined by the SDS-PAGE (Figure 8). Moreover, we found that tandem

mass spectrum of the doubly charged tryptic peptide at m/z 1024.543 from

SDS-PAGE of Gh-rTDH and a unique hit matching the 35VSDFWTNR42 of Gh-rTDH

peptide sequence was identified from the mass differences in the y-fragment ion series

of MALDI TOF/TOF spectrum (Figure 9).

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Figure 8 Purification and characterization of the Gh-rTDH protein. (A) Coomassie blue-stained SDS-PAGE of Gh-rTDH protein. Marker proteins (M): phosphorylase b

(97 kDa), albumin (66 kDa), ovalbumin (45 kDa), carbonic anhydrase (30 kDa),

trypsin inhibitor (20 kDa), α-lactoalbumin (14 kDa); lane 1: cell crude extract of

BL21(DE3) pLysS strain containing pCR2.1-TOPO plasmid alone; lane 2: crude

protein expressed from BL21(DE3) pLysS strain containing pCR2.1-TOPO-Gh-tdh

gene; lane 3 and 4: Phenyl Sepharose 6 Fast Flow purified protein showed a

homogenous protein with a molecular mass of ~ 22 kDa.

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Figure 9 Tandem mass spectrum of the doubly charged tryptic peptide at m/z 1024.543 from SDS-PAGE of Gh-rTDH. A unique hit matching the 35VSDFWTNR42

of Gh-rTDH peptide sequence was identified from the mass differences in the

y-fragment ion series of MALDI TOF/TOF spectrum.

3.2 Gh-rTDH caused in vitro liver cell damage

The morphology of liver cells was obviously changed after administrating with

1 μg/ml Gh-rTDH for 24 hours at 37 °C. The morphological changes included cell

detachment, and loss of cell cytoplasm with cell shrinkage (Figure 10A-D). The MTT

assay also revealed that the cytoviability of liver cells decreased in proportion to the

concentrations of Gh-rTDH in different treating durations. Moreover, we noted that

the Gh-rTDH damaged the liver cells in vitro when the concentration of Gh-rTDH

crossed 10-6 μg/ml (Figure 11). Moreover, in this study, primary human hepatocytes

(non-cancer liver cells) were used to demonstrate the toxicity of Gh-TDH via MTT

assay. These primary human hepatocytes were kindly provided by the liver

transplantation center of a medical center in central Taiwan under IRB permission

(IRB number: 120305). In this MTT assay, the Gh-TDH still caused obvious

hepatotoxicity in primary human hepatocytes (Figure 12).

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Figure 10 The morphology of liver cells (FL83B) was clearly changed after the administration of 1 μg/ml Gh-rTDH for 24 hours at 37 °C. The morphological

changes included cell detachment and a loss of cell cytoplasm with cell shrinkage;

they were the same cells recorded in different time points. (A) The liver cells before

exposure and (B) after exposure to the Gh-rTDH protein for 8 hours, (C) for 16 hours

and (D) for 24 hours.

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Figure 11 The MTT assay of mouse liver cells. The MTT assay revealed that the cytoviability of mouse liver cells decreased in proportion to the concentration of

Gh-rTDH over different treatment durations. Moreover, we noted that Gh-rTDH

damaged liver cells in vitro when the concentration of Gh-rTDH exceeded 10-6 μg/ml.

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Figure 12 The MTT assay of human liver cells. The MTT assay revealed that the cytoviability of primary human hepatocytes (non-cancer liver cells) decreased in

proportion to the concentration of Gh-rTDH over different treatment durations.

Moreover, we noted that Gh-rTDH damaged liver cells in vitro when the

concentration of Gh-rTDH exceeded 10-8μg/ml.

3.21 Gh-rTDH-FITC bound the margin of liver cells and invaded their nucleuses

Gh-rTDH-FITC was used to demonstrate the locations where the protein

invaded. The confocal microscopy with FITC filter revealed that Gh-rTDH-FITC

bound around the margin of liver cells after administrating with 10 μg/ml Gh-rTDH-FITC for 20 min at 26 °C (Figure 13 A-C). Moreover, Gh-rTDH-FITC

further located in the nucleus of liver cell after treating with Gh-rTDH-FITC for 40

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min at 26 °C and PI was also stained for confirming the location of nucleus (Figure 13

D-F).

Figure 13 Subcellular localization of Gh-rTDH. The liver cells respectively administrated with 10 μg/ml Gh-rTDH-FITC for 20 (A-C) and for 40 (D-F) min at 26

°C and were observed by confocal microscopy. (A) The liver cells were observed

without FICT filter (B) with FITC filter (C) merge A and C confirmed that

without FICT filter (B) with FITC filter (C) merge A and C confirmed that

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