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In contrast, apolipoprotein E-deficient mice fed western-type diet for the same period were resistant to diet-induced obesity, had normal plasma glucose, leptin and insulin levels, and ex

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Apolipoprotein E predisposes to obesity and related

metabolic dysfunctions in mice

Iordanes Karagiannides1,2, Rami Abdou1, Aikaterini Tzortzopoulou1, Peter J Voshol3

and Kyriakos E Kypreos1,4

1 Whitaker Cardiovascular Institute, Boston University School of Medicine, MA, USA

2 Beth Israel Deaconess Medical Center, Gastrointestinal Neuropeptide Center, Division of Gastroenterology, Harvard Medical School, Boston, MA, USA

3 Department of Endocrinology, Leiden University Medical Center, The Netherlands

4 Department of Medicine, Pharmacology Unit, University of Patras Medical School, Rio, Greece

Obesity and its related pathologies constitute a major

cause of death, with rates increasing at an alarming

pace [1] By the beginning of the millennium,

over-weight adults accounted for over 15% of the world’s

population (body mass index > 30, World Health

Organization) [2], with this number increasing to 50% within the USA and Europe [3] Obesity develops as a result of disruption of the homeostasis between food intake and energy expenditure, and therefore factors affecting these processes are the focus of extensive

Keywords

ApoE3knock-inmice; apolipoprotein E;

glucose intolerance; insulin resistance;

obesity

Correspondence

K E Kypreos, Department of Medicine,

Pharmacology Unit, University of Patras

Medical School, Panepistimioupolis, Rio,

TK 26500, Greece

Fax: +30 2610996103

Tel: +30 2610969120

E-mail: kkypreos@med.upatras.gr

(Received 23 February 2008, revised 25 July

2008, accepted 30 July 2008)

doi:10.1111/j.1742-4658.2008.06619.x

Obesity is a central feature of the metabolic syndrome and is associated with increased risk for insulin resistance and type II diabetes Here, we investigated the contribution of human apoliprotein E3 and mouse apoli-protein E to the development of diet-induced obesity in response to western-type diet Our data show that apolipoprotein E contributes to the development of obesity and other related metabolic disorders, and that human apolipoprotein E3 is more potent than mouse apolipoprotein E in promoting obesity in response to western-type diet Specifically, we found that apolipoprotein E3 knock-in mice fed western-type diet for 24 weeks became obese and developed hyperglycemia, hyperinsulinemia, hyperleptin-emia, glucose intolerance and insulin resistance that were more severe than

in C57BL/6 mice In contrast, apolipoprotein E-deficient mice fed western-type diet for the same period were resistant to diet-induced obesity, had normal plasma glucose, leptin and insulin levels, and exhibited normal responses to glucose tolerance and insulin resistance tests Furthermore, low-density lipoprotein receptor-deficient mice were more sensitive to the development of diet-induced obesity and insulin resistance than apolipo-protein E-deficient mice, but were still more resistant than C57BL/6 mice, raising the possibility that low-density lipoprotein receptor mediates, at least in part, the effects of apolipoprotein E on obesity Taken together, our findings suggest that, in addition to other previously identified mecha-nisms of obesity, apolipoprotein E and possibly the chylomicron pathway are also important contributors to the development of obesity and related metabolic dysfunctions in mice

Abbreviations

ApoE, apolipoprotein E; ApoE)/),ApoE-deficient; ApoE3knock-inmice, mice containing a targeted replacement of the mouse ApoE gene for the human ApoE3 gene; GTT, glucose tolerance test; IST, insulin sensitivity test; LDLr, low-density lipoprotein receptor; LDLr)/), LDLr-deficient; LpL, lipoprotein lipase; VLDL, very low-density lipoprotein.

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research for the development of effective antiobesity

drugs, with only limited success being achieved thus

far [4] Aging, hormonal imbalance and genetic

predis-position may also contribute to obesity [5–16]

How-ever, very few cases of human obesity are reported to

be caused by genetic factors [17], leaving western-type

diet and sedentary lifestyle, physical inactivity and

imbalance in caloric load as the most common

contrib-utors to the development of central obesity and the

metabolic syndrome [2,18]

The risk of developing all other components of the

metabolic syndrome increases with obesity, supporting

the hypothesis that obesity is the central feature of the

syndrome [19] It is well established that abdominal

obesity may result in insulin resistance and

hyperinsu-linemia [19,20] Epidemiological and population studies

have established a direct correlation between obesity

and the development of cardiovascular disease [21,22]

Despite the pivotal role of obesity and dyslipidemia in

the development of the metabolic syndrome and heart

disease, the functional interactions between adipose

tissue and the lipid and lipoprotein transport system

have not yet been investigated thoroughly

Apolipoprotein E (ApoE) is a 34.2 kDa glycoprotein

synthesized by the liver and other peripheral tissues In

humans, there are three major natural isoforms, ApoE2,

ApoE3, and ApoE4 [23], with ApoE3 being the most

common [23–29] ApoE is a major protein component

of chylomicron remnants and very low-density

lipopro-tein (VLDL) [23] The importance of this prolipopro-tein in the

maintenance of plasma lipid homeostasis and

athero-protection was first established with the generation of

the ApoE-deficient mouse [30,31], which develops

hypercholesterolemia and spontaneous atherosclerosis

[30,31] Lipid-bound ApoE is the natural ligand of the

low-density lipoprotein receptor (LDLr) [32–34], a cell

surface receptor that is responsible for the catabolism of

atherogenic lipoproteins [32,35–37]

In this study, we sought to determine the role of

ApoE in the development of diet-induced obesity,

glu-cose intolerance and insulin resistance in vivo To

address this question, female ApoE3knock-in, wild-type

C57BL/6, LDLr-deficient (LDLr)/)) and

ApoE-defi-cient (ApoE)/)) mice were fed western-type diet for a

period of 15 or 24 weeks, during which time their

plasma lipid and glucose levels, body weight, body

com-position, glucose tolerance and insulin sensitivity were

monitored We chose to study ApoE3 because it is the

most common ApoE isoform in humans [24–29] Our

data establish that expression of ApoE predisposes mice

to diet-induced obesity, hyperglycemia and insulin

resis-tance, whereas deficiency in ApoE renders mice resistant

to these conditions Human ApoE3 appeared to be more

potent than mouse ApoE in promoting obesity in response to western-type diet Furthermore, LDLr)/) mice were more sensitive to the development of diet-induced obesity and insulin resistance than ApoE)/) mice, but still more resistant than wild-type C57BL/6 mice in response to western-type diet Gavage adminis-tration of olive oil containing the nonhydrolyzable [3H]cholesteryl-hexadecyl-ether to mice suggested that deficiency in LDLr and ApoE reduces the direct delivery

of postprandial nonhydrolyzed lipids to the liver, one of the major tissues involved in glucose uptake from the circulation A similar trend was also observed in the delivery of nonhydrolyzed dietary lipids to adipose tissue Taken together, our data establish that ApoE is

a key mediator of diet-induced obesity in response to western-type diet

Results

ApoE promotes diet-induced weight gain in mice, whereas ApoE deficiency prevents it

To test the effects of ApoE on weight gain in mice, groups of 4–6 weeks old female ApoE3knock-in, ApoE)/) and wild-type C57BL/6 mice were placed on western-type or normal chow diet for a total period of

24 weeks Mice in each group were weighed immedi-ately before the initiation of the experiment (week 0) and every 6 weeks thereafter up to week 24, using a Mettler precision microscale

It became apparent that as early as 6 weeks on high-fat diet, ApoE3-expressing mice gained weight and were significantly heavier than the wild-type C57BL/6 mice on the same diet (Fig 1A) The weight of the ApoE3knock–in mice was 31.37 ± 3.98 g (115 ± 34% higher than their initial weight of 17.03 ± 0.94 g,

P < 0.05) (Fig 1A) During the same period, C57BL/

6 mice on a high-fat diet had an average body weight

of 26.08 ± 1.89 g (66 ± 8% higher than their initial weight of 16.26 ± 0.61 g, P < 0.05) (Fig 1A) There were no statistical differences between the weights of the ApoE3knock-in and C57BL/6 control groups fed chow diet for 6 weeks (data not shown)

At week 24 on high-fat diet, ApoE3-expressing mice showed a dramatic increase in body weight, with an average value of 50.23 ± 2.22 g (235 ± 32% higher than their starting weight at week 0, P < 0.05) (Fig 1A) The body weight of the C57BL/6 mice was 43.10 ± 0.94 g (164 ± 9% higher than their starting weight at week 0, P < 0.05) (Fig 1A) The control ApoE3knock-inand C57BL/6 mouse groups on chow diet showed a much smaller increase in body weight, ranging between 22 and 24 g (29 ± 6% increase as compared to

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their starting weight at week 0, P < 0.05) (data not

shown) In contrast to the ApoE3knock-in and the

C57BL/6 mice, ApoE)/)mice that were fed western-type

diet showed only a modest increase in body weight

dur-ing the course of the experiment (Fig 1A) At week 6 of

the experiment, the ApoE)/)mouse group had an

aver-age body weight of 20.36 ± 1.37 g (32 ± 6% increase

as compared to their starting weight of 16.26 ± 0.61 g

at week 0, P < 0.05) At week 24, their body weight

was 24.58 ± 1.07 g (41 ± 4% increase as compared to

their starting weight at week 0, P < 0.05) (Fig 1A)

Similar increases in body weight were observed in the

control ApoE)/)mice fed chow diet (data not shown)

To compare the steady-state plasma ApoE levels

between ApoE3knock-in and C57BL/6 mice, at week 0

of the experiment plasma samples were isolated from

three mice from each group and 5 lL of plasma was analyzed by western blotting using a polyclonal antibody that recognizes both human and mouse ApoE (Santa-Cruz Biotech, Santa Cruz, CA, USA; cat no sc-31821) This analysis showed that C57BL/6 mice had approximately four-fold higher steady-state plasma ApoE levels than ApoE3knock-in mice, suggest-ing that the increased sensitivity of ApoE3knock-in mice

to obesity is not due to higher plasma ApoE levels in these mice compared to C57BL/6 mice (Fig 1B)

To determine whether body weight differences among groups fed western-type diet could be explained

by differences in their average daily food consumption,

at week 12 of the experiment we determined the average daily food consumption for each mouse group

It was found that ApoE3knock-in mice consumed

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% of initial body-weight

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Fig 1 Percentage of initial body weight (A), plasma ApoE levels (B), plasma cholesterol levels (C) and plasma triglyceride levels (D) of C57BL/6, ApoE3knock-inand ApoE)/)mice fed western-type diet for a period of 24 weeks (E) Percentage body fat content of ApoE3knock-in, ApoE)/)and C57BL/6 mice at week 24 (F) Fasting plasma glucose levels of ApoE3 knock-in , ApoE)/)and C57BL/6 mice at weeks 0 and 24 Each point on the graphs represents the mean value of the group, and error bars indicate the SEM The statistical significance of the observed differences among groups at each time-point is as indicated (*P < 0.05; **P < 0.005) In (B), plasma ApoE levels in ApoE3knock-in and ApoE)/)mice were determined by western blot analysis using an antibody that recognizes equally mouse ApoE and human ApoE Ponceau S staining of the nitrocellulose membrane was used to confirm equal loading and efficient transfer of proteins to the membrane.

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3.04 ± 0.13 g per mouse per day, C57BL/6 mice

con-sumed 3.31 ± 0.15 g per mouse per day, and ApoE)/)

mice consumed 3.19 ± 0.17 g per mouse per day, and

there was no statistical difference among groups

(P > 0.05)

Plasma lipid levels of the mice fed western-type

diet for 24 weeks

During the 24-week period of feeding mice with

western-type diet, fasting plasma samples were taken

every 6 weeks, and cholesterol, triglyceride and free

fatty acid levels were measured as described in

Experimental procedures As shown in Fig 1C, at

week 24 both ApoE3knock in and C57BL/6 mice on

high-fat diet had slightly elevated fasting cholesterol

levels (138 ± 10 mgÆdL)1 and 188 ± 14 mgÆdL)1

respectively) as compared to their starting cholesterol

levels at week 0 (50 ± 3 mgÆdL)1 and 54 ±

6 mgÆdL)1, respectively), whereas their plasma

triglyc-eride levels remained normal (24 ± 7 mgÆdL)1 and

24 ± 5 mgÆdL)1, respectively) (Fig 1D) FPLC

anal-ysis of plasma from these mice showed that the

small increases in the cholesterol levels of these mice

at week 24 were due to a minor accumulation of

chylomicron and VLDL remnants (Fig 2A,B) In

contrast, ApoE)/) mice showed a dramatic increase

in their plasma cholesterol levels during the course

of the experiment (Fig 1C) At week 24 of the experiment, plasma cholesterol levels of the ApoE)/) mice were 1064 ± 198 mgÆdL)1 (Fig 1C), whereas their plasma triglyceride levels remained normal (52 ± 28 mgÆdL)1 at week 24) (Fig 1D) FPLC analysis showed that the hypercholesterolemia of these mice was due to increased accumulation of triglyceride-containing cholesterol-rich chylomicron remnants (Fig 2A,B) No significant difference in the plasma free fatty acid levels among groups was observed during the course of the experiment At week 24, plasma free fatty acid levels of the ApoE3knock-in, C57BL/6 and ApoE)/) mice were 0.89 ± 0.08 mmol equiv., 0.81 ± 0.05 mmol equiv., and 0.99 ± 0.08 mmol equiv., respectively

Body composition analysis of the mice fed western-type diet for 24 weeks

At the end of the 24-week period on western-type diet,

at least six mice from each group (ApoE3knock-in, ApoE)/) and C57BL/6 mice) were killed The total weight, dry weight, water content, lipid content and lean body mass of the mice were determined as described in Experimental procedures As shown in Fig 1E, this analysis established that ApoE3knock-in mice had a total body lipid content of 39 ± 4% The wild-type C57BL/6 mice had a significantly lower total body lipid content of 32 ± 3% (P < 0.05) Thus, the increased body weight of the ApoE3knock-in and C57BL/6 mice reflects excess accumulation of adipose tissue in these mice In contrast, ApoE)/) mice remained lean during the course of the experiment, with a total body fat content of 11 ± 1% (Fig 1E,

P < 0.005) The complete body composition analysis

of the mice fed western-type diet for 24 weeks is sum-marized in Table 1

Diet-induced obesity in ApoE3knock-inand C57BL/6 mice is associated with elevated plasma glucose, insulin and leptin levels

Epidemiological and animal studies have established that central obesity is associated with glucose intoler-ance and insulin resistintoler-ance [20] In addition, obesity is accompanied by increased levels of leptin [38], a hor-mone that reduces appetite and may function as the link between obesity and hypertension in individuals with the metabolic syndrome [39,40]

To determine whether the obesity observed in ApoE3knock-in and C57BL/6 mice is associated with

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Fig 2 Representative FPLC cholesterol (A) and triglyceride (B)

pro-files of C57BL/6, ApoE3 knock-in and ApoE)/)mice fed western-type

diet for a period of 24 weeks.

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hyperglycemia, at weeks 0 and 24 of the experiment

mice were fasted for 16 h and plasma glucose levels

were then measured Immediately prior to switching

mice to western-type diet (week 0), the fasting plasma

glucose levels of the ApoE3knock-in, ApoE)/) and

C57BL/6 mice were 84.2 ± 3.7 mgÆdL)1,

60.7 ± 2.8 mgÆdL)1, and 75.2 ± 3.5 mgÆdL)1,

respec-tively (Fig 1F, P< 0.05) After 24 weeks on

western-type diet, ApoE3knock-in and control C57BL/6

mice developed hyperglycemia with fasting

glucose levels of 146.5 ± 9.1 mgÆdL)1 (P < 0.05) and

135.4 ± 7.9 mgÆdL)1 (P < 0.05) respectively

(Fig 1F) In contrast, ApoE)/) mice, which remained

lean during the course of the experiment, had only

slightly elevated fasting glucose levels that were within

the physiological range (94.3 ± 3.22 mgÆdL)1,

P< 0.005) To determine plasma insulin and leptin

levels in these mice, serum samples were isolated at

weeks 0 and 24 of the experiment and analyzed for

insulin and leptin At week 0, all three mouse groups

had similar plasma insulin levels (0.17 ± 0.05

ngÆmL)1 for ApoE3knock-in mice, 0.12 ± 0.05 ngÆmL)1

for ApoE)/) mice, and 0.16 ± 0.01 ngÆmL)1 for

C57BL/6 mice) At week 24, ApoE3knock-in and

C57BL/6 mice had elevated plasma insulin levels, with

concentrations in the range of 4.73 ± 1.03 ngÆmL)1

(P < 0.05) and 1.28 ± 0.32 ngÆmL)1 (P < 0.05),

respectively In contrast, ApoE)/) mice fed

western-type diet for 24 weeks had insulin levels of

0.23 ± 0.07 ngÆmL)1, which were similar to the levels

of ApoE)/) mice (0.317 ± 0.17 ngÆmL)1) on chow

diet for the same period of time

Analysis of plasma leptin levels showed that at

week 0, mice had similar leptin levels: 5.90 ± 0.40

ngÆmL)1 for ApoE3knock-in mice, 4.51 ± 0.32 ngÆmL)1

for ApoE)/) mice, and 9.2 ± 0.30 ngÆmL)1 for

C57BL/6 mice At week 24, the plasma leptin levels

of the ApoE)/) mice were reduced to 2.1 ± 0.4

ngÆmL)1 In contrast, in ApoE3knock-in mice fed

western-type diet for 24 weeks, leptin levels increased

dramatically to 41.14 ± 1.20 ngÆmL)1 (P < 0.005) A

similar but lower increase was also observed in

the plasma leptin levels of C57BL/6 mice fed

western-type diet for 24 weeks (34.70 ± 1.50 ngÆmL)1,

P < 0.005)

Diet-induced obesity in ApoE3knock-inand C57BL/6 mice is associated with reduced glucose tolerance and insulin sensitivity

To determine the role of ApoE in the development of obesity-associated insulin resistance and glucose intol-erance, we performed the standard glucose tolerance test (GTT) and insulin sensitivity test (IST) The GTT established that at week 0 all three mouse groups (ApoE3knock-in, ApoE)/) and C57BL/6 mice) had simi-lar normal responses to intraperitoneal administration

of glucose (Fig 3A) However, at week 24 of the experiment, ApoE3knock-in mice showed a significant deterioration in their ability to clear plasma glucose, as compared to C57BL/6 and ApoE)/) mice (Fig 3B;

P < 0.05) A similar but less severe effect was also observed in the C57BL/6 mice (Fig 3A,B; P < 0.05) Remarkably, however, ApoE)/) mice (which are resis-tant to diet-induced obesity) fed western-type diet for

24 weeks cleared glucose from the circulation more efficiently than the two other groups, and there was no significant difference in their response to intraperito-neal glucose load between weeks 0 and 24 on western-type diet (compare Fig 3A,B, P > 0.05)

In a similar fashion, when an IST was performed at week 0, all three mouse groups exhibited a similar response to intraperitoneal administration of insulin (Fig 3C) However, at week 24, ApoE3knock-in mice fed western-type diet for 24 weeks displayed the poor-est response to insulin administration as compared to C57BL/6 and ApoE)/) mice (Fig 3D; P < 0.05) C57BL/6 mice also exhibited reduced insulin sensitivity

at week 24 that was less severe than in ApoE3knock-in mice (Fig 3D; P < 0.05) In contrast, ApoE)/) mice fed western-type diet for 24 weeks exhibited the highest sensitivity to insulin of all three mouse groups (P < 0.05) In addition, there was no significant differ-ence in their insulin sensitivity curves between weeks 0 and 24 of the experiment (compare Fig 3C,D;

P > 0.05)

Table 1 Body composition of ApoE3 knock-in , C57BL/6 and ApoE)/)mice fed western-type diet for 24 weeks Values are in grams expressed

as mean ± SEM.

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LDLr)/)mice are more sensitive to diet-induced

obesity and hyperglycemia than ApoE)/)mice

but less sensitive than C57BL/6 mice

Low-density lipoprotein receptor is the major receptor

involved in the clearance of ApoE-containing

lipopro-teins from the circulation [41] Therefore, one

mecha-nistic explanation for the role of ApoE in the

development of obesity would be that LDLr-mediated

uptake of ApoE-containing chylomicron remnants

promotes the direct deposition of dietary fat into the

adipose tissue If this was the case, deficiency in LDLr

would prevent obesity and hyperglycemia To address

this possibility, groups of 8–10 female LDLr)/) or

C57BL/6 or ApoE)/) mice were placed on

western-type diet for a period of 15 weeks, and their body

weight and composition, and plasma cholesterol,

tri-glyceride and glucose levels were determined during

the course of the experiment At week 5 of the

experi-ment, the average weight of the LDLr)/) mice was

22.28 ± 1.32 g (29.7 ± 4.1% higher than their initial

weight of 17.13 ± 0.65 g, P < 0.05) (Fig 4A) This

increase was comparable to the 27.4 ± 3.8% increase

observed in the ApoE)/)mice (from 16.83 ± 0.24 g to

21.43 ± 0.56 g, P < 0.05), but lower than the 55.4 ±

3.8% increase in the C57BL/6 mice (from

16.26 ± 0.28 g to 25.25 ± 0.50 g, P< 0.005)

(Fig 4A) At week 15 of the experiment, however,

LDLr)/) mice showed an 84.5 ± 8.7% increase in

body weight (with an average final weight of

31.63 ± 2.10 g, P < 0.05) (Fig 4A) This increase

was higher than the 51.4 ± 4.5% increase (P < 0.05)

observed in the weight of the ApoE)/) mice (with an average final weight of 25.46 ± 0.55 g) However, it was still significantly lower than the 119.8 ± 7.6% increase observed in the weight of C57BL/6 mice (with an aver-age final weight of 37.28 ± 0.72 g) fed western-type diet for the same period of time (Fig 4A; P < 0.05) Body composition analysis at the end of the experi-ment revealed that at week 15, LDLr)/) mice had a body fat content of 19.9 ± 1.2%, which was much higher than the body fat content of the ApoE)/) mice (13 ± 1.9%, P < 0.05) but still lower than the body fat content of the C57BL/6 mice (28.06 ± 3.92%,

P < 0.05) fed western-type diet for the same period of time (Fig 4D) The complete body composition analy-sis of the mice fed western-type diet for 15 weeks is summarized in Table 2

Plasma lipid and glucose analysis showed that during the 15-week period, LDLr)/) mice developed severe hypercholesterolemia (1338 ± 135 mgÆdL)1) that was accompanied by moderate hypertriglyceride-mia (242.6 ± 14.9 mgÆdL)1) (Fig 4B,C) Plasma glucose levels were increased moderately (from 71.3 ± 6.7 mgÆdL)1 to 101 ± 4.9 mgÆdL)1, P < 0.05) but were still lower than the levels of C57BL/6 mice (131 ± 5.3 mgÆdL)1) at week 15 of the experiment (Fig 4E; P < 0.005)

The GTT and IST revealed that the LDLr)/) mice fed western-type diet for 15 weeks had similar toler-ance to glucose and sensitivity to insulin as in their starting state (week 0) (Fig 5; P > 0.05) In addition, there was no significant difference in the response to intraperitoneal administration of glucose or insulin

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Fig 3 Glucose tolerance curves (A, B)

and insulin sensitivity curves (C, D) of

ApoE3knock-in, C57BL/6 and ApoE)/)mice

at weeks 0 and 24 Values indicate the

average plasma glucose levels expressed as

mean ± SEM The statistical significance of

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each time-point is as indicated (*P < 0.05).

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Fig 4 Percentage of initial body weight (A), plasma cholesterol levels (B) and plasma tri-glyceride levels (C) of C57BL/6, LDLr)/)and ApoE)/)mice fed western-type diet for a period of 15 weeks (D) Percentage body fat content of ApoE3knock-in, ApoE)/)and C57BL/6 mice at week 15 (E) Fasting plasma glucose levels of ApoE3 knock-in , ApoE)/)and C57BL/6 mice at weeks 0 and 15 Each point on the graphs represents the mean value of the group, and error bars indicate the SEM The statistical significance of the observed differences among groups at each time-point is indicated (*P < 0.05;

**P < 0.005).

Table 2 Body composition of LDLr)/), ApoE)/)and C57BL/6 mice fed western-type diet for 15 weeks Values are in grams expressed as mean ± SEM.

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Fig 5 Glucose tolerance curves (A, B) and insulin sensitivity curves (C, D) of LDLr)/) and C57BL/6 mice at weeks 0 and 15 Values indicate the average plasma glucose levels expressed as mean ± SEM The statistical significance of the observed differences among groups at each time-point is as indicated (*P < 0.05).

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between LDLr)/) and C57BL/6 mice fed western-type

diet for 15 weeks (Fig 5) Interestingly, in our studies,

feeding western-type diet to C57BL/6 for 15 weeks did

not result in insulin resistance or glucose intolerance

(Fig 5B,D)

Taken together, these data indicate that female

LDLr)/) mice fed western-type diet for 15 weeks

appear to be more sensitive than female ApoE)/)mice

but still more resistant than female C57BL/6 mice in

the development of diet-induced obesity and its related

disorders

ApoE promotes diet-induced accumulation of

excess triglycerides in the liver while ApoE or

LDLr deficiency does not

If ApoE and LDLr are involved in the direct delivery

of dietary lipids to tissues, one would expect that

feed-ing ApoE3knock-in and C57BL/6 mice western-type diet

would result in excess accumulation of triglycerides in

the liver, whereas ApoE)/) and LDLr)/) mice would

be resistant to excess hepatic triglyceride accumulation

To test this hypothesis, we isolated liver samples from

mice fed western-type diet for 15 weeks and

deter-mined their triglyceride content (Fig 6A)

Liver samples from ApoE)/) and LDLr)/) mice fed

western-type diet for 15 weeks had similar

triglycer-ide contents of 60.67 ± 4.12 mgÆg)1 and 58.40 ±

5.11 mgÆg)1 of hepatic tissue, respectively (Fig 6A,

P> 0.05) In contrast, ApoE3knock-in and C57BL/6

mice had a much higher hepatic triglyceride content

(215.00 ± 33.56 mgÆg)1 and 213.72 ± 11.89 mgÆg)1

respectively, P < 0.05), confirming that human ApoE3,

murine ApoE, and the LDLr contribute to the

accu-mulation of excess lipids in the liver in response to

western-type diet (Fig 6A)

Effects of ApoE and LDLr deficiency on the direct

delivery of dietary lipids to adipose tissue

As ApoE and LDLr play pivotal roles in the

catabo-lism of chylomicron remnants, we attempted to

evalu-ate the contribution of the direct delivery of

nonhydrolyzed postprandial lipids to the development

of obesity To address this question, groups of

ApoE3-knock-in, ApoE)/), LDLr)/) and C57BL/6 mice that

were maintained on western-type diet for 15 weeks

were gavaged with 0.5 mL of olive oil containing

15 lCi of the nonhydrolyzable [3

H]cholesteryl-hexade-cyl-ether [cholesteryl-1,2-3H(N)] Twenty-four hours

later, mice were killed, and visceral fat and liver

sam-ples were isolated, weighed, and homogenized Then,

the amount of3H radioactivity present in the

homoge-nized tissues was determined using a liquid scintillation counter

ApoE3knock-in mice showed a higher average accu-mulation of dietary [3H]cholesteryl-hexadecyl-ether in adipose tissue (1718 ± 492 c.p.m per gram of tissue) than C57BL/6 mice (1010 ± 202 c.p.m per gram of tissue) (Fig 6B), but this difference between the two groups did not reach statistical significance (P > 0.05) The hepatic accumulation of 3H-label was similar between these two animal groups (3181

± 585 c.p.m per gram of tissue for the ApoE3knock-in mice and 3281 ± 578 c.p.m per gram of tissue for the C57BL/6 mice) (Fig 6C) ApoE)/) and LDLr)/) mice showed lower average accumulation of 3H-label in their fat (618 ± 41 c.p.m per gram of tissue and

664 ± 65 c.p.m per gram of tissue, respectively) and hepatic tissues (1624 ± 209 c.p.m per gram of tissue

0

cpms per gram of hepatic tissue

B A

0

cpms per gram of adipose tissue

0 50 100

2500 2000 1500 1000 500

4000 3500 3000 2500 2000 1500 1000 500

150 200 250

300

apoE3knock-in apoE –/– LDLr –/– C57BL/6

*

*

*

*

C

Fig 6 (A) Hepatic triglyceride content of ApoE3 knock-in , ApoE)/), LDLr)/) and C57BL/6 mice fed western-type diet for 15 weeks The statistical significance of the observed differences is indicated (*P < 0.05; **P < 0.005) (B, C) Total accumulation of tritiated label expressed as counts per minute per gram of adipose (B) or hepatic (C) tissue, 24 h after gavage administration of 0.5 mL of olive oil containing 15 lCi of [ 3 H]hexadecyl-cholesteryl-ether, per mouse.

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and 1759 ± 752 c.p.m per gram of tissue,

respec-tively) (Fig 6B,C) No measurable radioactivity was

found in the blood of the tested mice at the time of

killing (not shown)

Discussion

ApoE is a major protein involved in the metabolism

of dietary lipids and the removal of atherogenic

lipo-proteins from the circulation Following a lipid-rich

meal, lipids are packaged into chylomicrons which,

following partial lipolysis by lipoprotein lipase (LpL),

are converted into chylomicron remnants and acquire

ApoE [23] Then, lipid-bound ApoE interacts with

LDLr, which mediates the removal of

ApoE-contain-ing lipoproteins from the circulation In the present

study, we show that ApoE3knock-in mice are more

sen-sitive to diet-induced obesity and related metabolic

dysfunctions than wild-type C57BL/6 mice, whereas

ApoE)/) mice are resistant to the development of

these conditions Furthermore, deficiency in LDLr

results in reduced sensitivity to obesity in response to

western-type diet, raising the possibility that the

effects of ApoE may be mediated, at least in part,

via its interactions with LDLr

Interestingly, there were no significant differences in

plasma free fatty acid levels among mouse groups

(ApoE3knock-in versus C57BL/6 versus LDLr)/) versus

ApoE)/)), although previous studies suggested that

increased plasma levels of free fatty acids are closely

associated with obesity-induced insulin resistance

[42,43] Moreover, daily food consumption of the

ApoE3knock-in, C57BL/6 and ApoE)/) mice was similar

among groups, suggesting that different responses to

western-type diet cannot be attributed to differences in

appetite

One possible explanation for the increased sensitivity

of the ApoE3knock-in mice to diet-induced obesity

would be that higher plasma ApoE levels in these mice

than in C57BL/6 mice are responsible for the enhanced

deposition of dietary lipids in adipose tissue To

address this possibility, we compared the ApoE levels

in plasma samples isolated from ApoE3knock-in and

C57BL/6 mice at week 0 of the experiment, using

western blotting This analysis showed that

steady-state plasma ApoE levels in the ApoE3knock-in mice

used in our study are approximately four times lower

than those in wild-type C57BL/6 mice Thus, the

increased sensitivity of ApoE3knock-in mice to

diet-induced obesity is not the result of elevated plasma

ApoE levels in these mice as compared to C57BL/6

mice, and the difference in the ability of human

ApoE3 and murine ApoE to promote obesity in

response to high-fat diet may be due to intrinsic differ-ences between these two peptides The data presented

in Fig 6B raise the possibility that chylomicron and VLDL remnants containing the human ApoE3 isoform are taken up more avidly by adipose tissue than the lipoproteins containing mouse ApoE

In a previous study, Sullivan et al [44] reported that ApoE3knock-in mice and C57BL/6 mice have similar plasma ApoE levels Furthermore, using northern blot analysis, they also showed that ApoE mRNA levels were indistinguishable between ApoE3knock-in mice and C57BL/6 mice, in all tissues tested except for the small intestine, where human ApoE3 mRNA expression was lower than mouse ApoE mRNA expression [44] How-ever, the ApoE3knock-inmice studied by these investiga-tors are different from the ApoE3knock-inmice tested in our experiments, because our ApoE3knock-in mice have been bred for nine generations to the C57BL/6 back-ground It is possible that back-crossing ApoE3knock-in mice to C57BL/6 mice for nine generations resulted in the reduced plasma human ApoE3 levels that we observed

Human ApoE has three natural isoforms in humans: ApoE2, ApoE3 and ApoE4 In vitro receptor binding studies established that lipid-bound ApoE3 and ApoE4 have a similar affinity for LDLr, whereas lipid-bound ApoE2 has a much lower affinity [45,46] In this study,

we focused on ApoE3, mainly because it is the most common ApoE genetic polymorphism in humans [24– 29] If the effects of ApoE3 on obesity are mediated by its lipid-lowering potential via LDLr, then we expect that both ApoE3 and ApoE4 will predispose to a simi-lar extent to diet-induced obesity and insulin resistance

in mice, whereas ApoE2 may have a much lower potential to promote these conditions Further studies are needed in order to address this point, and other mechanisms of ApoE-promoted diet-induced obesity should not be excluded

It is quite interesting that in all our experiments, plasma cholesterol levels correlated inversely with body weight gain and body fat accumulation (Figs 1 and 4)

In the ApoE)/)mice, failure to clear chylomicron rem-nants due to deficiency in ApoE resulted in a steady increase in plasma cholesterol levels and rendered these mice resistant to diet-induced obesity In contrast, in the ApoE3knock-in mice, the efficient catabolism of chylomicron remnants resulted in only slightly elevated plasma cholesterol levels, but promoted obesity, insulin resistance and glucose intolerance Similar to the ApoE3knock-in mice, C57BL/6 mice, which express the mouse ApoE, developed only mild hypercholesterol-emia but became obese and insulin resistant following western-type diet for 24 weeks

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Plasma triglyceride levels of the LDLr)/) mice were

moderately elevated at week 0, and remained elevated

during the 15 weeks on high-fat diet, whereas plasma

triglyceride levels of the other animal groups remained

normal for the duration of the experiment This is not

surprising, because in the absence of the LDLr,

reduced clearance of ApoE-containing lipoproteins

from the circulation results in elevated steady-state

plasma ApoE levels Since ApoE is a known inhibitor

of LpL [47], and plasma triglyceride levels correlate

with plasma ApoE levels [48], accumulation of ApoE

in the blood of LDLr)/) mice results in reduced

LpL-mediated lipolysis of plasma triglycerides and

hyper-triglyceridemia

In our studies, LDLr)/) mice became more obese

than ApoE)/) mice but less obese than C57BL/6

mice, raising the possibility that, in addition to

LDLr, other ApoE-recognizing receptors may also

promote the deposition of postprandial lipids in

adi-pose tissue, thus contributing to diet-induced obesity

and related metabolic dysfunctions Indeed, a recent

study showed that adipose-tissue-specific deletion of

the LDLr-related protein makes mice less sensitive to

obesity [49] In the case of the LDLr)/) mice,

LDLr-related protein and possibly other ApoE-recognizing

‘scavenger’ receptors may promote, to some extent,

delivery of ApoE-containing chylomicron remnants to

adipose tissue However, in the case of the ApoE)/)

mice, which lack the endogenous ApoE, all of these

ApoE-mediated receptor processes are blocked, and

ApoE)/) mice are more resistant to body fat gain

than LDLr)/) mice Even though the expression of

LDLr in adipose tissue is much lower than in liver,

our data support a functional role for this receptor

in the ApoE-mediated mechanism of diet-induced

obesity

In a previous study by Schreyer et al [50], it was

suggested that LDLr)/) mice fed a diabetogenic diet

for 16 weeks were more susceptible to diet-induced

obesity and hyperglycemia than C57BL/6 mice,

whereas ApoE)/) mice appeared to be as susceptible

to the development of these conditions as C57BL/6

mice Furthermore, in that study, LDLr)/) mice

developed severe hypertriglyceridemia during the

course of the experiment The diabetogenic diet used

in that study contained a very high fat content of

35.5% (derived mainly from lard) (Bioserve,

French-town, NJ, USA; cat no F1850) In all our

experi-ments, mice were fed the standard western-type diet

containing 21.1% fat (Harlan Teklad; cat no

TD88137) It is possible that the high fat content of

the diabetogenic diet predisposed ApoE)/), LDLr)/)

and C57BL6 mice to the development of diet-induced

obesity, and resulted in saturation of the metabolic pathways that control body fat deposition and plasma lipid and glucose homeostasis Under such conditions, ApoE or LDLr deficiency may not be sufficient to prevent obesity, as other mechanisms contributing to obesity may override the protective effect of ApoE or LDLr deletion that we observed in our experiments Our data on LDLr)/) mice are in agreement with the data of MacDonald et al [51], showing that female LDLr)/) mice do not become excessively obese, and do not develop hyperglycemia and glucose intolerance in response to western-type diet

In our experiments, we studied the role of ApoE in the development of obesity in response to dietary consumption of fat, one of the major causes of human obesity [2,18] Our findings are supported by previous observations by Gao et al [52] and Chiba

et al [53] showing that deficiency in ApoE renders genetically predisposed obese mice less sensitive to spontaneous development of obesity Furthermore,

in vitro studies suggested that ApoE promotes triglyc-eride uptake and deposition in in vitro differentiated adipocytes and in freshly isolated adipose tissue explants [54], whereas VLDL induces adipocyte differ-entiation in an ApoE-dependent manner [53] In the present study, we report for the first time that human ApoE3 increases susceptibility to diet-induced obesity

as compared to mouse ApoE The functional role of ApoE-containing chylomicron and VLDL remnants

in the development of diet-induced obesity is further supported by the observation that ApoE)/) mice remain lean when fed western-type diet for 15 or

24 weeks

One of the hallmarks of obesity-associated insulin resistance is the increase in the circulating levels of insulin [55] Such a change is also evident in our stud-ies, further supporting the hypothesis that the benefi-cial effects of ApoE deletion on weight loss extend to increased insulin sensitivity Furthermore, our data demonstrate a significant increase in plasma leptin levels in ApoE3knock-in and, to a lesser extent, in C57BL/6 mice, an observation that is also consistent with their increased adiposity [56]

ApoE has long been known to be atheroprotective, mainly because of its ability to clear plasma lipids However, our data show that if excess dietary lipids are present in the circulation, this atheroprotective property of ApoE may be counteracted by the enhanced deposition of lipids in adipose tissue Over-all, our findings identify ApoE expression as a key peripheral contributor to the development of obesity and related metabolic dysfunctions in mice

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