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Human adipocytes induce inflammation and atrophy in muscle cells during obesity

Pellegrinelli V1,2,3#, Rouault C1,2, 3, RodriguezCuenca S4, Albert V1,2,3, EdomVovard F5,6,7,8 ,VidalPuig A4, Clément K 1, 2, 3*, ButlerBrowne GS5,6,7,8* and Lacasa D1,2, 3* 1 INSERM, U1166 Nutriomique, Paris, F75006 France; 2 Sorbonne Universités, University Pierre et MarieCurieParis 6, UMR S 1166, Paris, F 75006 France; 3 Institut Cardiométabolisme et Nutrition, ICAN, Pitié Salpétrière Hospital, Paris, F75013 France;

4 Metabolic Research Laboratories, Wellcome TrustMRC Institute of Metabolic Science, University of Cambridge, Cambridge, United Kingdom;

5 Sorbonne Universités, University Pierre et MarieCurieParis6, Centre de recherches en Myologie UMR 974, F75005, Paris, France;

6 INSERM, U974, 75013, Paris, France;

7 CNRS FRE 3617, 75013, Paris, France;

8 Institut de Myologie, 75013, Paris, France.

Key Words: muscle cells atrophy, visceral adipocytes, human obesity. Running title: obese human adipocytes induce muscle cells atrophy * These authors share senior coauthorship # Corresponding author: Current address: Wellcome TrustMRC Institute of Metabolic Science, Metabolic Research Laboratories,University of Cambridge email address: [email protected] Phone+44 (0)1223 336786 Fax: +44 1223 330598 Word count of body: 4568 Word count of abstract: 206 Number of figures/Tables: 7 Supplementary data

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ABSTRACT

Inflammation and lipid accumulation are hallmarks of muscular pathologies resulting from

metabolic diseases such as obesity and type II diabetes. During obesity, the hypertrophy of

visceral adipose tissue (VAT) contributes to muscle dysfunctions particularly through

dysregulated production of adipokines.

We have investigated the crosstalk between human adipocytes and cells to

identify mechanisms linking adiposity and muscular dysfunctions.

First, we demonstrated that the secretome of obese adipocytes decreased the expression of

contractile in myotubes consequently inducing atrophy. Using a threedimensional co

culture of human myotubes and VAT adipocytes we showed the decreased expression of

corresponding to skeletal muscle contractility complex and . We demonstrated an

increased secretion by cocultured cells of cytokines and chemokines with IL6 and IL1β as key

contributors. Moreover, we gathered evidence showing that obese subcutaneous adipocytes were

less potent than VAT adipocytes in inducing these myotubes dysfunctions. Interestingly, the

atrophy induced by visceral adipocytes was corrected by IGFII/IGFBP5. Finally, we observed

that skeletal muscle of obese mice displayed decreased expression of muscular markers in

correlation with VAT hypertrophy and abnormal distribution of the muscle fiber size.

In summary, we show the negative impact of obese adipocytes on the muscle phenotype that

could contribute to muscle wasting associated with metabolic disorders.

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INTRODUCTION

During obesity, hypertrophy of white adipose tissue (WAT) is associated with fibroinflammation and cell dysfunctions. However, visceral depot (VAT) differs from subcutaneous (SAT) by its inflammatory status due to its high infiltration with immune cells such as macrophages, T lymphocytes and mast cells (1,2). Hence, VAT hypertrophy is considered an important contributor to obesity related metabolic and cardiovascular diseases (3). In obese subjects, hypertrophied adipocytes display abnormal secretion of leptin and adiponectin, an increased production of numerous inflammatory cytokines and chemokines, leading to a disrupted inter organ communication between VAT and important metabolic peripheral tissues such as liver and skeletal muscle (4). These organs are particularly exposed to free fatty acids and cytokines, mostly IL6, increasingly released by visceral fat of obese subjects (5,6). Skeletal muscles are one of the major metabolic tissues of the body, playing a pivotal role in glucose and lipid metabolism.

There is growing body of evidence showing the contribution of obesity on skeletal muscle dysfunctions, such as the development of insulin resistance, as attested by numerous studies performed in rodent models of obesity (7,8). Thus, it has been shown that obese rats display impaired activation of skeletal muscle synthesis in response to chronic lipid infiltration

(9). Ectopic lipid storage in muscle (e.i. within muscle cells and/or in adipocytes located between muscle fibers) represents a negative risk factor for development of type 2 diabetes related to muscle insulin resistance (10–13). Moreover, skeletal muscle of obese subjects also displays an impairment in oxidative capacity (14) and abnormal muscle fiber organization (15,16). Increased amount of VAT in obesity has been proposed to link obesity and muscle metabolic alterations, such as insulin resistance. Surgical removal of VAT in rats leads to reduction of systemic concentrations of cytokines (e.g. IL6, Fractalkine resistin) and improvement of skeletal muscle

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insulin sensitivity (17). Particularly, adipocytes have been identified as potential effectors of

muscle cells dysfunctions. Coculture system previously showed impact of adipocytes on muscle

cells insulin resistance and oxidative capacity (18–20). However, the impact of VAT adipocytes

on muscle structural organization and the molecular factors involved remains elusive.

Inflammatory mediators act synergistically to negatively impact regeneration capacity and insulin

sensitivity in muscle (7). For example, IL6 is overproduced by hypertrophied adipocytes and its

systemic concentration is increased in obesity. Increased IL6 is associated with altered insulin

signalling in myotubes and impaired myoblast differentiation/proliferation capacity leading to

muscle atrophy (7,8). At the molecular level, IL6 with IL1β downregulate IGFs/Akt signalling

decreasing muscle protein synthesis (21). Excessive production of various mediators by obese

VAT adipocytes could trigger a crosstalk with muscle cells, altering the production of myokines

(22) which could affect in turn VAT endocrine functions. This crosstalk associated with an

inflammatory microenvironment could be deleterious in perpetuating a vicious cycle leading to

muscle loss and wasting. To address this question, the secretome of obese adipocytes was tested

on human primary muscle cells and direct cocultures were performed.

The main aim of our study was to identify the major contributors of VAT adipocytes/skeletal

muscle crosstalk and their role in inducing muscle atrophy and inflammation, common disorders

associated with metabolic pathologies such as obesity and type 2 diabetes (4).

Research Design and Methods

The antibodies and recombinant proteins used in the study are listed in Table S1.

Mice studies

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C57BL/6N male mice were purchased from Charles River. Standard chow or 58 kcal% fat w/sucrose Surwit Diet (HFD, D12331, Research Diets) was given ad libitum to animals. Dietary intervention started at 8 weeks of age and continued for 12 or 16 weeks (n=89 per experimental group). The detailed procedures of the mice studies are provided in (23). Adipose tissues and skeletal muscles from 12 weeks chow/HFD mice (RNA extraction) and Gastrocnemius muscle of

16 weeks chow/HFD mice (histological analysis) were prepared as described in (23,24). Muscle fiber crosssection size (up to 201 muscle fibers/section) was automatically evaluated by determination of the Feret’s diameter (Image J software) in 3 independent fields in each section.

The variance coefficient of the Feret’s diameter was defined to evaluate the muscle fiber size variability between the experimental groups of mice and avoid experimental errors such as the orientation of the sectioning angle (25). Number of intermuscular adipocyte spots was assessed visually in the total longitudinal section biopsy.

Human studies

Subcutaneous (SAT) and VAT biopsies were obtained from morbid obese subjects (body mass index, BMI>40 kg/m2) (clinical parameters in Table S2) and SAT biopsies from lean female subjects (BMI<25 kg/m2) undergoing elective surgery. None of the lean subjects had diabetes or metabolic disorders, and none were taking medication. All clinical investigations were performed according to the Declaration of Helsinki and approved by the Ethical Committee of HôtelDieu

Hospital (Paris, France).

Isolation of mature adipocytes from human WAT and preparation of the 3D adipocyte cultures

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Tridimensional gels were prepared with mature adipocytes isolated from WAT of lean (SAT) or

obese (SAT and VAT) subjects as described in (26,27). Adipocytes were embedded in the

hydrogel (Puramatrix, BD Biosciences, Bedford, MA, USA). Hydrogel was diluted in 20%

sucrose solution at a concentration of 1x 105cells per 1mL of gel preparation into 24well plates

containing 1.5ml of DMEM/F12 (1% bovine albumin, 1% antibiotics). Culture medium was

changed after 1h with fresh medium containing human insulin (50 nM). Adipocytes from SAT or

VAT were incubated for 48h before collecting the conditioned media referred as lean SAT CM

(lean SAT adipocyte conditioned media) and obese SAT/VAT CM (obese SAT/VAT adipocyte

conditioned media), respectively. Samples were kept at 80°C prior experimental measurements.

Myoblast cultures, differentiation to myotubes and treatment with adipocyte conditioned

media

We used the human primary satellite cells (CHQ5B) originally isolated from the quadriceps of a

newborn child without indication of neuromuscular disease as previously described (28). The

cells were cultivated in a growth medium consisting of 4V DMEM, 1V M199 supplemented with

50 g/ml of gentamycin and 20% FBS. Cells were plated in 24 wellplates at a density of 1500

cells per cm2. Differentiation was induced at subconfluence by replacing the growth medium

with differentiation medium (DMEM supplemented with 50 g/ml of gentamycin and 10 g/ml

human insulin) (29). After 5 days of differentiation, the medium was replaced for 48h with

control DMEM/F12 culture medium or conditioned media from either adipocytes from lean SAT

(lean SAT CM ) or adipocytes from obese SAT/VAT (obese SAT/VAT CM). Then, the medium

was exchanged for fresh medium, for 24 h before being collected and stored at 80 C.

Quantification of the fusion index and measurement of myotube thickness

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Muscle cells were allowed to differentiate in serumfree medium for 6 days in the presence of lean SAT CM or obese SAT/VAT CM. After fixation with 4 % paraformaldehyde, the cells were incubated with an antibody specific for . Specific antibody binding was revealed using an

Alexa488 coupled goat anti mouse secondary antibody. Nuclei were visualized with Hoechst staining.

The efficiency of the fusion was assessed by counting the number of nuclei in myotubes (>2 myonuclei) as a percentage of the total number of nuclei (mononucleated and plurinucleated).

This percentage was determined by counting 1000 nuclei per dish on three independent cultures.

Myotubes thickness was quantified with Image J software measuring intensity of the desmin staining in ten random fields (X10) from three independent cultures.

The counting was performed with blind lectures by two different investigators (VA and FEV).

Direct cocultures of primary adipocytes and differentiated myoblasts in 3D hydrogels

Muscle cells were embedded in hydrogel prepared as below, at a concentration of 200.000 cells per 100µl of gel preparation containing 0.5 mg/ml collagen type 1 (BD Biosciences). The gel preparation was put into 96well plates containing 150 µl of growth medium. After 2 days in culture, this medium was replaced by differentiating medium for 3 days. We then added to this preparation a second hydrogel containing mature adipocytes isolated from VAT or SAT from obese subjects (10.000 cells per 100 µl of gel preparation). Adipocytes and differentiated myoblasts were then cocultured for 24h or 3 days in DMEM/F12 (1% antibiotics and 50 nM insulin).

Secretory function analysis and serum biochemistry

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Adipokine secretions were evaluated by ELISA assay, using secretory medium from 24hcultures

of 3D adipocytes (leptin, adiponectin) (Duoset, R&D Systems). Concentrations of human

cytokines were determined in either conditioned media from adipocytes, myotubes or cocultures

using human cytokine/chemokine Panel I 41plex from Millipore (Billirica, MA, USA) as

previously described in (30). IGFII levels were measured by ELISA assay in CM from myotubes

cocultured or not with 3D adipocytes during 48h incubation. Serum levels of the murine

cytokines IL6, IL1 β, IL10 and TNFα were analyzed using the MSD 7plex mouse

proinflammatory cytokine high sensitivity kit (product code K15012C2, MesoScale Discovery

(Gaithersburg, MD, USA) on the MesoScale Discovery Sector 6000 analyzer. Results were

calculated using the MSD Workbench software package. Lower limit of detection were the

following: CXCL1/2/3 (3.3 pg/ml), IL10 (5.0 pg/ml), IL1β (2.3 pg/ml), IL6 (16.8 pg/ml) and

TNFα (0.9 pg/ml).

Neutralizing experiments and treatment by recombinant proteins

Myotubes alone or cocultured cells were treated with IgG1 (3µg/mL) (MYO IgG, and

MYO+AD IgG) or with IL6 (2.5µg/mL) and IL1β (0.5µg/mL) neutralizing antibodies (MYO

abIL6/IL1β, MYO+AD abIL6/IL1β) during 3 days with renewal every day. Human

recombinant IL6 (10 ng/ml) and IL1β (1ng/ml) were added to myotubes cultured in the

hydrogel for 3 days with renewal every day. In another set of experiments, myotubes and co

cultured cells were treated with IGFII (50 ng/ml) and IGFBP5 (200 ng/ml), when indicated, for

3 days with every day change. After this period, cells and media were collected for

immunofluorescence and multiplex analysis, respectively.

Immunofluorescence analysis and confocal microscopy

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After fixation in 4 % paraformaldehyde, cells were processed for immunofluorescence analysis.

These samples were incubated with the appropriate primary antibody (overnight et 4°C), and then the corresponding Alexa488conjugated antimouse IgG or Alexa546conjugated antirabbit IgG interspaced by multiple washes in PBS, and followed by mounting coverslips. Sample examinations were performed at the imaging platform (CICC, Centre de Recherches des

Cordeliers, Paris, France) using a Zeiss 710 confocal laser scanning microscope (Carl Zeiss, Inc.,

Thornwood, NY, USA) fitted with a LD LCI PlanApochromat oilimmersion objective 25x or

63X. Images were captured and analyzed with ZEN 2009 imaging software (Carl Zeiss, Inc.).

Western blot analysis:

Total cell extracts were prepared in a buffer containing a cocktail of protease and phosphatase inhibitors (Roche Diagnostics, Mannheim, Germany) and were analyzed as previously described

(26). Apparent molecular sizes were estimated by using the SeeBlue® Plus2 PreStained Protein

Standard (Life Technologies, Foster City, CA, USA) as indicated on immunoblots.

RNA preparation and PCR array

Muscle cells and adipocytes cocultured in the 3D scaffolds were processed for RNA extraction using the RNeasy RNA mini kit (Qiagen, Courtaboeuf, France). After reverse transcription of the total RNAs (1 g), samples were analyzed by i) real time PCR and ii) PCR array using the “

Human myogenesis and myopathy PCR array” according to manufacturer’s instructions

(Qiagen). Table S3 presents the list of the primers used. Data were normalized according to the

RPLPO expression. Supplementary list presents the functional classification of the genes in the “Human myogenesis and myopathy PCR array”.

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Statistical analysis

The experiments were performed at least three times, using adipocytes from different human

subjects. Statistical analyses were performed with GraphPad Software (San Diego, CA, USA).

Values are expressed as means ± SEM of (n) independent experiments performed with different

adipocyte preparations. Comparisons between the two conditions in the in vitro experiments

(adipocytes and adipocytes/myotubes cultures) and in vivo studies (chow and HFD mice) were

analyzed using the Wilcoxon non parametric paired test. Comparisons between more than two

groups were carried out using one way analysis of variance (ANOVA) followed by posthoc

tests. Spearman coefficients were calculated to examine correlations. Differences were

considered significant when p<0.05.

RESULTS

Adipocyte secretions from obese VAT provoke muscle cells dysfunctions

Obesity and associated metabolic disorders such as type 2 diabetes are associated with muscle

dysfunctions characterized by inflammation and fat deposition (7). The muscle alterations are

associated with insulin resistance in link with an increase of visceral fat mass (10,20,31). We

sought to establish whether SAT/VAT from obese subjects could directly contribute to the

harmful phenotype observed in the muscle of these patients. Here, we take advantage of a well

characterized cohort of severe obese subjects showing chronic inflammation and marked insulin

resistance (1,32), as shown in Table S2.

Initially, we characterized the secretome of conditioned media from SAT and VAT adipocytes

(obese SAT CM and obese VAT CM, respectively) obtained from obese subjects and the media

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obtained from the adipocytes isolated from SAT of healthy lean subjects as control (lean SAT

CM). As expected, low adiponectin secretion was observed in obese SAT and VAT adipocytes compared to lean SAT ones (Figure S1A). We observed that the secretome of VAT adipocytes exhibited a proinflammatory profile with high levels of several cytokines (IL6 and GCSF), chemokines (IL8, CCL2, CCL5 and CXCL1/2/3) as well as the growth factor FGF2, compared to SAT adipocytes from either lean or obese subjects (Figure S1AB). Adipocytes from obese

SAT presented an intermediary inflammatory profile between lean SAT and obese VAT.

For this reason, we focused on obese VAT adipocytes and explored the impact of conditioned media (VAT CM) on myotubes morphology and contractile protein expression, in comparison to conditioned media from lean SAT adipocytes (lean SAT CM). We observed that myotubes were thinner in the presence of obese VAT CM than control and lean SAT CM conditions as estimated after immunofluorescence staining with desmin (a major component of architecture and contractility), which was also significantly lower in myotubes treated with obese VAT CM than with lean SAT CM (20%, p<0.01) (Figure 1AB). The number of nuclei in the differentiated cultures was not modified by the two conditioned media (Figure 1C). Western blot analysis showed that the heavy chain (MF20) and , two major components of the contractile apparatus in skeletal muscle, were also significantly decreased for both proteins in the presence of obese VAT CM compared to control and lean SAT CM conditions (Figure 1D). If lean SAT seems to impact myotube thickness and protein content, this was not statistically significant and remains much lower than the effect of obese VAT CM. These data suggest that the secretome of obese VAT adipocyte decrease myogenic capacity of the skeletal myoblasts.

VAT adipocytes directly induce muscle cells atrophy in a 3D scaffold

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We then examined the effect of obese SAT and VAT adipocytes on skeletal muscle cells by using

3D cocultures. Mature adipocytes and muscle cells were then embedded in separate hydrogels

(Figure S2AB), previously characterized by the ability to maintain altered functions of obese

adipocytes (26). Direct cocultures of human cells in a 3D setting are of great value because they

allow the study of a complex set from adipocytederived signals that regulate skeletal muscle

functions without confounding effects of other organ systems, while maintaining signals

including labile diffusible factors. The human myoblast differentiation and fusion processes were

not modified by cultivation in this 3D scaffold compared to 2D cultures (see MF20 and DAPI

staining, Figure S2C).

We used a skeletal muscle specific PCR array to determine the effects of the obese VAT

adipocytes on the expression of the musclespecific genes. Among the set of 86 genes screened

on the array, we detected significant underexpression of 10 genes in the muscle cells exposed to

obese VAT adipocytes. Especially, we found decreased level of myogenic transcription factors,

MyoD1 and Myogenin (MYOG), the growth factor IGFII and its binding partner IGFBP5, the

sarcomeric protein (TTN), a member of the complex, α (SGCA), the

musclespecific protein of caveolae, caveolin3 (CAV3) and an important component of the

neuromuscular junction, the musclespecific (MuSK) (Figure 2A). It should be noted

that ER stress and cytolysis pathways were not detected in the cocultured cells as assessed from

expression of ER stress markers (ATF4, HSPA5 and C/EBPζ) and lactate dehydrogenase

activity, respectively (Figures S3 and S4A).

By confocal analysis, we confirmed decreased expression of titin at protein level in myotubes

induced by VAT adipocytes (Figure 2B). Moreover, myotubes exposed to VAT adipocytes

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showed a reduced striated staining for titin suggesting a disturbed internal sarcomeric structure

(Figure 2C).

Importantly, when compared to obese VAT adipocytes, obese SAT adipocytes appeared less potent in inducing muscle cell atrophy as seen from myotube thickness and staining of troponin and titin (Figure 2DF).

Next, we quantified the levels of inflammationrelated proteins using a multiplex “human cytokines/chemokines” approach. Among the 41 screened proteins, 12 were significantly detected in VAT adipocytes and myocytes (level >15 pg/ml). CCL2, IL8, CXCLs, GCSF, IL6 and VEGF were highly secreted by the cocultured cells compared to VAT adipocytes grown alone in hydrogel (Figure S4B). For most of these inflammatory proteins, secretion was increased in an additive manner in the presence of myotubes and adipocytes. Moreover, a synergistic affect was observed for GCSF (75fold, p<0.05), IL6 (25fold, p<0.05) and CCL7 (10fold, p<0.01)

(Figure 3A). As observed above for muscle cell atrophy, adipocytes from obese SAT provoked a lower inflammatory status than those from obese VAT in the cocultured cells (Figure 3BC).

Considering the marked inflammatory profile of VAT adipocytes from obese subjects and their potent impact on muscle cell phenotype compared to lean and obese SAT ones, we then focus on the cross talk between muscle cells and obese VAT adipocytes.

IL6 and IL1βββ have relevant role in inflammation of myotubes/adipocytes

We tested the role of two proinflammatory cytokines IL6 and IL1β, as drivers of myotubes inflammation observed in cocultures based on previous reports (26,33,34). After 3 days of culture, in the presence of the neutralizing IL6/IL1β antibodies, the inflammatory response of

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cocultured cells was reduced with decreased levels of GCSF (71.4%), Fractalkine (58%), IL7

(56%) and the chemokines CXCL1/2/3 (53%), CCL7 (42%), IL8 (43.5%), IP10 (36%) and

CCL5 (38%,) (Figure 3D). To further confirm the role of IL6/IL1β, we treated myotubes with

human recombinant IL6 and IL1β which showed marked increased secretion of inflammatory

molecules such as GCSF (~6000fold), CXCL1/2/3 (~100fold) and IL8 (~100fold) (p<0.001)

(Figure 3E). These results indicate the key role played by the cytokines IL6 and IL1β in the

inflammatory environment of the cocultured cells. Strikingly, the combination of recombinant

IL6/IL1β or antibody neutralization of endogenous IL6/IL1β failed to influence myotube

thickness and titin staining (figure S5) suggesting an independent relationship between

inflammation and the atrophic phenotype observed in our experimental conditions.

IGFII and IGFBP5 correct the myotubes atrophy induced by obese VAT adipocytes

As shown in Figure 2A, myotubes cocultured with obese VAT adipocytes displayed a decreased

expression of IGFII and its binding partner IGFBP5 known to synergically promotes myogenic

action (35). In 3D cultures of myocytes, 3 days treatment with human recombinant IGFII (50

ng/ml) and IGFBP5 (200 ng/ml) impacts muscle cells by increasing titin content (Figure S6). We

then tested the potential correction of the atrophic phenotype of myotubes cocultured with VAT

adipocytes. Interestingly, chronic treatment with IGFII and IGFBP5 corrected the atrophic

aspect of the cocultured myotubes through increased thickness (Figure 4A) and production of

both titin and myosin heavy chain (Figure 4BD).

However, chronic treatment with IGFII/IGFBP5 failed to decrease the inflammatory secretome

of the cocultured cells (Figure S7). Conversely, cocultured cells treated by neutralizing

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antibodies for IL6 and IL1β or treatment of myotubes by recombinant IL6 and IL1β did not influence the production of IGFII (Figure S5A).

Downregulation of the signaling pathways controlling protein synthesis in myotubes exposed to VAT adipocytes.

Skeletal muscle mass is determined by the balance between protein synthesis and degradation.

The rate of protein synthesis is, at least in part, finely controlled by the PI3K/Akt/mTOR signaling pathway, activated by IGFs and amino acids (36). Here, we assessed the IGFII/IGFBP

5 activation of this pathway in cocultured atrophic myotubes by measuring the phosphorylation status of direct targets, Akt, p70S6K and 4EBP1. We showed a reduction of basal phosphorylation of Akt, S6K and 4EBP1 in cocultured myotubes compared to control ones as well as reduced AKT phosphorylation in response to IGFII/IGFBP5 (Figure 4EF), suggesting that signalling pathways (Akt/S6K/4EBP1) controlling protein synthesis are down regulated in cocultured muscle cells. Conversely, the of two atrophyrelated ubiquitin , atrogin1 and murf1, remained unchanged in muscle cells exposed to VAT adipocytes and not influenced by IGFII/IGFBP5 (Figure S8). Overall, our data suggest that the atrophy phenotype is likely related to decreased synthesis of muscle proteins.

Dietinduced obesity induces muscle atrophy in association with epididymal WAT hypertrophy and accumulation of intermuscular adipocytes in mice models.

In order to validate in vivo the relevance of the molecular candidates identified from our 3D co cultures, we used a dietinduced obese mouse model and screened expression of inflammatory/muscle markers in their skeletal muscles. After 12 weeks of HFD, mice displayed

WAT and systemic inflammation with a global decrease in gene expression of muscular markers such as MyoD (76%), IGFII (58%), β (43%) and PGC1β (30%) in the Gastrocnemius 15

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muscle (Figure 5AB, Figure S9A). Conversely, expression of the inflammatory markers IL1β

and CCL2 was significantly increased: 1.7 fold (p=0.0415) and 1.5 fold (p=0.0361), respectively.

In addition, the expression of skeletal muscle markers such as MyoD and IGFII was negatively

correlated with increased fat mass (% fat mass and leptin expression) and inflammation in

epididymal WAT (IL6 expression) (Table 1 and Table S4). However, we did not find any

correlations between IL6 expression in inguinal WAT and skeletal muscle markers, highlighting

the specificity of the crosstalk between muscle and the VAT depots (Table S5). Finally,

histological analysis of the Gastrocnemius of obese mice revealed pathological changes generally

observed in muscle of dystrophic mice (25) with abnormal distribution of the muscle fiber size in

crosssection samples (increased covariance coefficient of Feret’s diameter) and adipocytes

accumulation (Figure 5CE, Figure S9B). Accordingly, we observed in obese mice muscle an

increased expression of adipocyte markers (i.e. leptin and FABP4) in muscle reflecting increased

fat infiltration (Figure S9C). Interestingly, the expression of MyoD, IGFII and βactin in skeletal

muscle was negatively correlated with leptin expression in epididymal WAT and skeletal muscle

(Table 1). These results suggest a potential relationship between adipocyte accumulations and

muscle structure and dysfunction.

Discussion

We studied the adipocyte/muscle cell crosstalk participating in the complex intertissular

network of hypertrophied WAT with skeletal muscle. Here, using a 3D coculture system, we

provide evidence in vitro that adipocytes from obese VAT induce muscle cells inflammation and

atrophy by decreasing the expression of contractile proteins such as troponin, titin and myosin

heavy chain. Importantly, we showed that in obese states, VAT adipocytes are more potent than

their SAT counterparts in provoking deleterious effects in muscle cells such as atrophy and 16

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inflammation. These observations are probably linked to differences in the lineage specificity and tissular environment characterizing these two depots (1,2,37,38).

Coculture systems have been developed during the last decade to study the crosstalk between adipose tissue and muscle, particularly focusing on the impact of adipocytes on muscle insulin resistance and glucose uptake. Cocultures of human in vitro differentiated preadipocytes with skeletal muscle cells induce the insulin resistance of the latters (19) with impact on oxidative metabolism (20). Here we have used mature (i.e. unilocular) adipocytes isolated from human subjects. These cells display high metabolic (lipolytic activity) and secretory (production of leptin and adiponectin) functions when compared to in vitro differentiated preadipocytes. We believe that the results obtained using this 3D setting with mature adipocytes may be more patho physiologically relevant than previous studies using 2D coculture with primary human myoblasts (18–20,39). In addition this system allowed a longterm survey of muscle cells functions addressing the original question of the impact of obese VAT adipocytes on inflammation and myotubes structural organization.

Our results reveal that cocultures of obese VAT adipocytes with muscle cells enhanced the pro inflammatory environment with increased production of several cytokines (IL6 and GCSF) and chemokines (CCL2, IL8, CXCL1/2/3, CCL7 and Fractalkine). More importantly, we have clearly identified IL6 and IL1β playing key roles in this inflammatory loop. These cytokines which act in concert in various inflammatory processes are overproduced by adipose tissue during obesity and type 2 diabetes (40,41).

Muscles cells are characterized by their contractile capacity, a phenomenon depending of tissue physical properties. The hydrogel used in the present study is soft (~300 Pa, compared to skeletal

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muscle or culture well plates (~12kPa and ~100 kPa respectively, (42)), limiting the analysis of

myotubes contractility in our 3D cocultures. However, in this 3 D model maintaining adipocyte

viability, we were able to demonstrate a major role of obese VAT adipocytes on inducing muscle

cell atrophy phenotype. The myogenic program is controlled by transcription factors such as

MyoD and myogenin. Binding of these factors to specific sequences in the promoter of muscle

specific genes increases the expression of contractile proteins such as myosin heavy chain,

, troponin (43,44) and titin, which maintains integrity (45). At the

molecular level, we observed the downregulation of MyoD, myogenin, and

contractile/sarcomeric proteins (titin) and the dystrophin complex in myotubes cocultured with

the obese VAT adipocytes. These data provide new insights about which proteins are

preferentially targeted and participate to the atrophic phenotype of the muscle cells when co

cultured in this 3D setting.

In addition to the decreased gene expression of muscle specific structural proteins, we also

observed a decreased expression of the growth/myogenic factor IGFII and its binding partner

IGFBP5 (35). IGFII shares with IGFI the same receptor and protein signaling pathway

(Akt/mTOR/p70S6) which mediates hypertrophy in skeletal muscle (21). Importantly, the acute

activation by IGFII/IGFBP5 of this pathway, in particular Akt phosphorylation, was altered in

myotubes exposed to obese VAT adipocytes, which could be responsible for the observed muscle

cell atrophy phenotype. This is supported by the observations that chronic treatment by IGFII

and IGFBP5 were able to correct the muscle atrophy phenotype and restore titin and myosin

heavy chain production in the cocultured myotubes while having a minor influence on myotube

phenotype alone in control conditions.

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Finally, the pathophysiological relevance of our results was further supported in a rodent model of nutritionally induced obesity. Obese mice displayed abnormal distribution of crosssection muscle fiber size, adipocyte accumulation and decreased muscle markers expression.

Interestingly, expression of the muscle markers (i.e. MyoD, βActin, Titin, IGFII and IGFBP5) were negatively correlated with inflammation and epididymal fat expansion. We cannot exclude in our in vivo model, an additional role of the liver in promoting muscle dysfunctions. According to the “portal hypothesis”, the liver is directly exposed to VATderived factor which may induce inflammation, causing then skeletal muscle defects such as insulin resistance (46). Moreover, correlative observations made in skeletal muscle need further studies to firmly establish a potential role of intermuscular adipocytes in muscle dysfunctions.. Several clinical studies suggest that musclederived adipocytes through secretion of bioactive factors could play a role in muscle deteriorations by inducing insulinresistance and negatively affecting myogenesis and muscle performance (13,31,47). Moreover, a recent report showed a potential link between obesityinduced lipotoxicity and muscle insulin resistance through disruption of 4EBP1 phosphorylation and protein synthesis (48). Consequently, future goal will be to compare both

VAT and intermuscular adipocytes phenotypes; the latter still remains unknown.

To conclude, we provide evidence for a crosstalk between human obese adipocytes and muscle cells leading to muscle atrophy (Figure 6). The subset of underexpressed musclespecific genes could constitute a molecular signature of muscle damage induced by hypertrophy of obese WAT.

The identification of negative (IL6/IL1β) and beneficial (IGFII/IGFBP5) molecular actors involved in these dysfunctions open new avenues for therapeutic strategies of myopathies associated with metabolic disorders.

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Author contributions

VP, SRC and DL conceived and performed the experiments and analyzed the data.

CR performed and analyzed multiplex and PCR array experiments.

FEV and VA performed muscle cell culture.

KC contributed to clinical investigation and patients recruitment.

VP, KC, SRC, AVP, GBB and DL wrote the manuscript with the input of all the coauthors.

GBB and DL are the guarantors of this work, such as, had the full access to all data in the study

and take the responsibility for the integrity of the data and the accuracy of the data analysis.

Acknowledgments

The authors are very grateful to Dr Peter Van Der Ven (Institut for Cell Biology, Bonn,

Germany) for providing us the antihuman titin monoclonal antibody. We acknowledge patients,

the physician Dr Christine Poitou of the Nutrition Department of Pitié Salpétrière (Paris, France)

for patients’ recruitment. We also thank Christophe Klein from imaging facilities of Centre de

Recherches des Cordeliers. For cellular studies, ethical authorization was obtained from CPP

Pitié Salpêtrière. Human adipose tissues pieces were obtained thanks to Clinical Research

Contract (Assistance Publique/Direction de la Recherche Clinique AOR 02076).

Fundings

This work was supported by a grant from the European Community seventh framework program,

Adipokines as Drug to combat Adverse Effects of Excess Adipose tissue project (contract

number HEALTHFP22008201100), APHP Clinical Research Contract, Emergence program,

University Pierre et Marie Curie (to VP), Région Ile de France (FUIOSEO Sarcob), the

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Fondation pour la Recherche Médicale, grant number “DEQ20120323701” (to KC) as well as

French National Agency of Research (French government grant, Investments for the Future”; grant no. ANR10IAHU, ANR AdipoFib). This work was also financed by the EU FP7

Programme project MYOAGE (contract HEALTHF22009223576), the ANR Genopath

INAFIB, the AFLD, the CNRS and the AFM (Association Française contre les Myopathies).

SRC and AVP are supported by MRC and BHF programme grants.

Disclosure statement:

No conflicts of interest to this article are reported.

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31. Hilton TN, Tuttle LJ, Bohnert KL, Mueller MJ, Sinacore DR. Excessive adipose tissue infiltration in skeletal muscle in individuals with obesity, diabetes mellitus, and peripheral neuropathy: association with performance and function. Phys Ther. 2008 Nov;88(11):1336–44. 32. Dalmas E, Rouault C, Abdennour M, Rovere C, Rizkalla S, BarHen A, et al. Variations in circulating inflammatory factors are related to changes in calorie and carbohydrate intakes early in the course of surgeryinduced weight reduction. Am J Clin Nutr. 2011 Aug;94(2):450–8. 33. Spranger J, Kroke A, Möhlig M, Hoffmann K, Bergmann MM, Ristow M, et al. Inflammatory cytokines and the risk to develop type 2 diabetes: results of the prospective populationbased European Prospective Investigation into Cancer and Nutrition (EPIC)Potsdam Study. Diabetes. 2003 Mar;52(3):812–7. 34. Stapp JM, Sjoelund V, Lassiter HA, Feldhoff RC, Feldhoff PW. Recombinant rat IL 1beta and IL6 synergistically enhance C3 mRNA levels and complement component C3 secretion by H35 rat hepatoma cells. Cytokine. 2005 Apr 21;30(2):78–85. 35. Ren H, Yin P, Duan C. IGFBP5 regulates muscle cell differentiation by binding to IGFII and switching on the IGFII autoregulation loop. J Cell Biol. 2008 Sep 8;182(5):979–91. 36. Glass DJ. Skeletal muscle hypertrophy and atrophy signaling pathways. Int J Biochem Cell Biol. 2005 Oct;37(10):1974–84. 37. Chau YY, Bandiera R, Serrels A, MartínezEstrada OM, Qing W, Lee M, et al. Visceral and subcutaneous fat have different origins and evidence supports a mesothelial source. Nat Cell Biol. 2014 Apr;16(4):367–75. 38. Macotela Y, Emanuelli B, Mori MA, Gesta S, Schulz TJ, Tseng YH, et al. Intrinsic differences in adipocyte precursor cells from different white fat depots. Diabetes. 2012 Jul;61(7):1691–9. 39. Yu J, Shi L, Wang H, Bilan PJ, Yao Z, Samaan MC, et al. Conditioned medium from hypoxiatreated adipocytes renders muscle cells insulin resistant. Eur J Cell Biol. 2011 Dec;90(12):100015. 40. Kern PA, Ranganathan S, Li C, Wood L, Ranganathan G. Adipose tissue and interleukin6 expression in human obesity and insulin resistance. Am J Physiol Endocrinol Metab. 2001 May;280(5):E745–51. 41. Moschen AR, Molnar C, Enrich B, Geiger S, Ebenbichler CF, Tilg H. Adipose and liver expression of interleukin (IL)1 family members in morbid obesity and effects of weight loss. Mol Med. 2011;17(78):840–5. 42. Buxboim A, Ivanovska IL, Discher DE. Matrix elasticity, cytoskeletal forces and physics of the nucleus: how deeply do cells “feel” outside and in? J Cell Sci. 2010 Feb 1;123(Pt 3):297– 308. 43. Berkes CA, Tapscott SJ. MyoD and the transcriptional control of myogenesis. Semin Cell Dev Biol. 2005 Oct;16(45):585–95. 44. Le Grand F, Rudnicki MA. Skeletal muscle satellite cells and adult myogenesis. Curr Opin Cell Biol. 2007 Dec;19(6):628–33.

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45. Gautel M. Cytoskeletal protein : titin and its relations in mechanosensing. Pflugers Arch. 2011 Jul;462(1):119–34. 46. Cai D, Yuan M, Frantz DF, Melendez PA, Hansen L, Lee J, et al. Local and systemic insulin resistance resulting from hepatic activation of IKKbeta and NFkappaB. Nat Med. 2005 Feb;11(2):183–90. 47. Takegahara Y, Yamanouchi K, Nakamura K, Nakano SI, Nishihara M. Myotube formation is affected by adipogenic lineage cells in a celltocell contactindependent manner. Exp Cell Res. 2014 May 15;324(1):105–14. 48. Stephens FB, Chee C, Wall BT, Murton AJ, Shannon CE, van Loon LJC, et al. Lipid induced insulin resistance is associated with an impaired skeletal muscle protein synthetic response to amino acid ingestion in healthy young men. Diabetes. 2014 Dec 18;

LEGENDS TO FIGURES

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Figure 1: Human adipocyte secretions and myotube phenotype

A. Human differentiated muscle cells were cultured in the presence of lean SAT CM or obese

VAT CM for 5 days. Cells were incubated with an antibody specific for desmin, revealed using

an Alexa488 coupled goat anti mouse secondary antibody (green). Nuclei were visualized with

Hoechst staining (blue). A representative photomicrograph is presented. Scale bar=200 µm.

B. Myotube thickness was quantified using Image J software measuring intensity of the desmin

staining in ten random fields (X20) in three independent cultures.

C. Fusion index: the number of nuclei in differentiated myotubes (>2 myonuclei) were calculated

as a percentage of the total number of nuclei (mononucleated and plurinucleated). 1000 nuclei per

dish were counted in three independent cultures.

The counting was performed with blind lectures by different investigators (VA and FEV).

D. Cell lysates were immunoblotted to detect the heavy chain of myosin II (MF20, 200kDa),

troponin (24 kDa) and emerin (37 kDa, used as normalization control). Graphs represent the

quantification of the immunoblots.

Data are mean ± SEM of 5 independent experiments.

*p<0.05, **p<0.01, control vs. lean SAT CM or obese VAT CM.

Figure 2: Gene expression profile of myocytes cocultured with human adipocytes

Muscle cells were differentiated in the 3D hydrogel for 3 days and then VAT adipocytes in the

3D hydrogel were added for an additional period of 1 day. Cells were then collected for RNA

extraction to obtain after reverse transcription cDNAs.

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A. cDNAs were analyzed using “Human myogenesis and myopathy PCR array”.

Graph represents the percentage of decreased gene expression in myocytes cocultured with adipocytes (MYO+AD) compared to myocytes alone (control, MYO).

Data are presented as means ± SEM of 5 independent experiments.

**p<0.01, ***p<0.001 MYO+AD vs MYO

BC Cells were also fixed and stained using antibody directed against titin (green, Alexa488 conjugated antimouse IgG) and phalloidin (red, Alexa546 phalloidin). A representative photomicrograph from confocal microscopy is presented. (Figure 2C, scale bar = 20 µm and D, scale bar = 10 µm).

DF. Muscle cells were differentiated for 3 days and then 3D adipocytes from SAT or VAT from paired biopsies of obese subjects were added for an additional period of 3 days. Cells were fixed and stained using antibody directed against desmin, troponin or titin (green, Alexa488conjugated antimouse IgG). Graphs represent quantifications of myotube thickness (D), troponin staining

(E) and titin staining (F).

Data are the means ± SEM of 68 independent experiments.

*p<0.05 MYO+VAT AD vs MYO

Figure 3: Inflammatory profile of cocultured human adipocytes and myocytes: IL6 and

IL1βββ as key factors.

Muscle cells were differentiated in the 3D hydrogel for 3 days and then SAT or VAT adipocytes from obese subjects, cultured in the 3D hydrogel were added for an additional period of 3 days.

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Media were then collected for the measurement of cytokine and chemokine secretion by a

multiplex assay.

ASignificant changes in the secretory profile in the 3D cultures of muscle cells and obese VAT

adipocytes (AD+MYO) compared to AD as control and MYO are presented in the graph. Data

are expressed as fold variations between AD (white bars), AD+MYO (black bars) and MYO

(grey bars) to take into account human interindividual variations.

Data are presented as means ± SEM of 7 independent experiments.

*p<0.05, **p< 0.01 AD+MYO vs AD.

B Significant changes in the secretory profile in the 3D cultures of muscle cells and obese SAT

(MYO+ SAT AD) or obese VAT adipocytes (MYO+ VAT AD) are presented in the graph. Data

are expressed as fold variations between MYO (control) and MYO+ SAT AD (white bars) or

MYO+ VAT AD (black bars).

Data are presented as means ± SEM of 8 independent experiments.

C. Gene expression of IL6, IL8 and IL1β in muscle cells cultured alone (control, MYO, white

bars) or exposed to adipocytes from paired biopsies of obese SAT (MYO+ SAT AD, grey bars)

or VAT adipocytes (MYO+ VAT AD, black bars), estimated by real time PCR. Data (fold over

control, MYO) are presented as means ± SEM of 6 independent experiments performed with

different adipocytes preparations.

*p<0.05, **p<0.01 MYO+ VAT AD vs MYO.

#p<0.05 MYO+ SAT AD vs. MYO+ VAT AD

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D. 3D VAT adipocytes were added for an additional period of 3 days treated with neutralizing antibodies of IL6 (2.5 µg/ml) and IL1β (0.5 µg/ml) (MYO+AD+ abIL6/IL1β) IgG1 (control

MYO+AD IgG) in the 3D setting. Media were then collected for multiplex assay. Black bars represent the percentage decrease in secretions in MYO+AD+ abIL6/IL1β cocultures compared to control MYO+AD IgG cocultures.

Data are presented as means ± SEM of 5 independent experiments.

* P<0.05, **p<0.01, ***p<0.001, ns nonsignificant MYO+AD IgG vs. MYO+AD+ abIL6/IL

E. Multiplex assay of the inflammatory secretome of myocytes treated (MYO IL6/IL1β, black bars) or not (MYO, white bars) with recombinant IL6 (10 ng/ml) and IL1β (1 ng/ml) during 3 days.

Data are presented as means ± SEM of 5 independent experiments.

* P<0.05, **p<0.01, ***p<0.001 MYO IL6/IL1β vs. MYO

Figure 4: IGFII and IGFBP5 rescue the atrophy of myotubes induced by adipocytes

AD. Muscle cells were differentiated in the 3D hydrogel for 3 days without (MYO) or with 3D

VAT adipocytes (from obese subjects) (MYO+AD) were added for an additional period of 3 days in the presence or not of IGFII (50ng/ml) and IGFBP5 (200ng/ml) (MYO+AD+IGFII/IGFBP

5. Cells were fixed and stained with the corresponding antibodies.

A. Myotube thickness was quantified using Image J software measuring intensity of the desmin staining in ten random fields (X20) in 6 independent cultures.

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BD. Quantification of immunofluorescence was performed using Image J software measuring

intensity of the MF20 (B) and titin (C) staining in five random fields (X20).

D. Immunostaining of titin (green, Alexa488conjugated antimouse IgG) and nuclei (blue,

DAPI). A representative photomicrograph of titin staining is presented (scale bar = 50 µm).

Data are presented as means ± SEM of 6 independent experiments.

** p<0.01, MYO+AD vs MYO or MYO+AD+IGFII/IGFBP5.

EF. Differentiated muscle cells exposed or not to obese VAT adipocytes were stimulated with

IGFII (50 ng/ml) and IGFBP5 (200 ng/ml) for 10 min at 37°C. Serine 473 phosphorylation of

Akt (pS 473 Akt, 56kDa), Serine 65 of 4EBP1 (pS65 4EBP1, 21 kDa) and Serine 240 and 244

phosphorylation of S6 ribosomal protein (pS 240/244 S6 ribosomal protein, 32 kDa) were

detected using the corresponding antibodies. A representative Western blot is presented among

the five different experiments (E). The graph represents quantifications of the immunoblots in

IGFII/IGFBP5stimulated conditions normalized to total proteins (F).

Data are presented as means ± SEM of 57 independent experiments.

* p<0.05, **p<0.01, MYO+AD vs. MYO

#p<0.05, ##p<0.01, vs. + IGFII/IGFBP5 treatment.

Figure 5: Skeletal muscle characteristics of HFDinduced obese mice

AB. Serum cytokine level (A) and Gastrocnemius gene expression (B) evaluated in 20weekold

mice after 12 weeks of standard diet (chow, white bars) or High Fat Diet (HFD, black bars).

n=8 mice per experimental group

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CE. Histological analysis of the Gastrocnemius muscle obtained from 24weekold mice (n=9 mice per experimental group) after 16 weeks of standard diet (chow) or High Fat Diet (HFD).

C. Representative microphotographs of crosssection (right panel) and longitudinal section (left panel). Scale bar=100m.

D. Measurement of variance coefficients of the fiber size in the crosssection samples of Chow and HFD mice using Feret’s diameter as geometrical parameter. n=61 (minimum)–201

(maximum) fibers were analyzed for each sample (3 random fields/mouse, X10).

E. Quantification of adipocyte spots in the longitudinal section samples of chow and HFD mice

(total biopsy).

*p<0.05, ***p<0.001, ns, nonsignificant, chow vs. HFD mice.

Figure 6: Scheme representing the proposed crosstalk between obese adipocytes and muscle cells which could trigger inflammation and muscle dysfunctions.

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Table 1. Spearman correlations between the gene expression of muscle markers in the skeletal muscle (Gastrocnemius) and the expression of leptin/IL6 in both epididymal AT and skeletal muscle.

Epididymal Adipose Tissue Gastrocnemius

Fat mass % Leptin IL6 Leptin IL6

Gastrocnemius R p R p R p R p R p

MyoD 0.729 0.001 0.747 0.001 0.708 0.001 0.618 0.006 0.427 0.051

MyoG 0.279 0.147 0.053 0.424 0.030 0.454 0.053 0.424 0.009 0.489

βActin 0.415 0.056 0.788 0.000 0.720 0.001 0.435 0.047 0.271 0.155

Titin 0.5357 0.0396 0.4107 0.1283 0.6452 0.0094 0.3429 0.2109 0.1679 0.5499

IGFII 0.577 0.011 0.744 0.001 0.664 0.003 0.588 0.009 0.453 0.040 IGFBP5 0.6264 0.0165 0.6791 0.0076 0.6865 0.0067 0.5121 0.0612 0.4549 0.1022

PGC1α 0.068 0.410 0.152 0.303 0.213 0.230 0.020 0.476 0.160 0.292

PGC1β 0.350 0.101 0.646 0.006 0.459 0.042 0.332 0.113 0.418 0.061

20weekold mice (n=16).

Significant correlations are indicated in bold

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Diabetes Page 34 of 58 Figure 1

A

200µm control Lean SAT CM Obese VAT CM * B * C (fold (fold control) over Myotube thickness Myotube thickness

Control Lean SAT CM Obese VAT CM Control Lean SAT CM Obese VAT CM

D Control Lean SAT CM Obese VAT CM MF20 188 kDa Troponin 18 kDa

Emerin 38 kDa

** * *

Control Lean SAT CM Obese VAT CM Control Lean SAT CM Obese VAT CM PageFigure 35 of 58 2 Diabetes

0 A -20

-40

-60 MYO+AD MYO+AD ** **

(% control, MYO) (% control, ** -80 *** *** *** ** *** *** *** mRNA relative expression in expression mRNA relative -100

Phalloidin Titin Merge B C Titin MYO MYO MYO+AD MYO+AD MYO+AD MYO+AD

D E * * * *

F

* MYO MYO+SAT AD MYO+ VAT AD * titin Figure 3 Diabetes Page 36 of 58 AD MYO+ SAT AD A B # 125 * AD+MYO 1480 * MYO+VAT AD MYO # 75 * * 25 780 # ** # # * * 80 70 * ** 60 * * * 50 40 ** * ** 30 * * *

Secretion (fold over control, MYO) control, over (fold Secretion 20 Secretion (fold over control, AD) control, over (fold Secretion 10 * * * *

*

0 α α -8 -8 -6 -8 -8 -6

FRK FRK 1/2/3 FRK IL IL IL IL CCL7 FGF2 FGF2 CCL7 CCL7 FGF2 FGF2 VEGF VEGF CCL2 VEGF VEGF CCL2 G-CSF G-CSF G-CSF G-CSF MIP1 MIP1 CXCL

C α

D 1/2/3

MYO 10 -

-7 -8 β G-CSF FRK CXCL CCL7 IL IL IP CCL2 MIP1 CCL5 VEGF

MYO+SAT AD 1 - 0

* MYO+VAT AD )

 6/IL - IgG -15

-30 * ns  ns ** -45 ns  in AD+MYO ab IL ab AD+MYO in -60 * ** * (% control, AD+MYO control, (% -75 ** ** * IL-6 IL-8 IL-1 *** Secretion *** -90 E 19200 MYO *** *** MYO IL-6/IL-1β

13200 *** **

/ml) 7200 ***

**

pg ** ( 1200 600 *

Secretion ** 300 **

0 ** G-CSF FRK CXCL CCL7 IL-7 IL-8 IP-10 CCL2 MIP-1α CCL5 VEGF 1/2/3 PageFigure 37 of 58 4 Diabetes

A B C ** ** ** ** ** ** Myotube thickness Myotube thickness (fold over control, (fold control, over MYO) MYO MYO+AD MYO+AD MYO MYO+AD MYO+AD MYO MYO+AD MYO+AD +IGF-II/IGFBP-5 +IGF-II/IGFBP-5 +IGF-II/IGFBP-5 D Titin, nuclei MYO MYO+AD MYO+AD+IGF-II/IGFBP-5

50µm

E F MYO MYO+AD ** ** IGF-II/IGFBP-5 - + - + ** # ** pS473 AKT ## 49 kDa **

Total AKT 49 kDa - + - + IGF-II/IGFBP-5 65 - + - + pS 4E-BP1 IGF-II/IGFBP-5 17 kDa MYO * Total 4E-BP1 * MYO+AD 17 kDa

pS240/244 S6 28 kDa

Total S6 28 kDa - + - + IGF-II/IGFBP-5 Diabetes Page 38 of 58 Figure 5

A B

Chow 160.0 Chow HFD 100 HFD *

50 20 /ml) 60.0 40.0

pg 18 ( * * 16 30.0 14 12 20.0 10 Serum level Serum level 8

mRNA relative expression (A.U) expression mRNA relative 6 10.0 *** *** *** 4 *** * 2 0.0 0 IL-6 IL-1β TNFα CXCL IL-10 MyoD MyoG IGF-II β-Actin PGC1α PGC1β Titin IGFBP-5 1/2/3

C D Transverse Section Longitudinal Section 40 Chow *** HFD 30 20 Chow * 10

CV feret diameter (%) diameter CV feret 0 E 6 Chow * HFD 4 HFD * 2 * * * Adipocyte spots Adipocyte 0 PageFigure 39 of 58 6 Diabetes

IGF-II/IGFBP-5

Adipocyte Muscle cell

ATROPHIC PHENOTYPE (decreased myogenic and contractile protein expression)

Adipokines secretion

INFLAMMATION INFLAMMATION

IL-6/IL-1β

Cytokines/chemokines production

Recruitment/activation of immune cells Diabetes Page 40 of 58

SUPPLEMENTARY DATA

Human adipocytes induce inflammation and atrophy in muscle cells during obesity

Pellegrinelli V1, 2, 3#, Rouault C1, 2, 3*, RodriguezCuenca S4*, Albert V1, 2, 3, Edom Vovard F5, 6, 7, 8 , VidalPuig A4, Clément K 1, 2, 3 **, ButlerBrowne GS5, 6, 7, 8** and Lacasa D1, 2, 3 ** 1 INSERM, U1166 Nutriomique, Paris, F75006 France; 2 Sorbonne Universités, University Pierre et MarieCurieParis 6, UMR S 1166, Paris, F75006 France; 3 Institut Cardiométabolisme et Nutrition, ICAN), Pitié Salpétrière Hospital, Paris, F 75013 France;

4 Metabolic Research Laboratories, Wellcome TrustMRC Institute of Metabolic Science, University of Cambridge, Cambridge, United Kingdom;

5 Sorbonne Universités, University Pierre et MarieCurieParis6, Centre de recherches en Myologie UMR 974, F75005, Paris, France;

6 INSERM, U974, 75013, Paris, France;

7 CNRS FRE 3617, 75013, Paris, France;

8 Institut de Myologie, 75013, Paris, France.

# Corresponding author: Current address: Wellcome TrustMRC Institute of Metabolic Science, Metabolic Research Laboratories,University of Cambridge

This file includes: 9 supplementary figures 5 supplementary tables 1 supplementary list Supplemental references

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Figure S1

Secretory profile of human SAT and VAT adipocytes.

ASecretory profile of 48hconditioned media prepared from 3D human adipocytes from lean SAT (lean SAT CM) and obese SAT and VAT (obese SAT CM and obese VAT CM, respectively). Heatmap represents the secretory profile. Graded shades from green to red represent the secretion levels (pg/ml). Cytokines and chemokines were classified from high to low secretion.

BSecretory profile of 48hconditioned media prepared from human adipocytes from paired samples of obese SAT (obese SAT CM, white bars) and VAT (obese VAT CM, black bars). Results are expressed as fold variation over control, obese SAT CM.

The amounts of adipokines were determined by ELISA (leptin and adiponectin) and multiplex (cytokines/chemokines) analysis in 1014 experiments using different adipocyte preparations.

*p<0.05, ** p<0.01 obese VAT CM vs. obese SAT CM (B)

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Figure S2

Human SAT and VAT

Experimental procedure for 3D coculture of human adipocytes and myoblasts

A Muscle cells were embedded in hydrogel at the concentration of 200.000 cells per 100µl of gel preparation containing 0.5 mg/ml type 1collagen. The gel preparation was put into 96well plates containing 150µl of growth medium. After 2 days of culture, this medium was replaced by differentiating medium for 3 days. A second hydrogel containing mature adipocytes isolated from SAT or VAT of obese subjects (10.000 cells per 100µl of gel preparation) was then added. Adipocytes and differentiated myoblasts were then cocultured for 3 days in DMEM/F12 (1% antibiotics and 50 nM insulin). Culture medium was changed daily.

B Microphotographs showing the hydrogel containing the mature adipocytes (AD) and/or the differentiated myoblastes (MYO).

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C Immunofluorescence analysis of differentiated myoblastes cultured in 2D or 3D conditions using antibodies against MF20 (green, Cy2conjugated antimouse IgG), actin (red, Cy3phalloidin). Nuclei were stained with DAPI (blue).

Figure S3

ATF4

HSPA5

C/EBPζζζ

Gene expression of ER stress markers in cocultured myocytes and VAT adipocytes.

Muscle cells were differentiated in the 3D hydrogel for 3 days and then 3D adipocytes (from VAT of obese subjects) were added for an additional period of 1 day. Cells were then collected for RNA extraction. After reverse transcription of total RNAs, cDNAs were analyzed for ER stress markers.

Graph represents gene expression in myocytes cocultured with adipocytes (black bars, AD+MYO) compared to adipocytes alone (white bars, AD) and myocytes alone (grey bars, MYO).

Data (fold over control, AD) are presented as means ± SEM of 5 independent experiments.

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Figure S4

A B

Cytotoxicity and inflammation in cocultures and impact on myotubes thickness.

AMuscle cells were differentiated in the 3D hydrogel for 3 days and then adipocytes (from VAT of obese subjects) in another 3D hydrogel were added for an additional period of 1 day. Cytotoxicity was evaluated by measuring the activity of lactate dehydrogenase (LDH) released from damaged cells. Graph represents LDH activity in differentiated myocytes cocultured with adipocytes (black bars, AD+MYO) compared to adipocytes alone (white bars, AD) or differentiated myocytes alone (grey bars, MYO).

Data (fold over control, AD) are presented as means ± SEM of 5 independent experiments.

B Muscle cells were differentiated in the 3D hydrogel for 3 days and then inflammatory adipocytes in the 3D hydrogel were added for an additional period of 3 days. Media were then collected for the measurement of cytokine and chemokine secretion by a multiplex assay. Heatmap represents the secretory profile. Graded shades from green to red represent the secretion levels (pg/ml). Cytokines and chemokines were classified from high to low secretion. AD was used as control.

Data are presented as means ± SEM of 7 independent experiments.

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Figure S5

A.

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B.

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Muscle cells atrophy and inflammatory mediators IL6/IL1βββ.

A Muscle cells differentiated for 3 days were treated by IL6 (10 ng/ml) and IL1β (1 ng/ml) during 3 days. Cells were fixed and stained for desmin (thickness measurement) or titin (green, alexa488 conjugated antimouse IgG) and nuclei (blue, DAPI staining). Media were collected for IGFII ELISA assay.

Graph represents IGFII secretion, myotube thickness and titin staining in muscle cells (white bar, MYO) compared to muscle treated by recombinant IL6 and IL1β (black bar, MYO+IL6/IL1β).

B Muscle cells were differentiated for 3 days and then 3D adipocytes (from VAT of obese subjects) were added for an additional period of 3 days treated with IL6 and IL1β neutralizing antibody (MYO+AD+Neut Ab) compared to cocultures treated with IgG1 (control MYO+AD) in the 3D setting. Cells were fixed and stained for desmin (thickness measurement) or titin (green, alexa488 conjugated antimouse IgG) and nuclei (blue, DAPI staining). Media were collected and analyzed for IGFII ELISA assay.

Graph represents myotube thickness and titin staining in muscle cells (MYO, white bar) or cocultured with VAT adipocytes (black bar, MYO+AD) compared to myocytes cocultured with VAT adipocytes in the presence of neutralizing antibodies (grey bar, MYO+AD+Neut Ab).

Data are presented as means ± SEM of 48 independent experiments.

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Figure S6

Cell thickness, titin and troponin content in muscle cells treated by IgFII/IGFBP5

Muscle cells were differentiated in the 3D hydrogel for 3 days before 3 additional days of treatment with (MYO+IGFII/IGFBP5) or without (MYO) IGFII (50ng/ml) and IGFBP5 (200ng/ml). Cells were fixed and stained with the corresponding antibodies. Immunostaining of titin (green, Alexa488 conjugated antimouse IgG), troponin (red, Alexa555conjugated antimouse IgG) and nuclei (blue, DAPI) were performed. Representative photomicrographs are presented (scale bar = 50 µm). Myotube thickness was quantified using Image J software in five random fields in 6 independent cultures. Quantification of immunofluorescence was performed using Image J software measuring intensity of the titin and troponin staining in five random fields in 6 independent cultures. Data are presented as means ± SEM of 6 independent experiments.

* p<0.05, MYO vs MYO+IGFII/IGFBP5.

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Figure S7

G-CSF Fractalkine CXCL CCL7 IL-6 IL-8 CCL2 MIP-1VEGF

Inflammatory secretome of cocultured muscle cells treated by IGFII/IGFBP5.

Muscle cells were differentiated for 3 days and then 3D adipocytes (from VAT of obese subjects) were added for an additional period of 3 days treated by IGFII (50 ng/ml) and IGFBP5 (200 ng/ml) or not . Media were then collected and analyzed for multiplex assay.

Graph represents the percentage variation in secretions in differentiated myocytes cocultured with adipocytes in the presence of IGFII (50 ng/ml) and IGFBP5 (200 ng/ml) (black bars, % of MYO+AD) compared to control coculture of myocytes and adipocytes (MYO+AD).

Data are presented as means ± SEM of 6 independent experiments.

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Figure S8

Atrogin-1 Murf-1

Gene expression of atrophic markers in muscle cells exposed to VAT adipocytes.

Differentiated muscle cells were exposed to 3D adipocytes (from VAT of obese subjects) with or without IGF1 (10 ng/ml) or with IGFII (50 ng/ml) and IGFBP5 (200 ng/ml). Gene expression of atrogin1 and murf1 was estimated by real time PCR and normalized to 18S. The graph represents gene expression in myocytes cocultured with adipocytes (black bars, MYO+AD) compared to myocytes alone (white bars, MYO) with IGFI (dark grey bars, MYO+AD+IGFI) or IGFII/IGFBP5 (light grey, MYO+AD+IGFII/IGFBP5).

Data (fold over control, MYO) are presented as means ± SEM of 6 independent experiments performed with different adipocytes preparations.

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Figure S9 A

B

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C

Gene expression profile of muscle, adipose and inflammatory markers in skeletal muscle of obese mice

AGene expression profile in subcutaneous (SAT) and epididymal (GnAT) adipose tissue from 20 weekold mice after 12 weeks of standard diet (chow, white bars) or High Fat Diet (HFD, black bars). n=8 mice per experimental group *p<0.05, **p<0.01, ***p<0.001, ns, nonsignificant, chow vs HFD mice in SAT and GnAT ###p<0.001, SAT vs GnAT during HFD

BGene expression in the Gastrocnemius of 24weekold mice after 16 weeks of standard diet (chow, white bars) or High Fat Diet (HFD, black bars). n=9 mice per experimental group *p<0.05, **p<0.01, ***p<0.001, ns, nonsignificant, chow vs HFD mice

CGene expression of adipose markers in the Gastrocnemius of 20weekold mice after 12 weeks of standard diet (chow, white bars) or High Fat Diet (HFD, black bars). n=8 mice per experimental group

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Supplementary tables

Table S1. List of the antibodies, recombinant proteins and ELISA kits used in the study

Antibody Reference Provider

antihuman MF20 antibody MAB4470 Developmental Hybridoma Bank, Iowa City, IW, USA

antihuman emerin antibody NCLemerin Leica, Newcastle, UK

antihuman desmin monoclonal antibody M076 DAKO, Glostrup, Dk

antihuman troponin monoclonal antibody T6277 Sigma, St Louis, Mo, USA

rabbit antihuman phosphoAKT (Ser473) mAb 4060 Cell Signaling Technology, Denvers, CO, USA

rabbit antihuman totalAKT mAb 9272 Cell Signaling

rabbit antihuman phospho4EBP1(Ser65) mAb174A9 Cell Signaling

rabbit antihuman total4EBP1 mAb 53H11 Cell Signaling

rabbit antihuman phospho S6 ribosomal protein mAb D68F8 Cell Signaling (Ser240/244) rabbit antihuman total S6 ribosomal protein mAb 5G10 Cell Signaling

antirabbit IgG HRPlinked antibody P7074 Cell Signaling

mouse anti human IL6 neutralizing monoclonal MAB206 R&D Systems antibody mouse anti human IL1β neutralizing monoclonal MAB201 R&D Systems antibody Alexa 488conjugated antimouse antibody A21202 Life Technologies, Foster City, CA, USA

Alexa 546conjugated antirabbit antibody A22283 Life Technologies

phalloidin conjugate alexa®568 A22283 Molecular Probes, Eugene, Or, USA

antimouse conjugate alexa®488 A21202 Molecular Probes, Eugene, Or, USA

human recombinant protein IGFII 292G2050 R&D Systems

human recombinant protein IGFBP5 875B5025 R&D Systems

human recombinant protein IL6 20006 Peprotech ,Rocky Hill, NJ, USA

human recombinant protein IL1β 20001 Peprotech

human Insulin I91077C Sigma

ELISA assay for human IGFII CSB CUSABIO , Wuhan, P.R. China E04583h

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Table S2. Clinical parameters of obese subjects

Obese subjects Number of 41 subjects Age (years) 41.6 ±1.5

BMI (kg/m2) 47.74 ±1.2***

% of fat mass 43.22 ±5.32

% of lean mass 48.34±1.23 Adipocyte 800.7 ±94.6 volume (pL) Glycaemia 4.66 ±0.57 (mmol/L)

Insulinemia 15.39 ±2.65 (µU/mL) HOMAIR 3.75 ±0.65

Leptin 63.17 ±12.56 (ng/ml) Adiponectin 4.52 ±0.87 ( µg/mL) IL6 2.93±0.53 (pg/ml)

The obese subjects were not on a diet and their weights were stable before surgery. Regarding lean subjects, none had diabetes and metabolic disorders and none was taking medication. Informed personal consent was obtained from every participant. In obese subjects, body composition was estimated by wholebody fanbeam dual energy Xray absorptiometry scanning (Hologic Discovery W, software v12.6, 2; Hologic, Bedford, MA) (1). To determine body fat and lean mass repartition, we used specific measures and analyses as described (2). Blood samples were collected after an overnight fast of 12 h. Glycaemia was measured enzymatically. Serum insulin concentrations were measured using a commercial IRMA kit (Biinsuline IRMA CisBio International, France). Serum leptin and adiponectin were determined using a radioimmunoassay kit (Linco Research, St. Louis, MO), according to the manufacturer's recommendations, sensitivity: 0.5 ng/ml and 0.8 ng/ml for leptin and adiponectin respectively.

Data are presented as means ± SEM (standard error of the mean).

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Table S3

List of primer sequences used for realtime PCR

Gene Forward Reverse

h ATF4 ggtcagtccctccaacaaca ctatacccaacagggcatcc

h C/EBPζ aaggcactgagcgtatcatgt tgaagatacacttccttcttgaaca

h HSPA5 agctgtagcgtatggtgctg aaggggacatacatcaagcagt

h IGFII gctggcagaggagtgtcc gattcccattggtgtctgga

h IGFBP5 agagctaccgcgagcaagt gtaggtctcctcggccatct

h irisin tcgtggtcctgttcatgtg ggttcattgtccttgatgatgtc

h PGCα tgagagggccaagcaaag ataaatcacacggcgctctt

h PGC1β ggcaggcctcagatctaaaa tcatgggagccttcttgtct

h RPLPO acagggcgacctggaagt ggatctgctgcatctgctt

h titin tggccacactgatggtctta tcctacagtcaccgtcatcg

m βactin gctctggctcctagcaccat gccaccgatccacacagagt

m CCL2 ggctcagccagatgcagttaa cctactcattgggatcatcttgct

m IGFII cgcttcagtttgtctgttcg gcagcactcttccacgatg

mIGFBP5 aaagcagtgtaagccctccc tccccatccacgtactccat m IL6 gtctggaagactcgcctacg aagtgcatcatcgttgttcataca

m IL1β agttgacggaccccaaaag agctggatgctctcatcagg

m leptin agcatccactgctatggtagc tcttctagtcccaagcattttgg

m myo D agcactacagtggcgactca ggccgctgtaatccatca

m myogenin ccttgctcagctccctca tgggagttgcattcactgg

m PGC1α gaaagggccaaacagagaga gtaaatcacacggcgctctt

m PGC1β ctccagttccggctcctc ccctctgctctcacgtctg

m titin ` gaggtgccgaagaaacttgt ttgggtgcttccggtactt

h, human; m, mice

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Table S4. Spearman correlations between the gene expression of muscle markers in the quadriceps muscle and the expression of leptin/IL6 in both epididymal adipose tissue and skeletal muscle.

Epididymal Adipose Tissue Quadriceps

Fat mass % Leptin IL6 Leptin IL6

Quadriceps R p R p R p R p R p

MyoD 0.314 0.127 0.414 0.063 0.824 0.0001 0.433 0.053 0.293 0.144

MyoG 0.203 0.225 0.050 0.428 0.516 0.021 0.091 0.366 0.462 0.037

βActin 0.232 0.193 0.577 0.011 0.643 0.004 0.225 0.198 0.253 0.172

Titin 0.04706 0.8626 0.1059 0.6963 0.1947 0.47 0.4297 0.0967 0.08824 0.7452

IGFII 0.482 0.036 0.489 0.033 0.577 0.013 0.320 0.120 0.207 0.229

IGFBP5 0.2412 0.3682 0.2735 0.3053 0.4248 0.101 0.4032 0.1214 0.3176 0.2306

PGC1α 0.041 0.441 0.279 0.147 0.077 0.385 0.155 0.283 0.391 0.068

PGC1β 0.124 0.324 0.006 0.494 0.086 0.373 0.019 0.470 0.218 0.208

20weekold mice (n=16).

Significant correlations are indicated in bold

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Table S5. Spearman correlations between the gene expression of muscle markers in the Gastrocnemius muscle and the expression of leptin and IL6 in inguinal AT.

Inguinal Adipose Tissue

Leptin IL6 Gastrocnemius r p r p MyoD 0.7794 0.0004 0.01071 0.9698 MyoG 0.2029 0.4510 0.3750 0.1684 βActin 0.5000 0.0486 0.3250 0.2372 Titin 0.5 0.0577 0.2352 0.4183 IGFII 0.6588 0.0055 0.1714 0.5413 IGFBP5 0.6747 0.0081 0.1484 0.6286 PGC1α 0.03736 0.8991 0.5824 0.0367 PGC1β 0.4250 0.1143 0.08132 0.7823

20weekold mice (n=16).

Significant correlations are indicated in bold

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Supplemental list

List of the genes present in the “Human myogenesis and myopathy PCR array”

Skeletal Muscle Contractility: DystrophinGlycoprotein Complex: CAMK2G, CAPN3, CAV3, DAG1, DMD (Dystrophin), DYSF, LMNA, MAPK1, SGCA. Titin Complex: ACTA1, ACTN3, CAPN3, CRYAB, DES, LMNA, MAPK1, MSTN, MYH1, MYH2, MYOT, NEB, SGCA, TNNI2, TNNT1, TNNT3, TRIM63 (MuRF1), TTN. Energy Metabolism: CS, HK2, PDK4, SLC2A4 (GLUT4). FastTwitch Fibers: ATP2A1, MYH1, MYH2, TNNI2, TNNT3. SlowTwitch Fibers: MB, MYH1, TNNC1, TNNT1. Other: DMPK, IKBKB, RPS6KB1.

Skeletal Myogenesis: ACTA1, ADRB2, AGRN, BCL2, BMP4, CAPN2, CAST, CAV1, CTNNB1, DMD, HDAC5, IGF1, IGFBP3, IGFBP5, MEF2C, MSTN, MUSK, MYF5, MYF6, MYOD1, MYOG, PAX3, PAX7, PPP3CA (Calcineurin Aa), RHOA, RPS6KB1, UTRN.

Skeletal Muscle Hypertrophy: ACTA1, ACVR2B, ADRB2, IGF1, IGFBP5, MSTN, MYF6, MYOD1, RPS6KB1.

Skeletal Muscle Autocrine Signaling: ADIPOQ, FGF2, IGF1, IGF2, IL6, LEP, MSTN, TGFB1.

Diabetes/Metabolic Syndrome: ADIPOQ, LEP, PPARG, PPARGC1A (PGC1a), PPARGC1B (PGC1β), PRKAA1 (AMPK), PRKAB2, PRKAG1, PRKAG3, SLC2A4 (GLUT4).

Skeletal Muscle Wasting/Atrophy: Autophagy: CAPN2, CASP3, FBXO32 (Atrogin1), FOXO1, FOXO3, MSTN, NOS2, PPARGC1A (PGC1α), PPARGC1B (PGC1β), RPS6KB1, TRIM63 (MuRF1). Dystrophy: AKT1, AKT2, FBXO32 (Atrogin1), IL1B, MAPK1, MAPK14, MAPK3, MAPK8, MMP9, NFKB1, TNF, TRIM63 (MuRF1), UTRN.

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Supplemental references

1. Perlemuter G, Naveau S, BelleCroix F, Buffet C, Agostini H, Laromiguière M, et al. Independent and opposite associations of trunk fat and leg fat with liver levels. Liver Int. 2008 Dec;28(10):1381–8. 2. Ciangura C, Bouillot JL, LloretLinares C, Poitou C, Veyrie N, Basdevant A, et al. Dynamics of change in total and regional body composition after gastric bypass in obese patients. Obesity (Silver Spring). 2010 Apr;18(4):760–5.

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