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(1)

Figure S1. Two-variable Prediction model for high-risk IPMN – ROC curve.

Red dot marks the sensitivity and specificity of the radiographic MD-/BD- IPMN subtype alone.

(2)

Table S1. Complete list of multiplexed biomarker assays (87 markers).

Biological groups Markers

Tumor markers Alpha-fetoprotein (AFP), CA 19–9, CA 125,

CA 15–3, CA72–4, CEA

Hormones Adrenocorticotropic hormone (ACTH), follicle

stimulating hormone (FSH),

growth hormone (GH), insulin, luteinizing hormone (LH), prolactin (PRL),

parathyroid hormone (PTH), thyroid stimulating hormone (TSH), pancreatic polypeptide (PP), peptide YY (PYY, total) Apoptotic factors Cytokeratin 19 (Cyfra 21–1), DR5, sFas, sFasL

Cell Adhesion ICAM-1, VCAM-1, NCAM

Proteases/inhibitors Kallikrein 10, MMP-2, 3, 9, TIMP-1–4, Cathepsin D

Cytokines/chemokines/

receptors

CD40L, eotaxin-1, fractalkine, IFNγ, IL-10, IL-12p70, IL-13, IL-1β, IL-1Ra,

IL-2, IL-2R, sIL-2Rα, IL-4, IL-5, IL-6, IL-6R, IL-7, IL-8, IP-10, macrophage inhibitory factor (MIF),

MIP-1b, TGFα, TNFα, TNF-RI, TNF-RII Growth/angiogenesis

factors/receptors

Angiostatin, EGFR, endostatin, ErbB2, GM- CSF, IGFBP-1, thrombospondin, CNTF, NGF, BDNF

Adipokines Adiponectin, leptin

Apolipoproteins ApoAI, ApoAII, ApoB, ApoCII, ApoCIII,

ApoE

Other CRP, ghrelin (active), gastric inhibitory

polypeptide (GIP, total), glucagon like peptide 1(GLP-1, active), HE4, mesothelin, osteocalcin (OC), osteoprotegerin (OPG), osteopontin (OPN),

serum amyloid A (SAA), serum amyloid P (SAP), transglutaminase II (TG II), total plasminogen

activator inhibitor 1 (tPAI 1), myeloperoxidase (MPO)

(3)

Table S2. Cyst fluid proteins stratified by low-risk and high-risk groups.

Cyst fluid proteins n=78 Low-risk (n=48) High-risk (n=30) p-value

MIF, pg/ml 77 8163.6 (47) 17164 (30) 0.003

FASL, pg/ml 77 39 (47) 56.3 (30) 0.1

Fas, pg/ml 77 1947.6 (47) 6018.5 (30) 0.4

VCAM-1, pg/ml 78 12397.6 (48) 17091.2 (30) 0.1

ICAM-1, pg/ml 78 10140.2 (48) 12075.8 (30) 0.4

MMP-9, pg/ml 78 78728.1 (48) 156359.2 (30) 0.03

MPO, pg/ml 78 756696.40 (48) 1429481.3 (30) 0.009

tPAI-1, pg/ml 78 6696 (48) 8430.3 (30) 0.2

MIP-4, pg/ml 78 8466.8 (48) 9366.1 (30) 0.1

CathepsinD, pg/ml 78 401810.1 (48) 809082.5 (30) 0.01

NCAM, pg/ml 78 3222 (48) 9840.2 (30) 0.05

OPG, pg/ml 76 5840.3 (47) 12224.1 (29) 0.5

OC, pg/ml 76 3919.9 (47) 11140.2 (29) 0.01

OPN, pg/ml 76 10726.7 (47) 11399.8 (29) 0.9

PTH, pg/ml 76 109.9 (47) 326.2 (29) 0.001

Clusterin, pg/ml 78 9288731.2 (48) 13572936.1 (30) 0.5

M-CSF, pg/ml 76 58386.3 (48) 8962.6 (28) 0.5

Fractalkine, pg/ml 74 270.9 (46) 709.7 (28) 0.1

IFNα, pg/ml 74 40.9 (46) 86.7 (28) 0.1

IFNγ, pg/ml 74 6.8 (46) 27.5 (28) 0.003

IL-1α, pg/ml 74 79.1 (46) 694.7 (28) 0.02

IL-1β, pg/ml 74 92.9 (46) 707.1 (28) 0.001

IL-1Ra, pg/ml 74 482.8 (46) 1012.3 (28) 0.03

IL-2, pg/ml 74 2.8 (46) 15.2 (28) 0.001

IL-4, pg/ml 74 3.8 (46) 31 (28) 0.004

IL-5, pg/ml 74 0.5 (46) 6.8 (28) 0.001

IL-6, pg/ml 74 642.8 (46) 714.5 (28) 0.3

IL-7, pg/ml 74 14.9 (46) 31 (28) 0.1

CD40L, pg/ml 74 375.6 (46) 979.3 (28) 0.005

sIL-2Rα, pg/ml 74 13.1 (46) 86.5 (28) 0.003

TGFα, pg/ml 74 18.8 (46) 25.1 (28) 0.5

TNFα, pg/ml 74 24.6 (46) 117.6 (28) 0.1

TNFβ, pg/ml 74 2 (46) 20.5 (28) 0.013

Adiponectin, pg/ml 71 198940.5 (43) 476827.8 (28) 0.3

Lipocalin/NGAL, pg/ml 71 1610013 (43) 1469529.8 (28) 0.5

NGF, pg/ml 63 9.3 (39) 46.5 (24) 0.049

HGF, pg/ml 63 6756.1 (39) 4050 (24) 0.5

ACTH, pg/ml 71 9.6 (43) 17.3 (28) 0.2

CNTF, pg/ml 71 440.2 (43) 1269.5 (28) 0.03

FSH, uIU/ml 71 2657.4 (43) 4786.5 (28) 0.3

GH, pg/ml 71 72.5 (43) 209 (28) 0.08

LH, uIU/ml 71 402.5 (43) 1388.6 (28) 0.2

(4)

TSH, nIU/ml 71 469 (43) 5966.8 (28) 0.02

Prolactin, pg/ml 71 1037.5 (43) 8035.9 (28) 0.4

BDNF, pg/ml 71 107.7 (43) 335.2 (28) 0.05

Apolipoprotein Apo AII , pg/ml

78 2903.6 (48) 4942.9 (30) 0.5

Apolipoprotein Apo B, pg/ml

78 13165.4 (48) 15593.3 (30) 0.3

Apolipoprotein A1, pg/ml 78 113907.9 (48) 276089.3 (30) 0.3 Apolipoprotein Apo CIII,

pg/ml 78 675.9 (48) 2484.2 (30) 0.04

Apolipoprotein Apo E,

pg/ml 78 448.5 (48) 1665 (30) 0.5

CRP, pg/ml 78 294595 (48) 393225.2 (30) 0.1

SAA, pg/ml 78 98033.9 (48) 149759.9 (30) 0.1

SAP, pg/ml 78 783565.9 (48) 2221856.9 (30) 0.03

SCC, pg/ml 73 155586.7 (45) 1075347.2 (28) 0.1

TTR, pg/ml 73 4685380.5 (45) 7257130.9 (28) 0.5

CA72-4, U/ml 73 62.1 (45) 399.2 (28) 0.07

AFP, pg/ml 73 218.2 (45) 4837.9 (28) 0.009

Kallikrein, pg/ml 73 39373.8 (45) 42204.1 (28) 0.2

Mesothelin, pg/ml 73 92544.8 (45) 117811.9 (28) 0.5

IGFBP-1, pg/ml 73 198519.8 (45) 129304.8 (28) 0.8

Cytokeratin 19, pg/ml 73 191305.7 (45) 302619.5 (28) 0.02

CA-125, U/mL 73 486.8 (45) 371.3 (28) 0.7

CEA, pg/ml 73 382594.7 (45) 322379.4 (28) 0.2

ErbB2, pg/ml 73 4420.8 (45) 11458.6 (28) 0.01

EGFR, pg/ml 73 3430.8 (45) 15157.7 (28) 0.003

CA19-9, U/ml 73 372.5 (45) 234.5 (28) 0.11

MMP-2, pg/ml 75 25094.8 (47) 14786.3 (28) 0.9

MMP-3, pg/ml 75 2185.8 (47) 8824.3 (28) 0.2

TIMP-1, pg/ml 74 197682.3 (46) 242740.8 (28) 0.9

TIMP-2, pg/ml 74 38141.9 (46) 33539.5 (28) 0.5

TIMP-3, pg/ml 74 8070.5 (46) 9615.2 (28) 0.2

TIMP-4, pg/ml 74 358.8 (46) 619.3 (28) 0.8

TNF-R2, pg/ml 56 414.8 (34) 726.7 (22) 0.08

TNF-R1, pg/ml 56 715.8 (34) 1305.4 (22) 0.3

Eotaxin, pg/ml 56 18.6 (34) 33.9 (22) 0.8

MIP-1B, pg/ml 56 67.1 (34) 187.8 (22) 0.07

TNFα, pg/ml 56 7.5 (34) 35.6 (22) 0.02

IP-10, pg/ml 56 163.2 (34) 300.7 (22) 0.1

IL-2R, pg/ml 56 71.8 (34) 261.7 (22) 0.01

IL-8, pg/ml 56 10178.5 (34) 18625.5 (22) 0.4

Tg II, pg/ml 65 2372.3 (39) 4341.2 (26) 0.1

HE4, pg/ml 50 177274.9 (31) 149787.5 (19) 0.7

CA15-3, pg/ml 50 69.2 (31) 9.5 (19) 0.9

(5)

Thrombospondin, pg/ml 62 332915 (38) 397526 (24) 0.3

Endostatin, pg/ml 62 24188.2 (38) 30073 (24) 0.2

Angiostatin, pg/ml 62 118222.8 (38) 570947.6 (24) 0.3

Continous variables presented as: mean (number of patients).

Analysis of cyst fluid proteins was performed with Mann-Whitney test.

(6)

Table S3. Cyst fluid proteins stratified by Tumor-associated neutrophils (TAN)levels.

# Cyst fluid proteins2

TAN neg (n=52) TAN low (n=9) TAN high (n=16) p-

value1

1 IL-1β, pg/ml 1; 1-2 (50) 1; 1-235 (9) 19.5; 2-950 (14) 0.005

2 IL-1α, pg/ml 1; 1-9 (50) 1; 1-271 (9) 17; 1-760 (14) 0.03

3 IL-2, pg/ml 1; 1-1 (50) 1; 1-6.5 (9) 7.5; 2-26 (14) <0.001

4 sIL-2Rα, pg/ml 0; 0-1 (50) 1; 0-32 (9) 9.5; 1-120 (14) 0.004

5 IL-4, pg/ml 1; 1-2 (50) 4; 1.5-19 (9) 9; 1-35 (14) 0.004

6 IL-5, pg/ml 0; 0-0 (50) 0; 0-1 (9) 1; 0-3.5 (14) 0.004

7 TNFα, pg/ml 2.5; 1-19 (50) 5; 1-167 (9) 20.5; 3-123 (14) 0.02

8 TNFβ, pg/ml 1; 1-1 (50) 1; 1-1.5 (9) 1.5; 1-25 (14) 0.007

9 NGF, pg/ml 0; 0-5 (44) 0; 0-4 (6) 27; 0-113 (13) 0.02

10 NCAM, pg/ml 1500; 1300-2500 (52) 1600; 1400-13000 (9) 2200; 1600-26000 (16) 0.04 11 MPO, pg/ml 97000; 16000-

830000(52) 86000; 17000-

1500000 (9) 2000000; 125000-2700000

(16) 0.04

12 CNTF, pg/ml 177; 150-230 (47) 290; 150-900 (7) 430; 190-3000 (16) 0.02 13 TSH, nIU/ml 31; 26-36 (47) 32; 25-700 (7) 600; 30-25000 (16) 0.02 14 Cathepsin D,

pg/ml 130000; 90000-

430000 (52) 500000; 180000-

900000 (9) 460000; 210000-1200000

(16) 0.02

15 OPG, pg/ml 325; 134-1300 (51) 380; 40-2200 (8) 1700; 600-7200 (16) 0.04 16 OC, pg/ml 75; 50-2300 (51) 1800; 60-8000 (8) 8600; 800-26000 (16) 0.02

17 PTH, pg/ml 7; 4-42 (51) 63; 5-134 (8) 183; 34-685 (16) 0.001

18 IFNγ, pg/ml 2; 1-6 (50) 8; 2.5-13 (9) 10.5; 3-43 (14) 0.005

19 CD40L, pg/ml 16; 3-140 (50) 13; 7-2000 (9) 380; 50-560 (14) 0.04 20 CRP, pg/ml 40000; 18000-160000

(52)

140000; 16000- 500000 (9)

230000; 96000-680000 (16) 0.008 21 EGFR, pg/ml 514; 116-2000 (49) 3000; 200-7000 (7) 2400; 460-12000 (16) 0.02

Numbers presented as: median; interquartile range (number of patients).

1Analysis performed with Kruskal-Walis 1-way ANOVA.

2All proteins are CFIM except: TSH, OC, PTH.

(7)

Table S4. Differentially expressed cyst fluid proteins1 stratified by IPMN sub-type.

BD-IPMN (n=43)

# Cyst fluid proteins Functional group Low-risk, (n=35)

High-risk, (n=8)

p-value

1 MIF, pg/ml Immune modulator 1477; 530-550

(33)

4711; 2800- 18000 (7)

0.03

2 CathepsinD, pg/ml Protease 110000; 44000-

340000 (33) 312000;

165000-734000 (7)

0.04

3 CEA, pg/ml Tumor marker 412000; 95000-

630000 (33)

22000; 13000- 73000 (7)

0.01 4 ErbB2, ng/mL Growth factors/cell

cycle

700; 280-2373

(33) 2500; 788-5078

(7) 0.03

5 IL-2R, pg/ml Chemokines/cytokines 41; 41-46 (33) 51; 41-330 (7) 0.03 MD-IPMN (n=35)

# Cyst fluid protein Functional group Low-risk, (n=13)

High-risk, (n=22)

p-value 1 IFNγ, pg/ml Chemokines/cytokines 1.5; 1-2 (12) 10.5; 3-49 (20) <0.001 2 IL-1β, pg/ml Chemokines/cytokines 1; 1-2 (12) 19.5; 1-450 (20) 0.02 3 IL-2, pg/ml Chemokines/cytokines 1; 1-2 (12) 4.5; 1-22.75 (20) 0.01 4 IL-4, pg/ml Chemokines/cytokines 1.5; 1-5 (12) 12.5; 1.25-4.25

(20)

0.01 5 IL-5, pg/ml Chemokines/cytokines 0; 0-0 (12) 1; 0-5.75 (20) 0.009 6 CD-40L, pg/ml Chemokines/cytokines 58.5; 2-325 (12) 380; 43.75-786

(20)

0.04 7 sIL-2Rα, pg/ml Chemokines/cytokines 0.5; 0-1 (12) 9.5; 1-141.75

(20) 0.03

8 TNFβ, pg/ml Chemokines/cytokines 1; 1-1 (12) 1.5; 1-46.25 (20) 0.04 Analysis of cyst fluid proteins was performed with Mann-Whitney test. Presented as: median;

interquartile range (number of patients).

1All proteins are CFIM except: CEA.

(8)

Table S5. Multivariate Analysis of risk factors for high-risk IPMN.

Variable OR CI: 95% p-value

Standardized CA72-4

3.5 1.04-12.1 0.04

Standardized MMP9

- - NS

Radiographic

MD-/BD- IPMN 12.4 3.6-41.9 <0.001

Solid

component - - NS

Gender - - NS

Diabetes - - NS

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