#METABOLOMICS WORKBENCH mlhomme_20221213_043543 DATATRACK_ID:3642 STUDY_ID:ST002400 ANALYSIS_ID:AN003907 VERSION 1 CREATED_ON 12-16-2022 #PROJECT PR:PROJECT_TITLE Alcohol dehydrogenase 1B is crucial for adipocyte homeostasis. PR:PROJECT_SUMMARY Background. Alcohol dehydrogenase (ADH1B), encoded by the ADH1B gene, is a PR:PROJECT_SUMMARY cytosolic enzyme mainly known for its role in ethanol catabolism in the liver. A PR:PROJECT_SUMMARY few studies have paved the way to show an equally important role of ADH1B in PR:PROJECT_SUMMARY adipocytes. This study aimed to better identify the cellular mechanisms and PR:PROJECT_SUMMARY signaling pathways involving ADH1B in adipose tissue and to determine if ADH1B PR:PROJECT_SUMMARY variants might contribute to adipose tissue dysfunction. Results. We showed that PR:PROJECT_SUMMARY CRISPR-Cas9-mediated ADH1B knockout (KO) in human adipose stem cells (ASC) PR:PROJECT_SUMMARY abolished adipocyte differentiation and decreased insulin response. This was PR:PROJECT_SUMMARY accompanied by oxidative stress, altered mitochondrial functions, and cellular PR:PROJECT_SUMMARY senescence. Lipidomic analysis revealed that ADH1B deficiency results in a major PR:PROJECT_SUMMARY remodeling of lipid composition in ASC. An ADH1B homozygous loss-of-function PR:PROJECT_SUMMARY variant was also identified in a patient presenting with a lipodystrophic and PR:PROJECT_SUMMARY insulin resistant syndrome associated with major liver dysfunction, leading to PR:PROJECT_SUMMARY early death. Discussion. This translational study underlines the crucial role of PR:PROJECT_SUMMARY ADH1B in adipose tissue. It unveils cellular mechanisms accounting for its key PR:PROJECT_SUMMARY role in adipogenesis, and adipocyte homeostasis. This study also identifies PR:PROJECT_SUMMARY ADH1B as a candidate gene in monogenic forms of lipodystrophic and insulin PR:PROJECT_SUMMARY resistant syndromes. PR:INSTITUTE INSERM PR:LAST_NAME Gautheron PR:FIRST_NAME Jérémie PR:ADDRESS 27 rue Chaligny, 75012 Paris France PR:EMAIL jeremie.gautheron@gmail.com PR:PHONE +33623398373 PR:DOI http://dx.doi.org/10.21228/M8VM64 #STUDY ST:STUDY_TITLE Alcohol dehydrogenase 1B is crucial for adipocyte homeostasis ST:STUDY_SUMMARY Background. Alcohol dehydrogenase (ADH1B), encoded by the ADH1B gene, is a ST:STUDY_SUMMARY cytosolic enzyme mainly known for its role in ethanol catabolism in the liver. A ST:STUDY_SUMMARY few studies have paved the way to show an equally important role of ADH1B in ST:STUDY_SUMMARY adipocytes. This study aimed to better identify the cellular mechanisms and ST:STUDY_SUMMARY signaling pathways involving ADH1B in adipose tissue and to determine if ADH1B ST:STUDY_SUMMARY variants might contribute to adipose tissue dysfunction. Results. We showed that ST:STUDY_SUMMARY CRISPR-Cas9-mediated ADH1B knockout (KO) in human adipose stem cells (ASC) ST:STUDY_SUMMARY abolished adipocyte differentiation and decreased insulin response. This was ST:STUDY_SUMMARY accompanied by oxidative stress, altered mitochondrial functions, and cellular ST:STUDY_SUMMARY senescence. Lipidomic analysis revealed that ADH1B deficiency results in a major ST:STUDY_SUMMARY remodeling of lipid composition in ASC. An ADH1B homozygous loss-of-function ST:STUDY_SUMMARY variant was also identified in a patient presenting with a lipodystrophic and ST:STUDY_SUMMARY insulin resistant syndrome associated with major liver dysfunction, leading to ST:STUDY_SUMMARY early death. Discussion. This translational study underlines the crucial role of ST:STUDY_SUMMARY ADH1B in adipose tissue. It unveils cellular mechanisms accounting for its key ST:STUDY_SUMMARY role in adipogenesis, and adipocyte homeostasis. This study also identifies ST:STUDY_SUMMARY ADH1B as a candidate gene in monogenic forms of lipodystrophic and insulin ST:STUDY_SUMMARY resistant syndromes. ST:INSTITUTE INSERM ST:LAST_NAME Gautheron ST:FIRST_NAME Jérémie ST:ADDRESS 27 rue Chaligny ST:EMAIL jeremie.gautheron@gmail.com ST:PHONE +33623398373 ST:SUBMIT_DATE 2022-12-13 #SUBJECT SU:SUBJECT_TYPE Cultured cells SU:SUBJECT_SPECIES Homo sapiens #SUBJECT_SAMPLE_FACTORS: SUBJECT(optional)[tab]SAMPLE[tab]FACTORS(NAME:VALUE pairs separated by |)[tab]Additional sample data SUBJECT_SAMPLE_FACTORS ADH1B_KO_01_LIP329 ADH1B_KO_01_LIP329 Genotype:ADH1B_KO Batch=1; RAW_FILE_NAME=LIP329_C18_1x_KO_01; RAW_FILE_NAME=LIP329_C18_10x_KO_01; RAW_FILE_NAME=LIP329_long_1x_KO_01; RAW_FILE_NAME=LIP329_short_1x_KO_01; RAW_FILE_NAME=LIP329_short_20x_KO_01 SUBJECT_SAMPLE_FACTORS ADH1B_KO_02_LIP329 ADH1B_KO_02_LIP329 Genotype:ADH1B_KO Batch=1; RAW_FILE_NAME=LIP329_C18_1x_KO_02; RAW_FILE_NAME=LIP329_C18_10x_KO_02; RAW_FILE_NAME=LIP329_long_1x_KO_02; RAW_FILE_NAME=LIP329_short_1x_KO_02; RAW_FILE_NAME=LIP329_short_20x_KO_02 SUBJECT_SAMPLE_FACTORS ADH1B_KO_03_LIP329 ADH1B_KO_03_LIP329 Genotype:ADH1B_KO Batch=1; RAW_FILE_NAME=LIP329_C18_1x_KO_03; RAW_FILE_NAME=LIP329_C18_10x_KO_03; RAW_FILE_NAME=LIP329_long_1x_KO_03; RAW_FILE_NAME=LIP329_short_1x_KO_03; RAW_FILE_NAME=LIP329_short_20x_KO_03 SUBJECT_SAMPLE_FACTORS ADH1B_KO_04_LIP329 ADH1B_KO_04_LIP329 Genotype:ADH1B_KO Batch=1; RAW_FILE_NAME=LIP329_C18_1x_KO_04; RAW_FILE_NAME=LIP329_C18_10x_KO_04; RAW_FILE_NAME=LIP329_long_1x_KO_04; RAW_FILE_NAME=LIP329_short_1x_KO_04; RAW_FILE_NAME=LIP329_short_20x_KO_04 SUBJECT_SAMPLE_FACTORS ADH1B_KO_05_LIP329 ADH1B_KO_05_LIP329 Genotype:ADH1B_KO Batch=1; RAW_FILE_NAME=LIP329_C18_1x_KO_05; RAW_FILE_NAME=LIP329_C18_10x_KO_05; RAW_FILE_NAME=LIP329_long_1x_KO_05; RAW_FILE_NAME=LIP329_short_1x_KO_05; RAW_FILE_NAME=LIP329_short_20x_KO_05 SUBJECT_SAMPLE_FACTORS CTL_01_LIP329 CTL_01_LIP329 Genotype:CTL Batch=1; RAW_FILE_NAME=LIP329_C18_1x_CTL_01; RAW_FILE_NAME=LIP329_C18_10x_CTL_01; RAW_FILE_NAME=LIP329_long_1x_CTL_01; RAW_FILE_NAME=LIP329_short_1x_CTL_01; RAW_FILE_NAME=LIP329_short_20x_CTL_01 SUBJECT_SAMPLE_FACTORS CTL_02_LIP329 CTL_02_LIP329 Genotype:CTL Batch=1; RAW_FILE_NAME=LIP329_C18_1x_CTL_02; RAW_FILE_NAME=LIP329_C18_10x_CTL_02; RAW_FILE_NAME=LIP329_long_1x_CTL_02; RAW_FILE_NAME=LIP329_short_1x_CTL_02; RAW_FILE_NAME=LIP329_short_20x_CTL_02 SUBJECT_SAMPLE_FACTORS CTL_03_LIP329 CTL_03_LIP329 Genotype:CTL Batch=1; RAW_FILE_NAME=LIP329_C18_1x_CTL_03; RAW_FILE_NAME=LIP329_C18_10x_CTL_03; RAW_FILE_NAME=LIP329_long_1x_CTL_03; RAW_FILE_NAME=LIP329_short_1x_CTL_03; RAW_FILE_NAME=LIP329_short_20x_CTL_03 SUBJECT_SAMPLE_FACTORS CTL_04_LIP329 CTL_04_LIP329 Genotype:CTL Batch=1; RAW_FILE_NAME=LIP329_C18_1x_CTL_04; RAW_FILE_NAME=LIP329_C18_10x_CTL_04; RAW_FILE_NAME=LIP329_long_1x_CTL_04; RAW_FILE_NAME=LIP329_short_1x_CTL_04; RAW_FILE_NAME=LIP329_short_20x_CTL_04 SUBJECT_SAMPLE_FACTORS CTL_05_LIP329 CTL_05_LIP329 Genotype:CTL Batch=1; RAW_FILE_NAME=LIP329_C18_1x_CTL_05; RAW_FILE_NAME=LIP329_C18_10x_CTL_05; RAW_FILE_NAME=LIP329_long_1x_CTL_05; RAW_FILE_NAME=LIP329_short_1x_CTL_05; RAW_FILE_NAME=LIP329_short_20x_CTL_05 #COLLECTION CO:COLLECTION_SUMMARY ASC were isolated from surgical samples of subcutaneous abdominal adipose tissue CO:COLLECTION_SUMMARY from a 25-year-old healthy woman with a normal body mass index (BMI). Adipose CO:COLLECTION_SUMMARY tissue was enzymatically digested with collagenase B (0.2%). CO:SAMPLE_TYPE Adipose tissue #TREATMENT TR:TREATMENT_SUMMARY After centrifugation, stromal vascular fraction was filtered, rinsed, plated and TR:TREATMENT_SUMMARY cultured in α-MEM with 10% Fetal Calf Serum (FCS), 1% GlutaMAX (#35050061, TR:TREATMENT_SUMMARY Thermo Fisher Scientific), 1% Penicillin/streptomycin (PS - 10,000 UI/mL), 1% TR:TREATMENT_SUMMARY HEPES and Fibroblast Growth Factor-2 (FGF-2 -145 nmol/L). After 24 h, only ASC TR:TREATMENT_SUMMARY adhered to plastic surfaces, while other cells were removed after culture medium TR:TREATMENT_SUMMARY replacement. ASC were maintained in an undifferentiated state in α-MEM TR:TREATMENT_SUMMARY supplemented with 10 % newborn calf serum (#CA-1151500; Biosera, MI, USA), 1% TR:TREATMENT_SUMMARY GlutaMAX, HEPES and P/S, and FGF-2 (145 nmol/L). Adipocyte differentiation was TR:TREATMENT_SUMMARY induced by treating 2-day post-confluent cultures with high-glucose (25 mmol/L) TR:TREATMENT_SUMMARY DMEM supplemented with 10 % FCS, 1 % PS, 1 µmol/L dexamethasone (#D4902; TR:TREATMENT_SUMMARY Sigma-Aldrich, MI, USA), 1 µM rosiglitazone (#D4902; Sigma-Aldrich), 250 µM TR:TREATMENT_SUMMARY 3-isobutyl-1-methyl xanthine (IBMX) (#I7018; Sigma-Aldrich) and 0.17 µmol/L TR:TREATMENT_SUMMARY insulin (#I0516; Sigma-Aldrich) for ten days. The medium was then replaced with TR:TREATMENT_SUMMARY high-glucose DMEM supplemented with 10% FCS, 1 % PS, 1 µmol/L rosiglitazone and TR:TREATMENT_SUMMARY 0.17 µM insulin, and changed to fresh medium every 2 days until the 20th day. #SAMPLEPREP SP:SAMPLEPREP_SUMMARY Lipid extraction. Lipids were extracted from ASC cells according to a modified SP:SAMPLEPREP_SUMMARY Folch method. Cell pellets were resuspended in 100µl methanol and supplemented SP:SAMPLEPREP_SUMMARY with deuterated internal standards. Lipids were extracted with 1.5 mL chloroform SP:SAMPLEPREP_SUMMARY and 650 µL methanol, sonicated for 15 min. Phase separation was triggered by SP:SAMPLEPREP_SUMMARY addition of 450 µL of ammonium carbonate (250 mM). Lower organic phase was SP:SAMPLEPREP_SUMMARY dried and resuspended in 200 µL of liquid chromatography – mass spectrometry SP:SAMPLEPREP_SUMMARY (LC-MS) solvent. #CHROMATOGRAPHY CH:INSTRUMENT_NAME Shimadzu 20AD CH:COLUMN_NAME Phenomenex Kinetex HILIC (150 x 3mm,2.6um) CH:COLUMN_TEMPERATURE 45 CH:FLOW_GRADIENT - CH:FLOW_RATE 300ul/min CH:SOLVENT_A 100% water; 0.2% acetic acid; 30mM ammonium acetate CH:SOLVENT_B 100% acetonitrile; 0.2% acetic acid CH:CHROMATOGRAPHY_TYPE HILIC #ANALYSIS AN:ANALYSIS_TYPE MS #MS MS:INSTRUMENT_NAME ABI Sciex 4000 QTrap MS:INSTRUMENT_TYPE QTRAP MS:MS_TYPE ESI MS:MS_COMMENTS MRM acquisition of broad chromatographic peaks of low abundant phospho- and MS:MS_COMMENTS sphingolipid classes: PS, PA, LPC, LPE and some ceramides this acquisition is MS:MS_COMMENTS called "long_1x" MS:ION_MODE POSITIVE #MS_METABOLITE_DATA MS_METABOLITE_DATA:UNITS mol% of total lipids MS_METABOLITE_DATA_START Samples ADH1B_KO_01_LIP329 ADH1B_KO_02_LIP329 ADH1B_KO_03_LIP329 ADH1B_KO_04_LIP329 ADH1B_KO_05_LIP329 CTL_01_LIP329 CTL_02_LIP329 CTL_03_LIP329 CTL_04_LIP329 CTL_05_LIP329 Factors Genotype:ADH1B_KO Genotype:ADH1B_KO Genotype:ADH1B_KO Genotype:ADH1B_KO Genotype:ADH1B_KO Genotype:CTL Genotype:CTL Genotype:CTL Genotype:CTL Genotype:CTL LPC(16:0) 0.4605 0.4862 0.3549 0.3597 0.3847 0.3008 0.2893 0.2965 0.2718 0.2784 LPC(16:1) 0.0523 0.0613 0.0417 0.0340 0.0414 0.0307 0.0316 0.0366 0.0324 0.0323 LPC(18:0) 0.5466 0.5367 0.4829 0.5498 0.5094 0.2644 0.2558 0.2523 0.2401 0.2615 LPC(18:1) 0.2032 0.2137 0.1667 0.1261 0.1629 0.1160 0.1266 0.1406 0.1213 0.1269 LPC(18:2) 0.0088 0.0135 0.0104 0.0102 0.0109 0.0091 0.0096 LPC(20:3) 0.0182 0.0196 0.0131 0.0087 0.0132 0.0062 0.0059 0.0059 0.0059 LPC(20:4) 0.1111 0.1522 0.1006 0.0587 0.0958 0.0246 0.0299 0.0307 0.0282 0.0235 LPC(22:4) 0.0125 0.0081 0.0154 0.0059 0.0072 0.0066 0.0053 LPC(22:5) 0.0336 0.0386 0.0288 0.0160 0.0249 0.0115 0.0135 0.0170 0.0138 0.0119 LPC(22:6) 0.0274 0.0369 0.0265 0.0152 0.0223 0.0116 0.0135 0.0152 0.0133 0.0109 LPE(16:0) 0.0479 0.0551 0.0548 0.0398 0.0566 0.0642 0.0648 0.0627 0.0655 0.0514 LPE(16:1) 0.0218 0.0246 0.0163 0.0218 0.0155 0.0197 0.0166 0.0169 0.0144 LPE(18:0) 0.0696 0.0703 0.0705 0.0726 0.0648 0.0549 0.0569 0.0511 0.0530 0.0492 LPE(18:1) 0.3614 0.4264 0.4250 0.3154 0.3997 0.1835 0.2691 0.2068 0.2091 0.1971 LPE(18:2) 0.0326 0.0363 0.0384 0.0265 0.0335 0.0171 0.0238 0.0198 0.0179 0.0175 LPE(20:3) 0.0411 0.0533 0.0409 0.0353 0.0403 0.0171 0.0225 0.0187 0.0184 0.0170 LPE(20:4) 0.6046 0.7390 0.6820 0.4806 0.6349 0.2111 0.2959 0.2456 0.2331 0.1844 LPE(20:5) 0.0582 0.0656 0.0578 0.0480 0.0542 0.0261 0.0363 0.0297 0.0311 0.0245 LPE(22:3) 0.0093 0.0068 0.0103 0.0130 0.0187 0.0143 0.0127 0.0126 LPE(22:4) 0.1129 0.1428 0.1413 0.0928 0.1295 0.0884 0.1306 0.0959 0.1019 0.0871 LPE(22:5) 0.2082 0.2791 0.2624 0.1785 0.2413 0.1829 0.2549 0.2182 0.2112 0.1943 LPE(22:6) 0.2367 0.2800 0.2670 0.1801 0.2239 0.1700 0.2327 0.2041 0.1926 0.1771 PA(34:2) 0.0523 0.0979 0.0597 0.0818 0.0187 0.0584 0.0908 0.0580 0.0285 PA(36:1) 0.2963 0.6465 0.3532 0.3956 0.1670 0.1613 0.1597 PA(36:2) 0.1266 0.3772 0.2196 0.1597 0.0008 0.1341 0.1763 0.1188 0.0200 PA(36:3) 0.0524 0.1199 0.0834 0.0741 0.0149 0.0602 0.0794 0.0406 0.0256 PA(38:2) 0.0087 0.0585 0.0035 0.0186 0.0231 0.0149 0.0139 PA(38:3) 0.1052 0.2211 0.1379 0.1378 0.0632 0.0691 0.0667 0.0094 PA(38:4) 0.5620 0.9387 0.7992 0.2182 0.7288 0.1545 0.3355 0.3697 0.2673 0.1891 PA(38:5) 0.1179 0.2090 0.1476 0.0297 0.1375 0.0461 0.1021 0.1067 0.0748 0.0531 PA(38:6) 0.0522 0.0876 0.0648 0.0232 0.0512 0.0148 0.0342 0.0302 0.0264 0.0177 PA(40:5) 0.1262 0.1727 0.1837 0.0285 0.1309 0.0139 0.0689 0.0654 0.0650 0.0034 PS(34:0) 0.0119 0.0098 0.0129 0.0060 0.0097 0.0293 0.0483 0.0455 0.0397 0.0078 PS(34:1) 0.2952 0.4146 0.3405 0.1697 0.3077 0.1910 0.2946 0.2700 0.2303 0.0581 PS(34:2) 0.0241 0.0229 0.0193 0.0171 0.0194 0.0184 0.0228 0.0249 0.0231 0.0083 PS(34:3) 0.0028 0.0060 0.0022 0.0036 0.0006 0.0017 0.0029 0.0012 PS(36:0) 0.0424 0.0621 0.0488 0.0207 0.0427 0.0296 0.0455 0.0379 0.0427 0.0099 PS(36:1) 0.7754 1.0710 0.8474 0.4447 0.8769 0.5126 0.7803 0.6767 0.6433 0.1126 PS(36:2) 0.1876 0.2392 0.1864 0.1055 0.1879 0.1653 0.2498 0.2016 0.2104 0.0530 PS(36:3) 0.0288 0.0390 0.0340 0.0180 0.0327 0.0211 0.0279 0.0246 0.0242 0.0090 PS(36:4) 0.0192 0.0338 0.0221 0.0108 0.0216 0.0081 0.0132 0.0133 0.0108 0.0045 PS(38:2) 0.0260 0.0437 0.0320 0.0169 0.0292 0.0271 0.0438 0.0388 0.0332 0.0082 PS(38:3) 0.2968 0.3971 0.3022 0.1701 0.2954 0.1384 0.2259 0.1983 0.1823 0.0422 PS(38:4) 0.2596 0.3466 0.2743 0.1479 0.2644 0.1129 0.1694 0.1583 0.1418 0.0381 PS(38:5) 0.0457 0.0558 0.0462 0.0246 0.0452 0.0274 0.0361 0.0391 0.0361 0.0147 PS(38:6) 0.0590 0.0752 0.0515 0.0338 0.0525 0.0258 0.0375 0.0404 0.0367 0.0139 PS(40:4) 0.1210 0.1641 0.1228 0.0648 0.1362 0.0718 0.1154 0.0985 0.0866 0.0230 PS(40:5) 0.3066 0.4090 0.3204 0.1587 0.3515 0.1820 0.2775 0.2646 0.2289 0.0726 PS(40:6) 0.3459 0.4590 0.3500 0.1798 0.3410 0.1645 0.2368 0.2314 0.2084 0.0573 PS(40:7) 0.0248 0.0308 0.0231 0.0116 0.0212 0.0144 0.0211 0.0184 0.0186 0.0060 MS_METABOLITE_DATA_END #METABOLITES METABOLITES_START metabolite_name pubchem_id inchi_key kegg_id other_id other_id_type ri ri_type moverz_quant LPC(16:0) 10.1 LPC(16:1) 9.93 LPC(18:0) 9.91 LPC(18:1) 9.72 LPC(18:2) 9.84 LPC(20:3) 9.66 LPC(20:4) 9.63 LPC(22:4) 9.55 LPC(22:5) 9.57 LPC(22:6) 9.56 LPE(16:0) 7.85 LPE(16:1) 7.88 LPE(18:0) 7.38 LPE(18:1) 7.76 LPE(18:2) 7.81 LPE(20:3) 7.72 LPE(20:4) 7.69 LPE(20:5) 7.76 LPE(22:3) 7.63 LPE(22:4) 7.65 LPE(22:5) 7.67 LPE(22:6) 7.65 PA(34:2) 6.12 PA(36:1) 6 PA(36:2) 6 PA(36:3) 6.02 PA(38:2) 5.95 PA(38:3) 5.81 PA(38:4) 6 PA(38:5) 5.81 PA(38:6) 6 PA(40:5) 5.79 PS(34:0) 7.8 PS(34:1) 7.7 PS(34:2) 7.22 PS(34:3) 7.69 PS(36:0) 7.11 PS(36:1) 7.6 PS(36:2) 7.6 PS(36:3) 7.6 PS(36:4) 7.6 PS(38:2) 7.6 PS(38:3) 7.4 PS(38:4) 7.28 PS(38:5) 7.09 PS(38:6) 7.4 PS(40:4) 7.4 PS(40:5) 7.4 PS(40:6) 7.4 PS(40:7) 7.4 METABOLITES_END #END