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GM2-GM3 gangliosides ratio is dependent on GRP94 through down-regulation of GM2-AP cofactor in brain metastasis cells.

Bedia C, Badia M, Muixí L, Levade T, Tauler R, Sierra A.

Sci Rep. 2019 Oct 2;9(1):14241. doi: 10.1038/s41598-019-50761-5.


The TNF Paradox in Cancer Progression and Immunotherapy.

Montfort A, Colacios C, Levade T, Andrieu-Abadie N, Meyer N, Ségui B.

Front Immunol. 2019 Jul 31;10:1818. doi: 10.3389/fimmu.2019.01818. eCollection 2019. No abstract available.


Pregnancy outcome in Refsum disease: Affected fetuses and children born to an affected mother.

Dubot P, Astudillo L, Touati G, Baruteau J, Broué P, Roche S, Sabourdy F, Levade T.

JIMD Rep. 2019 Mar 14;46(1):11-15. doi: 10.1002/jmd2.12020. eCollection 2019 Mar.


Inherited monogenic defects of ceramide metabolism: Molecular bases and diagnoses.

Dubot P, Sabourdy F, Rybova J, Medin JA, Levade T.

Clin Chim Acta. 2019 Aug;495:457-466. doi: 10.1016/j.cca.2019.05.020. Epub 2019 May 22. Review.


Sphingomyelin Synthase 1 (SMS1) Downregulation Is Associated With Sphingolipid Reprogramming and a Worse Prognosis in Melanoma.

Bilal F, Montfort A, Gilhodes J, Garcia V, Riond J, Carpentier S, Filleron T, Colacios C, Levade T, Daher A, Meyer N, Andrieu-Abadie N, Ségui B.

Front Pharmacol. 2019 Apr 30;10:443. doi: 10.3389/fphar.2019.00443. eCollection 2019.


Activity-Based Imaging of Acid Ceramidase in Living Cells.

Ordóñez YF, Abad JL, Aseeri M, Casas J, Garcia V, Casasampere M, Schuchman EH, Levade T, Delgado A, Triola G, Fabrias G.

J Am Chem Soc. 2019 May 15;141(19):7736-7742. doi: 10.1021/jacs.8b11687. Epub 2019 May 2.


Allogeneic hematopoietic cell transplantation in Farber disease.

Ehlert K, Levade T, Di Rocco M, Lanino E, Albert MH, Führer M, Jarisch A, Güngör T, Ayuk F, Vormoor J.

J Inherit Metab Dis. 2019 Mar;42(2):286-294. doi: 10.1002/jimd.12043. Epub 2019 Feb 27.


Selective Targeting of the Interconversion between Glucosylceramide and Ceramide by Scaffold Tailoring of Iminosugar Inhibitors.

Baudoin-Dehoux C, Castellan T, Rodriguez F, Rives A, Stauffert F, Garcia V, Levade T, Compain P, Génisson Y.

Molecules. 2019 Jan 19;24(2). pii: E354. doi: 10.3390/molecules24020354.


Targeting the Sphingosine 1-Phosphate Axis Exerts Potent Antitumor Activity in BRAFi-Resistant Melanomas.

Garandeau D, Noujarède J, Leclerc J, Imbert C, Garcia V, Bats ML, Rambow F, Gilhodes J, Filleron T, Meyer N, Brayer S, Arcucci S, Tartare-Deckert S, Ségui B, Marine JC, Levade T, Bertolotto C, Andrieu-Abadie N.

Mol Cancer Ther. 2019 Feb;18(2):289-300. doi: 10.1158/1535-7163.MCT-17-1141. Epub 2018 Nov 27.


[Anti-TNFα antibodies in melanoma immunotherapy].

Montfort A, Colacios C, Levade T, Andrieu-Abadie N, Meyer N, Ségui B.

Med Sci (Paris). 2018 Oct;34(10):788-791. doi: 10.1051/medsci/2018202. Epub 2018 Nov 19. French. No abstract available.


Lysosomal and network alterations in human mucopolysaccharidosis type VII iPSC-derived neurons.

Bayó-Puxan N, Terrasso AP, Creyssels S, Simão D, Begon-Pescia C, Lavigne M, Salinas S, Bernex F, Bosch A, Kalatzis V, Levade T, Cuervo AM, Lory P, Consiglio A, Brito C, Kremer EJ.

Sci Rep. 2018 Nov 9;8(1):16644. doi: 10.1038/s41598-018-34523-3.


A new case of SMA phenotype without epilepsy due to biallelic variants in ASAH1.

Ame van der Beek N, Nelson I, Froissart R, Levade T, Garcia V, Lacene E, Boland A, Masson C, Romero NB, Stojkovic T, Bonne G, Béhin A.

Eur J Hum Genet. 2019 Mar;27(3):337-339. doi: 10.1038/s41431-018-0250-z. Epub 2018 Oct 5. No abstract available.


Lysosomal acid ceramidase ASAH1 controls the transition between invasive and proliferative phenotype in melanoma cells.

Leclerc J, Garandeau D, Pandiani C, Gaudel C, Bille K, Nottet N, Garcia V, Colosetti P, Pagnotta S, Bahadoran P, Tondeur G, Mograbi B, Dalle S, Caramel J, Levade T, Ballotti R, Andrieu-Abadie N, Bertolotto C.

Oncogene. 2019 Feb;38(8):1282-1295. doi: 10.1038/s41388-018-0500-0. Epub 2018 Sep 25.


Acid ceramidase deficiency: Farber disease and SMA-PME.

Yu FPS, Amintas S, Levade T, Medin JA.

Orphanet J Rare Dis. 2018 Jul 20;13(1):121. doi: 10.1186/s13023-018-0845-z. Review.


A novel adeno-associated virus capsid with enhanced neurotropism corrects a lysosomal transmembrane enzyme deficiency.

Tordo J, O'Leary C, Antunes ASLM, Palomar N, Aldrin-Kirk P, Basche M, Bennett A, D'Souza Z, Gleitz H, Godwin A, Holley RJ, Parker H, Liao AY, Rouse P, Youshani AS, Dridi L, Martins C, Levade T, Stacey KB, Davis DM, Dyer A, Clément N, Björklund T, Ali RR, Agbandje-McKenna M, Rahim AA, Pshezhetsky A, Waddington SN, Linden RM, Bigger BW, Henckaerts E.

Brain. 2018 Jul 1;141(7):2014-2031. doi: 10.1093/brain/awy126.


ASAH1-Related Disorders.

Dyment DA, Bennett SAL, Medin JA, Levade T.

In: Adam MP, Ardinger HH, Pagon RA, Wallace SE, Bean LJH, Stephens K, Amemiya A, editors. GeneReviews® [Internet]. Seattle (WA): University of Washington, Seattle; 1993-2019.
2018 Mar 29.


Report of the 12th Sphingolipid Club Meeting, Trabia, Italy (Sept. 7-10, 2017).

Levade T, Ghidoni R.

Naunyn Schmiedebergs Arch Pharmacol. 2018 Feb;391(2):111-113. doi: 10.1007/s00210-017-1452-6. Epub 2017 Dec 23. Review.


TNFα blockade overcomes resistance to anti-PD-1 in experimental melanoma.

Bertrand F, Montfort A, Marcheteau E, Imbert C, Gilhodes J, Filleron T, Rochaix P, Andrieu-Abadie N, Levade T, Meyer N, Colacios C, Ségui B.

Nat Commun. 2017 Dec 22;8(1):2256. doi: 10.1038/s41467-017-02358-7.


Chronic lung injury and impaired pulmonary function in a mouse model of acid ceramidase deficiency.

Yu FPS, Islam D, Sikora J, Dworski S, Gurka J, López-Vásquez L, Liu M, Kuebler WM, Levade T, Zhang H, Medin JA.

Am J Physiol Lung Cell Mol Physiol. 2018 Mar 1;314(3):L406-L420. doi: 10.1152/ajplung.00223.2017. Epub 2017 Nov 22.


LC-MS/MS multiplex analysis of lysosphingolipids in plasma and amniotic fluid: A novel tool for the screening of sphingolipidoses and Niemann-Pick type C disease.

Pettazzoni M, Froissart R, Pagan C, Vanier MT, Ruet S, Latour P, Guffon N, Fouilhoux A, Germain DP, Levade T, Vianey-Saban C, Piraud M, Cheillan D.

PLoS One. 2017 Jul 27;12(7):e0181700. doi: 10.1371/journal.pone.0181700. eCollection 2017.


PNPLA1 defects in patients with autosomal recessive congenital ichthyosis and KO mice sustain PNPLA1 irreplaceable function in epidermal omega-O-acylceramide synthesis and skin permeability barrier.

Pichery M, Huchenq A, Sandhoff R, Severino-Freire M, Zaafouri S, Opálka L, Levade T, Soldan V, Bertrand-Michel J, Lhuillier E, Serre G, Maruani A, Mazereeuw-Hautier J, Jonca N.

Hum Mol Genet. 2017 May 15;26(10):1787-1800. doi: 10.1093/hmg/ddx079.


Acid Ceramidase Deficiency in Mice Results in a Broad Range of Central Nervous System Abnormalities.

Sikora J, Dworski S, Jones EE, Kamani MA, Micsenyi MC, Sawada T, Le Faouder P, Bertrand-Michel J, Dupuy A, Dunn CK, Xuan ICY, Casas J, Fabrias G, Hampson DR, Levade T, Drake RR, Medin JA, Walkley SU.

Am J Pathol. 2017 Apr;187(4):864-883. doi: 10.1016/j.ajpath.2016.12.005.


[Papillon-Lefèvre syndrome: A new case].

Martinho S, Levade T, Fergelot P, Stephan JL.

Arch Pediatr. 2017 Apr;24(4):360-362. doi: 10.1016/j.arcped.2017.01.013. Epub 2017 Feb 24. French.


A Review of Gaucher Disease Pathophysiology, Clinical Presentation and Treatments.

Stirnemann J, Belmatoug N, Camou F, Serratrice C, Froissart R, Caillaud C, Levade T, Astudillo L, Serratrice J, Brassier A, Rose C, Billette de Villemeur T, Berger MG.

Int J Mol Sci. 2017 Feb 17;18(2). pii: E441. doi: 10.3390/ijms18020441. Review.


Liquid Chromatography-High Resolution Mass Spectrometry Method to Study Sphingolipid Metabolism Changes in Response to CD95L.

Bilal F, Pérès M, Le Faouder P, Dupuy A, Bertrand-Michel J, Andrieu-Abadie N, Levade T, Badran B, Daher A, Ségui B.

Methods Mol Biol. 2017;1557:213-217. doi: 10.1007/978-1-4939-6780-3_20.


Method to Measure Sphingomyelin Synthase Activity Changes in Response to CD95L.

Bilal F, Pérès M, Andrieu-Abadie N, Levade T, Badran B, Daher A, Ségui B.

Methods Mol Biol. 2017;1557:207-212. doi: 10.1007/978-1-4939-6780-3_19.


Acid Ceramidase Deficiency is characterized by a unique plasma cytokine and ceramide profile that is altered by therapy.

Dworski S, Lu P, Khan A, Maranda B, Mitchell JJ, Parini R, Di Rocco M, Hugle B, Yoshimitsu M, Magnusson B, Makay B, Arslan N, Guelbert N, Ehlert K, Jarisch A, Gardner-Medwin J, Dagher R, Terreri MT, Lorenco CM, Barillas-Arias L, Tanpaiboon P, Solyom A, Norris JS, He X, Schuchman EH, Levade T, Medin JA.

Biochim Biophys Acta Mol Basis Dis. 2017 Feb;1863(2):386-394. doi: 10.1016/j.bbadis.2016.11.031. Epub 2016 Dec 1.


Mitochondrial GSH replenishment as a potential therapeutic approach for Niemann Pick type C disease.

Torres S, Matías N, Baulies A, Nuñez S, Alarcon-Vila C, Martinez L, Nuño N, Fernandez A, Caballeria J, Levade T, Gonzalez-Franquesa A, Garcia-Rovés P, Balboa E, Zanlungo S, Fabrías G, Casas J, Enrich C, Garcia-Ruiz C, Fernández-Checa JC.

Redox Biol. 2017 Apr;11:60-72. doi: 10.1016/j.redox.2016.11.010. Epub 2016 Nov 20.


Downregulation of sphingosine kinase-1 induces protective tumor immunity by promoting M1 macrophage response in melanoma.

Mrad M, Imbert C, Garcia V, Rambow F, Therville N, Carpentier S, Ségui B, Levade T, Azar R, Marine JC, Diab-Assaf M, Colacios C, Andrieu-Abadie N.

Oncotarget. 2016 Nov 1;7(44):71873-71886. doi: 10.18632/oncotarget.12380.


Chromatin-Bound MDM2 Regulates Serine Metabolism and Redox Homeostasis Independently of p53.

Riscal R, Schrepfer E, Arena G, Cissé MY, Bellvert F, Heuillet M, Rambow F, Bonneil E, Sabourdy F, Vincent C, Ait-Arsa I, Levade T, Thibaut P, Marine JC, Portais JC, Sarry JE, Le Cam L, Linares LK.

Mol Cell. 2016 Jun 16;62(6):890-902. doi: 10.1016/j.molcel.2016.04.033. Epub 2016 Jun 2.


Brief Report: Peripheral Osteolysis in Adults Linked to ASAH1 (Acid Ceramidase) Mutations: A New Presentation of Farber's Disease.

Bonafé L, Kariminejad A, Li J, Royer-Bertrand B, Garcia V, Mahdavi S, Bozorgmehr B, Lachman RL, Mittaz-Crettol L, Campos-Xavier B, Nampoothiri S, Unger S, Rivolta C, Levade T, Superti-Furga A.

Arthritis Rheumatol. 2016 Sep;68(9):2323-7. doi: 10.1002/art.39659. Epub 2016 Jul 29.


Targeting TNF alpha as a novel strategy to enhance CD8+ T cell-dependent immune response in melanoma?

Bertrand F, Rochotte J, Colacios C, Montfort A, Andrieu-Abadie N, Levade T, Benoist H, Ségui B.

Oncoimmunology. 2015 Aug 12;5(1):e1068495. eCollection 2016.


[Hereditary peroxisomal diseases].

Astudillo L, Sabourdy F, Touati G, Levade T.

Presse Med. 2016 Mar;45(3):302-12. doi: 10.1016/j.lpm.2015.05.009. Epub 2016 Feb 18. Review. French.


Legionella pneumophila S1P-lyase targets host sphingolipid metabolism and restrains autophagy.

Rolando M, Escoll P, Nora T, Botti J, Boitez V, Bedia C, Daniels C, Abraham G, Stogios PJ, Skarina T, Christophe C, Dervins-Ravault D, Cazalet C, Hilbi H, Rupasinghe TW, Tull D, McConville MJ, Ong SY, Hartland EL, Codogno P, Levade T, Naderer T, Savchenko A, Buchrieser C.

Proc Natl Acad Sci U S A. 2016 Feb 16;113(7):1901-6. doi: 10.1073/pnas.1522067113. Epub 2016 Feb 1.


A sequential bioorthogonal dual strategy: ManNAl and SiaNAl as distinct tools to unravel sialic acid metabolic pathways.

Gilormini PA, Lion C, Vicogne D, Levade T, Potelle S, Mariller C, Guérardel Y, Biot C, Foulquier F.

Chem Commun (Camb). 2016 Feb 7;52(11):2318-21. doi: 10.1039/c5cc08838k.


Glucosylceramidases and malignancies in mammals.

Astudillo L, Therville N, Colacios C, Ségui B, Andrieu-Abadie N, Levade T.

Biochimie. 2016 Jun;125:267-80. doi: 10.1016/j.biochi.2015.11.009. Epub 2015 Nov 12. Review.


Acid ceramidase deficiency associated with spinal muscular atrophy with progressive myoclonic epilepsy.

Gan JJ, Garcia V, Tian J, Tagliati M, Parisi JE, Chung JM, Lewis R, Baloh R, Levade T, Pierson TM.

Neuromuscul Disord. 2015 Dec;25(12):959-63. doi: 10.1016/j.nmd.2015.09.007. Epub 2015 Sep 16.


A novel jaspine B-ceramide hybrid modulates sphingolipid metabolism.

Garcia V, Le Faouder P, Dupuy A, Levade T, Ballereau S, Génisson Y.

Chem Biodivers. 2015 Jul;12(7):1115-25. doi: 10.1002/cbdv.201400357.


Blocking Tumor Necrosis Factor α Enhances CD8 T-cell-Dependent Immunity in Experimental Melanoma.

Bertrand F, Rochotte J, Colacios C, Montfort A, Tilkin-Mariamé AF, Touriol C, Rochaix P, Lajoie-Mazenc I, Andrieu-Abadie N, Levade T, Benoist H, Ségui B.

Cancer Res. 2015 Jul 1;75(13):2619-28. doi: 10.1158/0008-5472.CAN-14-2524. Epub 2015 May 14.


Natural disease history and characterisation of SUMF1 molecular defects in ten unrelated patients with multiple sulfatase deficiency.

Sabourdy F, Mourey L, Le Trionnaire E, Bednarek N, Caillaud C, Chaix Y, Delrue MA, Dusser A, Froissart R, Garnotel R, Guffon N, Megarbane A, Ogier de Baulny H, Pédespan JM, Pichard S, Valayannopoulos V, Verloes A, Levade T.

Orphanet J Rare Dis. 2015 Mar 15;10:31. doi: 10.1186/s13023-015-0244-7.


Monogenic neurological disorders of sphingolipid metabolism.

Sabourdy F, Astudillo L, Colacios C, Dubot P, Mrad M, Ségui B, Andrieu-Abadie N, Levade T.

Biochim Biophys Acta. 2015 Aug;1851(8):1040-51. doi: 10.1016/j.bbalip.2015.01.010. Epub 2015 Feb 7. Review.


Refsum Disease Presenting with a Late-Onset Leukodystrophy.

Bompaire F, Marcaud V, Trionnaire EL, Sedel F, Levade T.

JIMD Rep. 2015;19:7-10. doi: 10.1007/8904_2014_355. Epub 2015 Jan 21.


Sphingolipids modulate the epithelial-mesenchymal transition in cancer.

Levade T, Andrieu-Abadie N, Micheau O, Legembre P, Ségui B.

Cell Death Discov. 2015 Oct 12;1:15001. doi: 10.1038/cddiscovery.2015.1. eCollection 2015. No abstract available.


Adult-onset genetic leukoencephalopathies: a MRI pattern-based approach in a comprehensive study of 154 patients.

Ayrignac X, Carra-Dalliere C, Menjot de Champfleur N, Denier C, Aubourg P, Bellesme C, Castelnovo G, Pelletier J, Audoin B, Kaphan E, de Seze J, Collongues N, Blanc F, Chanson JB, Magnin E, Berger E, Vukusic S, Durand-Dubief F, Camdessanche JP, Cohen M, Lebrun-Frenay C, Brassat D, Clanet M, Vermersch P, Zephir H, Outteryck O, Wiertlewski S, Laplaud DA, Ouallet JC, Brochet B, Goizet C, Debouverie M, Pittion S, Edan G, Deburghgraeve V, Le Page E, Verny C, Amati-Bonneau P, Bonneau D, Hannequin D, Guyant-Maréchal L, Derache N, Defer GL, Moreau T, Giroud M, Guennoc AM, Clavelou P, Taithe F, Mathis S, Neau JP, Magy L, Devoize JL, Bataillard M, Masliah-Planchon J, Dorboz I, Tournier-Lasserve E, Levade T, Boespflug Tanguy O, Labauge P.

Brain. 2015 Feb;138(Pt 2):284-92. doi: 10.1093/brain/awu353. Epub 2014 Dec 19.


Identification of novel CERT ligands as potential ceramide trafficking inhibitors.

Santos C, Rogriguez F, Garcia V, Moravčíková D, Berkeš D, Daïch A, Levade T, Baudoin-Dehoux C, Ballereau S, Génisson Y.

Chembiochem. 2014 Nov 24;15(17):2522-8. doi: 10.1002/cbic.201402366. Epub 2014 Sep 26.


New insights into the organ-specific adverse effects of fumonisin B1: comparison between lung and liver.

Loiseau N, Polizzi A, Dupuy A, Therville N, Rakotonirainy M, Loy J, Viadere JL, Cossalter AM, Bailly JD, Puel O, Kolf-Clauw M, Bertrand-Michel J, Levade T, Guillou H, Oswald IP.

Arch Toxicol. 2015 Sep;89(9):1619-29. doi: 10.1007/s00204-014-1323-6. Epub 2014 Aug 26.


Human genetic disorders of sphingolipid biosynthesis.

Astudillo L, Sabourdy F, Therville N, Bode H, Ségui B, Andrieu-Abadie N, Hornemann T, Levade T.

J Inherit Metab Dis. 2015 Jan;38(1):65-76. doi: 10.1007/s10545-014-9736-1. Epub 2014 Aug 21. Review.


On-tissue localization of ceramides and other sphingolipids by MALDI mass spectrometry imaging.

Jones EE, Dworski S, Canals D, Casas J, Fabrias G, Schoenling D, Levade T, Denlinger C, Hannun YA, Medin JA, Drake RR.

Anal Chem. 2014 Aug 19;86(16):8303-11. doi: 10.1021/ac501937d. Epub 2014 Aug 8.


Extensive Mongolian spots in 4p16.3 deletion (Wolf-Hirschhorn syndrome).

Ziegler A, Guichet A, Pinson L, Barth M, Levade T, Bonneau D, Colin E.

Clin Dysmorphol. 2014 Jul;23(3):109-10. doi: 10.1097/MCD.0000000000000041. No abstract available.


[Cerebrotendinous xanthomatosis: a multicentric retrospective study of 15 adults, clinical and paraclinical typical and atypical aspects].

Lionnet C, Carra C, Ayrignac X, Levade T, Gayraud D, Castelnovo G, Besson G, Androdias G, Vukusic S, Confavreux C, Zaenker C, De Seze J, Collongues N, Blanc F, Tranchant C, Wallon D, Hannequin D, Gerdelat-Mas A, Brassat D, Clanet M, Zephir H, Outteryck O, Vermersch P, Labauge P.

Rev Neurol (Paris). 2014 Jun-Jul;170(6-7):445-53. doi: 10.1016/j.neurol.2014.01.675. Epub 2014 Apr 16. French.


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