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GLP-1R agonists demonstrate potential to treat Wolfram syndrome in human preclinical models.
Gorgogietas V, Rajaei B, Heeyoung C, Santacreu BJ, Marín-Cañas S, Salpea P, Sawatani T, Musuaya A, Arroyo MN, Moreno-Castro C, Benabdallah K, Demarez C, Toivonen S, Cosentino C, Pachera N, Lytrivi M, Cai Y, Carnel L, Brown C, Urano F, Marchetti P, Gilon P, Eizirik DL, Cnop M, Igoillo-Esteve M. Gorgogietas V, et al. Among authors: brown c. Diabetologia. 2023 Jul;66(7):1306-1321. doi: 10.1007/s00125-023-05905-8. Epub 2023 Mar 30. Diabetologia. 2023. PMID: 36995380 Free PMC article.
Genotype and Clinical Characteristics of Patients with Wolfram Syndrome and WFS1-related Disorders.
Lee EM, Verma M, Palaniappan N, Pope EM, Lee S, Blacher L, Neerumalla P, An W, Campbell T, Brown C, Hurst S, Marshall B, Hershey T, Nunes V, de Heredia ML, Urano F. Lee EM, et al. Among authors: brown c. medRxiv [Preprint]. 2023 Feb 16:2023.02.15.23284904. doi: 10.1101/2023.02.15.23284904. medRxiv. 2023. PMID: 36824811 Free PMC article. Updated. Preprint.
Wolframin is a novel regulator of tau pathology and neurodegeneration.
Chen S, Acosta D, Li L, Liang J, Chang Y, Wang C, Fitzgerald J, Morrison C, Goulbourne CN, Nakano Y, Villegas NCH, Venkataraman L, Brown C, Serrano GE, Bell E, Wemlinger T, Wu M, Kokiko-Cochran ON, Popovich P, Flowers XE, Honig LS, Vonsattel JP, Scharre DW, Beach TG, Ma Q, Kuret J, Kõks S, Urano F, Duff KE, Fu H. Chen S, et al. Among authors: brown c. Acta Neuropathol. 2022 May;143(5):547-569. doi: 10.1007/s00401-022-02417-4. Epub 2022 Apr 7. Acta Neuropathol. 2022. PMID: 35389045
Multidimensional analysis and therapeutic development using patient iPSC-derived disease models of Wolfram syndrome.
Kitamura RA, Maxwell KG, Ye W, Kries K, Brown CM, Augsornworawat P, Hirsch Y, Johansson MM, Weiden T, Ekstein J, Cohen J, Klee J, Leslie K, Simeonov A, Henderson MJ, Millman JR, Urano F. Kitamura RA, et al. Among authors: brown cm. JCI Insight. 2022 Sep 22;7(18):e156549. doi: 10.1172/jci.insight.156549. JCI Insight. 2022. PMID: 36134655 Free PMC article.
A phase Ib/IIa clinical trial of dantrolene sodium in patients with Wolfram syndrome.
Abreu D, Stone SI, Pearson TS, Bucelli RC, Simpson AN, Hurst S, Brown CM, Kries K, Onwumere C, Gu H, Hoekel J, Tychsen L, Van Stavern GP, White NH, Marshall BA, Hershey T, Urano F. Abreu D, et al. Among authors: brown cm. JCI Insight. 2021 Aug 9;6(15):e145188. doi: 10.1172/jci.insight.145188. JCI Insight. 2021. PMID: 34185708 Free PMC article. Clinical Trial.
A calcium-dependent protease as a potential therapeutic target for Wolfram syndrome.
Lu S, Kanekura K, Hara T, Mahadevan J, Spears LD, Oslowski CM, Martinez R, Yamazaki-Inoue M, Toyoda M, Neilson A, Blanner P, Brown CM, Semenkovich CF, Marshall BA, Hershey T, Umezawa A, Greer PA, Urano F. Lu S, et al. Among authors: brown cm. Proc Natl Acad Sci U S A. 2014 Dec 9;111(49):E5292-301. doi: 10.1073/pnas.1421055111. Epub 2014 Nov 24. Proc Natl Acad Sci U S A. 2014. PMID: 25422446 Free PMC article.