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It significantly improves life expectancy and delays progression to end-stage kidney disease; however, it cannot prevent it. Unfortunately, compliance to therapy is often weak, particularly during adolescence. Therefore, finding better treatments is a priority in the field of cystinosis. Previously, we found that genistein, an isoflavone particularly enriched in soy, can revert part of the cystinotic cellular phenotype that is not sensitive to cysteamine in vitro. To test the effects of genistein in vivo, we fed 2-month-old wild-type and Ctns\u2212\/\u2212 female mice with either a control diet, a genistein-containing diet or a cysteamine-containing diet for 14\u00a0months. Genistein (160\u00a0mg\/kg\/day) did not affect the growth of the mice or hepatic functionality. Compared with untreated mice at 16\u00a0months, Ctns\u2212\/\u2212 mice fed with genistein had lower cystine concentrations in their kidneys, reduced formation of cystine crystals, a smaller number of LAMP1-positive structures and an overall better-preserved parenchymal architecture. Cysteamine (400\u00a0mg\/kg\/day) was efficient in reverting the lysosomal phenotype and in preventing the development of renal lesions. These preclinical data indicate that genistein ameliorates kidney injury resulting from cystinosis with no side effects. Genistein therapy represents a potential treatment to improve the outcome for patients with cystinosis.<\/jats:p>","DOI":"10.1093\/hmg\/ddac266","type":"journal-article","created":{"date-parts":[[2022,10,27]],"date-time":"2022-10-27T07:27:56Z","timestamp":1666855676000},"page":"1090-1101","source":"Crossref","is-referenced-by-count":12,"title":["Genistein improves renal disease in a mouse model of nephropathic cystinosis: a comparison study with cysteamine"],"prefix":"10.1093","volume":"32","author":[{"given":"Ester","family":"De Leo","sequence":"first","affiliation":[{"name":"Renal Diseases Research Unit, Genetics and Rare Diseases Research Area, Bambino Ges\u00f9 Children\u2019s Hospital, IRCCS , 00146 Rome , Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anna","family":"Taranta","sequence":"additional","affiliation":[{"name":"Renal Diseases Research Unit, Genetics and Rare Diseases Research Area, Bambino Ges\u00f9 Children\u2019s Hospital, IRCCS , 00146 Rome 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