{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,12,10]],"date-time":"2025-12-10T12:39:49Z","timestamp":1765370389097,"version":"3.40.4"},"reference-count":17,"publisher":"S. Karger AG","issue":"10","license":[{"start":{"date-parts":[[2024,4,24]],"date-time":"2024-04-24T00:00:00Z","timestamp":1713916800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/karger.com\/pages\/terms-and-conditions"},{"start":{"date-parts":[[2024,4,24]],"date-time":"2024-04-24T00:00:00Z","timestamp":1713916800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/karger.com\/pages\/terms-and-conditions"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Nephron"],"abstract":"<jats:p>Introduction: Cystinuria (CYS) is the most common monogenic kidney stone disease. Methods: Starting from an unusual case of CYS associated to primary sclerosing cholangitis, inflammatory bowel disease (IBD), and autoimmune hepatitis in a young male, we carefully review the literature and propose here a working hypothesis regarding the potential risk of cystinuric patients to develop conditions due to immune system dysregulation. To corroborate this hypothesis, we retrospectively evaluate the frequency of dysimmunity in a monocentric cohort including 36 cystinuric patients compared to healthy and disease controls. Results: CYS patients have an increased prevalence of atopic disease compared to disease controls (p\u00a0= 0.03) and 16.7% of CYS subjects were diagnosed with allergic disease to a variety of antigens. Conclusion: Further studies are needed to define the relationship between proximal tubular transport defect of CYS and dysregulated immunity. <\/jats:p>","DOI":"10.1159\/000538213","type":"journal-article","created":{"date-parts":[[2024,4,24]],"date-time":"2024-04-24T21:00:41Z","timestamp":1713992441000},"page":"687-692","source":"Crossref","is-referenced-by-count":1,"title":["Renal Cystinuria and Immune Cells (T Lymphocytes) Dysfunction: What We Know about?"],"prefix":"10.1159","volume":"148","author":[{"given":"Francesca","family":"Caprio","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Giuseppe","family":"Orefice","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Floriana","family":"Secondulfo","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Filippo","family":"Carone Fabiani","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Anna","family":"Iervolino","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Giovambattista","family":"Capasso","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mariadelina","family":"Simeoni","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Miriam","family":"Zacchia","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Francesco","family":"Trepiccione","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Giovanna","family":"Capolongo","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"127","published-online":{"date-parts":[[2024,4,24]]},"reference":[{"key":"ref1","doi-asserted-by":"publisher","DOI":"10.1007\/s00467-021-05342-y"},{"key":"ref2","doi-asserted-by":"publisher","DOI":"10.1038\/ng0494-420"},{"key":"ref3","doi-asserted-by":"publisher","DOI":"10.1007\/s12041-011-0045-2"},{"key":"ref4","doi-asserted-by":"publisher","DOI":"10.1007\/s00240-018-1101-7"},{"key":"ref5","doi-asserted-by":"publisher","DOI":"10.1016\/j.kint.2020.06.035"},{"key":"ref6","doi-asserted-by":"publisher","DOI":"10.1111\/j.1399-0004.2011.01638.x"},{"key":"ref7","doi-asserted-by":"publisher","DOI":"10.1038\/s41467-020-18184-3"},{"key":"ref8","doi-asserted-by":"publisher","DOI":"10.1016\/j.ucl.2007.04.006"},{"key":"ref9","doi-asserted-by":"publisher","DOI":"10.1038\/nrneph.2010.69"},{"key":"ref10","doi-asserted-by":"publisher","DOI":"10.1053\/j.gastro.2007.11.001"},{"key":"ref11","doi-asserted-by":"publisher","DOI":"10.1042\/BJ20150846"},{"key":"ref12","doi-asserted-by":"publisher","DOI":"10.1016\/s0006-291x(03)00808-8"},{"key":"ref13","doi-asserted-by":"publisher","DOI":"10.1158\/0008-5472.CAN-09-2587"},{"key":"ref14","doi-asserted-by":"publisher","DOI":"10.1016\/j.immuni.2014.04.007"},{"key":"ref15","doi-asserted-by":"publisher","DOI":"10.1111\/cea.12030"},{"key":"ref16","doi-asserted-by":"publisher","DOI":"10.1111\/bjd.20628"},{"key":"ref17","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2230.2010.03840.x"}],"container-title":["Nephron"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/karger.com\/nef\/article-pdf\/148\/10\/687\/4287203\/000538213.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/karger.com\/nef\/article-pdf\/148\/10\/687\/4287203\/000538213.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,4,23]],"date-time":"2025-04-23T15:10:51Z","timestamp":1745421051000},"score":1,"resource":{"primary":{"URL":"https:\/\/karger.com\/article\/doi\/10.1159\/000538213"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,4,24]]},"references-count":17,"journal-issue":{"issue":"10","published-online":{"date-parts":[[2024,10,8]]},"published-print":{"date-parts":[[2024,9,9]]}},"URL":"https:\/\/doi.org\/10.1159\/000538213","archive":["Portico"],"relation":{},"ISSN":["1660-8151","2235-3186"],"issn-type":[{"type":"print","value":"1660-8151"},{"type":"electronic","value":"2235-3186"}],"subject":[],"published":{"date-parts":[[2024,4,24]]}}}