{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,8]],"date-time":"2026-07-08T03:07:17Z","timestamp":1783480037740,"version":"3.55.0"},"reference-count":0,"publisher":"SAGE Publications","issue":"1","license":[{"start":{"date-parts":[[2003,1,1]],"date-time":"2003-01-01T00:00:00Z","timestamp":1041379200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Ann Clin Biochem"],"published-print":{"date-parts":[[2003,1,1]]},"abstract":"<jats:p>There is no definitive treatment protocol for alkaptonuria. A patient with alkaptonuria was treated with ascorbic acid (0\u00b75 g\/day) from the age of 4 years. He developed episodes of severe recurrent joint pain at 9\u00b75 years of age after which a protein-restricted diet (1\u00b73 g\/kg\/day) was started. Protein restriction in combination with ascorbic acid therapy (1 g\/day in two divided doses) resulted in a significant decrease but not a normalization of the urinary homogentisic acid excretion. Joint pain resolved and the radiological evidence of 'moth-eaten' irregularities on the articular surface in both knees disappeared. He is currently well, growing normally and in nitrogen balance. Our findings document a reversal of bone abnormalities and clinical symptoms in a case of alkaptonuria. The results should be confirmed in a larger study. We suggest that protein restriction should be applied in combination with ascorbic acid in affected patients as soon as joint pain occurs.<\/jats:p>","DOI":"10.1258\/000456303321016268","type":"journal-article","created":{"date-parts":[[2003,1,23]],"date-time":"2003-01-23T22:39:03Z","timestamp":1043361543000},"page":"108-111","source":"Crossref","is-referenced-by-count":27,"title":["Reversal of clinical symptoms and radiographic abnormalities with protein restriction and ascorbic acid in alkaptonuria"],"prefix":"10.1177","volume":"40","author":[{"given":"Eva","family":"Morava","sequence":"first","affiliation":[{"name":"University of P\u00e9cs, Department of Medical Genetics and Child Development, P\u00e9cs, Hungary & Departments of Pediatrics and Neurology, University Medical Centre Nijmegen, Nijmegen, The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gy\u00f6rgy","family":"Kosztol\u00e1nyi","sequence":"additional","affiliation":[{"name":"University of P\u00e9cs, Department of Medical Genetics and Child Development, P\u00e9cs, Hungary"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Udo F.H.","family":"Engelke","sequence":"additional","affiliation":[{"name":"Departments of Pediatrics and Neurology, University Medical Centre Nijmegen, Nijmegen, The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ron A.","family":"Wevers","sequence":"additional","affiliation":[{"name":"Departments of Pediatrics and Neurology, University Medical Centre Nijmegen, Nijmegen, The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2003,1,1]]},"container-title":["Annals of Clinical Biochemistry: International Journal of Laboratory Medicine"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1258\/000456303321016268","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1258\/000456303321016268","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,4,28]],"date-time":"2026-04-28T11:52:34Z","timestamp":1777377154000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.sagepub.com\/doi\/10.1258\/000456303321016268"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2003,1,1]]},"references-count":0,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2003,1,1]]}},"alternative-id":["10.1258\/000456303321016268"],"URL":"https:\/\/doi.org\/10.1258\/000456303321016268","relation":{},"ISSN":["0004-5632","1758-1001"],"issn-type":[{"value":"0004-5632","type":"print"},{"value":"1758-1001","type":"electronic"}],"subject":[],"published":{"date-parts":[[2003,1,1]]}}}