{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,8,29]],"date-time":"2025-08-29T00:03:13Z","timestamp":1756425793247,"version":"3.44.0"},"reference-count":33,"publisher":"BMJ","issue":"10","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Case Rep"],"accepted":{"date-parts":[[2023,10,8]]},"published-print":{"date-parts":[[2023,10]]},"abstract":"<jats:p>Alkaptonuria is a very rare disorder in which homogentisic acid accumulates due to a deficiency in the activity of homogentisic acid 1,2 dioxygenase. This deficiency results in deposition of a yellowish\u2013brown pigment in connective tissue. Such deposition is termed \u2018ochronosis\u2019 and leads to deterioration in the formation and structure of proteoglycans in hyaline cartilage. These actions lead to fragmentation and rapid destructive arthritis. Often, ochronotic arthritis appears at 40\u201360 years of age, and many patients are treated symptomatically. Here, we report two patients (three ankles) with ochronotic arthritis who were treated with ankle arthrodesis. In all cases, the postoperative clinical score improved, but the time needed for fusion was prolonged and symptomatic subtalar arthropathy developed in the early postoperative period.<\/jats:p>","DOI":"10.1136\/bcr-2022-254300","type":"journal-article","created":{"date-parts":[[2023,10,25]],"date-time":"2023-10-25T21:30:17Z","timestamp":1698269417000},"page":"e254300","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":0,"title":["Arthroscopic ankle arthrodesis in two alkaptonuria patients"],"prefix":"10.1136","volume":"16","author":[{"given":"Seiya","family":"Tomonaga","sequence":"first","affiliation":[{"name":"Department of Orthopedic Surgery, Faculty of Medicine, Fukuoka University, Fukuoka, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7829-3611","authenticated-orcid":false,"given":"Ichiro","family":"Yoshimura","sequence":"additional","affiliation":[{"name":"Faculty of Sports and Health Science, Fukuoka University, Fukuoka, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kazuki","family":"Kanazawa","sequence":"additional","affiliation":[{"name":"Department of Orthopedic Surgery, Fukuoka Seisyukai Hospital, Fukuoka, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Takuaki","family":"Yamamoto","sequence":"additional","affiliation":[{"name":"Department of Orthopedic Surgery, Faculty of Medicine, Fukuoka University, Fukuoka, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"239","published-online":{"date-parts":[[2023,10,25]]},"reference":[{"key":"2025082800564866000_16.10.e254300.1","doi-asserted-by":"crossref","DOI":"10.1097\/MD.0000000000016837","article-title":"Ochronotic arthritis and Ochronotic Achilles tendon rupture in Alkaptonuria: A 6 years follow-up case report in China","volume":"98","author":"Jiang","year":"2019","journal-title":"Medicine (Baltimore)"},{"key":"2025082800564866000_16.10.e254300.2","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa021736"},{"key":"2025082800564866000_16.10.e254300.3","doi-asserted-by":"crossref","first-page":"776","DOI":"10.1002\/jimd.12152","article-title":"Ochronotic Pigmentation is caused by Homogentisic acid and is the key event in Alkaptonuria leading to the destructive consequences of the disease-A review","volume":"42","author":"Ranganath","year":"2019","journal-title":"J Inherit Metab Dis"},{"key":"2025082800564866000_16.10.e254300.4","doi-asserted-by":"crossref","first-page":"1389","DOI":"10.1007\/s10067-014-2557-7","article-title":"Ochronotic Arthropathy as a paradigm of Metabolically induced degenerative joint disease. 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