{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,30]],"date-time":"2026-10-30T21:10:51Z","timestamp":1793394651523,"version":"4.5.0"},"reference-count":26,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2019,10,3]],"date-time":"2019-10-03T00:00:00Z","timestamp":1570060800000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2019,10,3]],"date-time":"2019-10-03T00:00:00Z","timestamp":1570060800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Arthroplasty"],"published-print":{"date-parts":[[2019,12]]},"abstract":"<jats:title>Abstract<\/jats:title>\n              <jats:sec>\n                <jats:title>Background<\/jats:title>\n                <jats:p>The objective of this study is to share our experience in total hip replacement for the treatment of ochronotic hip arthritis, in particular to report how to establish the diagnosis and some tips to limit complications.<\/jats:p>\n              <\/jats:sec>\n              <jats:sec>\n                <jats:title>Method<\/jats:title>\n                <jats:p>A cohort comprised of 10 patients (12 hips) with alkaptonuric hip arthritis. There were six men and four women with the mean age of 62.80\u2009\u00b1\u20097.57\u2009years. All patients had a stiff spine, grossly restricted movements of hip joints, and severely limited daily routine activities. Total hip replacement was performed in all patients. The patients were evaluated at 6, 12, and 24\u2009months after surgery, as well as every 4\u2009years thereafter. Harris hip score was used to assess the functional outcome. The level of significance was set at <jats:italic>p<\/jats:italic>\u2009&lt;\u20090.05.<\/jats:p>\n              <\/jats:sec>\n              <jats:sec>\n                <jats:title>Results<\/jats:title>\n                <jats:p>The mean follow-up lasted 16.70\u2009\u00b1\u20096.82\u2009years (3 to 24\u2009years). At the final available follow-up, nine patients returned to work, ambulate without an orthosis, and achieve\u00a0complete pain relief. Harris hip score was improved from poor to excellent. One patient died 16\u2009years after surgery due to breast cancer. No complication relating to prosthetic failures was detected.<\/jats:p>\n              <\/jats:sec>\n              <jats:sec>\n                <jats:title>Conclusion<\/jats:title>\n                <jats:p>Total hip replacement gives long-term satisfactory results in patients with alkaptonuric hip arthritis, resulting in comparable function of the hips in patients who\u00a0undergo primary osteoarthrosis.<\/jats:p>\n              <\/jats:sec>","DOI":"10.1186\/s42836-019-0010-8","type":"journal-article","created":{"date-parts":[[2019,10,2]],"date-time":"2019-10-02T23:09:02Z","timestamp":1570057742000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":9,"title":["Primary hip arthroplasty for\u00a0the treatment of alkaptonuric hip arthritis: 3- to 24-year follow-ups"],"prefix":"10.1186","volume":"1","author":[{"given":"Javahir A.","family":"Pachore","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Vikram Indrajit","family":"Shah","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2640-0147","authenticated-orcid":false,"given":"Sachin","family":"Upadhyay","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kalpesh","family":"Shah","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ashish","family":"Sheth","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Amish","family":"Kshatriya","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2019,10,3]]},"reference":[{"key":"10_CR1","first-page":"81","volume":"25","author":"JM Keller","year":"2005","unstructured":"Keller JM, Macaulay W, Nercessian OA, Jaffe IA. New developments in ochronosis: review of the literature. Rheumatol lnt. 2005;25:81\u20135.","journal-title":"Rheumatol lnt"},{"key":"10_CR2","doi-asserted-by":"publisher","first-page":"355","DOI":"10.1080\/00016470410001321","volume":"75","author":"JM Spencer","year":"2004","unstructured":"Spencer JM, Gibbons CL, Sharp RJ, Carr AJ, Athanasou NA. Arthroplasty for ochronotic arthritis: no failure of 11 replacements in 3 patients followed 6-12 years. Acta Orthop Scand. 2004;75:355\u20138.","journal-title":"Acta Orthop Scand"},{"key":"10_CR3","first-page":"41","volume":"73","author":"EJ Collins","year":"2005","unstructured":"Collins EJ, Hand R. Alkaptonuric ochronosis: a case report. AANA J. 2005;73:41\u20136.","journal-title":"AANA J"},{"key":"10_CR4","doi-asserted-by":"publisher","first-page":"205","DOI":"10.1007\/s00296-002-0175-1","volume":"21","author":"P Borman","year":"2002","unstructured":"Borman P, Bodur H, Ciliz D. Ochronotic arthropathy. Rheumatol Int. 2002;21:205\u20139.","journal-title":"Rheumatol Int"},{"key":"10_CR5","doi-asserted-by":"publisher","first-page":"78","DOI":"10.1007\/s002960100136","volume":"21","author":"G Sahin","year":"2001","unstructured":"Sahin G, Milcan A, Bagis S, Kokturk A, Pata C, Erdogan C. A case of ochronosis: upper extremity involvement. Rheumatol Int. 2001;21:78\u201380.","journal-title":"Rheumatol Int"},{"key":"10_CR6","doi-asserted-by":"publisher","first-page":"883","DOI":"10.1302\/0301-620X.85B6.13662","volume":"85","author":"RV Kumar","year":"2003","unstructured":"Kumar RV, Rajasekaran S. Spontaneous tendon ruptures in alkaptonuria. J Bone Joint Surg (Br). 2003;85:883\u20136.","journal-title":"J Bone Joint Surg (Br)"},{"key":"10_CR7","doi-asserted-by":"publisher","first-page":"209","DOI":"10.3121\/cmr.2.4.209","volume":"2","author":"AA Fisher","year":"2004","unstructured":"Fisher AA, Davis MW. Alkaptonuric ochronosis with aortic valve and joint replacements and femoral fracture: a case report and literature review. Clin Med Res. 2004;2:209\u201315.","journal-title":"Clin Med Res"},{"key":"10_CR8","doi-asserted-by":"publisher","first-page":"437","DOI":"10.1007\/s10067-003-0760-z","volume":"22","author":"S Demir","year":"2003","unstructured":"Demir S. Alkaptonuric ochronosis: a case with multiple joint replacement arthroplasties. Clin Rheumatol. 2003;22:437.","journal-title":"Clin Rheumatol"},{"key":"10_CR9","doi-asserted-by":"publisher","first-page":"51","DOI":"10.1007\/8904_2016_554","volume":"31","author":"G Jacomelli","year":"2016","unstructured":"Jacomelli G, Micheli V, Bernardini G, Millucci L, Santucci A. Quick diagnosis of alkaptonuria by homogentisic acid determination in urine paper spots. JIMD Rep. 2016;31:51\u20136.","journal-title":"JIMD Rep"},{"key":"10_CR10","doi-asserted-by":"publisher","first-page":"5","DOI":"10.1006\/geno.1994.1003","volume":"19","author":"S Janocha","year":"1994","unstructured":"Janocha S, Wolz W, Srsen S, Srsnova K, Montagutelli X, Guenet JL, Grimm T, Kress W, Mueller CR. The human gene for alkaptonuria (AKU) maps to chromosome 3q. Genomics. 1994;19:5\u20138.","journal-title":"Genomics."},{"key":"10_CR11","doi-asserted-by":"publisher","first-page":"170","DOI":"10.1007\/s10067-002-8277-4","volume":"21","author":"K Nas","year":"2002","unstructured":"Nas K, G\u00fcr A, Akdeniz S, Cevik R, Harman M, Sara\u00e7 AJ. Ochronosis: a case of severe ochronoticarthropathy. Clin Rheumatol. 2002;21:170\u20132.","journal-title":"Clin Rheumatol"},{"key":"10_CR12","doi-asserted-by":"publisher","first-page":"732","DOI":"10.1016\/j.ejcts.2008.12.032","volume":"35","author":"P Wauthy","year":"2009","unstructured":"Wauthy P, Seghers V, Mathonet P, Deuvaert FE. Cardiac ochronosis: not so benign. Eur J Cardiothorac Surg. 2009;35:732\u20133.","journal-title":"Eur J Cardiothorac Surg"},{"key":"10_CR13","doi-asserted-by":"publisher","first-page":"274","DOI":"10.2106\/00004623-199502000-00016","volume":"77","author":"N Kusakabe","year":"1995","unstructured":"Kusakabe N, Tsuzuki N, Sonada M. Compression of the cervical cord due to alcaptonuric arthropathy of the atlanto-axial joint. A case report. J Bone Joint Surg Am. 1995;77:274\u20137.","journal-title":"J Bone Joint Surg Am"},{"key":"10_CR14","doi-asserted-by":"publisher","first-page":"2111","DOI":"10.1056\/NEJMoa021736","volume":"347","author":"C Phornphutkul","year":"2002","unstructured":"Phornphutkul C, Introne WJ, Perry MB, Bernardini I, Murphey MD, Fitzpatrick DL, Anderson PD, Huizing M, Anikster Y, Gerber LH, Gahl WA. Natural history of alkaptonuria. N Engl J Med. 2002;347:2111\u201321.","journal-title":"N Engl J Med"},{"key":"10_CR15","doi-asserted-by":"publisher","first-page":"653","DOI":"10.1302\/0301-620X.52B4.653","volume":"52","author":"FH Laskar","year":"1970","unstructured":"Laskar FH, Sargison KD. Ochronotic arthropathy. A review with four case reports. J Bone Joint Surg. 1970;52:653\u201366.","journal-title":"J Bone Joint Surg"},{"key":"10_CR16","doi-asserted-by":"publisher","first-page":"1141","DOI":"10.1007\/s10545-011-9374-9","volume":"34","author":"LR Ranganath","year":"2011","unstructured":"Ranganath LR, Cox TF. Natural history of alkaptonuria revisited: analyses based on scoring systems. J Inherit Metab Dis. 2011;34:1141\u201351.","journal-title":"J Inherit Metab Dis"},{"key":"10_CR17","doi-asserted-by":"publisher","first-page":"79","DOI":"10.1001\/archinte.1950.00230130101006","volume":"86","author":"EISENBERG H","year":"1950","unstructured":"EISENBERG H, et al. Arch Intern Med. 1950;86:79.","journal-title":"Arch Intern Med"},{"key":"10_CR18","doi-asserted-by":"publisher","first-page":"1169","DOI":"10.2106\/00004623-195941070-00001","volume":"41","author":"J Cervenansky","year":"1959","unstructured":"Cervenansky J, Sitaj S, Urbanek T. Alkaptonuria and ochronosis. J Bone Joint Surg. 1959;41:1169.","journal-title":"J Bone Joint Surg"},{"key":"10_CR19","first-page":"1787","volume-title":"Diagnosis of bone and joint disorders","author":"D Resnick","year":"1988","unstructured":"Resnick D. Alkaptonuria. In: Resnick D, Niwayama G, editors. Diagnosis of bone and joint disorders. 2nd ed. Philadelphia: W. B. Saunders; 1988. p. 1787\u2013803.","edition":"2"},{"key":"10_CR20","doi-asserted-by":"publisher","first-page":"813","DOI":"10.1016\/0002-9343(63)90089-5","volume":"34","author":"W O\u2019Brien","year":"1963","unstructured":"O\u2019Brien W, La DB, Bunim JJ. Biochemical, pathologic and clinical aspects of alcaptonuria, ochronosis, and chronotic arthropathy. Am J Med. 1963;34:813\u201338.","journal-title":"Am J Med"},{"key":"10_CR21","doi-asserted-by":"publisher","first-page":"481","DOI":"10.1002\/bjs.18004418708","volume":"44","author":"GK Rose","year":"1957","unstructured":"Rose GK. Ochronosis. Br J Surg. 1957;44:481.","journal-title":"Br J Surg"},{"key":"10_CR22","doi-asserted-by":"publisher","first-page":"27","DOI":"10.12659\/AJCR.890008","volume":"15","author":"O Cebesoy","year":"2014","unstructured":"Cebesoy O, Isik M, Subasi M, Kaya A, Bilgin F, Kaya O. Total hip replacement for an ochronotic patient: a technical trick. Am J Case Rep. 2014;15:27\u201330.","journal-title":"Am J Case Rep"},{"key":"10_CR23","doi-asserted-by":"publisher","first-page":"539","DOI":"10.1054\/arth.2000.4228","volume":"15","author":"S Aydogdu","year":"2000","unstructured":"Aydogdu S, Cullu E, Ozsoy MH, Sur H. Cementless total knee arthroplasty in ochronotic arthropathy: a case report with a 4-year follow-up. J Arthroplast. 2000;15:539\u201343.","journal-title":"J Arthroplast"},{"key":"10_CR24","first-page":"1005","volume":"19","author":"DA Carrier","year":"1990","unstructured":"Carrier DA, Harris CM. Bilateral hip and bilateral knee arthroplasties in a patient with ochronotic arthropathy. Orthop Rev. 1990;19:1005\u20139.","journal-title":"Orthop Rev"},{"key":"10_CR25","doi-asserted-by":"publisher","first-page":"138","DOI":"10.1097\/RHU.0b013e31819e6b41","volume":"15","author":"K Araki","year":"2009","unstructured":"Araki K, Sudo A, Hasegawa M, Uchida A. Devastating ochronotic arthropathy with successful bilateral hip and knee arthroplasties. J Clin Rheumatol. 2009;15:138\u201340.","journal-title":"J Clin Rheumatol"},{"key":"10_CR26","first-page":"26","volume":"50","author":"A Moslavac","year":"2003","unstructured":"Moslavac A, Moslavac S, Cop R. Case report of a patient with ochronosis and arthroplasty of the hip and both knees. Reumatizam. 2003;50:26\u20138.","journal-title":"Reumatizam."}],"container-title":["Arthroplasty"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/link.springer.com\/content\/pdf\/10.1186\/s42836-019-0010-8.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/link.springer.com\/article\/10.1186\/s42836-019-0010-8\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/link.springer.com\/content\/pdf\/10.1186\/s42836-019-0010-8.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2020,10,1]],"date-time":"2020-10-01T23:22:13Z","timestamp":1601594533000},"score":1,"resource":{"primary":{"URL":"https:\/\/arthroplasty.biomedcentral.com\/articles\/10.1186\/s42836-019-0010-8"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2019,10,3]]},"references-count":26,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2019,12]]}},"alternative-id":["10"],"URL":"https:\/\/doi.org\/10.1186\/s42836-019-0010-8","relation":{},"ISSN":["2524-7948"],"issn-type":[{"value":"2524-7948","type":"electronic"}],"subject":[],"published":{"date-parts":[[2019,10,3]]},"assertion":[{"value":"22 March 2019","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"12 August 2019","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"3 October 2019","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"The study was approved by the Scientific Review Committee and the institutional review board of the participating Health Service. Written Informed consent (about the surgical technique, risks and potential complications) was provided according to the Declaration of Helsinki and obtained from all participating patients.","order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"Informed consent was obtained from the patient\u2019s for publication of their case records for providing evidence-based scientific literature for further research.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors declare that they have no competing interests.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"8"}}