{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,18]],"date-time":"2026-09-18T04:29:06Z","timestamp":1789705746148,"version":"4.0.1"},"reference-count":10,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2018,9,19]],"date-time":"2018-09-19T00:00:00Z","timestamp":1537315200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Journal of Forensic Sciences"],"published-print":{"date-parts":[[2019,5]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Ochronosis is the blue\u2010gray discoloration of collagen\u2010containing tissues due to homogentisic acid (<jats:styled-content style=\"fixed-case\">HGA<\/jats:styled-content>) deposition, secondary to endogenous alkaptonuria or exogenous enzyme inhibition. In renal disease, accumulation of <jats:styled-content style=\"fixed-case\">HGA<\/jats:styled-content> in serum can cause methemoglobinemia. A 60\u2010year\u2010old woman with renal disease and anemia presented with 3 days of weakness and months of gray skin discoloration. Her hemoglobin was 8.1g\/dl with 24.5% methemoglobin. Despite treatment with methylene blue, exchange transfusion, and continuous renal replacement therapy, the patient died. Autopsy revealed gray discoloration and ochronotic pigment in the ribs and cartilage. Based on these findings, the patient was diagnosed with ochronosis, suggestive of alkaptonuria, complicated by methemoglobinemia. The differential diagnosis for blue\u2010gray skin discoloration includes argyria, methemoglobinemia, and ochronosis. This patient's clinical and autopsy findings suggested alkaptonuria complicated by methemoglobinemia due to progressive renal dysfunction. Development of methemoglobinemia in the setting of chronic skin discoloration and renal failure should prompt consideration of alkaptonuria.<\/jats:p>","DOI":"10.1111\/1556-4029.13907","type":"journal-article","created":{"date-parts":[[2018,9,19]],"date-time":"2018-09-19T10:41:25Z","timestamp":1537353685000},"page":"913-916","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":10,"title":["Ochronosis Presenting as Methemoglobinemia"],"prefix":"10.1111","volume":"64","author":[{"given":"Sarah B.","family":"Hugar","sequence":"first","affiliation":[{"name":"Department of Pathology University of Pittsburgh Medical Center Pittsburgh PA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joshua","family":"Shulman","sequence":"additional","affiliation":[{"name":"Department of Emergency Medicine University of Pittsburgh Medical Center Pittsburgh PA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joseph","family":"Yanta","sequence":"additional","affiliation":[{"name":"Department of Emergency Medicine University of Pittsburgh Medical Center Pittsburgh PA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jeffrey","family":"Nine","sequence":"additional","affiliation":[{"name":"Department of Pathology University of Pittsburgh Medical Center Pittsburgh PA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2018,9,19]]},"reference":[{"key":"e_1_2_5_2_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa021736"},{"key":"e_1_2_5_3_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-4632.2007.03351.x"},{"key":"e_1_2_5_4_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-4632.2008.02719.x"},{"key":"e_1_2_5_5_1","doi-asserted-by":"publisher","DOI":"10.2169\/internalmedicine.53.1938"},{"key":"e_1_2_5_6_1","doi-asserted-by":"publisher","DOI":"10.1055\/s-2008-1039313"},{"key":"e_1_2_5_7_1","doi-asserted-by":"publisher","DOI":"10.1093\/ckj\/sfu121"},{"key":"e_1_2_5_8_1","doi-asserted-by":"publisher","DOI":"10.1007\/s12024-018-9965-y"},{"key":"e_1_2_5_9_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10545-015-9902-0"},{"key":"e_1_2_5_10_1","doi-asserted-by":"publisher","DOI":"10.4103\/0971-4065.134681"},{"key":"e_1_2_5_11_1","doi-asserted-by":"publisher","DOI":"10.1097\/01.paf.0000203265.61137.88"}],"container-title":["Journal of Forensic Sciences"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2F1556-4029.13907","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/1556-4029.13907","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/1556-4029.13907","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/1556-4029.13907","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,9]],"date-time":"2023-09-09T17:05:47Z","timestamp":1694279147000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/1556-4029.13907"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2018,9,19]]},"references-count":10,"journal-issue":{"issue":"3","published-print":{"date-parts":[[2019,5]]}},"alternative-id":["10.1111\/1556-4029.13907"],"URL":"https:\/\/doi.org\/10.1111\/1556-4029.13907","archive":["Portico"],"relation":{},"ISSN":["0022-1198","1556-4029"],"issn-type":[{"value":"0022-1198","type":"print"},{"value":"1556-4029","type":"electronic"}],"subject":[],"published":{"date-parts":[[2018,9,19]]},"assertion":[{"value":"2018-05-15","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2018-08-21","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2018-09-19","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}