A Rare Case of Acquired Methemoglobinemia Associated with Alkaptonuria
We herein present a rare case of acquired methemoglobinemia associated with alkaptonuria. Alkaptonuria is a congenital error of metabolism caused by the deficiency of homogentisic acid oxidase, which subsequently results in the accumulation of homogentisic acid (HGA) in body tissues. As renal dysfun...
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| Veröffentlicht in: | Internal Medicine 2014, Vol.53(16), pp.1797-1800 |
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| container_title | Internal Medicine |
| container_volume | 53 |
| creator | Isa, Yasuki Nihei, Shun-ichi Irifukuhama, Yuna Ikeda, Tomoya Matsumoto, Hiroyuki Nagata, Keiji Harayama, Nobuya Aibara, Keiji Kamochi, Masayuki |
| description | We herein present a rare case of acquired methemoglobinemia associated with alkaptonuria. Alkaptonuria is a congenital error of metabolism caused by the deficiency of homogentisic acid oxidase, which subsequently results in the accumulation of homogentisic acid (HGA) in body tissues. As renal dysfunction progresses, the level of HGA excretion in the urine decreases and the blood concentration of HGA increases. HGA oxidizes oxyhemoglobin to methemoglobin, which can induce multiple organ failure accompanied by tissue hypoxia, intravascular hemolysis and metabolic acidosis. The mortality of this disease is high when alkaptonuria is associated with the presence of methemoglobinemia; therefore, treatment should be carefully planned in such cases. |
| doi_str_mv | 10.2169/internalmedicine.53.1938 |
| format | Article |
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Alkaptonuria is a congenital error of metabolism caused by the deficiency of homogentisic acid oxidase, which subsequently results in the accumulation of homogentisic acid (HGA) in body tissues. As renal dysfunction progresses, the level of HGA excretion in the urine decreases and the blood concentration of HGA increases. HGA oxidizes oxyhemoglobin to methemoglobin, which can induce multiple organ failure accompanied by tissue hypoxia, intravascular hemolysis and metabolic acidosis. The mortality of this disease is high when alkaptonuria is associated with the presence of methemoglobinemia; therefore, treatment should be carefully planned in such cases.</description><identifier>ISSN: 0918-2918</identifier><identifier>EISSN: 1349-7235</identifier><identifier>DOI: 10.2169/internalmedicine.53.1938</identifier><identifier>PMID: 25130113</identifier><language>eng</language><publisher>Japan: The Japanese Society of Internal Medicine</publisher><subject>acute kidney injury ; Aged ; alkaptonuria ; Alkaptonuria - complications ; Alkaptonuria - diagnosis ; Alkaptonuria - metabolism ; Fatal Outcome ; Female ; hemolysis ; homogentisic acid ; Homogentisic Acid - metabolism ; Humans ; Kidney Failure, Chronic - etiology ; methemoglobinemia ; Methemoglobinemia - diagnosis ; Methemoglobinemia - drug therapy ; Methemoglobinemia - etiology ; methylene blue ; Rare Diseases</subject><ispartof>Internal Medicine, 2014, Vol.53(16), pp.1797-1800</ispartof><rights>2014 by The Japanese Society of Internal Medicine</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c527t-9f91b11a24092f787a0af0c2b99de1e53ec34ccf087a08829464fc57bcbe8dcd3</citedby><cites>FETCH-LOGICAL-c527t-9f91b11a24092f787a0af0c2b99de1e53ec34ccf087a08829464fc57bcbe8dcd3</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>314,776,780,1877,27901,27902</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/25130113$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Isa, Yasuki</creatorcontrib><creatorcontrib>Nihei, Shun-ichi</creatorcontrib><creatorcontrib>Irifukuhama, Yuna</creatorcontrib><creatorcontrib>Ikeda, Tomoya</creatorcontrib><creatorcontrib>Matsumoto, Hiroyuki</creatorcontrib><creatorcontrib>Nagata, Keiji</creatorcontrib><creatorcontrib>Harayama, Nobuya</creatorcontrib><creatorcontrib>Aibara, Keiji</creatorcontrib><creatorcontrib>Kamochi, Masayuki</creatorcontrib><title>A Rare Case of Acquired Methemoglobinemia Associated with Alkaptonuria</title><title>Internal Medicine</title><addtitle>Intern. Med.</addtitle><description>We herein present a rare case of acquired methemoglobinemia associated with alkaptonuria. Alkaptonuria is a congenital error of metabolism caused by the deficiency of homogentisic acid oxidase, which subsequently results in the accumulation of homogentisic acid (HGA) in body tissues. As renal dysfunction progresses, the level of HGA excretion in the urine decreases and the blood concentration of HGA increases. HGA oxidizes oxyhemoglobin to methemoglobin, which can induce multiple organ failure accompanied by tissue hypoxia, intravascular hemolysis and metabolic acidosis. The mortality of this disease is high when alkaptonuria is associated with the presence of methemoglobinemia; therefore, treatment should be carefully planned in such cases.</description><subject>acute kidney injury</subject><subject>Aged</subject><subject>alkaptonuria</subject><subject>Alkaptonuria - complications</subject><subject>Alkaptonuria - diagnosis</subject><subject>Alkaptonuria - metabolism</subject><subject>Fatal Outcome</subject><subject>Female</subject><subject>hemolysis</subject><subject>homogentisic acid</subject><subject>Homogentisic Acid - metabolism</subject><subject>Humans</subject><subject>Kidney Failure, Chronic - etiology</subject><subject>methemoglobinemia</subject><subject>Methemoglobinemia - diagnosis</subject><subject>Methemoglobinemia - drug therapy</subject><subject>Methemoglobinemia - etiology</subject><subject>methylene blue</subject><subject>Rare Diseases</subject><issn>0918-2918</issn><issn>1349-7235</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2014</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNptkMtOwzAURC0EoqXwC8hLNil-xEm8jCpeUlElBGvLcW5alzxa2xHi70nU0gViM3cxZ-5IgxCmZM5oIu9tG8C1um6gtMa2MBd8TiXPztCU8lhGKePiHE2JpFnEBpmgK--3hPAslewSTZignFDKp-gxx2_aAV5oD7ircG72vXVQ4lcIG2i6dd0VQ0FjNc6974zVYTC_bNjgvP7Uu9C1vbP6Gl1UuvZwc7wz9PH48L54jparp5dFvoyMYGmIZCVpQalmMZGsSrNUE10RwwopS6AgOBgeG1OR0ckyJuMkroxIC1NAVpqSz9Dd4e_OdfsefFCN9QbqWrfQ9V5RIeI4YSRNBzQ7oMZ13juo1M7ZRrtvRYkaV1R_V1SCq3HFIXp7bOmLwTwFf2cbgNUB2Pqg13ACtAvW1PD_52TUY8WJNBvtFLT8BwYhkLg</recordid><startdate>20140101</startdate><enddate>20140101</enddate><creator>Isa, Yasuki</creator><creator>Nihei, Shun-ichi</creator><creator>Irifukuhama, Yuna</creator><creator>Ikeda, Tomoya</creator><creator>Matsumoto, Hiroyuki</creator><creator>Nagata, Keiji</creator><creator>Harayama, Nobuya</creator><creator>Aibara, Keiji</creator><creator>Kamochi, Masayuki</creator><general>The Japanese Society of Internal Medicine</general><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7X8</scope></search><sort><creationdate>20140101</creationdate><title>A Rare Case of Acquired Methemoglobinemia Associated with Alkaptonuria</title><author>Isa, Yasuki ; Nihei, Shun-ichi ; Irifukuhama, Yuna ; Ikeda, Tomoya ; Matsumoto, Hiroyuki ; Nagata, Keiji ; Harayama, Nobuya ; Aibara, Keiji ; Kamochi, Masayuki</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c527t-9f91b11a24092f787a0af0c2b99de1e53ec34ccf087a08829464fc57bcbe8dcd3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2014</creationdate><topic>acute kidney injury</topic><topic>Aged</topic><topic>alkaptonuria</topic><topic>Alkaptonuria - complications</topic><topic>Alkaptonuria - diagnosis</topic><topic>Alkaptonuria - metabolism</topic><topic>Fatal Outcome</topic><topic>Female</topic><topic>hemolysis</topic><topic>homogentisic acid</topic><topic>Homogentisic Acid - metabolism</topic><topic>Humans</topic><topic>Kidney Failure, Chronic - etiology</topic><topic>methemoglobinemia</topic><topic>Methemoglobinemia - diagnosis</topic><topic>Methemoglobinemia - drug therapy</topic><topic>Methemoglobinemia - etiology</topic><topic>methylene blue</topic><topic>Rare Diseases</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Isa, Yasuki</creatorcontrib><creatorcontrib>Nihei, Shun-ichi</creatorcontrib><creatorcontrib>Irifukuhama, Yuna</creatorcontrib><creatorcontrib>Ikeda, Tomoya</creatorcontrib><creatorcontrib>Matsumoto, Hiroyuki</creatorcontrib><creatorcontrib>Nagata, Keiji</creatorcontrib><creatorcontrib>Harayama, Nobuya</creatorcontrib><creatorcontrib>Aibara, Keiji</creatorcontrib><creatorcontrib>Kamochi, Masayuki</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>MEDLINE - Academic</collection><jtitle>Internal Medicine</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Isa, Yasuki</au><au>Nihei, Shun-ichi</au><au>Irifukuhama, Yuna</au><au>Ikeda, Tomoya</au><au>Matsumoto, Hiroyuki</au><au>Nagata, Keiji</au><au>Harayama, Nobuya</au><au>Aibara, Keiji</au><au>Kamochi, Masayuki</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>A Rare Case of Acquired Methemoglobinemia Associated with Alkaptonuria</atitle><jtitle>Internal Medicine</jtitle><addtitle>Intern. Med.</addtitle><date>2014-01-01</date><risdate>2014</risdate><volume>53</volume><issue>16</issue><spage>1797</spage><epage>1800</epage><pages>1797-1800</pages><issn>0918-2918</issn><eissn>1349-7235</eissn><abstract>We herein present a rare case of acquired methemoglobinemia associated with alkaptonuria. Alkaptonuria is a congenital error of metabolism caused by the deficiency of homogentisic acid oxidase, which subsequently results in the accumulation of homogentisic acid (HGA) in body tissues. As renal dysfunction progresses, the level of HGA excretion in the urine decreases and the blood concentration of HGA increases. HGA oxidizes oxyhemoglobin to methemoglobin, which can induce multiple organ failure accompanied by tissue hypoxia, intravascular hemolysis and metabolic acidosis. The mortality of this disease is high when alkaptonuria is associated with the presence of methemoglobinemia; therefore, treatment should be carefully planned in such cases.</abstract><cop>Japan</cop><pub>The Japanese Society of Internal Medicine</pub><pmid>25130113</pmid><doi>10.2169/internalmedicine.53.1938</doi><tpages>4</tpages><oa>free_for_read</oa></addata></record> |
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| ispartof | Internal Medicine, 2014, Vol.53(16), pp.1797-1800 |
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| language | eng |
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| subjects | acute kidney injury Aged alkaptonuria Alkaptonuria - complications Alkaptonuria - diagnosis Alkaptonuria - metabolism Fatal Outcome Female hemolysis homogentisic acid Homogentisic Acid - metabolism Humans Kidney Failure, Chronic - etiology methemoglobinemia Methemoglobinemia - diagnosis Methemoglobinemia - drug therapy Methemoglobinemia - etiology methylene blue Rare Diseases |
| title | A Rare Case of Acquired Methemoglobinemia Associated with Alkaptonuria |
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