Febuxostat for the Prevention of Recurrent 2,8-dihydroxyadenine Nephropathy due to Adenine Phosphoribosyltransferase Deficiency Following Kidney Transplantation
Adenine phosphoribosyltransferase (APRT) deficiency is a rare autosomal recessive disorder that results in irreversible renal damage due to 2,8-dihydroxyadenine (DHA) nephropathy. A 28-year-old man underwent living-related kidney transplantation for chronic kidney disease of unknown etiology. Numero...
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| Veröffentlicht in: | Internal Medicine 2017/06/01, Vol.56(11), pp.1387-1391 |
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| container_title | Internal Medicine |
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| creator | Nanmoku, Koji Kurosawa, Akira Shinzato, Takahiro Shimizu, Toshihiro Kimura, Takaaki Yagisawa, Takashi |
| description | Adenine phosphoribosyltransferase (APRT) deficiency is a rare autosomal recessive disorder that results in irreversible renal damage due to 2,8-dihydroxyadenine (DHA) nephropathy. A 28-year-old man underwent living-related kidney transplantation for chronic kidney disease of unknown etiology. Numerous spherical brownish crystals observed in his urinary sediment on postoperative day 3 and were observed within the tubular lumen of renal allograft biopsy specimens on postoperative day 7. After a genetic diagnosis, febuxostat treatment was started on postoperative day 7, with the dosage gradually increased to 80 mg/day until complete the disappearance of 2,8-DHA crystals. Febuxostat prevented secondary 2,8-DHA nephropathy after kidney transplantation. |
| doi_str_mv | 10.2169/internalmedicine.56.8142 |
| format | Article |
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A 28-year-old man underwent living-related kidney transplantation for chronic kidney disease of unknown etiology. Numerous spherical brownish crystals observed in his urinary sediment on postoperative day 3 and were observed within the tubular lumen of renal allograft biopsy specimens on postoperative day 7. After a genetic diagnosis, febuxostat treatment was started on postoperative day 7, with the dosage gradually increased to 80 mg/day until complete the disappearance of 2,8-DHA crystals. Febuxostat prevented secondary 2,8-DHA nephropathy after kidney transplantation.</description><identifier>ISSN: 0918-2918</identifier><identifier>EISSN: 1349-7235</identifier><identifier>DOI: 10.2169/internalmedicine.56.8142</identifier><identifier>PMID: 28566603</identifier><language>eng</language><publisher>Japan: The Japanese Society of Internal Medicine</publisher><subject>2,8-dihydroxyadenine nephropathy ; Adenine - analogs & derivatives ; Adenine - metabolism ; Adenine Phosphoribosyltransferase - deficiency ; Adult ; Case Report ; febuxostat ; Febuxostat - administration & dosage ; Gout Suppressants - administration & dosage ; Humans ; kidney transplantation ; Kidney Transplantation - methods ; Male ; Metabolism, Inborn Errors - prevention & control ; nephrolithiasis ; Urolithiasis - prevention & control ; xanthine dehydrogenase inhibitor</subject><ispartof>Internal Medicine, 2017/06/01, Vol.56(11), pp.1387-1391</ispartof><rights>2017 by The Japanese Society of Internal Medicine</rights><rights>Copyright © 2017 by The Japanese Society of Internal Medicine 2017</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c639t-c4fd409b7db7d620ade8f4e69129ac7f8f8e41b70c164914588cba5e26a2fdea3</citedby><cites>FETCH-LOGICAL-c639t-c4fd409b7db7d620ade8f4e69129ac7f8f8e41b70c164914588cba5e26a2fdea3</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC5498204/pdf/$$EPDF$$P50$$Gpubmedcentral$$Hfree_for_read</linktopdf><linktohtml>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC5498204/$$EHTML$$P50$$Gpubmedcentral$$Hfree_for_read</linktohtml><link.rule.ids>230,314,723,776,780,881,1877,27901,27902,53766,53768</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/28566603$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Nanmoku, Koji</creatorcontrib><creatorcontrib>Kurosawa, Akira</creatorcontrib><creatorcontrib>Shinzato, Takahiro</creatorcontrib><creatorcontrib>Shimizu, Toshihiro</creatorcontrib><creatorcontrib>Kimura, Takaaki</creatorcontrib><creatorcontrib>Yagisawa, Takashi</creatorcontrib><title>Febuxostat for the Prevention of Recurrent 2,8-dihydroxyadenine Nephropathy due to Adenine Phosphoribosyltransferase Deficiency Following Kidney Transplantation</title><title>Internal Medicine</title><addtitle>Intern. Med.</addtitle><description>Adenine phosphoribosyltransferase (APRT) deficiency is a rare autosomal recessive disorder that results in irreversible renal damage due to 2,8-dihydroxyadenine (DHA) nephropathy. A 28-year-old man underwent living-related kidney transplantation for chronic kidney disease of unknown etiology. Numerous spherical brownish crystals observed in his urinary sediment on postoperative day 3 and were observed within the tubular lumen of renal allograft biopsy specimens on postoperative day 7. After a genetic diagnosis, febuxostat treatment was started on postoperative day 7, with the dosage gradually increased to 80 mg/day until complete the disappearance of 2,8-DHA crystals. Febuxostat prevented secondary 2,8-DHA nephropathy after kidney transplantation.</description><subject>2,8-dihydroxyadenine nephropathy</subject><subject>Adenine - analogs & derivatives</subject><subject>Adenine - metabolism</subject><subject>Adenine Phosphoribosyltransferase - deficiency</subject><subject>Adult</subject><subject>Case Report</subject><subject>febuxostat</subject><subject>Febuxostat - administration & dosage</subject><subject>Gout Suppressants - administration & dosage</subject><subject>Humans</subject><subject>kidney transplantation</subject><subject>Kidney Transplantation - methods</subject><subject>Male</subject><subject>Metabolism, Inborn Errors - prevention & control</subject><subject>nephrolithiasis</subject><subject>Urolithiasis - prevention & control</subject><subject>xanthine dehydrogenase inhibitor</subject><issn>0918-2918</issn><issn>1349-7235</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2017</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNptUdGO1CAUbYzGHVd_wfDogx2B0hZeTDaro8aNuzHrM6H0MmXTgQp03f6NnyqTGSdqTMgl5J57zrmcokAErylpxBvrEgSnxh30VlsH67pZc8Loo2JFKibKllb142KFBeElzeWseBbjHcYVbwV9WpxRXjdNg6tV8XMD3fzgY1IJGR9QGgDdBLgHl6x3yBv0FfQcQn4j-pqXvR2WPviHRfXgsjL6AtMQ_KTSsKB-BpQ8uji2bgYfp8EH2_m4jCkoFw0EFQG9A5N9g9ML2vhx9D-s26LPtnewoNs9bhqVy5ayhefFE6PGCC-O93nxbfP-9vJjeXX94dPlxVWpm0qkUjPTMyy6ts-noTjb44ZBIwgVSreGGw6MdC3WpGGCsJpz3akaaKOo6UFV58XbA-80d_lbdV44qFFOwe5UWKRXVv7dcXaQW38vayY4xSwTvDoSBP99hpjkzkYNY94E_BwlEZgJ3BLGM5QfoDr4GAOYkwzBch-w_DdgWTdyH3AeffmnzdPg70Qz4PoAuMuZbuEEUCFZPcJ_mQnZ16PECakHFSS46hdmwsve</recordid><startdate>20170101</startdate><enddate>20170101</enddate><creator>Nanmoku, Koji</creator><creator>Kurosawa, Akira</creator><creator>Shinzato, Takahiro</creator><creator>Shimizu, Toshihiro</creator><creator>Kimura, Takaaki</creator><creator>Yagisawa, Takashi</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><scope>5PM</scope></search><sort><creationdate>20170101</creationdate><title>Febuxostat for the Prevention of Recurrent 2,8-dihydroxyadenine Nephropathy due to Adenine Phosphoribosyltransferase Deficiency Following Kidney Transplantation</title><author>Nanmoku, Koji ; Kurosawa, Akira ; Shinzato, Takahiro ; Shimizu, Toshihiro ; Kimura, Takaaki ; Yagisawa, Takashi</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c639t-c4fd409b7db7d620ade8f4e69129ac7f8f8e41b70c164914588cba5e26a2fdea3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2017</creationdate><topic>2,8-dihydroxyadenine nephropathy</topic><topic>Adenine - analogs & derivatives</topic><topic>Adenine - metabolism</topic><topic>Adenine Phosphoribosyltransferase - deficiency</topic><topic>Adult</topic><topic>Case Report</topic><topic>febuxostat</topic><topic>Febuxostat - administration & dosage</topic><topic>Gout Suppressants - administration & dosage</topic><topic>Humans</topic><topic>kidney transplantation</topic><topic>Kidney Transplantation - methods</topic><topic>Male</topic><topic>Metabolism, Inborn Errors - prevention & control</topic><topic>nephrolithiasis</topic><topic>Urolithiasis - prevention & control</topic><topic>xanthine dehydrogenase inhibitor</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Nanmoku, Koji</creatorcontrib><creatorcontrib>Kurosawa, Akira</creatorcontrib><creatorcontrib>Shinzato, Takahiro</creatorcontrib><creatorcontrib>Shimizu, Toshihiro</creatorcontrib><creatorcontrib>Kimura, Takaaki</creatorcontrib><creatorcontrib>Yagisawa, Takashi</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><collection>PubMed Central (Full Participant titles)</collection><jtitle>Internal Medicine</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Nanmoku, Koji</au><au>Kurosawa, Akira</au><au>Shinzato, Takahiro</au><au>Shimizu, Toshihiro</au><au>Kimura, Takaaki</au><au>Yagisawa, Takashi</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Febuxostat for the Prevention of Recurrent 2,8-dihydroxyadenine Nephropathy due to Adenine Phosphoribosyltransferase Deficiency Following Kidney Transplantation</atitle><jtitle>Internal Medicine</jtitle><addtitle>Intern. Med.</addtitle><date>2017-01-01</date><risdate>2017</risdate><volume>56</volume><issue>11</issue><spage>1387</spage><epage>1391</epage><pages>1387-1391</pages><issn>0918-2918</issn><eissn>1349-7235</eissn><abstract>Adenine phosphoribosyltransferase (APRT) deficiency is a rare autosomal recessive disorder that results in irreversible renal damage due to 2,8-dihydroxyadenine (DHA) nephropathy. A 28-year-old man underwent living-related kidney transplantation for chronic kidney disease of unknown etiology. Numerous spherical brownish crystals observed in his urinary sediment on postoperative day 3 and were observed within the tubular lumen of renal allograft biopsy specimens on postoperative day 7. After a genetic diagnosis, febuxostat treatment was started on postoperative day 7, with the dosage gradually increased to 80 mg/day until complete the disappearance of 2,8-DHA crystals. Febuxostat prevented secondary 2,8-DHA nephropathy after kidney transplantation.</abstract><cop>Japan</cop><pub>The Japanese Society of Internal Medicine</pub><pmid>28566603</pmid><doi>10.2169/internalmedicine.56.8142</doi><tpages>5</tpages><oa>free_for_read</oa></addata></record> |
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| source | MEDLINE; J-STAGE (Japan Science & Technology Information Aggregator, Electronic) Freely Available Titles - Japanese; PubMed Central; PubMed Central Open Access |
| subjects | 2,8-dihydroxyadenine nephropathy Adenine - analogs & derivatives Adenine - metabolism Adenine Phosphoribosyltransferase - deficiency Adult Case Report febuxostat Febuxostat - administration & dosage Gout Suppressants - administration & dosage Humans kidney transplantation Kidney Transplantation - methods Male Metabolism, Inborn Errors - prevention & control nephrolithiasis Urolithiasis - prevention & control xanthine dehydrogenase inhibitor |
| title | Febuxostat for the Prevention of Recurrent 2,8-dihydroxyadenine Nephropathy due to Adenine Phosphoribosyltransferase Deficiency Following Kidney Transplantation |
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