The Impact of Early Indication of Living Donor Liver Transplantation on the Outcomes of Patients With Propionic Acidemia: A Single‐Center Experience
ABSTRACT Background Liver transplantation has been indicated for propionic acidemia (PA) patients with frequent metabolic decompensation and performed as an enzyme replacement therapy. We retrospectively evaluated the outcomes of patients with PA and analyze the appropriate timing of living donor li...
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| Veröffentlicht in: | Pediatric transplantation 2024-12, Vol.28 (8), p.e14886-n/a |
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| creator | Nakao, Toshimasa Sakamoto, Seisuke Shimizu, Seiichi Fukuda, Akinari Uchida, Hajime Yanagi, Yusuke Komine, Ryuji Kodama, Tasuku Ninomiya, Aoi Yamada, Masaki Ono, Hiroshi Nosaka, Shunsuke Horikawa, Reiko Kasahara, Mureo |
| description | ABSTRACT
Background
Liver transplantation has been indicated for propionic acidemia (PA) patients with frequent metabolic decompensation and performed as an enzyme replacement therapy. We retrospectively evaluated the outcomes of patients with PA and analyze the appropriate timing of living donor liver transplantation (LDLT).
Methods
We reviewed 12 children with PA who underwent LDLT, who were divided into early (period from the first episode of metabolic decompensation to LDLT 1 year; n = 6) indication groups depending on the timing of LDLT.
Results
The patient and graft survival rates were 100% in 12 children with PA, and the median observation period was 61 months (5–193 months). None of the patients experienced any episodes of metabolic decompensation after LDLT. The timing of LDLT did not influence the incidence of surgical complications. Two patients in the late indication group had episodes of cardiac arrest and long QT syndrome before LDLT, and one patient showed prolongation of QT interval after LDLT. Two of the six patients in the late indication group had findings of metabolic stroke of the brain on MRI before LDLT. Although LDLT improved the findings of metabolic stroke, a decrease in development quotient score was shown in the post‐LDLT course.
Conclusions
LDLT may be an effective therapeutic option for improving metabolic control. Early LDLT might be help prevent cardiomyopathy and neurological impairment. |
| doi_str_mv | 10.1111/petr.14886 |
| format | Article |
| fullrecord | <record><control><sourceid>proquest_cross</sourceid><recordid>TN_cdi_proquest_miscellaneous_3125495304</recordid><sourceformat>XML</sourceformat><sourcesystem>PC</sourcesystem><sourcerecordid>3125495304</sourcerecordid><originalsourceid>FETCH-LOGICAL-c2826-aed583137157742bb68ffe4ae863b9b520b68cff123ff84304d05e0439e13cca3</originalsourceid><addsrcrecordid>eNp9kcGO1CAYx4nRuOvqxQcwJF6MSXeh0JZ6m4yjO8kkO9ExHhtKP1w2LVSg6tx8BE8-oE-ydGf0sAe5AB-_7xfgj9BzSs5pGhcjRH9OuRDlA3RKWV1njPDy4d26yhjl-Ql6EsINIbTkgj9GJ6wuiCBVeYp-764Br4dRqoidxivp-z1e284oGY2zc21jvhn7Bb911vl5Ax7vvLRh7KWNR8rimDxXU1RugDB3bdMJ2BjwZxOv8da7MYFG4YUyHQxGvsEL_DF5e_jz89cykUm7-jGCT10KnqJHWvYBnh3nM_Tp3Wq3vMw2V-_Xy8UmU7nIy0xCVwhGWUWLquJ525ZCa-ASRMnaui1ykipKa5ozrQVP39KRAghnNVCmlGRn6NXBO3r3dYIQm8EEBX16GrgpNIzmBa-L1JjQl_fQGzd5m26XKMYIK4uCJur1gVLeheBBN6M3g_T7hpJmTquZ02ru0krwi6Nyagfo_qF_40kAPQDfTQ_7_6ia7Wr34SC9BdzooUA</addsrcrecordid><sourcetype>Aggregation Database</sourcetype><iscdi>true</iscdi><recordtype>article</recordtype><pqid>3133036551</pqid></control><display><type>article</type><title>The Impact of Early Indication of Living Donor Liver Transplantation on the Outcomes of Patients With Propionic Acidemia: A Single‐Center Experience</title><source>MEDLINE</source><source>Wiley Online Library</source><creator>Nakao, Toshimasa ; Sakamoto, Seisuke ; Shimizu, Seiichi ; Fukuda, Akinari ; Uchida, Hajime ; Yanagi, Yusuke ; Komine, Ryuji ; Kodama, Tasuku ; Ninomiya, Aoi ; Yamada, Masaki ; Ono, Hiroshi ; Nosaka, Shunsuke ; Horikawa, Reiko ; Kasahara, Mureo</creator><creatorcontrib>Nakao, Toshimasa ; Sakamoto, Seisuke ; Shimizu, Seiichi ; Fukuda, Akinari ; Uchida, Hajime ; Yanagi, Yusuke ; Komine, Ryuji ; Kodama, Tasuku ; Ninomiya, Aoi ; Yamada, Masaki ; Ono, Hiroshi ; Nosaka, Shunsuke ; Horikawa, Reiko ; Kasahara, Mureo</creatorcontrib><description>ABSTRACT
Background
Liver transplantation has been indicated for propionic acidemia (PA) patients with frequent metabolic decompensation and performed as an enzyme replacement therapy. We retrospectively evaluated the outcomes of patients with PA and analyze the appropriate timing of living donor liver transplantation (LDLT).
Methods
We reviewed 12 children with PA who underwent LDLT, who were divided into early (period from the first episode of metabolic decompensation to LDLT < 1 year; n = 6) and late (> 1 year; n = 6) indication groups depending on the timing of LDLT.
Results
The patient and graft survival rates were 100% in 12 children with PA, and the median observation period was 61 months (5–193 months). None of the patients experienced any episodes of metabolic decompensation after LDLT. The timing of LDLT did not influence the incidence of surgical complications. Two patients in the late indication group had episodes of cardiac arrest and long QT syndrome before LDLT, and one patient showed prolongation of QT interval after LDLT. Two of the six patients in the late indication group had findings of metabolic stroke of the brain on MRI before LDLT. Although LDLT improved the findings of metabolic stroke, a decrease in development quotient score was shown in the post‐LDLT course.
Conclusions
LDLT may be an effective therapeutic option for improving metabolic control. Early LDLT might be help prevent cardiomyopathy and neurological impairment.</description><identifier>ISSN: 1397-3142</identifier><identifier>ISSN: 1399-3046</identifier><identifier>EISSN: 1399-3046</identifier><identifier>DOI: 10.1111/petr.14886</identifier><identifier>PMID: 39508076</identifier><language>eng</language><publisher>Denmark: Wiley Subscription Services, Inc</publisher><subject>Adolescent ; Cardiomyopathy ; Child ; Child, Preschool ; Early experience ; Female ; Follow-Up Studies ; Graft Survival ; Humans ; Infant ; Liver Transplantation ; Liver transplants ; living donor liver transplantation (LDLT) ; Living Donors ; Long QT syndrome ; Male ; Metabolism ; Neurological complications ; Patients ; Postoperative Complications - epidemiology ; Propionic acidemia ; Propionic Acidemia - complications ; Propionic Acidemia - surgery ; Retrospective Studies ; Stroke ; the appropriate timing of LDLT ; Time Factors ; Treatment Outcome</subject><ispartof>Pediatric transplantation, 2024-12, Vol.28 (8), p.e14886-n/a</ispartof><rights>2024 Wiley Periodicals LLC.</rights><rights>2024 Wiley Periodicals, LLC.</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><cites>FETCH-LOGICAL-c2826-aed583137157742bb68ffe4ae863b9b520b68cff123ff84304d05e0439e13cca3</cites><orcidid>0000-0003-3031-7319 ; 0000-0003-2547-4790 ; 0000-0002-1651-0430 ; 0000-0003-2441-5158 ; 0009-0005-1067-1697 ; 0000-0002-3393-6943 ; 0000-0003-4684-0446 ; 0000-0003-1003-6800 ; 0000-0003-3669-5548</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://onlinelibrary.wiley.com/doi/pdf/10.1111%2Fpetr.14886$$EPDF$$P50$$Gwiley$$H</linktopdf><linktohtml>$$Uhttps://onlinelibrary.wiley.com/doi/full/10.1111%2Fpetr.14886$$EHTML$$P50$$Gwiley$$H</linktohtml><link.rule.ids>314,780,784,1417,27924,27925,45574,45575</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/39508076$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Nakao, Toshimasa</creatorcontrib><creatorcontrib>Sakamoto, Seisuke</creatorcontrib><creatorcontrib>Shimizu, Seiichi</creatorcontrib><creatorcontrib>Fukuda, Akinari</creatorcontrib><creatorcontrib>Uchida, Hajime</creatorcontrib><creatorcontrib>Yanagi, Yusuke</creatorcontrib><creatorcontrib>Komine, Ryuji</creatorcontrib><creatorcontrib>Kodama, Tasuku</creatorcontrib><creatorcontrib>Ninomiya, Aoi</creatorcontrib><creatorcontrib>Yamada, Masaki</creatorcontrib><creatorcontrib>Ono, Hiroshi</creatorcontrib><creatorcontrib>Nosaka, Shunsuke</creatorcontrib><creatorcontrib>Horikawa, Reiko</creatorcontrib><creatorcontrib>Kasahara, Mureo</creatorcontrib><title>The Impact of Early Indication of Living Donor Liver Transplantation on the Outcomes of Patients With Propionic Acidemia: A Single‐Center Experience</title><title>Pediatric transplantation</title><addtitle>Pediatr Transplant</addtitle><description>ABSTRACT
Background
Liver transplantation has been indicated for propionic acidemia (PA) patients with frequent metabolic decompensation and performed as an enzyme replacement therapy. We retrospectively evaluated the outcomes of patients with PA and analyze the appropriate timing of living donor liver transplantation (LDLT).
Methods
We reviewed 12 children with PA who underwent LDLT, who were divided into early (period from the first episode of metabolic decompensation to LDLT < 1 year; n = 6) and late (> 1 year; n = 6) indication groups depending on the timing of LDLT.
Results
The patient and graft survival rates were 100% in 12 children with PA, and the median observation period was 61 months (5–193 months). None of the patients experienced any episodes of metabolic decompensation after LDLT. The timing of LDLT did not influence the incidence of surgical complications. Two patients in the late indication group had episodes of cardiac arrest and long QT syndrome before LDLT, and one patient showed prolongation of QT interval after LDLT. Two of the six patients in the late indication group had findings of metabolic stroke of the brain on MRI before LDLT. Although LDLT improved the findings of metabolic stroke, a decrease in development quotient score was shown in the post‐LDLT course.
Conclusions
LDLT may be an effective therapeutic option for improving metabolic control. Early LDLT might be help prevent cardiomyopathy and neurological impairment.</description><subject>Adolescent</subject><subject>Cardiomyopathy</subject><subject>Child</subject><subject>Child, Preschool</subject><subject>Early experience</subject><subject>Female</subject><subject>Follow-Up Studies</subject><subject>Graft Survival</subject><subject>Humans</subject><subject>Infant</subject><subject>Liver Transplantation</subject><subject>Liver transplants</subject><subject>living donor liver transplantation (LDLT)</subject><subject>Living Donors</subject><subject>Long QT syndrome</subject><subject>Male</subject><subject>Metabolism</subject><subject>Neurological complications</subject><subject>Patients</subject><subject>Postoperative Complications - epidemiology</subject><subject>Propionic acidemia</subject><subject>Propionic Acidemia - complications</subject><subject>Propionic Acidemia - surgery</subject><subject>Retrospective Studies</subject><subject>Stroke</subject><subject>the appropriate timing of LDLT</subject><subject>Time Factors</subject><subject>Treatment Outcome</subject><issn>1397-3142</issn><issn>1399-3046</issn><issn>1399-3046</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2024</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNp9kcGO1CAYx4nRuOvqxQcwJF6MSXeh0JZ6m4yjO8kkO9ExHhtKP1w2LVSg6tx8BE8-oE-ydGf0sAe5AB-_7xfgj9BzSs5pGhcjRH9OuRDlA3RKWV1njPDy4d26yhjl-Ql6EsINIbTkgj9GJ6wuiCBVeYp-764Br4dRqoidxivp-z1e284oGY2zc21jvhn7Bb911vl5Ax7vvLRh7KWNR8rimDxXU1RugDB3bdMJ2BjwZxOv8da7MYFG4YUyHQxGvsEL_DF5e_jz89cykUm7-jGCT10KnqJHWvYBnh3nM_Tp3Wq3vMw2V-_Xy8UmU7nIy0xCVwhGWUWLquJ525ZCa-ASRMnaui1ykipKa5ozrQVP39KRAghnNVCmlGRn6NXBO3r3dYIQm8EEBX16GrgpNIzmBa-L1JjQl_fQGzd5m26XKMYIK4uCJur1gVLeheBBN6M3g_T7hpJmTquZ02ru0krwi6Nyagfo_qF_40kAPQDfTQ_7_6ia7Wr34SC9BdzooUA</recordid><startdate>202412</startdate><enddate>202412</enddate><creator>Nakao, Toshimasa</creator><creator>Sakamoto, Seisuke</creator><creator>Shimizu, Seiichi</creator><creator>Fukuda, Akinari</creator><creator>Uchida, Hajime</creator><creator>Yanagi, Yusuke</creator><creator>Komine, Ryuji</creator><creator>Kodama, Tasuku</creator><creator>Ninomiya, Aoi</creator><creator>Yamada, Masaki</creator><creator>Ono, Hiroshi</creator><creator>Nosaka, Shunsuke</creator><creator>Horikawa, Reiko</creator><creator>Kasahara, Mureo</creator><general>Wiley Subscription Services, Inc</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>7T5</scope><scope>H94</scope><scope>K9.</scope><scope>NAPCQ</scope><scope>7X8</scope><orcidid>https://orcid.org/0000-0003-3031-7319</orcidid><orcidid>https://orcid.org/0000-0003-2547-4790</orcidid><orcidid>https://orcid.org/0000-0002-1651-0430</orcidid><orcidid>https://orcid.org/0000-0003-2441-5158</orcidid><orcidid>https://orcid.org/0009-0005-1067-1697</orcidid><orcidid>https://orcid.org/0000-0002-3393-6943</orcidid><orcidid>https://orcid.org/0000-0003-4684-0446</orcidid><orcidid>https://orcid.org/0000-0003-1003-6800</orcidid><orcidid>https://orcid.org/0000-0003-3669-5548</orcidid></search><sort><creationdate>202412</creationdate><title>The Impact of Early Indication of Living Donor Liver Transplantation on the Outcomes of Patients With Propionic Acidemia: A Single‐Center Experience</title><author>Nakao, Toshimasa ; Sakamoto, Seisuke ; Shimizu, Seiichi ; Fukuda, Akinari ; Uchida, Hajime ; Yanagi, Yusuke ; Komine, Ryuji ; Kodama, Tasuku ; Ninomiya, Aoi ; Yamada, Masaki ; Ono, Hiroshi ; Nosaka, Shunsuke ; Horikawa, Reiko ; Kasahara, Mureo</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c2826-aed583137157742bb68ffe4ae863b9b520b68cff123ff84304d05e0439e13cca3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2024</creationdate><topic>Adolescent</topic><topic>Cardiomyopathy</topic><topic>Child</topic><topic>Child, Preschool</topic><topic>Early experience</topic><topic>Female</topic><topic>Follow-Up Studies</topic><topic>Graft Survival</topic><topic>Humans</topic><topic>Infant</topic><topic>Liver Transplantation</topic><topic>Liver transplants</topic><topic>living donor liver transplantation (LDLT)</topic><topic>Living Donors</topic><topic>Long QT syndrome</topic><topic>Male</topic><topic>Metabolism</topic><topic>Neurological complications</topic><topic>Patients</topic><topic>Postoperative Complications - epidemiology</topic><topic>Propionic acidemia</topic><topic>Propionic Acidemia - complications</topic><topic>Propionic Acidemia - surgery</topic><topic>Retrospective Studies</topic><topic>Stroke</topic><topic>the appropriate timing of LDLT</topic><topic>Time Factors</topic><topic>Treatment Outcome</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Nakao, Toshimasa</creatorcontrib><creatorcontrib>Sakamoto, Seisuke</creatorcontrib><creatorcontrib>Shimizu, Seiichi</creatorcontrib><creatorcontrib>Fukuda, Akinari</creatorcontrib><creatorcontrib>Uchida, Hajime</creatorcontrib><creatorcontrib>Yanagi, Yusuke</creatorcontrib><creatorcontrib>Komine, Ryuji</creatorcontrib><creatorcontrib>Kodama, Tasuku</creatorcontrib><creatorcontrib>Ninomiya, Aoi</creatorcontrib><creatorcontrib>Yamada, Masaki</creatorcontrib><creatorcontrib>Ono, Hiroshi</creatorcontrib><creatorcontrib>Nosaka, Shunsuke</creatorcontrib><creatorcontrib>Horikawa, Reiko</creatorcontrib><creatorcontrib>Kasahara, Mureo</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>Immunology Abstracts</collection><collection>AIDS and Cancer Research Abstracts</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Nursing & Allied Health Premium</collection><collection>MEDLINE - Academic</collection><jtitle>Pediatric transplantation</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Nakao, Toshimasa</au><au>Sakamoto, Seisuke</au><au>Shimizu, Seiichi</au><au>Fukuda, Akinari</au><au>Uchida, Hajime</au><au>Yanagi, Yusuke</au><au>Komine, Ryuji</au><au>Kodama, Tasuku</au><au>Ninomiya, Aoi</au><au>Yamada, Masaki</au><au>Ono, Hiroshi</au><au>Nosaka, Shunsuke</au><au>Horikawa, Reiko</au><au>Kasahara, Mureo</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>The Impact of Early Indication of Living Donor Liver Transplantation on the Outcomes of Patients With Propionic Acidemia: A Single‐Center Experience</atitle><jtitle>Pediatric transplantation</jtitle><addtitle>Pediatr Transplant</addtitle><date>2024-12</date><risdate>2024</risdate><volume>28</volume><issue>8</issue><spage>e14886</spage><epage>n/a</epage><pages>e14886-n/a</pages><issn>1397-3142</issn><issn>1399-3046</issn><eissn>1399-3046</eissn><abstract>ABSTRACT
Background
Liver transplantation has been indicated for propionic acidemia (PA) patients with frequent metabolic decompensation and performed as an enzyme replacement therapy. We retrospectively evaluated the outcomes of patients with PA and analyze the appropriate timing of living donor liver transplantation (LDLT).
Methods
We reviewed 12 children with PA who underwent LDLT, who were divided into early (period from the first episode of metabolic decompensation to LDLT < 1 year; n = 6) and late (> 1 year; n = 6) indication groups depending on the timing of LDLT.
Results
The patient and graft survival rates were 100% in 12 children with PA, and the median observation period was 61 months (5–193 months). None of the patients experienced any episodes of metabolic decompensation after LDLT. The timing of LDLT did not influence the incidence of surgical complications. Two patients in the late indication group had episodes of cardiac arrest and long QT syndrome before LDLT, and one patient showed prolongation of QT interval after LDLT. Two of the six patients in the late indication group had findings of metabolic stroke of the brain on MRI before LDLT. Although LDLT improved the findings of metabolic stroke, a decrease in development quotient score was shown in the post‐LDLT course.
Conclusions
LDLT may be an effective therapeutic option for improving metabolic control. Early LDLT might be help prevent cardiomyopathy and neurological impairment.</abstract><cop>Denmark</cop><pub>Wiley Subscription Services, Inc</pub><pmid>39508076</pmid><doi>10.1111/petr.14886</doi><tpages>11</tpages><orcidid>https://orcid.org/0000-0003-3031-7319</orcidid><orcidid>https://orcid.org/0000-0003-2547-4790</orcidid><orcidid>https://orcid.org/0000-0002-1651-0430</orcidid><orcidid>https://orcid.org/0000-0003-2441-5158</orcidid><orcidid>https://orcid.org/0009-0005-1067-1697</orcidid><orcidid>https://orcid.org/0000-0002-3393-6943</orcidid><orcidid>https://orcid.org/0000-0003-4684-0446</orcidid><orcidid>https://orcid.org/0000-0003-1003-6800</orcidid><orcidid>https://orcid.org/0000-0003-3669-5548</orcidid><oa>free_for_read</oa></addata></record> |
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| ispartof | Pediatric transplantation, 2024-12, Vol.28 (8), p.e14886-n/a |
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| source | MEDLINE; Wiley Online Library |
| subjects | Adolescent Cardiomyopathy Child Child, Preschool Early experience Female Follow-Up Studies Graft Survival Humans Infant Liver Transplantation Liver transplants living donor liver transplantation (LDLT) Living Donors Long QT syndrome Male Metabolism Neurological complications Patients Postoperative Complications - epidemiology Propionic acidemia Propionic Acidemia - complications Propionic Acidemia - surgery Retrospective Studies Stroke the appropriate timing of LDLT Time Factors Treatment Outcome |
| title | The Impact of Early Indication of Living Donor Liver Transplantation on the Outcomes of Patients With Propionic Acidemia: A Single‐Center Experience |
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