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Long-term outcome of Japanese children with complicated minimal change nephrotic syndrome treated with mycophenolate mofetil after cyclosporine
Background Although recent studies have shown that more than half of children with steroid-dependent nephrotic syndrome (SDNS) may continue to have active disease beyond childhood, the long-term outcome in this cohort treated with mycophenolate mofetil (MMF) after cyclosporine remains unknown, parti...
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| Published in: | Pediatric nephrology (Berlin, West) West), 2019-11, Vol.34 (11), p.2417-2421 |
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| container_issue | 11 |
| container_start_page | 2417 |
| container_title | Pediatric nephrology (Berlin, West) |
| container_volume | 34 |
| creator | Fujinaga, Shuichiro Hirano, Daishi Nishino, Tomohiko Umeda, Chisato Watanabe, Yoshitaka Nakagawa, Mayu |
| description | Background
Although recent studies have shown that more than half of children with steroid-dependent nephrotic syndrome (SDNS) may continue to have active disease beyond childhood, the long-term outcome in this cohort treated with mycophenolate mofetil (MMF) after cyclosporine remains unknown, particularly in adulthood.
Methods
We conducted a retrospective study of 44 adult patients (median age, 22.3 years) who received MMF for complicated SDNS (median age at MMF initiation, 13.3 years) at a single center. Complicated SDNS was defined as the case continuing to relapse after cyclosporine (CsA) treatment. When patients experienced relapses despite MMF initiation, they additionally received a rituximab infusion. The primary endpoint was the probability of achieving treatment-free remission for > 2 years.
Results
Prior to MMF initiation, all patients received CsA for a median of 46 months and 19 received the 12-week cyclophosphamide. After switching from CsA to MMF, only four patients did not relapse during a median follow-up period of 9.6 years. At the last visit, only 15 of the 44 patients achieved treatment-free sustained remission. Multivariate analysis revealed that young age ( |
| doi_str_mv | 10.1007/s00467-019-04339-y |
| format | article |
| fullrecord | <record><control><sourceid>gale_proqu</sourceid><recordid>TN_cdi_proquest_miscellaneous_2306215629</recordid><sourceformat>XML</sourceformat><sourcesystem>PC</sourcesystem><galeid>A602851665</galeid><sourcerecordid>A602851665</sourcerecordid><originalsourceid>FETCH-LOGICAL-c511t-ef39de9b668bd67ba791948bbf06023119093d0ab15fd877e52ca0ec6af1cf893</originalsourceid><addsrcrecordid>eNp9ks9q3DAQxk1paLZpX6CHIiiUXpxK8lq2jiH0Lwu5pJCbkOXRWkGWXEkm-Cn6ytXuJk1TlqKDYOb3jWZGX1G8IficYNx8jBivWVNiwku8ripeLs-KFVlXtCS8vXlerDCvSE6Rm9PiZYy3GOO2btmL4rTKVN3QelX82ni3LROEEfk5KT8C8hp9l5N0EAGpwdg-gEN3Jg0opydrlEzQo9E4M0qbCem2gBxMQ_DJKBQX14ddnRRgT-6l46L8NIDzNsfQ6DUkY5HU-WWkFmV9nHwwDl4VJ1raCK_v77Pix-dP15dfy83Vl2-XF5tS1YSkEnTFe-AdY23Xs6aTDSd83XadxgzTihCeZ--x7Eit-7ZpoKZKYlBMaqJ0y6uz4sOh7hT8zxliEqOJCqzNc_s5ClphRknN6A599w966-fgcneC0qYhDcOMPFJbaUEYp30KUu2KiovcUlsTxupMlUeoLTgI0noH2uTwE_78CJ9PD6NRRwXv_xIMIG0aordzMt7FpyA9gCr4GANoMYX8oWERBIudvcTBXiLbS-ztJZYsenu_irkbof8jefBTBqoDEHMq-yI87uo_ZX8DXjHcNw</addsrcrecordid><sourcetype>Aggregation Database</sourcetype><iscdi>true</iscdi><recordtype>article</recordtype><pqid>2277176061</pqid></control><display><type>article</type><title>Long-term outcome of Japanese children with complicated minimal change nephrotic syndrome treated with mycophenolate mofetil after cyclosporine</title><source>Springer Nature</source><creator>Fujinaga, Shuichiro ; Hirano, Daishi ; Nishino, Tomohiko ; Umeda, Chisato ; Watanabe, Yoshitaka ; Nakagawa, Mayu</creator><creatorcontrib>Fujinaga, Shuichiro ; Hirano, Daishi ; Nishino, Tomohiko ; Umeda, Chisato ; Watanabe, Yoshitaka ; Nakagawa, Mayu</creatorcontrib><description>Background
Although recent studies have shown that more than half of children with steroid-dependent nephrotic syndrome (SDNS) may continue to have active disease beyond childhood, the long-term outcome in this cohort treated with mycophenolate mofetil (MMF) after cyclosporine remains unknown, particularly in adulthood.
Methods
We conducted a retrospective study of 44 adult patients (median age, 22.3 years) who received MMF for complicated SDNS (median age at MMF initiation, 13.3 years) at a single center. Complicated SDNS was defined as the case continuing to relapse after cyclosporine (CsA) treatment. When patients experienced relapses despite MMF initiation, they additionally received a rituximab infusion. The primary endpoint was the probability of achieving treatment-free remission for > 2 years.
Results
Prior to MMF initiation, all patients received CsA for a median of 46 months and 19 received the 12-week cyclophosphamide. After switching from CsA to MMF, only four patients did not relapse during a median follow-up period of 9.6 years. At the last visit, only 15 of the 44 patients achieved treatment-free sustained remission. Multivariate analysis revealed that young age (< 6 years) at onset of nephrotic syndrome (odds ratio, 11.3) and the experience of steroid dependency during initial CsA treatment (odds ratio, 29.8) were the independent risk factors of active disease into adulthood after MMF initiation.
Conclusions
Although none developed renal insufficiency and severe adverse effects of therapy, the introduction of MMF after CsA treatment may not be necessarily associated with improved long-term outcome of children with complicated SDNS.</description><identifier>ISSN: 0931-041X</identifier><identifier>EISSN: 1432-198X</identifier><identifier>DOI: 10.1007/s00467-019-04339-y</identifier><identifier>PMID: 31435725</identifier><language>eng</language><publisher>Berlin/Heidelberg: Springer Berlin Heidelberg</publisher><subject>Age ; Brief Report ; Children ; Cyclophosphamide ; Cyclosporins ; Diagnosis ; Health aspects ; Kidney diseases ; Medicine ; Medicine & Public Health ; Monoclonal antibodies ; Multivariate analysis ; Mycophenolate mofetil ; Mycophenolic acid ; Nephrology ; Nephrotic syndrome ; Patient outcomes ; Pediatric research ; Pediatrics ; Remission ; Renal insufficiency ; Risk factors ; Rituximab ; Urology</subject><ispartof>Pediatric nephrology (Berlin, West), 2019-11, Vol.34 (11), p.2417-2421</ispartof><rights>IPNA 2019</rights><rights>COPYRIGHT 2019 Springer</rights><rights>Pediatric Nephrology is a copyright of Springer, (2019). All Rights Reserved.</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c511t-ef39de9b668bd67ba791948bbf06023119093d0ab15fd877e52ca0ec6af1cf893</citedby><cites>FETCH-LOGICAL-c511t-ef39de9b668bd67ba791948bbf06023119093d0ab15fd877e52ca0ec6af1cf893</cites><orcidid>0000-0002-2957-3705</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>314,780,784,27924,27925</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/31435725$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Fujinaga, Shuichiro</creatorcontrib><creatorcontrib>Hirano, Daishi</creatorcontrib><creatorcontrib>Nishino, Tomohiko</creatorcontrib><creatorcontrib>Umeda, Chisato</creatorcontrib><creatorcontrib>Watanabe, Yoshitaka</creatorcontrib><creatorcontrib>Nakagawa, Mayu</creatorcontrib><title>Long-term outcome of Japanese children with complicated minimal change nephrotic syndrome treated with mycophenolate mofetil after cyclosporine</title><title>Pediatric nephrology (Berlin, West)</title><addtitle>Pediatr Nephrol</addtitle><addtitle>Pediatr Nephrol</addtitle><description>Background
Although recent studies have shown that more than half of children with steroid-dependent nephrotic syndrome (SDNS) may continue to have active disease beyond childhood, the long-term outcome in this cohort treated with mycophenolate mofetil (MMF) after cyclosporine remains unknown, particularly in adulthood.
Methods
We conducted a retrospective study of 44 adult patients (median age, 22.3 years) who received MMF for complicated SDNS (median age at MMF initiation, 13.3 years) at a single center. Complicated SDNS was defined as the case continuing to relapse after cyclosporine (CsA) treatment. When patients experienced relapses despite MMF initiation, they additionally received a rituximab infusion. The primary endpoint was the probability of achieving treatment-free remission for > 2 years.
Results
Prior to MMF initiation, all patients received CsA for a median of 46 months and 19 received the 12-week cyclophosphamide. After switching from CsA to MMF, only four patients did not relapse during a median follow-up period of 9.6 years. At the last visit, only 15 of the 44 patients achieved treatment-free sustained remission. Multivariate analysis revealed that young age (< 6 years) at onset of nephrotic syndrome (odds ratio, 11.3) and the experience of steroid dependency during initial CsA treatment (odds ratio, 29.8) were the independent risk factors of active disease into adulthood after MMF initiation.
Conclusions
Although none developed renal insufficiency and severe adverse effects of therapy, the introduction of MMF after CsA treatment may not be necessarily associated with improved long-term outcome of children with complicated SDNS.</description><subject>Age</subject><subject>Brief Report</subject><subject>Children</subject><subject>Cyclophosphamide</subject><subject>Cyclosporins</subject><subject>Diagnosis</subject><subject>Health aspects</subject><subject>Kidney diseases</subject><subject>Medicine</subject><subject>Medicine & Public Health</subject><subject>Monoclonal antibodies</subject><subject>Multivariate analysis</subject><subject>Mycophenolate mofetil</subject><subject>Mycophenolic acid</subject><subject>Nephrology</subject><subject>Nephrotic syndrome</subject><subject>Patient outcomes</subject><subject>Pediatric research</subject><subject>Pediatrics</subject><subject>Remission</subject><subject>Renal insufficiency</subject><subject>Risk factors</subject><subject>Rituximab</subject><subject>Urology</subject><issn>0931-041X</issn><issn>1432-198X</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2019</creationdate><recordtype>article</recordtype><recordid>eNp9ks9q3DAQxk1paLZpX6CHIiiUXpxK8lq2jiH0Lwu5pJCbkOXRWkGWXEkm-Cn6ytXuJk1TlqKDYOb3jWZGX1G8IficYNx8jBivWVNiwku8ripeLs-KFVlXtCS8vXlerDCvSE6Rm9PiZYy3GOO2btmL4rTKVN3QelX82ni3LROEEfk5KT8C8hp9l5N0EAGpwdg-gEN3Jg0opydrlEzQo9E4M0qbCem2gBxMQ_DJKBQX14ddnRRgT-6l46L8NIDzNsfQ6DUkY5HU-WWkFmV9nHwwDl4VJ1raCK_v77Pix-dP15dfy83Vl2-XF5tS1YSkEnTFe-AdY23Xs6aTDSd83XadxgzTihCeZ--x7Eit-7ZpoKZKYlBMaqJ0y6uz4sOh7hT8zxliEqOJCqzNc_s5ClphRknN6A599w966-fgcneC0qYhDcOMPFJbaUEYp30KUu2KiovcUlsTxupMlUeoLTgI0noH2uTwE_78CJ9PD6NRRwXv_xIMIG0aordzMt7FpyA9gCr4GANoMYX8oWERBIudvcTBXiLbS-ztJZYsenu_irkbof8jefBTBqoDEHMq-yI87uo_ZX8DXjHcNw</recordid><startdate>20191101</startdate><enddate>20191101</enddate><creator>Fujinaga, Shuichiro</creator><creator>Hirano, Daishi</creator><creator>Nishino, Tomohiko</creator><creator>Umeda, Chisato</creator><creator>Watanabe, Yoshitaka</creator><creator>Nakagawa, Mayu</creator><general>Springer Berlin Heidelberg</general><general>Springer</general><general>Springer Nature B.V</general><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>3V.</scope><scope>7QP</scope><scope>7RV</scope><scope>7X7</scope><scope>7XB</scope><scope>88E</scope><scope>8AO</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>AZQEC</scope><scope>BENPR</scope><scope>CCPQU</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>K9-</scope><scope>K9.</scope><scope>KB0</scope><scope>M0R</scope><scope>M0S</scope><scope>M1P</scope><scope>NAPCQ</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>7X8</scope><orcidid>https://orcid.org/0000-0002-2957-3705</orcidid></search><sort><creationdate>20191101</creationdate><title>Long-term outcome of Japanese children with complicated minimal change nephrotic syndrome treated with mycophenolate mofetil after cyclosporine</title><author>Fujinaga, Shuichiro ; Hirano, Daishi ; Nishino, Tomohiko ; Umeda, Chisato ; Watanabe, Yoshitaka ; Nakagawa, Mayu</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c511t-ef39de9b668bd67ba791948bbf06023119093d0ab15fd877e52ca0ec6af1cf893</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2019</creationdate><topic>Age</topic><topic>Brief Report</topic><topic>Children</topic><topic>Cyclophosphamide</topic><topic>Cyclosporins</topic><topic>Diagnosis</topic><topic>Health aspects</topic><topic>Kidney diseases</topic><topic>Medicine</topic><topic>Medicine & Public Health</topic><topic>Monoclonal antibodies</topic><topic>Multivariate analysis</topic><topic>Mycophenolate mofetil</topic><topic>Mycophenolic acid</topic><topic>Nephrology</topic><topic>Nephrotic syndrome</topic><topic>Patient outcomes</topic><topic>Pediatric research</topic><topic>Pediatrics</topic><topic>Remission</topic><topic>Renal insufficiency</topic><topic>Risk factors</topic><topic>Rituximab</topic><topic>Urology</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Fujinaga, Shuichiro</creatorcontrib><creatorcontrib>Hirano, Daishi</creatorcontrib><creatorcontrib>Nishino, Tomohiko</creatorcontrib><creatorcontrib>Umeda, Chisato</creatorcontrib><creatorcontrib>Watanabe, Yoshitaka</creatorcontrib><creatorcontrib>Nakagawa, Mayu</creatorcontrib><collection>PubMed</collection><collection>CrossRef</collection><collection>ProQuest Central (Corporate)</collection><collection>Calcium & Calcified Tissue Abstracts</collection><collection>Nursing & Allied Health Database</collection><collection>ProQuest_Health & Medical Collection</collection><collection>ProQuest Central (purchase pre-March 2016)</collection><collection>Medical Database (Alumni Edition)</collection><collection>ProQuest Pharma Collection</collection><collection>Hospital Premium Collection</collection><collection>Hospital Premium Collection (Alumni Edition)</collection><collection>ProQuest Central (Alumni) (purchase pre-March 2016)</collection><collection>ProQuest Central (Alumni)</collection><collection>ProQuest Central</collection><collection>ProQuest Central Essentials</collection><collection>AUTh Library subscriptions: ProQuest Central</collection><collection>ProQuest One Community College</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>Consumer Health Database (Alumni Edition)</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Nursing & Allied Health Database (Alumni Edition)</collection><collection>Consumer Health Database</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>PML(ProQuest Medical Library)</collection><collection>Nursing & Allied Health Premium</collection><collection>ProQuest One Academic Eastern Edition (DO NOT USE)</collection><collection>ProQuest One Academic</collection><collection>ProQuest One Academic UKI Edition</collection><collection>MEDLINE - Academic</collection><jtitle>Pediatric nephrology (Berlin, West)</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Fujinaga, Shuichiro</au><au>Hirano, Daishi</au><au>Nishino, Tomohiko</au><au>Umeda, Chisato</au><au>Watanabe, Yoshitaka</au><au>Nakagawa, Mayu</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Long-term outcome of Japanese children with complicated minimal change nephrotic syndrome treated with mycophenolate mofetil after cyclosporine</atitle><jtitle>Pediatric nephrology (Berlin, West)</jtitle><stitle>Pediatr Nephrol</stitle><addtitle>Pediatr Nephrol</addtitle><date>2019-11-01</date><risdate>2019</risdate><volume>34</volume><issue>11</issue><spage>2417</spage><epage>2421</epage><pages>2417-2421</pages><issn>0931-041X</issn><eissn>1432-198X</eissn><abstract>Background
Although recent studies have shown that more than half of children with steroid-dependent nephrotic syndrome (SDNS) may continue to have active disease beyond childhood, the long-term outcome in this cohort treated with mycophenolate mofetil (MMF) after cyclosporine remains unknown, particularly in adulthood.
Methods
We conducted a retrospective study of 44 adult patients (median age, 22.3 years) who received MMF for complicated SDNS (median age at MMF initiation, 13.3 years) at a single center. Complicated SDNS was defined as the case continuing to relapse after cyclosporine (CsA) treatment. When patients experienced relapses despite MMF initiation, they additionally received a rituximab infusion. The primary endpoint was the probability of achieving treatment-free remission for > 2 years.
Results
Prior to MMF initiation, all patients received CsA for a median of 46 months and 19 received the 12-week cyclophosphamide. After switching from CsA to MMF, only four patients did not relapse during a median follow-up period of 9.6 years. At the last visit, only 15 of the 44 patients achieved treatment-free sustained remission. Multivariate analysis revealed that young age (< 6 years) at onset of nephrotic syndrome (odds ratio, 11.3) and the experience of steroid dependency during initial CsA treatment (odds ratio, 29.8) were the independent risk factors of active disease into adulthood after MMF initiation.
Conclusions
Although none developed renal insufficiency and severe adverse effects of therapy, the introduction of MMF after CsA treatment may not be necessarily associated with improved long-term outcome of children with complicated SDNS.</abstract><cop>Berlin/Heidelberg</cop><pub>Springer Berlin Heidelberg</pub><pmid>31435725</pmid><doi>10.1007/s00467-019-04339-y</doi><tpages>5</tpages><orcidid>https://orcid.org/0000-0002-2957-3705</orcidid></addata></record> |
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| ispartof | Pediatric nephrology (Berlin, West), 2019-11, Vol.34 (11), p.2417-2421 |
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| source | Springer Nature |
| subjects | Age Brief Report Children Cyclophosphamide Cyclosporins Diagnosis Health aspects Kidney diseases Medicine Medicine & Public Health Monoclonal antibodies Multivariate analysis Mycophenolate mofetil Mycophenolic acid Nephrology Nephrotic syndrome Patient outcomes Pediatric research Pediatrics Remission Renal insufficiency Risk factors Rituximab Urology |
| title | Long-term outcome of Japanese children with complicated minimal change nephrotic syndrome treated with mycophenolate mofetil after cyclosporine |
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