Pediatric Recurrent Pericarditis: Appropriateness of the Standard of Care and Response to IL-1 Blockade
To analyze, in a cohort of pediatric patients with recurrent pericarditis undergoing anti-interleukin (IL)-1 treatment: the agent and dosing used as first-line treatment, the long-term efficacy of IL-1 blockers, the percentage of patients achieving a drug-free remission, and the presence of variable...
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| Veröffentlicht in: | The Journal of pediatrics 2023-05, Vol.256, p.18-26.e8 |
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| creator | Caorsi, Roberta Insalaco, Antonella Bovis, Francesca Martini, Giorgia Cattalini, Marco Chinali, Marcello Rimini, Alessandro Longo, Chiara Federici, Silvia Celani, Camilla Filocamo, Giovanni Consolini, Rita Maggio, Maria Cristina Fadanelli, Gloria Licciardi, Francesco Romano, Micol Teruzzi, Barbara Lia Taddio, Andrea Miniaci, Angela La Torre, Francesco De Fanti, Alessandro Cavalli, Giulio Bigucci, Barbara Gallizzi, Romina Chinello, Matteo Imazio, Massimo Brucato, Antonio Cimaz, Rolando De Benedetti, Fabrizio Gattorno, Marco |
| description | To analyze, in a cohort of pediatric patients with recurrent pericarditis undergoing anti-interleukin (IL)-1 treatment: the agent and dosing used as first-line treatment, the long-term efficacy of IL-1 blockers, the percentage of patients achieving a drug-free remission, and the presence of variables associated with drug-free remission.
Data were collected from patients’ charts. The annualized relapse rate (ARR) was used for evaluation of treatment efficacy, and bivariate logistic regression analysis was used for variables associated with drug-free remission.
Fifty-eight patients, treated between 2008 and 2018, were included in the study (mean follow-up. 2.6 years). Of the 56 patients treated with first-line drugs, 14 not responsive patients were underdosed. Fifty-seven patients were treated with anakinra: the ARR before and during daily treatment was 3.05 and 0.28, respectively (P |
| doi_str_mv | 10.1016/j.jpeds.2022.11.034 |
| format | Article |
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Data were collected from patients’ charts. The annualized relapse rate (ARR) was used for evaluation of treatment efficacy, and bivariate logistic regression analysis was used for variables associated with drug-free remission.
Fifty-eight patients, treated between 2008 and 2018, were included in the study (mean follow-up. 2.6 years). Of the 56 patients treated with first-line drugs, 14 not responsive patients were underdosed. Fifty-seven patients were treated with anakinra: the ARR before and during daily treatment was 3.05 and 0.28, respectively (P < .0001); an increase to 0.83 was observed after the reduction/withdrawal of treatment (P < .0001). The switch from anakinra to canakinumab (5 patients) was associated to an increase of the ARR (0.49 vs 1.46), but without statistical significance (P = .215). At last follow-up, only 9 of the 58 patients had withdrawn all treatments. With the limits of a retrospective study and the heterogeneity between the patients enrolled in the study, a shorter duration of treatment with anakinra was the only variable associated with drug-free remission.
This study shows that most pediatric patients with recurrent pericarditis needing IL-1 blockade received an inadequate treatment with first-line agents. The effectiveness of anakinra is supported by this study, but few patients achieved drug-free remission. The different rate of response to anakinra and canakinumab may suggest a possible role of IL-1α in the pathogenesis of recurrent pericarditis.</description><identifier>ISSN: 0022-3476</identifier><identifier>EISSN: 1097-6833</identifier><identifier>DOI: 10.1016/j.jpeds.2022.11.034</identifier><identifier>PMID: 36470465</identifier><language>eng</language><publisher>United States: Elsevier Inc</publisher><subject>anti-inflammatory agents nonsteroidal ; Child ; colchicine ; Humans ; Interleukin 1 Receptor Antagonist Protein - therapeutic use ; interleukin-1 ; Interleukin-1 - therapeutic use ; pericarditis ; Pericarditis - drug therapy ; Recurrence ; Retrospective Studies ; Standard of Care ; steroids ; therapy ; Treatment Outcome</subject><ispartof>The Journal of pediatrics, 2023-05, Vol.256, p.18-26.e8</ispartof><rights>2022 The Authors</rights><rights>Copyright © 2022 The Authors. Published by Elsevier Inc. All rights reserved.</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c404t-28173188a7f1dcf444e42f1e3786ea714fae8eb322e9c6cbe005458194b518103</citedby><cites>FETCH-LOGICAL-c404t-28173188a7f1dcf444e42f1e3786ea714fae8eb322e9c6cbe005458194b518103</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktohtml>$$Uhttps://www.sciencedirect.com/science/article/pii/S0022347622010952$$EHTML$$P50$$Gelsevier$$Hfree_for_read</linktohtml><link.rule.ids>314,776,780,3537,27901,27902,65534</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/36470465$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Caorsi, Roberta</creatorcontrib><creatorcontrib>Insalaco, Antonella</creatorcontrib><creatorcontrib>Bovis, Francesca</creatorcontrib><creatorcontrib>Martini, Giorgia</creatorcontrib><creatorcontrib>Cattalini, Marco</creatorcontrib><creatorcontrib>Chinali, Marcello</creatorcontrib><creatorcontrib>Rimini, Alessandro</creatorcontrib><creatorcontrib>Longo, Chiara</creatorcontrib><creatorcontrib>Federici, Silvia</creatorcontrib><creatorcontrib>Celani, Camilla</creatorcontrib><creatorcontrib>Filocamo, Giovanni</creatorcontrib><creatorcontrib>Consolini, Rita</creatorcontrib><creatorcontrib>Maggio, Maria Cristina</creatorcontrib><creatorcontrib>Fadanelli, Gloria</creatorcontrib><creatorcontrib>Licciardi, Francesco</creatorcontrib><creatorcontrib>Romano, Micol</creatorcontrib><creatorcontrib>Teruzzi, Barbara Lia</creatorcontrib><creatorcontrib>Taddio, Andrea</creatorcontrib><creatorcontrib>Miniaci, Angela</creatorcontrib><creatorcontrib>La Torre, Francesco</creatorcontrib><creatorcontrib>De Fanti, Alessandro</creatorcontrib><creatorcontrib>Cavalli, Giulio</creatorcontrib><creatorcontrib>Bigucci, Barbara</creatorcontrib><creatorcontrib>Gallizzi, Romina</creatorcontrib><creatorcontrib>Chinello, Matteo</creatorcontrib><creatorcontrib>Imazio, Massimo</creatorcontrib><creatorcontrib>Brucato, Antonio</creatorcontrib><creatorcontrib>Cimaz, Rolando</creatorcontrib><creatorcontrib>De Benedetti, Fabrizio</creatorcontrib><creatorcontrib>Gattorno, Marco</creatorcontrib><title>Pediatric Recurrent Pericarditis: Appropriateness of the Standard of Care and Response to IL-1 Blockade</title><title>The Journal of pediatrics</title><addtitle>J Pediatr</addtitle><description>To analyze, in a cohort of pediatric patients with recurrent pericarditis undergoing anti-interleukin (IL)-1 treatment: the agent and dosing used as first-line treatment, the long-term efficacy of IL-1 blockers, the percentage of patients achieving a drug-free remission, and the presence of variables associated with drug-free remission.
Data were collected from patients’ charts. The annualized relapse rate (ARR) was used for evaluation of treatment efficacy, and bivariate logistic regression analysis was used for variables associated with drug-free remission.
Fifty-eight patients, treated between 2008 and 2018, were included in the study (mean follow-up. 2.6 years). Of the 56 patients treated with first-line drugs, 14 not responsive patients were underdosed. Fifty-seven patients were treated with anakinra: the ARR before and during daily treatment was 3.05 and 0.28, respectively (P < .0001); an increase to 0.83 was observed after the reduction/withdrawal of treatment (P < .0001). The switch from anakinra to canakinumab (5 patients) was associated to an increase of the ARR (0.49 vs 1.46), but without statistical significance (P = .215). At last follow-up, only 9 of the 58 patients had withdrawn all treatments. With the limits of a retrospective study and the heterogeneity between the patients enrolled in the study, a shorter duration of treatment with anakinra was the only variable associated with drug-free remission.
This study shows that most pediatric patients with recurrent pericarditis needing IL-1 blockade received an inadequate treatment with first-line agents. The effectiveness of anakinra is supported by this study, but few patients achieved drug-free remission. The different rate of response to anakinra and canakinumab may suggest a possible role of IL-1α in the pathogenesis of recurrent pericarditis.</description><subject>anti-inflammatory agents nonsteroidal</subject><subject>Child</subject><subject>colchicine</subject><subject>Humans</subject><subject>Interleukin 1 Receptor Antagonist Protein - therapeutic use</subject><subject>interleukin-1</subject><subject>Interleukin-1 - therapeutic use</subject><subject>pericarditis</subject><subject>Pericarditis - drug therapy</subject><subject>Recurrence</subject><subject>Retrospective Studies</subject><subject>Standard of Care</subject><subject>steroids</subject><subject>therapy</subject><subject>Treatment Outcome</subject><issn>0022-3476</issn><issn>1097-6833</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2023</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNp9kM1OGzEURi1EVQLtE1RCXrKZ6b22M_YgsQgRpZEigWi7thzPnTIhmRlsB4m3r0Nol11Zn3zu32HsC0KJgNXXdbkeqYmlACFKxBKkOmIThFoXlZHymE0g_xRS6eqEnca4BoBaAXxkJ7JSGlQ1nbDf99R0LoXO8wfyuxCoT_yecnah6VIXL_lsHMMwhkxRTzHyoeXpkfiP5PomQ_s8d4F4jrlHHIc-Ek8DXywL5NebwT-5hj6xD63bRPr8_p6xX99ufs6_F8u728V8tiy8ApUKYVBLNMbpFhvfKqVIiRZJalOR06haR4ZWUgiqfeVXBDBVU4O1Wk3RIMgzdnHom3d-3lFMdttFT5uN62nYRSu00kIbUWNG5QH1YYgxUGvzkVsXXi2C3Ru2a_tm2O4NW0SbDeeq8_cBu9WWmn81f5Vm4OoAUD7zpaNgo--o99lzIJ9sM3T_HfAH8JKMww</recordid><startdate>202305</startdate><enddate>202305</enddate><creator>Caorsi, Roberta</creator><creator>Insalaco, Antonella</creator><creator>Bovis, Francesca</creator><creator>Martini, Giorgia</creator><creator>Cattalini, Marco</creator><creator>Chinali, Marcello</creator><creator>Rimini, Alessandro</creator><creator>Longo, Chiara</creator><creator>Federici, Silvia</creator><creator>Celani, Camilla</creator><creator>Filocamo, Giovanni</creator><creator>Consolini, Rita</creator><creator>Maggio, Maria Cristina</creator><creator>Fadanelli, Gloria</creator><creator>Licciardi, Francesco</creator><creator>Romano, Micol</creator><creator>Teruzzi, Barbara Lia</creator><creator>Taddio, Andrea</creator><creator>Miniaci, Angela</creator><creator>La Torre, Francesco</creator><creator>De Fanti, Alessandro</creator><creator>Cavalli, Giulio</creator><creator>Bigucci, Barbara</creator><creator>Gallizzi, Romina</creator><creator>Chinello, Matteo</creator><creator>Imazio, Massimo</creator><creator>Brucato, Antonio</creator><creator>Cimaz, Rolando</creator><creator>De Benedetti, Fabrizio</creator><creator>Gattorno, Marco</creator><general>Elsevier Inc</general><scope>6I.</scope><scope>AAFTH</scope><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>202305</creationdate><title>Pediatric Recurrent Pericarditis: Appropriateness of the Standard of Care and Response to IL-1 Blockade</title><author>Caorsi, Roberta ; Insalaco, Antonella ; Bovis, Francesca ; Martini, Giorgia ; Cattalini, Marco ; Chinali, Marcello ; Rimini, Alessandro ; Longo, Chiara ; Federici, Silvia ; Celani, Camilla ; Filocamo, Giovanni ; Consolini, Rita ; Maggio, Maria Cristina ; Fadanelli, Gloria ; Licciardi, Francesco ; Romano, Micol ; Teruzzi, Barbara Lia ; Taddio, Andrea ; Miniaci, Angela ; La Torre, Francesco ; De Fanti, Alessandro ; Cavalli, Giulio ; Bigucci, Barbara ; Gallizzi, Romina ; Chinello, Matteo ; Imazio, Massimo ; Brucato, Antonio ; Cimaz, Rolando ; De Benedetti, Fabrizio ; Gattorno, Marco</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c404t-28173188a7f1dcf444e42f1e3786ea714fae8eb322e9c6cbe005458194b518103</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2023</creationdate><topic>anti-inflammatory agents nonsteroidal</topic><topic>Child</topic><topic>colchicine</topic><topic>Humans</topic><topic>Interleukin 1 Receptor Antagonist Protein - 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Data were collected from patients’ charts. The annualized relapse rate (ARR) was used for evaluation of treatment efficacy, and bivariate logistic regression analysis was used for variables associated with drug-free remission.
Fifty-eight patients, treated between 2008 and 2018, were included in the study (mean follow-up. 2.6 years). Of the 56 patients treated with first-line drugs, 14 not responsive patients were underdosed. Fifty-seven patients were treated with anakinra: the ARR before and during daily treatment was 3.05 and 0.28, respectively (P < .0001); an increase to 0.83 was observed after the reduction/withdrawal of treatment (P < .0001). The switch from anakinra to canakinumab (5 patients) was associated to an increase of the ARR (0.49 vs 1.46), but without statistical significance (P = .215). At last follow-up, only 9 of the 58 patients had withdrawn all treatments. With the limits of a retrospective study and the heterogeneity between the patients enrolled in the study, a shorter duration of treatment with anakinra was the only variable associated with drug-free remission.
This study shows that most pediatric patients with recurrent pericarditis needing IL-1 blockade received an inadequate treatment with first-line agents. The effectiveness of anakinra is supported by this study, but few patients achieved drug-free remission. The different rate of response to anakinra and canakinumab may suggest a possible role of IL-1α in the pathogenesis of recurrent pericarditis.</abstract><cop>United States</cop><pub>Elsevier Inc</pub><pmid>36470465</pmid><doi>10.1016/j.jpeds.2022.11.034</doi><oa>free_for_read</oa></addata></record> |
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| ispartof | The Journal of pediatrics, 2023-05, Vol.256, p.18-26.e8 |
| issn | 0022-3476 1097-6833 |
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| source | MEDLINE; Elsevier ScienceDirect Journals Complete |
| subjects | anti-inflammatory agents nonsteroidal Child colchicine Humans Interleukin 1 Receptor Antagonist Protein - therapeutic use interleukin-1 Interleukin-1 - therapeutic use pericarditis Pericarditis - drug therapy Recurrence Retrospective Studies Standard of Care steroids therapy Treatment Outcome |
| title | Pediatric Recurrent Pericarditis: Appropriateness of the Standard of Care and Response to IL-1 Blockade |
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