{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,12]],"date-time":"2026-10-12T11:51:36Z","timestamp":1791805896519,"version":"4.3.4"},"reference-count":21,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2018,9,11]],"date-time":"2018-09-11T00:00:00Z","timestamp":1536624000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Pediatric Blood &amp; Cancer"],"published-print":{"date-parts":[[2019,1]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Purpose<\/jats:title><jats:p>We analyzed central diabetes insipidus (CDI) development in pediatric patients with Langerhans cell histiocytosis (LCH) treated according to the Japan LCH Study Group (JLSG) regimen, which is the combination chemotherapy including cytarabine (Ara\u2010C).<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Retrospective data from 317 patients (multisystem disease (MS), <jats:italic>n<\/jats:italic>\u00a0=\u00a0206; multiple focal bone (MFB), <jats:italic>n<\/jats:italic>\u00a0=\u00a0111) treated according to the JLSG\u201096\/02 regimens were analyzed.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>The median follow\u2010up duration was 10.6 years (range, 0.1\u201321.1). A total of 50\/317 (15.8%) patients developed CDI (MFB, <jats:italic>n<\/jats:italic>\u00a0=\u00a04; MS, <jats:italic>n<\/jats:italic>\u00a0=\u00a046). Of the 50 cases, CDI was already present at the time of LCH diagnosis (pre\u2010CDI) in 25, and it newly developed after the diagnosis and initiation of treatment (post\u2010CDI) in the other 25 cases. The cumulative incidence of post\u2010CDI at 10\u2010year calculated by Kaplan\u2013Meier analysis was 9.0% for total and 12.0% for MS patients. A positive correlation with LCH lesions at the CNS risk sites at diagnosis was found in pre\u2010CDI cases (17\/164\u00a0vs 8\/171; <jats:italic>P<\/jats:italic>\u00a0=\u00a00.0359), but not in post\u2010CDI cases (14\/129\u00a0vs 11\/163; <jats:italic>P<\/jats:italic> =\u00a00.254). Multivariate analysis showed that relapse at the CNS risk sites was significantly associated with post\u2010CDI development (hazard ratio: 4.70; 95% CI, 1.29\u201317.1, <jats:italic>P<\/jats:italic> &lt;\u00a00.05).<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>In the JLSG\u201096\/02 studies, CDI developed in 15.8% of the cohort in which half as pre\u2010 and the other half as post\u2010CDI. Relapse, particularly at the CNS risk sites, was linked with the development of post\u2010CDI.<\/jats:p><\/jats:sec>","DOI":"10.1002\/pbc.27454","type":"journal-article","created":{"date-parts":[[2018,9,12]],"date-time":"2018-09-12T05:54:39Z","timestamp":1536731679000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":18,"title":["Central diabetes insipidus in pediatric patients with Langerhans cell histiocytosis: Results from the JLSG\u201096\/02 studies"],"prefix":"10.1002","volume":"66","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-1499-538X","authenticated-orcid":false,"given":"Kenichi","family":"Sakamoto","sequence":"first","affiliation":[{"name":"Department of Pediatrics Graduate School of Medical Science Kyoto Prefectural University of Medicine  Kyoto Japan"},{"name":"Children's Cancer Center National Center for Child Health and Development  Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Akira","family":"Morimoto","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Jichi Medical University School of Medicine  Shimotsuke Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yoko","family":"Shioda","sequence":"additional","affiliation":[{"name":"Children's Cancer Center National Center for Child Health and Development  Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5727-4470","authenticated-orcid":false,"given":"Toshihiko","family":"Imamura","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Graduate School of Medical Science Kyoto Prefectural University of Medicine  Kyoto Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shinsaku","family":"Imashuku","sequence":"additional","affiliation":[{"name":"Division of Laboratory Medicine Uji\u2010Tokushukai Medical Center  Uji Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"name":"on behalf of the Japan LCH Study Group (JLSG)","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2018,9,11]]},"reference":[{"key":"e_1_2_7_2_1","doi-asserted-by":"publisher","DOI":"10.1111\/ped.12380"},{"key":"e_1_2_7_3_1","doi-asserted-by":"publisher","DOI":"10.1111\/bjh.13247"},{"key":"e_1_2_7_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1470-2045(17)30031-1"},{"key":"e_1_2_7_5_1","doi-asserted-by":"publisher","DOI":"10.1067\/mpd.2001.111331"},{"key":"e_1_2_7_6_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2007-08-106211"},{"key":"e_1_2_7_7_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2012-09-455774"},{"key":"e_1_2_7_8_1","doi-asserted-by":"publisher","DOI":"10.1002\/cncr.21985"},{"key":"e_1_2_7_9_1","doi-asserted-by":"publisher","DOI":"10.1007\/s12185-016-1993-3"},{"key":"e_1_2_7_10_1","article-title":"Intensification of induction therapy and prolongation of maintenance therapy did not improve the outcome of pediatric Langerhans cell histiocytosis with single\u2010system multifocal bone lesions: results of the Japan Langerhans Cell Histiocytosis Study Group\u201002 Protocol Study","author":"Morimoto ASY","journal-title":"Int J Hematol"},{"key":"e_1_2_7_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpeds.2003.12.030"},{"key":"e_1_2_7_12_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.20003"},{"key":"e_1_2_7_13_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.20425"},{"key":"e_1_2_7_14_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.24367"},{"key":"e_1_2_7_15_1","doi-asserted-by":"publisher","DOI":"10.1007\/s12185-011-0965-x"},{"key":"e_1_2_7_16_1","doi-asserted-by":"publisher","DOI":"10.1002\/mpo.2950210406"},{"key":"e_1_2_7_17_1","doi-asserted-by":"publisher","DOI":"10.1007\/s12185-014-1717-5"},{"key":"e_1_2_7_18_1","doi-asserted-by":"publisher","DOI":"10.1200\/JCO.2015.62.8289"},{"key":"e_1_2_7_19_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMra1406184"},{"key":"e_1_2_7_20_1","doi-asserted-by":"publisher","DOI":"10.1200\/JCO.2006.09.9861"},{"key":"e_1_2_7_21_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0006-8993(99)02089-2"},{"key":"e_1_2_7_22_1","doi-asserted-by":"publisher","DOI":"10.1215\/S1152851703000516"}],"container-title":["Pediatric Blood &amp; 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