TY - JOUR
T1 - Analysis of 43 cases of Langerhans cell histiocytosis (LCH)-induced central diabetes insipidus registered in the JLSG-96 and JLSG-02 studies in Japan
AU - Shioda, Yoko
AU - Adachi, Souichi
AU - Imashuku, Shinsaku
AU - Kudo, Kazuko
AU - Imamura, Toshihiko
AU - Morimoto, Akira
N1 - Funding Information:
Acknowledgments This work was supported by a Grant for Clinical Cancer Research and Research on Measures for Intractable Diseases from the Japanese Ministry of Health, Labour, and Welfare. The authors thank all physicians for registering their patients and for providing data for the study. Yasuko Hashimoto at the registration office is also thanked for her assistance in preparing the manuscript.
Copyright:
Copyright 2012 Elsevier B.V., All rights reserved.
PY - 2011/12
Y1 - 2011/12
N2 - To determine the ability of recent systemic chemotherapy protocols to reduce the incidence of central diabetes insipidus (CDI) in Langerhans cell histiocytosis (LCH), 43 CDI cases that belonged to a cohort of 348 pediatric patients with multi-focal LCH who were treated with the JLSG-96/-02 protocols were analyzed. The overall incidence of CDI was 12.4%, but in 24 cases CDI was already present at the time LCH was diagnosed. Thus, CDI developed during or after systemic chemotherapy over a follow-up period of 5.0 (0.2-14.7) years in only 19 patients (5.9%), with 7.4% at 5-year cumulative risk by Kaplan-Meier analysis. In two cases, complete resolution of CDI was noted. Anterior pituitary hormone deficiency was detected in 13 cases, while CDI-associated neurodegenerative disease was observed in six cases. The JLSG-96/-02 protocol appears to effectively reduce the occurrence of CDI. However, novel therapeutic measures are required to reverse pre-existing CDI and to prevent CDI-associated neurological complications.
AB - To determine the ability of recent systemic chemotherapy protocols to reduce the incidence of central diabetes insipidus (CDI) in Langerhans cell histiocytosis (LCH), 43 CDI cases that belonged to a cohort of 348 pediatric patients with multi-focal LCH who were treated with the JLSG-96/-02 protocols were analyzed. The overall incidence of CDI was 12.4%, but in 24 cases CDI was already present at the time LCH was diagnosed. Thus, CDI developed during or after systemic chemotherapy over a follow-up period of 5.0 (0.2-14.7) years in only 19 patients (5.9%), with 7.4% at 5-year cumulative risk by Kaplan-Meier analysis. In two cases, complete resolution of CDI was noted. Anterior pituitary hormone deficiency was detected in 13 cases, while CDI-associated neurodegenerative disease was observed in six cases. The JLSG-96/-02 protocol appears to effectively reduce the occurrence of CDI. However, novel therapeutic measures are required to reverse pre-existing CDI and to prevent CDI-associated neurological complications.
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U2 - 10.1007/s12185-011-0965-x
DO - 10.1007/s12185-011-0965-x
M3 - Article
C2 - 22139588
AN - SCOPUS:84355165245
VL - 94
SP - 545
EP - 551
JO - International Journal of Hematology
JF - International Journal of Hematology
SN - 0925-5710
IS - 6
ER -