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Original Article
A Study of Central Diabetes Insipidus in Head and Neck Langerhans Cell Histiocytosis: A Single Center Experience
Clin Pediatr Hematol Oncol 2018;25:136-41.
Published online October 31, 2018
© 2018 Korean Society of Pediatric Hematology-Oncology and Korean Society for Pediatric Neuro-Oncology

Mi Jin Lee, M.D., Byeong Sub Park, M.D., Yeong Myong Yoo, M.D., Hyun Joo Jung, M.D. and Jun Eun Park, M.D., Ph.D.

Department of Pediatrics, Ajou University School of Medicine, Suwon, Korea
Correspondence to: Jun Eun Park
Department of Pediatrics, Ajou University School of Medicine, 164 Worldcup-ro, Yeongtong-gu, Suwon 16499, Korea
Tel: +82-31-219-5167
Fax: +82-31-219-5169
E-mail: pedpje@ajou.ac.kr
ORCID ID: orcid.org/0000-0003-4292-3500
Received September 17, 2018; Revised September 26, 2018; Accepted October 2, 2018.
This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.
Abstract
Background: Langerhans cell histiocytosis (LCH) frequently involves the head and neck and increases the risk of central nervous system (CNS) involvement of LCH, such as central diabetes insipidus (CDI), when the craniofacial bones are involved. We analyzed risk factors and clinical features of CDI among patients with LCH involving the head and neck.
Methods: From January 1, 2000 to May 1, 2018, 63 patients with histologically confirmed LCH in the Department of Pediatrics, Ajou University Hospital were retrospectively analyzed.
Results: Forty eight cases (76.2%) of patients had head and neck involvement, and 9 cases (14.3%) in craniofacial bones at the time of initial diagnosis of LCH. CDI was found in 6 cases (9.5%) among all LCH patients, 6 cases (12.2%) among patients with head and neck involvement, and 3 cases (33.3%) among patients with craniofacial bone involvement. Three cases of CDI occurred at the time of initial LCH diagnosis, and another 3 cases occurred at the time of 2, 4, and 8 years after initial LCH diagnosis. Of the 6 CDI patients, 3 had CNS risk lesions and 3 had no CNS risk lesions, but all had multisystem involvement of LCH.
Conclusion: CDI can occur even in patients with head and neck LCH without CNS risk lesions, if there are multisystem involvement of LCH. Patients with head and neck LCH may develop CDI over time, so continuous observations should be done while considering the occurrence of CDI.
Keywords: Langerhans cell histiocytosis, Head and neck, Central diabetes insipidus


October 2018, 25 (2)
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  • Jun Eun Park