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Central diabetes insipidus as a very late relapse limited to the pituitary stalk in Langerhans cell histiocytosis

  • Shunsuke Nakagawa EMAIL logo , Yuichi Shinkoda , Daisuke Hazeki , Mari Imamura , Yasuhiro Okamoto , Kiyoshi Kawakami and Yoshifumi Kawano
Published/Copyright: April 18, 2016

Abstract

Central diabetes insipidus (CDI) and relapse are frequently seen in multifocal Langerhans cell histiocytosis (LCH). We present two females with multifocal LCH who developed CDI 9 and 5 years after the initial diagnosis, respectively, as a relapse limited to the pituitary stalk. Combination chemotherapy with cytarabine reduced the mass in the pituitary stalk. Although CDI did not improve, there has been no anterior pituitary hormone deficiency (APHD), neurodegenerative disease in the central nervous system (ND-CNS) or additional relapse for 2 years after therapy. It was difficult to predict the development of CDI in these cases. CDI might develop very late in patients with multifocal LCH, and therefore strict follow-up is necessary, especially with regard to symptoms of CDI such as polydipsia and polyuria. For new-onset CDI with LCH, chemotherapy with cytarabine might be useful for preventing APHD and ND-CNS.

  1. Author contributions: All the authors have accepted responsibility for the entire content of this submitted manuscript and approved submission.

  2. Research funding: None declared.

  3. Employment or leadership: None declared.

  4. Honorarium: None declared.

  5. Competing interests: The funding organization(s) played no role in the study design; in the collection, analysis, and interpretation of data; in the writing of the report; or in the decision to submit the report for publication.

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Received: 2015-9-29
Accepted: 2016-3-7
Published Online: 2016-4-18
Published in Print: 2016-7-1

©2016 by De Gruyter

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