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Serial assessments of anterior pituitary hormones in a case of mixed histiocytosis representing Langerhans cell histiocytosis overlapping with Erdheim–Chester disease

SUMMARY: A 61-year-old man developed central diabetes insipidus caused by mixed histiocytosis (MH) representing Langerhans cell histiocytosis overlapping with Erdheim–Chester disease. Bone, skin, vascular, and retroperitoneal involvements were also observed. Dynamic hormonal testing showed normal re...

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Autores principales: Ito, Kei, Ito, Jun, Yamamoto, Yuki, Nakajima, Rikako, Fujii, Masanao, Katakura, Yukino, Muramatsu, Aiko, Takayashiki, Norio, Toyama, Kazuhiro, Kurokawa, Mineo, Yagyu, Hiroaki
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Bioscientifica Ltd 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8185526/
https://www.ncbi.nlm.nih.gov/pubmed/33982663
http://dx.doi.org/10.1530/EDM-21-0030
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author Ito, Kei
Ito, Jun
Yamamoto, Yuki
Nakajima, Rikako
Fujii, Masanao
Katakura, Yukino
Muramatsu, Aiko
Takayashiki, Norio
Toyama, Kazuhiro
Kurokawa, Mineo
Yagyu, Hiroaki
author_facet Ito, Kei
Ito, Jun
Yamamoto, Yuki
Nakajima, Rikako
Fujii, Masanao
Katakura, Yukino
Muramatsu, Aiko
Takayashiki, Norio
Toyama, Kazuhiro
Kurokawa, Mineo
Yagyu, Hiroaki
author_sort Ito, Kei
collection PubMed
description SUMMARY: A 61-year-old man developed central diabetes insipidus caused by mixed histiocytosis (MH) representing Langerhans cell histiocytosis overlapping with Erdheim–Chester disease. Bone, skin, vascular, and retroperitoneal involvements were also observed. Dynamic hormonal testing showed normal responses for anterior pituitary hormones, except for impaired secretion of growth hormone (GH). MRI of the brain showed thickening of the pituitary stalk with slightly reduced signal hyperintensity in the posterior pituitary lobe on T1-weighted imaging. During 2 years of follow-up without radical treatment for MH, imaging studies suggested extension of vascular and retroperitoneal involvements. In contrast, brain MRI did not show any particular interval changes, except for the disappearance of hyperintense signalling in the posterior pituitary lobe. Moreover, no other anterior pituitary dysfunctions beyond GH deficiency emerged during the 2 years of follow-up. The natural history of MH in this case is described, focusing on serial assessments of pituitary functions using dynamic tests. LEARNING POINTS: Erdheim–Chester disease and Langerhans cell histiocytosis overlapping as MH was described, focusing on pituitary functions. MH caused both GH deficiency and central diabetes insipidus. Despite a lack of radical therapy for MH, no other anterior pituitary dysfunctions emerged for 2 years. Radiological images showed no particular interval changes in pituitary stalk lesions, while vascular and retroperitoneal involvements extended.
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spelling pubmed-81855262021-06-10 Serial assessments of anterior pituitary hormones in a case of mixed histiocytosis representing Langerhans cell histiocytosis overlapping with Erdheim–Chester disease Ito, Kei Ito, Jun Yamamoto, Yuki Nakajima, Rikako Fujii, Masanao Katakura, Yukino Muramatsu, Aiko Takayashiki, Norio Toyama, Kazuhiro Kurokawa, Mineo Yagyu, Hiroaki Endocrinol Diabetes Metab Case Rep Insight into Disease Pathogenesis or Mechanism of Therapy SUMMARY: A 61-year-old man developed central diabetes insipidus caused by mixed histiocytosis (MH) representing Langerhans cell histiocytosis overlapping with Erdheim–Chester disease. Bone, skin, vascular, and retroperitoneal involvements were also observed. Dynamic hormonal testing showed normal responses for anterior pituitary hormones, except for impaired secretion of growth hormone (GH). MRI of the brain showed thickening of the pituitary stalk with slightly reduced signal hyperintensity in the posterior pituitary lobe on T1-weighted imaging. During 2 years of follow-up without radical treatment for MH, imaging studies suggested extension of vascular and retroperitoneal involvements. In contrast, brain MRI did not show any particular interval changes, except for the disappearance of hyperintense signalling in the posterior pituitary lobe. Moreover, no other anterior pituitary dysfunctions beyond GH deficiency emerged during the 2 years of follow-up. The natural history of MH in this case is described, focusing on serial assessments of pituitary functions using dynamic tests. LEARNING POINTS: Erdheim–Chester disease and Langerhans cell histiocytosis overlapping as MH was described, focusing on pituitary functions. MH caused both GH deficiency and central diabetes insipidus. Despite a lack of radical therapy for MH, no other anterior pituitary dysfunctions emerged for 2 years. Radiological images showed no particular interval changes in pituitary stalk lesions, while vascular and retroperitoneal involvements extended. Bioscientifica Ltd 2021-04-26 /pmc/articles/PMC8185526/ /pubmed/33982663 http://dx.doi.org/10.1530/EDM-21-0030 Text en >© The authors https://creativecommons.org/licenses/by-nc-nd/4.0/ This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License. (https://creativecommons.org/licenses/by-nc-nd/4.0/) .
spellingShingle Insight into Disease Pathogenesis or Mechanism of Therapy
Ito, Kei
Ito, Jun
Yamamoto, Yuki
Nakajima, Rikako
Fujii, Masanao
Katakura, Yukino
Muramatsu, Aiko
Takayashiki, Norio
Toyama, Kazuhiro
Kurokawa, Mineo
Yagyu, Hiroaki
Serial assessments of anterior pituitary hormones in a case of mixed histiocytosis representing Langerhans cell histiocytosis overlapping with Erdheim–Chester disease
title Serial assessments of anterior pituitary hormones in a case of mixed histiocytosis representing Langerhans cell histiocytosis overlapping with Erdheim–Chester disease
title_full Serial assessments of anterior pituitary hormones in a case of mixed histiocytosis representing Langerhans cell histiocytosis overlapping with Erdheim–Chester disease
title_fullStr Serial assessments of anterior pituitary hormones in a case of mixed histiocytosis representing Langerhans cell histiocytosis overlapping with Erdheim–Chester disease
title_full_unstemmed Serial assessments of anterior pituitary hormones in a case of mixed histiocytosis representing Langerhans cell histiocytosis overlapping with Erdheim–Chester disease
title_short Serial assessments of anterior pituitary hormones in a case of mixed histiocytosis representing Langerhans cell histiocytosis overlapping with Erdheim–Chester disease
title_sort serial assessments of anterior pituitary hormones in a case of mixed histiocytosis representing langerhans cell histiocytosis overlapping with erdheim–chester disease
topic Insight into Disease Pathogenesis or Mechanism of Therapy
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8185526/
https://www.ncbi.nlm.nih.gov/pubmed/33982663
http://dx.doi.org/10.1530/EDM-21-0030
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