Endocrinology and Metabolism

Case Reports

A Case of Diabetes Insipidus with Langerhans Cell Histiocytosis in Adult.: Jae Hoon Chung, Kwang Won Kim, Kyu Jeung Ahn, Yong Ki Min, Myung Shik Lee, Moon Kyu Lee, Eun Mi Koh, Choon Kwan Kim, Mann Pyo Jung; J Korean Endocr Soc. 1996;11(3):330-335. Published online November 7, 2019

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Abstract PDF: In Langerhans cell histiocytosis, diabetes insipidus is the most common endocrinologic complication. We experienced a case of Langerhans cell histiocytosis, involving pituitary stalk and lung. The patient was a 43 year old male with complaint of polyuria and polydipsia. The water deprivation test was carried out to confirm the diagnosis of diabetes insipidus. We found multiple small cysts and nodules in HRCT of lung, and diagnosed Langerhans cell histiocytosis by transbronchial lung biopsy, The patient was managed conservatively with DDAVP nasal spray. The polyuria,polydipsia was relieved completely. After that, we follow up and observe closely the patients lung and pituitary lesion.

Langerhans Cell Histiocytosis in the Thyroid and Draining Lymph Nodes: A Case Report.: Dong Hae Chung, Seung Yeon Ha, Hyun Yee Cho, Na Rae Kim, Jung Suk An, Young Don Lee, Sanghui Park; Endocrinol Metab. 2012;27(2):138-141. Published online June 20, 2012; DOI: https://doi.org/10.3803/EnM.2012.27.2.138

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A 53-year-old woman was presented with several 0.3-0.6 cm-sized nodules within the right lobe of the thyroid. Histologic sections of the thyroid demonstrated multiple papillary microcarcinomas in the background of lymphocytic thyroiditis, with a small focus of Langerhans cell histiocytosis (LCH). Small LCH nodules were also found in the draining cervical lymph nodes. Although the association of LCH with papillary thyroid carcinoma in the thyroid has been reported, their co-existence with LCH in the draining lymph nodes is very rare.

Citations

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Thyroid Langerhans cell histiocytosis concurrent with papillary thyroid carcinoma: A case report and literature review
Bin Mi, Di Wu, Yue Fan, Benjamin Ka Seng Thong, Yudong Chen, Xue Wang, Chaofu Wang
Frontiers in Medicine.2023;[Epub] CrossRef
Adult Langerhans cell histiocytosis of skull in a patient with synchronous papillary thyroid carcinoma and Castleman disease
In Kyeong Kim, Kyoung Yul Lee
BMJ Case Reports.2021; 14(1): e239341. CrossRef
Langerhans cell histiocytosis of the thyroid together with papillary thyroid carcinoma
Hatice Ozisik, Banu Sarer Yurekli, Derya Demir, Yesim Ertan, Ilgın Yildirim Simsir, Murat Ozdemir, Mehmet Erdogan, Sevki Cetinkalp, Gokhan Ozgen, Fusun Saygili
Hormones.2020; 19(2): 253. CrossRef
BRAF gene mutations in synchronous papillary thyroid carcinoma and Langerhans cell histiocytosis co-existing in the thyroid gland: a case report and literature review
Mohammad A. Al Hamad, Hassan M. Albisher, Weam R. Al Saeed, Ahmed T. Almumtin, Fatimah M. Allabbad, Mohammed A. Shawarby
BMC Cancer.2019;[Epub] CrossRef
Adult Multisystem Langerhans Cell Histiocytosis Presenting with Central Diabetes Insipidus Successfully Treated with Chemotherapy
Jung-Eun Choi, Hae Ri Lee, Jung Hun Ohn, Min Kyong Moon, Juri Park, Seong Jin Lee, Moon-Gi Choi, Hyung Joon Yoo, Jung Han Kim, Eun-Gyoung Hong
Endocrinology and Metabolism.2014; 29(3): 394. CrossRef

Original Article

Clinical Characteristics of Langerhans Cell Histiocytosis with Hypothalamo-Pituitary Involvement.: Eun Shil Hong, Jung Hun Ohn, Jung Hee Kim, Yul Hwang-Bo, Jin Joo Kim, Jung Hee Kwon, Jung Won Lee, Se Youn Choi, Eun Kyung Lee, Sun Wook Cho, Chan Soo Shin, Kyong Soo Park, Hak Chul Jang, Bo Youn Cho, Hong Kyu Lee, Choong Ho Shin, Sei Won Yang, Seong Yeon Kim; Endocrinol Metab. 2011;26(1):38-43. Published online March 1, 2011; DOI: https://doi.org/10.3803/EnM.2011.26.1.38

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Abstract PDF

BACKGROUND
Langerhans cell histiocytosis (LCH) is a rare disease that involves a clonal proliferation of Langerhans cells. LCH has a predilection for hypothalamo-pituitary axis (HPA) dysfunction, and this leads to diabetes insipidus (DI) and/or anterior pituitary dysfunction. Here, we describe the endocrine dysfunction and clinical characteristics of adult patients with LCH and we analyzed the differences between an adult-onset type and a childhood-onset type. METHODS: The data was obtained from a retrospective chart review of the patients with LCH that involved the HPA and who attended Seoul National University Hospital. The patients were classified into the adult-onset type (age at the time of diagnosis > or = 16) and the childhood-onset type (age at the time of diagnosis < or = 15). RESULTS: Ten patients (9 males and 1 female) were diagnosed with LCH involving the HPA. Five patients were classified as an adultonset type and the other five patients were classified as a childhood-onset type. The median follow-up duration was 6 (3-12) years for the adult-onset type and 16 (15-22) years for the childhood-onset type. All the patients presented with DI as the initial manifestation of HPA involvement. Four adult-onset patients and three childhood-onset patients had a multi-system disease. Panhypopituitarism developed in three adult-onset patients and in one childhood-onset patient. The pituitary lesion of the three adult-onset patients had spread to the brain during the follow-up duration. In contrast, the pituitary lesion of the other two adult-onset patients without panhypopituitarism and all the childhood-onset patients had not changed. CONCLUSION: DI was the initial presentation symptom of HPA involvement. Anterior pituitary hormone deficiency followed in some patients. Compared with the childhood-onset patients, the adult-onset patients were more likely to have panhypopituitarism and a poor prognosis.

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Pulmonary Langerhans Cell Histiocytosis in an Adult Male Presenting with Central Diabetes Insipidus and Diabetes Mellitus: A Case Report
Yeun Seoung Choi, Jung Soo Lim, Woocheol Kwon, Soon-Hee Jung, Il Hwan Park, Myoung Kyu Lee, Won Yeon Lee, Suk Joong Yong, Seok Jeong Lee, Ye-Ryung Jung, Jiwon Choi, Ji Sun Choi, Joon Taek Jeong, Jin Sae Yoo, Sang-Ha Kim
Tuberculosis and Respiratory Diseases.2015; 78(4): 463. CrossRef
Adult Multisystem Langerhans Cell Histiocytosis Presenting with Central Diabetes Insipidus Successfully Treated with Chemotherapy
Jung-Eun Choi, Hae Ri Lee, Jung Hun Ohn, Min Kyong Moon, Juri Park, Seong Jin Lee, Moon-Gi Choi, Hyung Joon Yoo, Jung Han Kim, Eun-Gyoung Hong
Endocrinology and Metabolism.2014; 29(3): 394. CrossRef

Case Report

A Case of Langerhans Cell Histiocytosis Presented with Central Diabetes Insipidus.: Byung Chul Kim, Mi Yeoun Lee, Young Kue Kim, Ok Jun Lee, Sung Soo Koong, Tae Keun Oh; J Korean Endocr Soc. 2005;20(5):513-518. Published online October 1, 2005; DOI: https://doi.org/10.3803/jkes.2005.20.5.513

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Abstract PDF

Langerhans cell histiocytosis can cause central diabetes insipidus. Here, a case of Langerhans cell histiocytosis invading the pituitary stalk was experienced. The patient was 15 years old boy, with complaint of polydipsia and polyuria. A water deprivation test was carried out, and the urine osmolarity was increased from 165 to 469 mosm/kg following an injection of AVP to confirm the diagnosis of central diabetes insipidus. A pituitary function stimulation test gave a normal response. A sellar MRI was performed, which showed a thickened pituitary stalk mass (about 5.7mm), with an increased size, 6.9 mm, on a second MRI 2 month later. A tissue biopsy was performed, which showed aggregates of histiocytes and inflammatory cells, with prominent eosinophils (H&E), and also revealed strong reactivity to anti-CD1a antibody on the immunohistochemistry. After confirmative tissue diagnosis, the patient received radiotherapy (900 cGy). The thickened mass of the pituitary stalk disappeared on the MRI following the radiotherapy. The patient was managed with DDAVP nasal spray, after which the polyuric symptoms were completely relieved.

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Citations to this article as recorded by

Adult Multisystem Langerhans Cell Histiocytosis Presenting with Central Diabetes Insipidus Successfully Treated with Chemotherapy
Jung-Eun Choi, Hae Ri Lee, Jung Hun Ohn, Min Kyong Moon, Juri Park, Seong Jin Lee, Moon-Gi Choi, Hyung Joon Yoo, Jung Han Kim, Eun-Gyoung Hong
Endocrinology and Metabolism.2014; 29(3): 394. CrossRef
Clinical Characteristics of Langerhans Cell Histiocytosis with Hypothalamo-Pituitary Involvement
Eun Shil Hong, Jung Hun Ohn, Jung Hee Kim, Yul Hwang-Bo, Jin Joo Kim, Jung Hee Kwon, Jung Won Lee, Se Youn Choi, Eun Kyung Lee, Sun Wook Cho, Chan Soo Shin, Kyong Soo Park, Hak Chul Jang, Bo Youn Cho, Hong Kyu Lee, Choong Ho Shin, Sei Won Yang, Seong Yeon
Endocrinology and Metabolism.2011; 26(1): 38. CrossRef
A Case of Idiopathic Central Diabetes Insipidus together with Primary Empty Sella and Combined Pituitary Hormone Deficiency
Sun Young Ahn, Kyu Hwan Bae, Myung Hwan Kim, Ji Hyun Lee, Ho Sang Shon, Eui Dal Jung
Journal of Korean Endocrine Society.2007; 22(4): 272. CrossRef

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