Endocrinology and Metabolism

Case Reports

Adrenal gland
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; Endocrinol Metab. 2014;29(3):394-399. Published online September 25, 2014; DOI: https://doi.org/10.3803/EnM.2014.29.3.394

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We report the rare case of an adult who was diagnosed with recurrent multisystem Langerhans cell histiocytosis (LCH) involving the pituitary stalk and lung who present with central diabetes insipidus and was successfully treated with systemic steroids and chemotherapy. A 49-year-old man visited our hospital due to symptoms of polydipsia and polyuria that started 1 month prior. Two years prior to presentation, he underwent excision of right 6th and 7th rib lesions for the osteolytic lesion and chest pain, which were later confirmed to be LCH on pathology. After admission, the water deprivation test was done and the result indicated that he had central diabetes insipidus. Sella magnetic resonance imaging showed a mass on the pituitary stalk with loss of normal bright spot at the posterior lobe of the pituitary. Multiple patchy infiltrations were detected in both lung fields by computed tomography (CT). He was diagnosed with recurrent LCH and was subsequently treated with inhaled desmopressin, systemic steroids, vinblastine, and mercaptopurine. The pituitary mass disappeared after two months and both lungs were clear on chest CT after 11 months. Although clinical remission in multisystem LCH in adults is reportedly rare, our case of adult-onset multisystem LCH was treated successfully with systemic chemotherapy using prednisolone, vinblastine, and 6-mercaptopurine, which was well tolerated.

Citations

Citations to this article as recorded by

Adult localized Langerhans cell histiocytosis: A case report
Pan-Pan Yang, Su-Ye Hu, Xu-Ya Chai, Xiao-Meng Shi, Li-Xia Liu, Ling-E Li
World Journal of Clinical Cases.2023; 11(34): 8164. CrossRef
Adult Langerhans Cell Histiocytosis Masquerading as Hidradenitis Suppurativa
Jason Chertoff, Julian Chung, Ali Ataya
American Journal of Respiratory and Critical Care Medicine.2017; 195(8): e34. CrossRef
Articles in 'Endocrinology and Metabolism' in 2014
Won-Young Lee
Endocrinology and Metabolism.2015; 30(1): 47. CrossRef
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-onset Langerhans cell histiocytosis presenting with adipsic diabetes insipidus, diabetes mellitus and hypopituitarism: A case report and review of literature
Erick S. Mendoza, Amy A. Lopez, Valerie Ann U. Valdez, Jean D. Uy-Ho, Sjoberg A. Kho
Journal of Clinical and Translational Endocrinology: Case Reports.2015; 1(1): 1. CrossRef

A Case of Thyrotoxic Periodic Paralysis with Rhabdomyolysis.: Seo Hee Lee, Seong Yeol Kim, Hae Ri Lee, Jun Goo Kang, Ohk Hyun Ryu, Chul Sik Kim, Byung Wan Lee, Seong Jin Lee, Eun Gyoung Hong, Hyeon Kyu Kim, Doo Man Kim, Jae Myung Yu, Sung Hee Ihm, Moon Gi Choi, Hyung Joon Yoo; J Korean Endocr Soc. 2008;23(6):425-429. Published online December 1, 2008; DOI: https://doi.org/10.3803/jkes.2008.23.6.425

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Abstract PDF: Hyperthyroidism combined with rhabdomyolysis is extremely rare. There are only 6 reported cases of hyperthyroidism accompanied with rhabdomyolysis in the medical literature. Rhabdomyolysis is a syndrome involving the breakdown of skeletal muscle, and this causes myoglobin and intracellular protein to leak into the circulation. The causes of rhabdomyolysis include trauma, electrolyte abnormality, infection, drug, toxin and hypothyroidism. We report here on a patient who presented with thyrotoxic periodic paralysis and rhabdomyolysis with hypokalemia. He complained of lower leg paralysis along with muscle tenderness, and the laboratory findings showed elevated creatine kinase (CK) levels. After treatment by hydration, potassium replacement and drug medication, including propylthiouracil and beta-blocker, his CK levels were normalized and his symptoms were much improved. For patient with thyrotoxic periodic paralysis and muscle tenderness, the possibility of rhabdomyolysis should be clarified by examining the CK levels.

A Case of Unilateral Exophthalmos Caused by a Dural Arteriovenous Malformation in Thyroid-Associated Ophthalmopathy.: Sun Ryoung Choi, Seong Jin Lee, Hae Ri Lee, Jun Goo Kang, Ohk Hyun Ryu, Chul Sik Kim, Byung Wan Lee, Eun Gyung Hong, Hyeon Kyu Kim, Doo Man Kim, Jae Myung Yoo, Sung Hee Ihm, Moon Gi Choi, Hyung Joon Yoo; J Korean Endocr Soc. 2008;23(1):51-55. Published online February 1, 2008; DOI: https://doi.org/10.3803/jkes.2008.23.1.51

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Abstract PDF

Thyroid-associated ophthalmopathy is associated with thyroid dysfunction, particularly Graves' disease, and is manifested as eye signs, including proptosis. In cases of unilateral exophthalmos with thyroid-associated ophthalmopathy, other causes such as orbital neoplasm, carotid-cavernous fistula, cavernous sinus thrombosis, and dural arteriovenous malformation (AVM) should be excluded. Dural AVM, an abnormal dural arteriovenous connection, is a rare neurovascular entity that mimics thyroid-associated ophthalmopathy. When eye involvement is unilateral or asymmetric, dural AVM can be considered in the differential diagnosis of thyroid-associated ophthalmopathy. A twenty-six year-old woman presented with unilateral exophthalmos in Graves' disease. By orbital magnetic resonance imaging and cerebral angiography, thyroid-associated ophthalmopathy and dural AVM were diagnosed. The unilateral exophthalmos improved after coil embolization of the dural AVM. In summary, we report the first case of a dural AVM with Graves' disease and thyroid-associated ophthalmopathy.

Citations

Citations to this article as recorded by

Proptosis as a Primary Symptom of Brain Arteriovenous Malformation
Jong Eun Lee, Jin Sook Yoon, Keun Young Park
Ophthalmic Plastic & Reconstructive Surgery.2020; 36(2): e53. CrossRef
Ophthalmopathy Induced by Bilateral Carotid Cavernous Fistula in a Patient with Graves' Disease
Jong Kun Ha, Ji Hye Suk, A Ra Jo, Chan Woo Jung, Bong Jae Kim, Seong Oh Park, Sang Su Kim, Mi Kyung Kim
Endocrinology and Metabolism.2011; 26(4): 335. CrossRef

Original Article

The Clinical Significance of Retinoic Acid Receptor beta Expressions in Primary and Recurred Metastatic Lymph Node Papillary Thyroid Carcinomas.: Jae Pil Han, Seong Jin Lee, Kyung Chan Choi, Young Euy Park, Hae Ri Lee, Jun Goo Kang, Ohk Hyun Ryu, Chul Sik Kim, Byung Wan Lee, Eun Gyung Hong, Hyeon Kyu Kim, Doo Man Kim, Jae Myung Yoo, Sung Hee Ihm, Hyung Joon Yoo, Moon Gi Choi; J Korean Endocr Soc. 2007;22(6):419-427. Published online December 1, 2007; DOI: https://doi.org/10.3803/jkes.2007.22.6.419

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Abstract PDF: BACKGROUND
The present study was designed to investigate the correlations of retinoic acid receptor beta(RARbeta) expression for primary and recurred metastatic lymph node (LN) papillary thyroid carcinoma (PTC) tissues and the correlations of RARbeta expression with the uptake of I(131) as detected on a whole body scan (WBS). METHODS: Primary and metastatic LN PTC tissues were examined by immunohistochemical methods. Staining positivity was calculated, and staining intensity was graded as negative (0), weak (1+), moderate (2+) and strong (3+). Nuclear staining intensity (NSI) of cells from tissues was also examined. RESULTS: Seventeen patients who had regional cervical LN metastasis without distant metastasis were included in the study, and 13 patients had the abnormal uptake of I(131) as detected on a WBS. In primary PTC tissues, RARbeta staining positivity and intensity of carcinoma cells were significantly higher than those of normal cells but NSI was significantly higher in normal cells than carcinoma cells. Between primary and metastatic LN PTC tissues, RARbeta staining intensity was correlated after controlling for age. Primary PTC tissues from 14 (82.4%) out of 17 patients were concordant between NSI and the uptake of I(131) as detected on a WBS. NSI predicted the I(131) uptake as detected on a WBS with 81.3% positive predicted value (PPV) and 100% negative predicted value. Metastatic LN PTC tissues from 13 (76.5%) out of 17 patients were concordant between NSI and the uptake of I(131) as detected on a WBS. NSI predicted the uptake of I(131) as detected on a WBS with 76.5% PPV. When the results of NSI taken either as positive or negative were correlated with those of the uptake of I(131) as detected on a WBS in primary and metastatic LN PTC tissues, the correlation was not significant after controlling for age. CONCLUSION: Our results demonstrate that nuclear RARbeta expression may be decreased in PTC tissues than normal thyroid tissues, and RARbeta expression in primary PTC tissues as well as in recurred metastatic LN PTC tissues may predict the uptake of I(131) as detected on a WBS.

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