Endocrinology and Metabolism

Special Article

Miscellaneous
COVID-19 Vaccination for Endocrine Patients: A Position Statement from the Korean Endocrine Society: Cheol Ryong Ku, Kyong Yeun Jung, Chang Ho Ahn, Jun Sung Moon, Ju Hee Lee, Eun Heui Kim, Hyemi Kwon, Hee Kyung Kim, Sunghwan Suh, Sangmo Hong, Jeonghoon Ha, Eun Roh, Jin Hwa Kim, Mi-kyung Kim, the Committee of Clinical Practice Guideline of the Korean Endocrine Society; Endocrinol Metab. 2021;36(4):757-765. Published online August 17, 2021; DOI: https://doi.org/10.3803/EnM.2021.404

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Since the first outbreak of coronavirus disease 2019 (COVID-19), ongoing efforts have been made to discover an efficacious vaccine against COVID-19 to combat the pandemic. In most countries, both mRNA and DNA vaccines have been administered, and their side effects have also been reported. The clinical course of COVID-19 and the effects of vaccination against COVID-19 are both influenced by patients’ health status and involve a systemic physiological response. In view of the systemic function of endocrine hormones, endocrine disorders themselves and the therapeutics used to treat them can influence the outcomes of vaccination for COVID-19. However, there are very limited data to support the development of clinical guidelines for patients with specific medical backgrounds based on large clinical trials. In the current severe circumstances of the COVID-19 pandemic, position statements made by clinical specialists are essential to provide appropriate recommendations based on both medical evidence and clinical experiences. As endocrinologists, we would like to present the medical background of COVID-19 vaccination, as well as precautions to prevent the side effects of COVID-19 vaccination in patients with specific endocrine disorders, including adrenal insufficiency, diabetes mellitus, osteoporosis, autoimmune thyroid disease, hypogonadism, and pituitary disorders.

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COVID-19 mRNA vaccine may trigger subacute thyroiditis
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David Tak Wai Lui, Chi Ho Lee, Yu Cho Woo, Ivan Fan Ngai Hung, Karen Siu Ling Lam
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Adult-Onset Type 1 Diabetes Development Following COVID-19 mRNA Vaccination
Hyeyeon Moon, Sunghwan Suh, Mi Kyoung Park
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Prior immunization status of COVID-19 patients and disease severity: A multicenter retrospective cohort study assessing the different types of immunity
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Mortality and Severity of Coronavirus Disease 2019 in Patients with Long-Term Glucocorticoid Therapy: A Korean Nationwide Cohort Study
Eu Jeong Ku, Keeho Song, Kyoung Min Kim, Gi Hyeon Seo, Soon Jib Yoo
Endocrinology and Metabolism.2023; 38(2): 253. CrossRef
Pituitary Diseases and COVID-19 Outcomes in South Korea: A Nationwide Cohort Study
Jeonghoon Ha, Kyoung Min Kim, Dong-Jun Lim, Keeho Song, Gi Hyeon Seo
Journal of Clinical Medicine.2023; 12(14): 4799. CrossRef
Inactivated SARS-CoV-2 vaccination does not disturb the clinical course of Graves’ disease: An observational cohort study
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Vaccine.2023; 41(38): 5648. CrossRef
Adrenal Crisis Associated With COVID-19 Vaccination in Patients With Adrenal Insufficiency
Yukako Kurematsu, Takako Mohri, Sadanori Okada, Yutaka Takahashi
JCEM Case Reports.2023;[Epub] CrossRef
Adverse Events Associated with COVID-19 Vaccination in Adolescents with Endocrinological Disorders: A Cross-Sectional Study
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Neue Aspekte der Glukokortikoidsubstitution bei Nebennierenrindeninsuffizienz
Tina Kienitz, Gesine Meyer
Der Internist.2022; 63(1): 12. CrossRef
Endocrine Follow-up During Post-Acute COVID-19: Practical Recommendations Based on Available Clinical Evidence
Rimesh Pal, Ameya Joshi, Sanjay K. Bhadada, Mainak Banerjee, Suresh Vaikkakara, Satinath Mukhopadhyay
Endocrine Practice.2022; 28(4): 425. CrossRef
Safety of Inactivated and mRNA COVID-19 Vaccination Among Patients Treated for Hypothyroidism: A Population-Based Cohort Study
Xi Xiong, Carlos King Ho Wong, Ivan Chi Ho Au, Francisco Tsz Tsun Lai, Xue Li, Eric Yuk Fai Wan, Celine Sze Ling Chui, Esther Wai Yin Chan, Franco Wing Tak Cheng, Kristy Tsz Kwan Lau, Chi Ho Lee, Yu Cho Woo, David Tak Wai Lui, Ian Chi Kei Wong
Thyroid.2022; 32(5): 505. CrossRef
The New Entity of Subacute Thyroiditis amid the COVID-19 Pandemic: From Infection to Vaccine
Mihaela Popescu, Adina Ghemigian, Corina Maria Vasile, Andrei Costache, Mara Carsote, Alice Elena Ghenea
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Adrenal Crisis Secondary to COVID-19 Vaccination in a Patient With Hypopituitarism
Nikolina Markovic, Anila Faizan, Chirag Boradia, Sridhar Nambi
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Yue Ma, Shui Qiu, Renyi Zhou
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Tania Pilli, Cristina Dalmiglio, Gilda Dalmazio, Alfonso Sagnella, Raffaella Forleo, Lucia Brilli, Fabio Maino, Cristina Ciuoli, Maria Grazia Castagna
European Journal of Endocrinology.2022; 187(1): K7. CrossRef
Diabetes and COVID-19 Vaccination
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The Journal of Korean Diabetes.2021; 22(4): 221. CrossRef

Review Article

Miscellaneous
Clinical Characteristics, Management, and Potential Biomarkers of Endocrine Dysfunction Induced by Immune Checkpoint Inhibitors: Shintaro Iwama, Tomoko Kobayashi, Hiroshi Arima; Endocrinol Metab. 2021;36(2):312-321. Published online April 27, 2021; DOI: https://doi.org/10.3803/EnM.2021.1007

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Immune-related adverse events (irAEs) affecting the endocrine glands are among the most frequent irAEs induced by immune checkpoint inhibitors (ICIs) and include hypopituitarism, primary adrenal insufficiency, thyrotoxicosis, hypothyroidism, hypoparathyroidism, and type 1 diabetes mellitus. Since the incidence and clinical features of endocrine irAEs vary according to the ICI used, it is important to understand the characteristics of these irAEs and to manage each one appropriately. Since some endocrine irAEs, including adrenal crisis and diabetic ketoacidosis, are potentially life-threatening, predicting the risk of endocrine irAEs before their onset is critical. Several autoantibodies have been detected in patients who develop endocrine irAEs, among which anti-thyroid antibodies may be predictive biomarkers of thyroid dysfunction. In this review, we describe the clinical features of each endocrine irAE induced by ICIs and discuss their potential biomarkers, including autoantibodies.

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Clinical characteristics and potential biomarkers of thyroid and pituitary immune-related adverse events
Tomoko Kobayashi, Shintaro Iwama, Hiroshi Arima
Endocrine Journal.2024; 71(1): 23. CrossRef
A case of rapidly progressive insulin-dependent diabetes mellitus without islet autoantibodies developed over two years after the first dose of nivolumab
Kota Nishihama, Yuko Okano, Chisa Inoue, Kanako Maki, Kazuhito Eguchi, Soichiro Tanaka, Atsuro Takeshita, Mei Uemura, Taro Yasuma, Toshinari Suzuki, Esteban C. Gabazza, Yutaka Yano
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Endocrinopathies associated with immune checkpoint inhibitor use
Anupam Kotwal, Randol Kennedy, Nupur Kikani, Sonali Thosani, Whitney Goldner, Afreen Shariff
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Recovery from insulin dependence in immune checkpoint inhibitor‐associated diabetes mellitus: A case report
Marie Okubo, Yuji Hataya, Kanta Fujimoto, Toshio Iwakura, Naoki Matsuoka
Journal of Diabetes Investigation.2023; 14(1): 147. CrossRef
Case Report: A Rising Cause of New-Onset Endocrinopathies After Immunotherapy
Charity Tan, Sarah Hendricks, Kristina Hernandez, Martha Benavides, Rupinderjit Samra
The Journal for Nurse Practitioners.2023; 19(5): 104582. CrossRef
Risk of Thyroid Dysfunction in PD-1 Blockade Is Stratified by the Pattern of TgAb and TPOAb Positivity at Baseline
Xin Zhou, Shintaro Iwama, Tomoko Kobayashi, Masahiko Ando, Hiroshi Arima
The Journal of Clinical Endocrinology & Metabolism.2023; 108(10): e1056. CrossRef
Severe thyrotoxicosis induced by tislelizumab: a case report and literature review
Liman Huo, Chao Wang, Haixia Ding, Xuelian Shi, Bin Shan, Ruoying Zhou, Ping Liang, Juan Hou
Frontiers in Oncology.2023;[Epub] CrossRef
Life-Threatening Endocrinological Immune-Related Adverse Events of Immune Checkpoint Inhibitor Therapy
Aleksandra Basek, Grzegorz K. Jakubiak, Grzegorz Cieślar, Agata Stanek
Cancers.2023; 15(24): 5786. CrossRef
Increased Risk of Thyroid Dysfunction by PD-1 and CTLA-4 Blockade in Patients Without Thyroid Autoantibodies at Baseline
Shintaro Iwama, Tomoko Kobayashi, Yoshinori Yasuda, Takayuki Okuji, Masaaki Ito, Masahiko Ando, Xin Zhou, Ayana Yamagami, Takeshi Onoue, Yohei Kawaguchi, Takashi Miyata, Mariko Sugiyama, Hiroshi Takagi, Daisuke Hagiwara, Hidetaka Suga, Ryoichi Banno, Tets
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Biomarkers and risk factors for the early prediction of immune-related adverse events: a review
Ying Zhang, Xiaoling Zhang, Weiling Li, Yunyi Du, Wenqing Hu, Jun Zhao
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Immune Checkpoint Inhibitors as a Threat to the Hypothalamus–Pituitary Axis: A Completed Puzzle
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Cancers.2022; 14(4): 1057. CrossRef
Elevated TSH Level, TgAb, and Prior Use of Ramucirumab or TKIs as Risk Factors for Thyroid Dysfunction in PD-L1 Blockade
Tomoko Kobayashi, Shintaro Iwama, Ayana Yamagami, Yoshinori Yasuda, Takayuki Okuji, Masaaki Ito, Xin Zhou, Masahiko Ando, Takeshi Onoue, Takashi Miyata, Mariko Sugiyama, Daisuke Hagiwara, Hidetaka Suga, Ryoichi Banno, Tetsunari Hase, Masahiro Morise, Taka
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Preconditioning of the immune system modulates the response of papillary thyroid cancer to immune checkpoint inhibitors
Fabiana Pani, Yoshinori Yasuda, Sylvie T Rousseau, Kevin C Bermea, Solmaz Roshanmehr, Rulin Wang, Srinivasan Yegnasubramanian, Patrizio Caturegli, Luigi Adamo
Journal for ImmunoTherapy of Cancer.2022; 10(12): e005538. CrossRef
Survival benefit of endocrine dysfunction following immune checkpoint inhibitors for nonthyroidal cancers
Anupam Kotwal, Mabel Ryder
Current Opinion in Endocrinology, Diabetes & Obesity.2021; 28(5): 517. CrossRef

Special Article

Miscellaneous
Diagnosis and Treatment of Growth Hormone Deficiency: A Position Statement from Korean Endocrine Society and Korean Society of Pediatric Endocrinology: Jung Hee Kim, Hyun Wook Chae, Sang Ouk Chin, Cheol Ryong Ku, Kyeong Hye Park, Dong Jun Lim, Kwang Joon Kim, Jung Soo Lim, Gyuri Kim, Yun Mi Choi, Seong Hee Ahn, Min Ji Jeon, Yul Hwangbo, Ju Hee Lee, Bu Kyung Kim, Yong Jun Choi, Kyung Ae Lee, Seong-Su Moon, Hwa Young Ahn, Hoon Sung Choi, Sang Mo Hong, Dong Yeob Shin, Ji A Seo, Se Hwa Kim, Seungjoon Oh, Sung Hoon Yu, Byung Joon Kim, Choong Ho Shin, Sung-Woon Kim, Chong Hwa Kim, Eun Jig Lee; Endocrinol Metab. 2020;35(2):272-287. Published online June 24, 2020; DOI: https://doi.org/10.3803/EnM.2020.35.2.272

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Growth hormone (GH) deficiency is caused by congenital or acquired causes and occurs in childhood or adulthood. GH replacement therapy brings benefits to body composition, exercise capacity, skeletal health, cardiovascular outcomes, and quality of life. Before initiating GH replacement, GH deficiency should be confirmed through proper stimulation tests, and in cases with proven genetic causes or structural lesions, repeated GH stimulation testing is not necessary. The dosing regimen of GH replacement therapy should be individualized, with the goal of minimizing side effects and maximizing clinical improvements. The Korean Endocrine Society and the Korean Society of Pediatric Endocrinology have developed a position statement on the diagnosis and treatment of GH deficiency. This position statement is based on a systematic review of evidence and expert opinions.

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Once-Weekly Somapacitan as an Alternative Management of Growth Hormone Deficiency in Prepubertal Children: A Systematic Review and Meta-Analysis of Randomized Controlled Trial
Ghina Tsurayya, Cut Alifiya Nazhifah, Muhammad Rahmat Pirwanja, Putri Oktaviani Zulfa, Muhammad Raihan Ramadhan Tatroman, Fajar Fakri, Muhammad Iqhrammullah
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Evaluation of Adult Height in Patients with Non-Permanent Idiopathic GH Deficiency
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The effect of hypothalamic involvement and growth hormone treatment on cardiovascular risk factors during the transition period in patients with childhood-onset craniopharyngioma
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Annals of Pediatric Endocrinology & Metabolism.2023; 28(2): 107. CrossRef
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International Journal of Cardiology Cardiovascular Risk and Prevention.2023; 19: 200221. CrossRef
Diagnosis of GH Deficiency Without GH Stimulation Tests
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Metabolic Impacts of Discontinuation and Resumption of Recombinant Human Growth Hormone Treatment during the Transition Period in Patients with Childhood-Onset Growth Hormone Deficiency
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A Radiomics-Based Model with the Potential to Differentiate Growth Hormone Deficiency and Idiopathic Short Stature on Sella MRI
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Original Articles

A Clinical Study on Hypopitutiarism: Significance of Combined Pituitary Stimulation Test.: Bo Youn Cho, Hong Gyu Lee, Chang Soon Koh, Hyung Kyu Park, Sook Kyung Kim, Chan Soo Shin, Seong Yeon Kim, Jae Seok Chun, Kyung Soo Park, Hyeon Kyu Kim, Sun Wook Kim; J Korean Endocr Soc. 1996;11(3):268-276. Published online November 7, 2019

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Abstract PDF: Background
Hypopituitarism can be caused by various diseases. Its clinical manifestations vary, depending on the extent and severity of the pituitary hormone deficiency. And some patients may initially present with SIADH-like features. Combined pituitary stimulation test has been used for the diagnosis of hypopituitarism and subsequent hormone replacement therapy. However, the test is laborious, expensive and uncomfortable to the patients, So we performed this study to know whether combined pituitary stimulation test can be replaced with clinical features and simple basal hormone concentrations. Methods: Fifty-four patients who were diagnosed as hypopituitarism by combined pituitary stimulation test were included in this study. Clinical features and basal hormone data were compared with the results of combined pituitary stimulation test for the evaluation of pituitary-gonadal, pituitary-thyroid, and pituitary-adrenal axes, using X2 test. Results: 1) In pituitary-gonadal axis, the evaluation of clinical features and basal gonad hormone concentrations were significantly consistent with stimulation test(p<0.05), 2) In pituitary-thyroid axis, the evaluation of basal thyroid hormone concentrations were more helpful than stimulation test though results of the two tests were not consistent. 3) In pituitary-adrenal axis, all patients whose basal cortisol concentrations were low showed inadequate responses to stimulation test. However, stimulation test revealed adrenal insufficiency in some patients with normal basal cortisol concentrations. 4) 9 patients who presented with SIADH-like features were older than the others and had all corticotropin deficiency. Conclusion: In patients with suspected hypopituitarism, the evaluation of clinical features and basal hormone concentrations can be sufficient for the diagnosis of hypopituitarism and hormone replacement therapy. However, stimulation test is necessary to investigate adrenal function in patients with normal basal cortisol concentrations. And hypopituitarism should be considered in old patients who present with SIADH-like features.

Clinical Characteristics of Patients with Adrenal Insufficiency in a General Hospital: Ye Yeon Lee, Nan Hee Cho, Jong Won Lee, Nam Kyung Kim, Hye Soon Kim, Mi-Kyung Kim; Endocrinol Metab. 2017;32(1):83-89. Published online February 28, 2017; DOI: https://doi.org/10.3803/EnM.2017.32.1.83

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Background

Adrenal insufficiency (AI) is a life-threatening disorder caused by the deficiency of adrenal steroid hormones. This retrospective cross-sectional study investigated the characteristics of patients with AI in Korea.

Methods

All consecutive patients with suspected AI who received care at a tertiary referral center in Korea in 2014 and underwent adrenocorticotropic hormone stimulation or insulin-tolerance testing were identified through a review of medical charts. Patients diagnosed with AI were enrolled. Their demographic, clinical, and treatment details were extracted.

Results

Of 771 patients with suspected AI, 183 (23.7%) received a definitive diagnosis. The most common reason for testing was the presence of suspicious AI-related symptoms (30.0%), followed by a history of steroid medications (23.5%). Their mean age was 66.7 years, and females predominated (67.8%). The most common symptoms were general weakness, anorexia, arthralgia, and fever. Approximately half (53.6%) had a history of steroid use. Hydrocortisone was the most common treatment (71.6%), with most patients taking a 30 mg dose (44.2%). The most common dose frequency was twice a day (78.6%). Fourteen patients were treated for adrenal crisis (n=10, 5.5%) or an intercurrent illness (n=4, 2.2%).

Conclusion

AI may have been caused by steroid medication use in many of the patients included in this study. The detection of AI can be improved by careful history-taking and being alert to the possibility that a patient has used steroids.

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Risk of Mental Illnesses in Patients With Hypopituitarism: A Nationwide Population-Based Cohort Study
I-Hua Wei, Chih-Chia Huang
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Bradley D. Menz, Jacinta L. Johnson, Davina F. Gillard, William Chong, Michael B. Ward
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Clinical and biochemical factors to predict biochemical adrenal insufficiency in hospitalized patients with indeterminate cortisol levels: a retrospective study
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Sunao Matsubayashi, Nobuhiro Nakatake, Takeshi Hara
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Eun-Jung Rhee, Jung Hee Kim, Sun Joon Moon, Won-Young Lee
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Glucocorticoid management of adrenal insufficiency in the United Kingdom: assessment using real-world data
Kamran Iqbal, Kate Halsby, Robert D Murray, Paul V Carroll, Robert Petermann
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Clinical Study
Electrolyte Imbalance in Patients with Sheehan's Syndrome: Chur Hoan Lim, Ji Hyun Han, Joon Jin, Ji Eun Yu, Jin Ook Chung, Dong Hyeok Cho, Dong Jin Chung, Min Young Chung; Endocrinol Metab. 2015;30(4):502-508. Published online December 31, 2015; DOI: https://doi.org/10.3803/EnM.2015.30.4.502

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Background

We investigated the prevalence of electrolyte imbalance and the relationship between serum electrolyte and anterior pituitary hormone levels in patients with Sheehan's syndrome.

Methods

In a retrospective study, we investigated 78 patients with Sheehan's syndrome. We also included 95 normal control subjects who underwent a combined anterior pituitary hormone stimulation test and showed normal hormonal responses.

Results

In patients with Sheehan's syndrome, the serum levels of sodium, potassium, ionized calcium, magnesium, and inorganic phosphate were significantly lower than those in control subjects. The prevalence of hyponatremia, hypokalemia, hypocalcemia, hypomagnesemia, and hypophosphatemia in patients with Sheehan's syndrome was 59.0% (n=46), 26.9% (n=21), 35.9% (n=28), 47.4% (n=37), and 23.1% (n=18), respectively. Levels of sodium and ionized calcium in serum were positively correlated with levels of all anterior pituitary hormones (all P<0.05). Levels of potassium in serum were positively correlated with adrenocorticotrophic hormone (ACTH) and growth hormone (GH) levels (all P<0.05). Levels of inorganic phosphate in serum were positively correlated with levels of thyroid-stimulating hormone, prolactin, and GH (all P<0.05), and levels of magnesium in serum were positively correlated with delta ACTH (P<0.01).

Conclusion

Electrolyte imbalance was common in patients with Sheehan's syndrome. Furthermore, the degree of anterior pituitary hormone deficiency relates to the degree of electrolyte disturbance in patients with this disease.

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Review Article

Adrenal gland
Diagnosis and Treatment of Hypopituitarism: Seong Yeon Kim; Endocrinol Metab. 2015;30(4):443-455. Published online December 31, 2015; DOI: https://doi.org/10.3803/EnM.2015.30.4.443

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Hypopituitarism is a chronic endocrine illness that caused by varied etiologies. Clinical manifestations of hypopituitarism are variable, often insidious in onset and dependent on the degree and severity of hormone deficiency. However, it is associated with increased mortality and morbidity. Therefore, early diagnosis and prompt treatment is necessary. Hypopituitarism can be easily diagnosed by measuring basal pituitary and target hormone levels except growth hormone (GH) and adrenocorticotropic hormone (ACTH) deficiency. Dynamic stimulation tests are indicated in equivocal basal hormone levels and GH/ACTH deficiency. Knowledge of the use and limitations of these stimulation tests is mandatory for proper interpretation. It is necessary for physicians to inform their patients that they may require lifetime treatment. Hormone replacement therapy should be individualized according to the specific needs of each patient, taking into account possible interactions. Long-term endocrinological follow-up of hypopituitary patients is important to monitor hormonal replacement regimes and avoid under- or overtreatment.

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Original Article

Adrenal gland
Clinical Characteristics, Management, and Outcome of 22 Cases of Primary Hypophysitis: Sun Mi Park, Ji Cheol Bae, Ji Young Joung, Yoon Young Cho, Tae Hun Kim, Sang-Man Jin, Sunghwan Suh, Kyu Yeon Hur, Kwang-Won Kim; Endocrinol Metab. 2014;29(4):470-478. Published online December 29, 2014; DOI: https://doi.org/10.3803/EnM.2014.29.4.470

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Background

Primary hypophysitis causes varying degrees of endocrine dysfunction and mass effect. The natural course and best treatment have not been well established.

Methods

Medical records of 22 patients who had been diagnosed with primary hypophysitis between January 2001 and March 2013 were retrospectively reviewed. Based on the anatomical location, we classified the cases as adenohypophysitis (AH), infundibuloneurohypophysitis (INH), and panhypophysitis (PH). Clinical presentation, endocrine function, pathologic findings, magnetic resonance imaging findings, and treatment courses were reviewed.

Results

Among 22 patients with primary hypophysitis, 81.8% (18/22) had involvement of the posterior pituitary lobe. Two patients of the AH (2/3, 66.6%) and three patients of the PH (3/10, 30%) groups initially underwent surgical mass reduction. Five patients, including three of the PH (3/10, 33.3%) group and one from each of the AH (1/3, 33.3%) and INH (1/9, 11.1%) groups, initially received high-dose glucocorticoid treatment. Nearly all of the patients treated with surgery or high-dose steroid treatment (9/11, 82%) required continuous hormone replacement during the follow-up period. Twelve patients received no treatment for mass reduction due to the absence of acute symptoms and signs related to a compressive mass effect. Most of them (11/12, 92%) did not show disease progression, and three patients recovered partially from hormone deficiency.

Conclusion

Deficits of the posterior pituitary were the most common features in our cases of primary hypophysitis. Pituitary endocrine defects responded less favorably to glucocorticoid treatment and surgery. In the absence of symptoms related to mass effect and with the mild defect of endocrine function, it may not require treatment to reduce mass except hormone replacement.

Citations

Citations to this article as recorded by

Secondary xanthogranulomatous hypophysitis mimicking a pituitary macroadenoma: a case report
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Fabienne Langlois, Elena V Varlamov, Maria Fleseriu
The Journal of Clinical Endocrinology & Metabolism.2022; 107(1): 10. CrossRef
Outcomes of Initial Management Strategies in Patients With Autoimmune Lymphocytic Hypophysitis: A Systematic Review and Meta-analysis
Diane Donegan, Zeb Saeed, Danae A Delivanis, Mohammad Hassan Murad, Juergen Honegger, Felix Amereller, Seda Hanife Oguz, Dana Erickson, Irina Bancos
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Early Pulse Glucocorticoid Therapy and Improved Hormonal Outcomes in Primary Hypophysitis
Brijesh Krishnappa, Ravikumar Shah, Vijaya Sarathi, Anurag Ranjan Lila, Manjeet Kaur Sehemby, Virendra A. Patil, Shilpa Sankhe, Nalini Shah, Tushar Bandgar
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Case Reports

Pancytopenia Associated with Hypopituitarism in an Acromegaly Patient: A Case Report and a Review of the Literature.: Jung Hee Koh, Yong Jae Lee, Ji Hyun Kang, Bo Kwang Choi, Yun Kyung Jeon, Sang Soo Kim, Bo Hyun Kim, In Joo Kim; Endocrinol Metab. 2012;27(4):308-313. Published online December 20, 2012; DOI: https://doi.org/10.3803/EnM.2012.27.4.308

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Abstract PDF

We present the case of a patient with acromegaly who had pancytopenia with hypopituitarism secondary to the excision of a pituitary macroadenoma and radiation therapy. A 28-year-old man presented with pancytopenia and serum electrolyte abnormalities. He was diagnosed with acromegaly and underwent surgery and gamma-knife radiotherapy for a pituitary macroadenoma at the age of 22 years. A recent brain magnetic resonance imaging showed an empty sella, and the basal hormonal profile demonstrated deficiencies of pituitary hormones except thyrotropin. As presenting pancytopenia, his bone marrow biopsy showed hypocellular marrow. The total number of hemocytes increased after hydrocortisone replacement. Hypopituitarism was a possible cause of pancytopenia, and glucocorticoids had crucial effects on converting pancytopenia to normal in this case.

Citations

Citations to this article as recorded by

A 24-Year-Old Male with Gigantism, Growth Hormone Deficiency, Suspected Clivus Chordoma, Primary Hypothyroidism, Hypogonadism and Pancytopenia
W.A. Arsana, M.I. Diah Pramudianti
INDONESIAN JOURNAL OF CLINICAL PATHOLOGY AND MEDICAL LABORATORY.2019; 26(2): 249. CrossRef
Hormones and the Bone Marrow: Panhypopituitarism and Pancytopenia in a Man with a Pituitary Adenoma
Dianna Lang, Jennifer S. Mead, David B. Sykes
Journal of General Internal Medicine.2015; 30(5): 692. CrossRef

A Case of Sheehan's Syndrome with Pancytopenia.: Hyun Suk Lee, Byung Woon Kwon, Jin Hyung Han, Hee Jin Kim; Endocrinol Metab. 2012;27(1):54-58. Published online March 1, 2012; DOI: https://doi.org/10.3803/EnM.2012.27.1.54

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23 Download

Abstract PDF: Sheehan's syndrome is characterized by varying degrees of anterior pituitary dysfunction due to postpartum ischemic necrosis of the pituitary gland after massive bleeding. The spectrum of clinical presentation of Sheehan's syndrome is broad, with changes from nonspecific complaints, such as weakness, fatigue, and anemia, to severe pituitary insufficiency resulting in coma and death. Normochromic anemia is commonly associated with Sheehan's syndrome, but pancytopenia is rarely observed in patients with Sheehan's syndrome. We describe a 57-year-old woman with Sheehan's syndrome who presented with pancytopenia that was treated by hormone replacement with levothyroxine and glucocorticoid.

A Case of Pituitary Abscess that was Difficult to Diagnose due to Repeated Symptomatic Responses to Every Corticosteroid Administration.: Jin Sun Jang, Jae Seung Yun, Jung Ah Shin, Min Hee Kim, Dong Jun Lim, Jae Hyung Cho, Kun Ho Yoon, Moo Il Kang, Bong Yun Cha, Ho Young Son, Yong Kil Hong; Endocrinol Metab. 2011;26(1):72-77. Published online March 1, 2011; DOI: https://doi.org/10.3803/EnM.2011.26.1.72

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Abstract PDF: Pituitary abscess is a rare pathology, but it is a potentially life-threatening condition. Therefore, timely intervention, including antibiotics and an operation, can prevent the morbidity and mortality in such cases. A 31-year-old woman, who was 16 months after her second delivery, presented with intermittent headache for 3 months. Amenorrhea, polyuria and polydipsia were noticed and the endocrinological hormone studies were compatible with panhypopituitarism and diabetes insipidus. Pituitary MRI demonstrated a 2.3 cm sized cystic mass with an upper small nodular lesion. Her symptoms such as headache and fever were repeatedly improved whenever corticosteroid was administered, which led us to suspect the diagnosis of an inflammatory condition like lymphocytic hypophysitis. During the hormone replacement therapy, her cystic pituitary mass had grown and her symptoms progressively worsened for another two months. The patient underwent trans-sphenoidal exploration and she turned out to have a pituitary abscess. At the 3-month follow-up, amenorrhea was noticed and her residual function of the pituitary was tested by a combined pituitary stimulation test. The results were compatible with panhypopituitarism. She received levothyroxine 100 microg, prednisolone 5 mg and desmopressin spray and she is being observed at the out-patient clinic. The authors experienced a patient with primary pituitary abscess that was confirmed pathologically and we report on its clinical course with a literature review.

A Case of Ectopic Neurohypophysis Presenting with Hypogonadism.: In Woon Baek, Ji Hyun Kim, Guk Jin Lee, Kyoung Eun Lee, Hae Lim Lee, Hye Won Lee, Nam Yong Kim, Yon Kwon Ihn, Seung Hyun Ko, Seung Hwan Lee, Je Ho Han; Endocrinol Metab. 2011;26(1):67-71. Published online March 1, 2011; DOI: https://doi.org/10.3803/EnM.2011.26.1.67

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Pituitary stalk interruption and ectopic neurohypophysis seen on magnetic resonance Imaging (MRI) are often associated with either isolated growth hormone (GH) deficiency or combined anterior pituitary hormone deficiency, but their pathogenesis is not clear and the clinical data regarding these anatomical defect is limited. We experienced a 23-year-old male with the absence of secondary sexual characteristics and this was accompanied with pituitary stalk dysgenesis and ectopic neurohypophysis. He received growth hormone for a year when he was 12 years old due to his short stature. Sella MRI showed no visible pituitary stalk with minimal high signal change, suggesting ectopic neurohypophysis. The combined pituitary stimulation test revealed blunted responses of growth hormone, follicle stimulating hormone and luteinizing hormone. For the hypogonadotropic hypogonadism, the patient was given testosterone intramuscularly and he gradually developed secondary sexual characteristics. We concluded that the hypogonadism and growth hormone deficiency in this patient was caused by hypopituitarism due to pituitary stalk dysgenesis and ecopic nuerohypophysis.

Citations

Citations to this article as recorded by

MRI of ectopic posterior pituitary gland with dysgenesis of pituitary stalk in a patient with hypogonadotropic hypogonadism
Ashim Kumar Lahiri, Ramanivas Sundareyan, David Jenkins, Anjumara Nilak
Radiology Case Reports.2018; 13(4): 764. CrossRef
Hypothalamic Hypopituitarism Caused by Pituitary Stalk Dysgenesis
Seong-Ju Lee, Hye-Jin Yoon, A-Reum Cho, Yoo-Jin Um, Keun-Young Park, Dong-Mee Lim, Byung-Joon Kim
Korean Journal of Medicine.2013; 85(4): 420. CrossRef

Original Articles

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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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

Correlation between Pituitary Insufficiency and Magnetic Resonance Imaging Finding in Non-Functioning Pituitary Adenomas.: Hyun Min Kim, Cheol Ryong Ku, Eun Young Lee, Woo Kyung Lee, Jung Soo Lim, Sena Hwang, Mi Jung Lee, Seung Ku Lee, Sun Ho Kim, Eun Jig Lee; Endocrinol Metab. 2010;25(4):310-315. Published online December 1, 2010; DOI: https://doi.org/10.3803/EnM.2010.25.4.310

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BACKGROUND
Non-functioning pituitary adenomas (NFPAs) are characterized by the absence of clinical and biochemical evidence of pituitary hormone hypersecretion, and these tumors constitute approximately one third of all the tumors of the anterior pituitary. Recently, hormonal deficiencies have gradually evolved to become the leading presenting signs and symptoms in patients with NFPAs. We investigated pituitary hormonal insufficiencies according to the magnetic resonance imaging (MRI) findings in patients with NFPA. METHODS: We evaluated the patients who were newly diagnosed with NFPA from 1997 through 2009. Among them, we analyzed 387 patients who were tested for their combined pituitary function and who underwent MRI. The severity of the hypopituitarism was determined by the number of deficient axes of the pituitary hormones. On the MRI study, the maximal diameter of the tumor, Hardy's classification, the thickness of the pituitary gland and the presence of stalk compression were evaluated. RESULTS: The mean age was 46.85 +/- 12.93 years (range: 15-86) and 186 patients (48.1%) were male. As assessed on MRI, the tumor diameter was 27.87 +/- 9.93 mm, the thickness of the normal pituitary gland was 1.42 +/- 2.07 mm and stalk compression was observed in 201 patients (51.9%). Hypopituitarism was observed in 333 patients (86.0%). Deficiency for each pituitary hormone was most severe in the patients with Hardy type IIIA. Hypopituitarism was severe in the older age patients (P = 0.001) and the patients with a bigger tumor size (P < 0.001) and the presence of stalk compression (P < 0.001). However, the patients who had a thicker pituitary gland showed less severe hypopituitarism (P < 0.001). Multivariate analysis showed that age, tumor diameter and the thickness of pituitary gland were important determinants for pituitary deficiency (P = 0.004, P < 0.001, P = 0.022, respectively). CONCLUSION: The results suggest that the hormonal deficiencies in patient with NFPA were correlated with the MRI findings, and especially the tumor diameter and preservation of the pituitary gland.

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Clinical Parameters to Distinguish Silent Corticotroph Adenomas from Other Nonfunctioning Pituitary Adenomas
Daham Kim, Cheol Ryong Ku, Se Hee Park, Ju Hyung Moon, Eui Hyun Kim, Sun Ho Kim, Eun Jig Lee
World Neurosurgery.2018; 115: e464. CrossRef

Case Report

A Case of Insulin Autoimmune Syndrome in a Patient with Partial Hypopituitarism.: Obin Kwon, Eun Young Park, Jin Young Yoon, Kwang Joon Kim, Yong ho Lee, Jae Won Hong, Eun Jig Lee, Sung Kil Lim, Hyun Chul Lee, Bong Soo Cha; J Korean Endocr Soc. 2009;24(4):281-286. Published online December 1, 2009; DOI: https://doi.org/10.3803/jkes.2009.24.4.281

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Abstract PDF: Insulin autoimmune syndrome is one of the rare causes of hypoglycemia, and characterized by hyperinsulinemic hypoglycemia associated with high titer of antibodies to endogenous insulin. We report a case of insulin autoimmune syndrome in a 57-year-old woman, presenting with mental changes due to hypoglycemia. She had no history of diabetes or insulin administration. The serum C-peptide level was 4.69 ng/mL and the insulin concentration was 229.55 microU/mL, when fasting plasma glucose level was 32 mg/dL. The insulin-to-glucose ratio was 7.17, while there was no radiologic evidence of insulinoma. The insulin antibody level was over 100 microU/mL, resulting in the diagnosis of insulin autoimmune syndrome. Hormonal studies revealed partial hypopituitarism and a lack of glucagon-response to hypoglycemia. Hypoglycemia disappeared with replacement of prednisolone with levothyroxine therapy. Under secretion of growth hormone and of adrenocorticotropic hormone due to hypopituitarism were associated with insufficient counterregulation to hypoglycemia. One should keep in mind that insulin autoimmune syndrome or hypopituitarism is one cause of hypoglycemia in patients with no history of diabetes, and corticosteroid can be an effective treatment for both diseases.

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