Endocrinology and Metabolism

Original Articles

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
Salma Salhi, Ibtissem Oueslati, Yasmine Mouelhi, Alia Zehani, Nidhameddine Kchir, Elyes Kamoun, Meriem Yazidi, Melika Chihaoui
Journal of International Medical Research.2024;[Epub] CrossRef
Glucocorticoid therapy as first-line treatment in primary hypophysitis: a systematic review and individual patient data meta-analysis
Brijesh Krishnappa, Ravikumar Shah, Saba Samad Memon, Chakra Diwaker, Anurag R Lila, Virendra A Patil, Nalini S Shah, Tushar R Bandgar
Endocrine Connections.2023;[Epub] CrossRef
Hypophysitis, the Growing Spectrum of a Rare Pituitary Disease
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
The Journal of Clinical Endocrinology & Metabolism.2022; 107(4): 1170. CrossRef
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
Neuroendocrinology.2022; 112(2): 186. CrossRef
Preoperative differentiation of hypophysitis and pituitary adenomas using a novel clinicoradiologic scoring system
Kyla Wright, Hyon Kim, Travis Hill, Matthew Lee, Cordelia Orillac, Nikita Mogar, Donato Pacione, Nidhi Agrawal
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Hypophysitis
Diane Donegan, Jürgen Honegger
Endocrine Practice.2022; 28(9): 901. CrossRef
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David P. Bray, C. Arturo Solares, Nelson M. Oyesiku
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Diabetes insipidus secondary to sellar/parasellar lesions
Anna Angelousi, Chrysoula Mytareli, Paraskevi Xekouki, Eva Kassi, Konstantinos Barkas, Ashley Grossman, Gregory Kaltsas
Journal of Neuroendocrinology.2021;[Epub] CrossRef
Clinical presentation and management of hypophysitis: An observational study of case series
Marouan Karrou, Salma Benyakhlef, Achwak Alla, Najoua Messaoudi, Asmae Oulad Amar, Siham Rouf, Imane Kamaoui, Noureddine Oulali, Faycal Moufid, Naima Abda, Hanane Latrech
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Clinical aspects of autoimmune hypothalamitis, a variant of autoimmune hypophysitis: Experience from one center
Qian Wei, Guoqing Yang, Zhaohui Lue, Jingtao Dou, Li Zang, Yijun Li, Jin Du, Weijun Gu, Yiming Mu
Journal of International Medical Research.2020; 48(3): 030006051988783. CrossRef
Primary and Ipilimumab-induced Hypophysitis: A Single-center Case Series
Paul Atkins, Ehud Ur
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Hypophysitis (Including IgG4 and Immunotherapy)
Anna Angelousi, Krystallenia Alexandraki, Marina Tsoli, Gregory Kaltsas, Eva Kassi
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Hypophysitis in the era of immune checkpoint inhibitors and immunoglobulin G4-related disease
Leen Wehbeh, Sama Alreddawi, Roberto Salvatori
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Immune check point inhibitors-induced hypophysitis: a retrospective analysis of the French Pharmacovigilance database
Julie Garon-Czmil, Nadine Petitpain, Franck Rouby, Marion Sassier, Samy Babai, Mélissa Yéléhé-Okouma, Georges Weryha, Marc Klein, Pierre Gillet
Scientific Reports.2019;[Epub] CrossRef
Clinical Features, Magnetic Resonance Imaging, and Treatment Experience of 20 Patients with Lymphocytic Hypophysitis in a Single Center
Qiang Zhu, Ke Qian, Guijun Jia, Gang Lv, Jisheng Wang, Liyong Zhong, Shuqing Yu
World Neurosurgery.2019; 127: e22. CrossRef
Idiopathic granulomatous hypophysitis presenting with galactorrhea, headache, and nausea in a woman: a case report and review of the literature
Guive Sharifi, Mohammad Reza Mohajeri-Tehrani, Behrouz Navabakhsh, Bagher Larijani, Touraj Valeh
Journal of Medical Case Reports.2019;[Epub] CrossRef
Paciente de 31 años con polidipsia
A.R. Benavides Aramburu, M. Seguí Díaz
Medicina de Familia. SEMERGEN.2018; 44(2): e77. CrossRef
Primary hypophysitis and other autoimmune disorders of the sellar and suprasellar regions
Sriram Gubbi, Fady Hannah-Shmouni, Constantine A. Stratakis, Christian A. Koch
Reviews in Endocrine and Metabolic Disorders.2018; 19(4): 335. CrossRef
Primary lymphocytic hypophysitis: Clinical characteristics and treatment of 50 cases in a single centre in China over 18 years
Shuchang Wang, Linjie Wang, Yong Yao, Feng Feng, Hongbo Yang, Zhiyong Liang, Kan Deng, Hui You, Jian Sun, Bing Xing, Zimeng Jin, Renzhi Wang, Hui Pan, Huijuan Zhu
Clinical Endocrinology.2017; 87(2): 177. CrossRef
Clinical presentation and outcome of children with central diabetes insipidus associated with a self‐limited or transient pituitary stalk thickening, diagnosed as infundibuloneurohypophysitis
J. Schaefers, M. Cools, K. De Waele, I. Gies, V. Beauloye, P. Lysy, I. Francois, D. Beckers, J. De Schepper
Clinical Endocrinology.2017; 87(2): 171. CrossRef
Intrachiasmatic abscess caused by IgG4-related hypophysitis
Georgios F. Hadjigeorgiou, Eva Løbner Lund, Lars Poulsgaard, Ulla Feldt-Rasmussen, Åse Krogh Rasmussen, Marianne Wegener, Kåre Fugleholm
Acta Neurochirurgica.2017; 159(11): 2229. CrossRef
Granulomatous and lymphocytic hypophysitis – are they immunologically distinct?
Shilpa Rao, Anita Mahadevan, Tanmoy Maiti, Manish Ranjan, Shivayogi Durgad Shwetha, Arimappamagan Arivazhagan, Jitender Saini
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Articles in 'Endocrinology and Metabolism' in 2014
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Adrenal gland
Using Growth Hormone Levels to Detect Macroadenoma in Patients with Acromegaly: Ji Young Park, Jae Hyeon Kim, Sun Wook Kim, Jae Hoon Chung, Yong-Ki Min, Myung-Shik Lee, Moon-Kyu Lee, Kwang-Won Kim; Endocrinol Metab. 2014;29(4):450-456. Published online December 29, 2014; DOI: https://doi.org/10.3803/EnM.2014.29.4.450

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Abstract PDF PubReader

Background

The aim of this study was to assess the clinical differences between acromegalic patients with microadenoma and patients with macroadenoma, and to evaluate the predictive value of growth hormone (GH) levels for early detection of macroadenoma.

Methods

We performed a retrospective analysis of 215 patients diagnosed with a GH-secreting pituitary adenoma. The patients were divided into two groups: the microadenoma group and the macroadenoma group, and the clinical parameters were compared between these two groups. The most sensitive and specific GH values for predicting macroadenoma were selected using receiver operating characteristic (ROC) curves.

Results

Compared with the microadenoma group, the macroadenoma group had a significantly younger age, higher body mass index, higher prevalence of hyperprolactinemia and hypogonadism, and a lower proportion of positive suppression to octreotide. However, there were no significant differences in the gender or in the prevalence of diabetes between the two groups. The tumor diameter was positively correlated with all GH values during the oral glucose tolerance test (OGTT). All GH values were significantly higher in the macroadenoma group than the microadenoma group. Cut-off values for GH levels at 0, 30, 60, 90, and 120 minutes for optimal discrimination between macroadenoma and microadenoma were 5.6, 5.7, 6.3, 6.0, and 5.8 ng/mL, respectively. ROC curve analysis revealed that the GH value at 30 minutes had the highest area under the curve.

Conclusion

The GH level of 5.7 ng/mL or higher at 30 minutes during OGTT could provide sufficient information to detect macroadenoma at the time of diagnosis.

Citations

Citations to this article as recorded by

Sex differences in acromegaly at diagnosis: A nationwide cohort study and meta‐analysis of the literature
Jakob Dal, Benedikte G. Skov, Marianne Andersen, Ulla Feldt‐Rasmussen, Claus L. Feltoft, Jesper Karmisholt, Eigil H. Nielsen, Olaf M. Dekkers, Jens Otto L. Jørgensen
Clinical Endocrinology.2021; 94(4): 625. CrossRef
Pretreatment serum GH levels and cardio-metabolic comorbidities in acromegaly; analysis of data from Iran Pituitary Tumor Registry
Leila Hedayati Zafarghandi, Mohammad Ebrahim Khamseh, Milad Fooladgar, Shahrzad Mohseni, Mostafa Qorbani, Nahid Hashemi Madani, Mahboobeh Hemmatabadi, MohammadReza Mohajeri-Tehrani, Nooshin Shirzad
Journal of Diabetes & Metabolic Disorders.2020; 19(1): 319. CrossRef
Increased serum nesfatin-1 levels in patients with acromegaly
Yakun Yang, Song Han, Zuocheng Yang, Pengfei Wang, Chang-Xiang Yan, Ning Liu
Medicine.2020; 99(40): e22432. CrossRef
Articles in 'Endocrinology and Metabolism' in 2014
Won-Young Lee
Endocrinology and Metabolism.2015; 30(1): 47. CrossRef

Adrenal gland
Genetic Analysis of Multiple Endocrine Neoplasia Type 1 (MEN1) Leads to Misdiagnosis of an Extremely Rare Presentation of Intrasellar Cavernous Hemangioma as MEN1: Dong Min Lee, Seung Hee Yu, Hyun Hwa Yoon, Kang Lock Lee, Young Sil Eom, Kiyoung Lee, Byung-Joon Kim, Yeun Sun Kim, Ie Byung Park, Kwang-Won Kim, Sihoon Lee; Endocrinol Metab. 2014;29(2):146-153. Published online June 26, 2014; DOI: https://doi.org/10.3803/EnM.2014.29.2.146

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Background

Multiple endocrine neoplasia type 1 (MEN1) is a rare inherited disorder characterized by the simultaneous occurrence of endocrine tumors in target tissues (mainly the pituitary, endocrine pancreas, and parathyroid glands). MEN1 is caused by mutations in the MEN1 gene, which functions as a tumor suppressor and consists of one untranslated exon and nine exons encoding the menin protein. This condition is usually suspected when we encounter patients diagnosed with tumors in multiple endocrine organs, as mentioned above.

Methods

A 65-year-old woman who underwent surgery for a pancreatic tumor (serous cystadenoma) 5 years previously was referred to our hospital due to neurologic symptoms of diplopia and left ptosis. Brain magnetic resonance imaging revealed a 3.4-cm lesion originating from the cavernous sinus wall and extending into the sellar region. It was thought to be a nonfunctioning tumor from the results of the combined pituitary function test. Incidentally, we found that she also had a pancreatic tumor, indicating the necessity of genetic analysis for MEN1.

Results

Genomic analysis using peripheral leukocytes revealed a heterozygous c.1621G>A mutation in the MEN1 gene that was previously reported to be either a pathogenic mutation or a simple polymorphism. We pursued a stereotactic approach to the pituitary lesion, and microscopic findings of the tumor revealed it to be an intrasellar cavernous hemangioma, a rare finding in the sellar region and even rarer in relation to oculomotor palsy. The patient recovered well from surgery, but refused further evaluation for the pancreatic lesion.

Conclusion

There is great emphasis placed on genetic testing in the diagnosis of MEN1, but herein we report a case where it did not assist in diagnosis, hence, further discussion on the role of genetic testing in this disease is needed. Also, in cases of pituitary tumor with cranial nerve palsy, despite its low prevalence, intrasellar cavernous hemangioma could be suspected.

Citations

Citations to this article as recorded by

Diffuse cavernous hemangioma of the skull misdiagnosed as skull metastasis in breast cancer patient: one case report and literature review
Huizhi Liu, Xiaojing Chang, Hua Shang, Feng Li, Huandi Zhou, Xiaoying Xue
BMC Cancer.2019;[Epub] CrossRef
Articles in 'Endocrinology and Metabolism' in 2014
Won-Young Lee
Endocrinology and Metabolism.2015; 30(1): 47. CrossRef

Case Report

Thyroid
Steroid Responsive Xanthomatous Hypophysitis Associated with Autoimmune Thyroiditis: A Case Report: Ji Young Joung, Hyemin Jeong, Yoon Young Cho, Kyoungmin Huh, Yeon-Lim Suh, Kwang-Won Kim, Ji Cheol Bae; Endocrinol Metab. 2013;28(1):65-69. Published online March 25, 2013; DOI: https://doi.org/10.3803/EnM.2013.28.1.65

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Abstract PDF PubReader

We report the case of a 36-year-old woman who presented with headache, fever, and amenorrhea. Laboratory analysis revealed hypopituitarism and autoimmune thyroiditis, while a cerebrospinal fluid study suggested concurrent aseptic meningitis. A magnetic resonance image (MRI) scan revealed a 1.0×0.9 cm cystic mass enlarging the sella turcica. Surgical resection via an endoscopic transsphenoidal route was performed. The histological finding of the excised tissue revealed foamy histiocytes with vacuolated cytoplasm, supporting the diagnosis of xanthomatous hypophysitis. Although a residual soft lesion was observed on the MRI image postoperatively, the patient's headache and fever improved. Ten months after surgery, the patient complained of visual impairment and headache, and the residual mass had enlarged into the suprasellar area. High dose (500 mg intravenous) methylprednisolone was administered for 3 days. During the methylprednisolone pulse therapy, the patient's visual acuity and headache improved. A follow-up MRI taken after methylprednisolone therapy showed a marked mass reduction. Our case supports an autoimmune pathophysiology for xanthomatous hypophysitis and suggests that high dose glucocorticoid therapy as a treatment option.

Citations

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Xanthomatous hypophysitis causing hypogonadotropic hypogonadism resulting in delayed presentation of slipped capital femoral epiphysis
Kirit Singh, Avinash Kumar Kanodia, Peter Ross, Antonia Torgersen, Jamie Maclean, Graham Leese, Kismet Hossain-Ibrahim
British Journal of Neurosurgery.2022; 36(2): 286. CrossRef
Full recovery from chronic headache and hypopituitarism caused by lymphocytic hypophysitis: A case report
Mao-Guang Yang, Han-Qing Cai, Si-Si Wang, Lin Liu, Chun-Mei Wang
World Journal of Clinical Cases.2022; 10(3): 1041. CrossRef
Xanthomatous Hypophysitis Secondary to a Ruptured Rathke’s Cleft Cyst: A Case Report
Emre Gezer, Burak Çabuk, Büşra Yaprak Bayrak, Zeynep Cantürk, Berrin Çetinarslan, Alev Selek, Mehmet Sözen, Damla Köksalan, Savaş Ceylan
Brain Tumor Research and Treatment.2022; 10(1): 48. CrossRef
Successful immunomodulatory treatment for recurrent xanthogranulomatous hypophysitis in an adolescent: illustrative case
Sarah DeCou, Pablo F. Recinos, Richard A. Prayson, Christopher Karakasis, Anzar Haider, Neha Patel
Journal of Neurosurgery: Case Lessons.2022;[Epub] CrossRef
Xanthogranulomatous hypophysitis: A rare presentation in a young female patient
Mohammad Ali Yaghoubi, Samira Zabihyan, Amin Saeidinia, Masoumeh Gharib, Ramin Ghiyasi Moghaddam
Clinical Case Reports.2022;[Epub] CrossRef
Xanthomatous Hypophysitis Presenting in an Adolescent Girl: A Long-Term Follow-Up of a Rare Case and Review of the Literature
Jeanne Sze Lyn Wong, Azraai Bahari Nasruddin, Nalini M. Selveindran, Kartikasalwah Abd Latif, Fauziah Kassim, Sukanya Banerjee Nair, Janet Y.H. Hong
AACE Clinical Case Reports.2021; 7(3): 220. CrossRef
Xanthomatous Hypophysitis: A Case Report and Comprehensive Literature Review
Jianyu Zhu, Zhicheng Wang, Wenze Wang, Jinghua Fan, Yi Zhang, Xiaoxu Li, Jie Liu, Shenzhong Jiang, Kan Deng, Lian Duan, Yong Yao, Huijuan Zhu
Frontiers in Endocrinology.2021;[Epub] CrossRef
Recurring Primary Xanthomatous Hypophysitis Behaving Like Pituitary Adenoma: Additional Case and Literature Review
Mansour Mathkour, Tyler Zeoli, Cassidy Werner, Tyler Scullen, Juanita Garces, Joseph Keen, Marcus Ware
World Neurosurgery.2020; 138: 27. CrossRef
Imaging findings in hypophysitis: a review
Ferdinando Caranci, Giuseppe Leone, Andrea Ponsiglione, Massimo Muto, Fabio Tortora, Mario Muto, Sossio Cirillo, Luca Brunese, Alfonso Cerase
La radiologia medica.2020; 125(3): 319. CrossRef
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Expert Review of Endocrinology & Metabolism.2019; 14(3): 167. CrossRef
Xanthomatous Hypophysitis Is Associated with Ruptured Rathke’s Cleft Cyst
Kai Duan, Sylvia L. Asa, Daniel Winer, Zadeh Gelareh, Fred Gentili, Ozgur Mete
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