Endocrinology and Metabolism

Original Articles

Clinical Study
Characteristics of Korean Patients with Antithyroid Drug-Induced Agranulocytosis: A Multicenter Study in Korea: Hee Kyung Kim, Jee Hee Yoon, Min Ji Jeon, Tae Yong Kim, Young Kee Shong, Min Jin Lee, Bo Hyun Kim, In Joo Kim, Ji Young Joung, Sun Wook Kim, Jae Hoon Chung, Ho-Cheol Kang; Endocrinol Metab. 2015;30(4):475-480. Published online December 31, 2015; DOI: https://doi.org/10.3803/EnM.2015.30.4.475

4,225 View
59 Download
16 Web of Science
16 Crossref

Background

Antithyroid drugs (ATDs) can lead to the development of agranulocytosis, which is the most serious adverse effect. Characteristics of ATD-induced agranulocytosis (AIA) have seldom been reported due to the rarity. In this study, we characterized the clinical features for AIA in Korean patients.

Methods

We retrospectively reviewed data from patients with AIA diagnosed between 1997 and 2014 at four tertiary hospitals. Agranulocytosis was defined as an absolute neutrophil count (ANC) below 500/mm³.

Results

The mean age of the patients (11 males, 43 females) was 38.2±14.9 years. Forty-eight patients (88.9%) with AIA had fever and sore throat on initial presentation, 20.4% of patients developed AIA during the second course of treatment, and 75.9% of patients suffered AIA within 3 months after initiation of ATD. The patients taking methimazole (n=39) showed lower levels of ANC and more frequent use of granulocyte-macrophage colony-stimulating factor than propylthiouracil (n=15) users. The median duration of agranulocytosis was 5.5 days (range, 1 to 20). No differences were observed between the long (≥6 days) and short recovery time (≤5 days) groups in terms of age, gender, ATDs, duration of ATDs, or initial ANC levels. Four patients (7.4%) who were taking ATDs for less than 2 months died of sepsis on the first or second day of hospitalization.

Conclusion

The majority of AIA incidents occur in the early treatment period. Considering the high fatality rate of AIA, an early aggressive therapeutic approach is critical and patients should be well informed regarding the warning symptoms of the disease.

Citations

Citations to this article as recorded by

Novel Association of KLRC4-KLRK1 Gene Polymorphisms with Susceptibility and Progression of Antithyroid Drug-Induced Agranulocytosis
Yayi He, Pan Ma, Yuanlin Luo, Xiaojuan Gong, Jiayang Gao, Yuxin Sun, Pu Chen, Suliang Zhang, Yuxin Tian, Bingyin Shi, Bao Zhang
Experimental and Clinical Endocrinology & Diabetes.2024; 132(01): 17. CrossRef
A Disproportionality Analysis of the Adverse Effect Profiles of Methimazole and Propylthiouracil in Patients with Hyperthyroidism Using the Japanese Adverse Drug Event Report Database
Masanori Arai, Takahiro Tsuno, Hiromi Konishi, Kuniyuki Nishiyama, Yasuo Terauchi, Ryota Inoue, Jun Shirakawa
Thyroid®.2023; 33(7): 804. CrossRef
The Current Status of Hyperthyroidism in Korea
Hyemi Kwon
Endocrinology and Metabolism.2023; 38(4): 392. CrossRef
Clinical characteristics of neutropenic patients under antithyroid drug: Twelve-year experience in a medical center
Chih-Hsueh Tseng, Chi-Lung Tseng, Harn-Shen Chen, Pei-Lung Chen, Chun-Jui Huang
Journal of the Chinese Medical Association.2023; 86(9): 826. CrossRef
Association of MICA gene polymorphisms with thionamide-induced agranulocytosis
P. Ma, P. Chen, J. Gao, H. Guo, S. Li, J. Yang, J. Lai, X. Yang, B. Zhang, Y. He
Journal of Endocrinological Investigation.2021; 44(2): 363. CrossRef
Efficacy and adverse events related to the initial dose of methimazole in children and adolescents with Graves’ disease
Hyun Gyung Lee, Eun Mi Yang, Chan Jong Kim
Annals of Pediatric Endocrinology & Metabolism.2021; 26(3): 199. CrossRef
MICA polymorphisms associated with antithyroid drug‐induced agranulocytosis in the Chinese Han population
Xiaojuan Gong, Pu Chen, Pan Ma, Jiayang Gao, Jingsi Yang, Hui Guo, Chunxia Yan, Bao Zhang, Yayi He
Immunity, Inflammation and Disease.2020; 8(4): 695. CrossRef
The Management of Thyroid Disease in COVID-19 Pandemic
Won Sang Yoo, Hyun-Kyung Chung
International Journal of Thyroidology.2020; 13(2): 65. CrossRef
Increased Risk of Antithyroid Drug Agranulocytosis Associated with Amiodarone-Induced Thyrotoxicosis: A Population-Based Cohort Study
Michal Gershinsky, Walid Saliba, Idit Lavi, Chen Shapira, Naomi Gronich
Thyroid.2019; 29(2): 193. CrossRef
A Case of Acute Supraglottitis Following Anti-Thyroid Drug-Induced Agranulocytosis
Jung Jun Lee, Dong Young Kim, Jeon Yeob Jang
Journal of The Korean Society of Laryngology, Phoniatrics and Logopedics.2019; 30(2): 128. CrossRef
Association of HLA-B∗38:02 with Antithyroid Drug-Induced Agranulocytosis in Kinh Vietnamese Patients
Mai Phuong Thao, Pham Vo Anh Tuan, Le Gia Hoang Linh, Lam Van Hoang, Phan Huu Hen, Le Tuyet Hoa, Hoang Anh Vu, Do Duc Minh
International Journal of Endocrinology.2018; 2018: 1. CrossRef
Severe Gingival Ulceration and Necrosis Caused by an Antithyroid Drug: One Case Report and Proposed Clinical Approach
Ying‐Ying Chang, Chih‐Wen Tseng, Kuo Yuan
Clinical Advances in Periodontics.2018; 8(1): 11. CrossRef
Emphasis on the early diagnosis of antithyroid drug-induced agranulocytosis: retrospective analysis over 16 years at one Chinese center
Y. He, J. Li, J. Zheng, Z. Khan, W. Qiang, F. Gao, Y. Zhao, B. Shi
Journal of Endocrinological Investigation.2017; 40(7): 733. CrossRef
Association of HLA-B and HLA-DRB1 polymorphisms with antithyroid drug-induced agranulocytosis in a Han population from northern China
Yayi He, Jie Zheng, Qian Zhang, Peng Hou, Feng Zhu, Jian Yang, Wenhao Li, Pu Chen, Shu Liu, Bao Zhang, Bingyin Shi
Scientific Reports.2017;[Epub] CrossRef
Use of granulocyte colony‐stimulating factor in the treatment of methimazole‐induced agranulocytosis: a case report
Asha Birmingham, Carissa Mancuso, Craig Williams
Clinical Case Reports.2017; 5(10): 1701. CrossRef
2016 American Thyroid Association Guidelines for Diagnosis and Management of Hyperthyroidism and Other Causes of Thyrotoxicosis
Douglas S. Ross, Henry B. Burch, David S. Cooper, M. Carol Greenlee, Peter Laurberg, Ana Luiza Maia, Scott A. Rivkees, Mary Samuels, Julie Ann Sosa, Marius N. Stan, Martin A. Walter
Thyroid.2016; 26(10): 1343. CrossRef

Thyroid
Weight Changes in Patients with Differentiated Thyroid Carcinoma during Postoperative Long-Term Follow-up under Thyroid Stimulating Hormone Suppression: Seo Young Sohn, Ji Young Joung, Yoon Young Cho, Sun Mi Park, Sang Man Jin, Jae Hoon Chung, Sun Wook Kim; Endocrinol Metab. 2015;30(3):343-351. Published online August 4, 2015; DOI: https://doi.org/10.3803/EnM.2015.30.3.343

4,092 View
60 Download
10 Web of Science
14 Crossref

Abstract PDF PubReader

Background

There are limited data about whether patients who receive initial treatment for differentiated thyroid cancer (DTC) gain or lose weight during long-term follow-up under thyroid stimulating hormone (TSH) suppression. This study was aimed to evaluate whether DTC patients under TSH suppression experience long-term weight gain after initial treatment. We also examined the impact of the radioactive iodine ablation therapy (RAIT) preparation method on changes of weight, comparing thyroid hormone withdrawal (THW) and recombinant human TSH (rhTSH).

Methods

We retrospectively reviewed 700 DTC patients who underwent a total thyroidectomy followed by either RAIT and levothyroxine (T4) replacement or T4 replacement alone. The control group included 350 age-matched patients with benign thyroid nodules followed during same period. Anthropometric data were measured at baseline, 1 to 2 years, and 3 to 4 years after thyroidectomy. Comparisons were made between weight and body mass index (BMI) at baseline and follow-up.

Results

Significant gains in weight and BMI were observed 3 to 4 years after initial treatment for female DTC but not in male patients. These gains among female DTC patients were also significant compared to age-matched control. Women in the THW group gained a significant amount of weight and BMI compared to baseline, while there was no increase in weight or BMI in the rhTSH group. There were no changes in weight and BMI in men according to RAIT preparation methods.

Conclusion

Female DTC patients showed significant gains in weight and BMI during long-term follow-up after initial treatment. These changes were seen only in patients who underwent THW for RAIT.

Citations

Citations to this article as recorded by

Impact of a mobile health intervention based on multi-theory model of health behavior change on self-management in patients with differentiated thyroid cancer: protocol for a randomized controlled trial
Yang Jiang, Xiangju Sun, Maomin Jiang, Hewei Min, Jing Wang, Xinghua Fu, Jiale Qi, Zhenjie Yu, Xiaomei Zhu, Yibo Wu
Frontiers in Public Health.2024;[Epub] CrossRef
Thyroidectomy Effects on the Body Mass Index and Thyroid-Stimulating Hormone: A Systematic Review and Meta-Analysis
Hyder Mirghani, Ahmad M Fnjan, Abdullah F Almalki, Ali F Almadan, Omar Abdullah M Alammar, Abdulaziz S Alhwiati, Amer A Laradhi, Ahmed M Bakour, Mohamad A Aljahed, Abdulmajeed M Alzahrani
Cureus.2024;[Epub] CrossRef
Pre-surgery dietician counseling can prevent post-thyroidectomy body weight gain: results of an intervention trial
Laura Croce, Cristina Pallavicini, Noemi Busca, Benedetto Calì, Giuseppe Bellastella, Francesca Coperchini, Flavia Magri, Luca Chiovato, Hellas Cena, Mario Rotondi
Endocrine.2023; 81(2): 246. CrossRef
Determinants and mediating mechanisms of quality of life and disease-specific symptoms among thyroid cancer patients: the design of the WaTCh study
Floortje Mols, Dounya Schoormans, Romana Netea-Maier, Olga Husson, Sandra Beijer, Katrijn Van Deun, Wouter Zandee, Marleen Kars, Pleun C. M. Wouters van Poppel, Suat Simsek, Patrick van Battum, Jérôme M. H. Kisters, Jan Paul de Boer, Elske Massolt, Rachel
Thyroid Research.2023;[Epub] CrossRef
Effects of a low-iodine diet in post-thyroidectomy thyroid cancer patients undergoing I131 therapy at the Vietnam National Cancer Hospital
Bach Viet Hoang, Tien Thi Hong Nguyen, Yen Thi Duong, Hoa Thi Thanh Nguyen, Thu Ha Nguyen, Thanh Thi Nguyen, Lieu Thi Thu Nguyen, Huong Thi Le
Nutrition and Health.2023;[Epub] CrossRef
Positive effects of thyroid replacement therapy on assisted reproductive technology outcomes in women with subclinical hypothyroidism with positive thyroid peroxidase autoantibodies
Himanshu Arora, Ineabelle Collazo, Katherine L. Palmerola, Madhumita Parmar, Manish Narasimman, Nicholas Hendon, Juergen Eisermann, Maria Bustillo
F&S Reports.2022; 3(1): 32. CrossRef
Weight Gain After Thyroidectomy: A Systematic Review and Meta-Analysis
Christine N Huynh, Janina V Pearce, Le Kang, Francesco S Celi
The Journal of Clinical Endocrinology & Metabolism.2021; 106(1): 282. CrossRef
Weight change in patients with differentiated thyroid carcinoma after total thyroidectomy versus lobectomy
Hae-Ryong Cho, Ra-Yeong Song, Kyung Ho Kang
Korean Journal of Clinical Oncology.2020; 16(2): 127. CrossRef
Postthyroidectomy obesity in a Korean population: does the extent of surgery matter?
Min-Young Park, Sang Eun Nam, Kyoung Sik Park, Madhuri Saindane, Young-Bum Yoo, Jung-Hyun Yang, Ah-Leum Ahn, Jae-Kyung Choi, Won Seo Park
Annals of Surgical Treatment and Research.2019; 97(3): 119. CrossRef
Body weight change is unpredictable after total thyroidectomy
Ron Glick, Paula Chang, Peter Michail, Jonathan W. Serpell, Simon Grodski, James C. Lee
ANZ Journal of Surgery.2018; 88(3): 162. CrossRef
Weight Changes After Thyroid Surgery for Patients with Benign Thyroid Nodules and Thyroid Cancer: Population-Based Study and Systematic Review and Meta-Analysis
Naykky Singh Ospina, Ana Castaneda-Guarderas, Oksana Hamidi, Oscar J. Ponce, Wang Zhen, Larry Prokop, Victor M. Montori, Juan P. Brito
Thyroid.2018; 28(5): 639. CrossRef
Does the Risk of Metabolic Syndrome Increase in Thyroid Cancer Survivors?
Min-Hee Kim, Jin-young Huh, Dong-jun Lim, Moo-Il Kang
Thyroid.2017; 27(7): 936. CrossRef
Thyroid hormone and its metabolites in relation to quality of life in patients treated for differentiated thyroid cancer
E.T. Massolt, M. van der Windt, T.I.M. Korevaar, B.L.R. Kam, J.W. Burger, G.J.H. Franssen, I. Lehmphul, J. Köhrle, W.E. Visser, R.P. Peeters
Clinical Endocrinology.2016; 85(5): 781. CrossRef
High Serum Levels of Thyroid-Stimulating Hormone and Sustained Weight Gain in Patients with Thyroid Cancer Undergoing Radioiodine Therapy
Hyo Jung Seo, June-Key Chung, Keon Wook Kang, E. Edmund Kim, Gi Jeong Cheon, Jin Chul Paeng, Dong Soo Lee, Young Joo Park, Do Joon Park, Jae Gol Choe
International Journal of Thyroidology.2016; 9(1): 19. CrossRef

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

4,248 View
41 Download
26 Web of Science
24 Crossref

Abstract PDF PubReader

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
Pituitary.2022; 25(4): 602. CrossRef
Hypophysitis
Diane Donegan, Jürgen Honegger
Endocrine Practice.2022; 28(9): 901. CrossRef
Rare Case of a Disappearing Pituitary Adenoma During the Coronavirus Disease 2019 (COVID-19) Pandemic
David P. Bray, C. Arturo Solares, Nelson M. Oyesiku
World Neurosurgery.2021; 146: 148. CrossRef
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
Surgical Neurology International.2021; 12: 304. CrossRef
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
Endocrine Research.2020; 45(4): 246. CrossRef
Hypophysitis (Including IgG4 and Immunotherapy)
Anna Angelousi, Krystallenia Alexandraki, Marina Tsoli, Gregory Kaltsas, Eva Kassi
Neuroendocrinology.2020; 110(9-10): 822. CrossRef
Hypophysitis in the era of immune checkpoint inhibitors and immunoglobulin G4-related disease
Leen Wehbeh, Sama Alreddawi, Roberto Salvatori
Expert Review of Endocrinology & Metabolism.2019; 14(3): 167. CrossRef
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
APMIS.2016; 124(12): 1072. 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

3,484 View
30 Download
16 Crossref

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

Citations to this article as recorded by

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
Hypophysitis in the era of immune checkpoint inhibitors and immunoglobulin G4-related disease
Leen Wehbeh, Sama Alreddawi, Roberto Salvatori
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
Endocrine Pathology.2017; 28(1): 83. CrossRef
Xanthomatous Hypophysitis Presenting with Diabetes Insipidus Completely Cured Through Transsphenoidal Surgery: Case Report and Literature Review
Wei Lin, Lu Gao, Xiaopeng Guo, Wenze Wang, Bing Xing
World Neurosurgery.2017; 104: 1051.e7. CrossRef
Review of xanthomatous lesions of the sella
B.K. Kleinschmidt‐DeMasters, Kevin O. Lillehei, Todd C. Hankinson
Brain Pathology.2017; 27(3): 377. CrossRef
Xanthomatous hypophysitis
Bishoy Hanna, Yan M. Li, Timothy Beutler, Parul Goyal, Walter A. Hall
Journal of Clinical Neuroscience.2015; 22(7): 1091. CrossRef
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
Endocrinology and Metabolism.2014; 29(4): 470. CrossRef
Brief Review of Articles in 'Endocrinology and Metabolism' in 2013
Won-Young Lee
Endocrinology and Metabolism.2014; 29(3): 251. CrossRef

First
Prev
Page of 1
Next
Last

Search