Endocrinology and Metabolism

Original Articles

Thyroid
Long-Term Changes in the Mortality Rates of Thyroid Cancer in Korea: Analysis of Korean National Data from 1985 to 2020: Yun Mi Choi, Min-Ju Kim, Jiwoo Lee, Mi Kyung Kwak, Min Ji Jeon, Tae Yong Kim, Eun-Gyoung Hong, Won Bae Kim, Won Gu Kim; Endocrinol Metab. 2023;38(5):588-595. Published online September 8, 2023; DOI: https://doi.org/10.3803/EnM.2023.1723

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Background
Thyroid cancer mortality has been largely overlooked as relatively stable given the large gap between thyroid cancer incidence and mortality. This study evaluated long-term trends in age-standardized mortality rates (ASMRs) throughout Korea and compared them with mortality data reported by the Surveillance, Epidemiology, and End Results (SEER).
Methods
Cancer-specific mortality data from 1985 to 2020 were obtained from Statistics Korea. ASMRs from thyroid cancer were calculated based on the Korean mid-year resident registration population of 2005. We assessed SEER*Explorer and downloaded the mortality data.
Results
The ASMR increased from 0.19 to 0.77/100,000 between 1985 and 2002 but decreased continuously to 0.36/100,000 in 2020. The annual percent change (APC) in the ASMR between 1985 and 2003 and between 2003 and 2020 was 6.204 and −4.218, respectively, with similar patterns observed in both men and women. The ASMR of the SEER showed a modest increase from 1988 to 2016 and then stabilized. In subgroup analysis, the ASMR of the old age group (≥55 years) increased significantly from 0.82 in 1985 to 3.92/100,000 in 2002 (APC 6.917) but then decreased again to 1.86/100,000 in 2020 (APC −4.136). ASMRs according to the age group in the SEER showed a relatively stable trend even in the elderly group.
Conclusion
The ASMR of thyroid cancer in Korea had increased from 1985 to 2002 but has since been steadily decreasing. This trend was mainly attributed to elderly people aged 55 or over. The absolute APC value of Korea was much higher than that of the SEER.

Citations

Citations to this article as recorded by

It Is Time to Understand the Additional Benefits of Active Surveillance for Low-Risk Papillary Thyroid Carcinoma
Kyeong Jin Kim
Endocrinology and Metabolism.2024; 39(1): 95. CrossRef
A Clinical Audit of Thyroid Hormonal Replacement After Total Thyroidectomy
Islam Mansy, Abdelfatah M Elsenosy, Eslam M Hassan, Mujtaba Zakria
Cureus.2023;[Epub] CrossRef

Diabetes, obesity and metabolism
Efficacy of Gemigliptin Add-on to Dapagliflozin and Metformin in Type 2 Diabetes Patients: A Randomized, Double-Blind, Placebo-Controlled Study (SOLUTION): Byung Wan Lee, KyungWan Min, Eun-Gyoung Hong, Bon Jeong Ku, Jun Goo Kang, Suk Chon, Won-Young Lee, Mi Kyoung Park, Jae Hyeon Kim, Sang Yong Kim, Keeho Song, Soon Jib Yoo; Endocrinol Metab. 2023;38(3):328-337. Published online June 28, 2023; DOI: https://doi.org/10.3803/EnM.2023.1688

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Abstract PDF Supplementary Material PubReader ePub: Background
This study evaluated the efficacy and safety of add-on gemigliptin in patients with type 2 diabetes mellitus (T2DM) who had inadequate glycemic control with metformin and dapagliflozin.
Methods
In this randomized, placebo-controlled, parallel-group, double-blind, phase III study, 315 patients were randomized to receive either gemigliptin 50 mg (n=159) or placebo (n=156) with metformin and dapagliflozin for 24 weeks. After the 24-week treatment, patients who received the placebo were switched to gemigliptin, and all patients were treated with gemigliptin for an additional 28 weeks.
Results
The baseline characteristics were similar between the two groups, except for body mass index. At week 24, the least squares mean difference (standard error) in hemoglobin A1c (HbA1c) changes was –0.66% (0.07) with a 95% confidence interval of –0.80% to –0.52%, demonstrating superior HbA1c reduction in the gemigliptin group. After week 24, the HbA1c level significantly decreased in the placebo group as gemigliptin was administered, whereas the efficacy of HbA1c reduction was maintained up to week 52 in the gemigliptin group. The safety profiles were similar: the incidence rates of treatment-emergent adverse events up to week 24 were 27.67% and 29.22% in the gemigliptin and placebo groups, respectively. The safety profiles after week 24 were similar to those up to week 24 in both groups, and no new safety findings, including hypoglycemia, were noted.
Conclusion
Add-on gemigliptin was well tolerated, providing comparable safety profiles and superior efficacy in glycemic control over placebo for long-term use in patients with T2DM who had poor glycemic control with metformin and dapagliflozin.

Thyroid Big Data Articles (National Health Insurance Service Database)
Recent Changes in the Incidence of Thyroid Cancer in Korea between 2005 and 2018: Analysis of Korean National Data: Yun Mi Choi, Jiwoo Lee, Mi Kyung Kwak, Min Ji Jeon, Tae Yong Kim, Eun-Gyoung Hong, Won Bae Kim, Won Gu Kim; Endocrinol Metab. 2022;37(5):791-799. Published online October 11, 2022; DOI: https://doi.org/10.3803/EnM.2022.1533

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Background
In this study, we evaluated the recent changes in the standardized, age-specific, stage-specific incidence rates (IRs) of thyroid cancer in Korea and compared them with the incidence data reported by the Surveillance, Epidemiology, and End Results Program.
Methods
The analysis was conducted using the incidence data (2005 to 2018) from the Statistics Korea and Korea Central Cancer Registry.
Results
The age-standardized IR (SIR) of thyroid cancer increased from 24.09 per 100,000 in 2005 to 74.83 in 2012 (annual percent change [APC], 14.5). From 2012 to 2015, the SIR decreased to 42.52 (APC, –17.9) and then remained stable until 2018 (APC, 2.1). This trend was similar in both men and women. Regarding age-specific IRs, the IRs for ages of 30 years and older showed a trend similar to that of the SIR; however, for ages below 30 years, no significant reduction was observed from the vertex of IR in 2015. Regarding stage-specific IRs, the increase was more prominent in those with regional disease (APC, 17.4) than in those with localized disease until 2012; then, the IR decreased until 2015 (APC, –16.1). The average APC from 2005 to 2018 increased in men, those under the age of 30 years, and those with regional disease.
Conclusion
The SIR in Korea peaked in 2012 and decreased until 2015 and then remained stable until 2018. However, in young individuals under the age of 30 years, the IR did not significantly decrease but tended to increase again. In terms of stage-specific IRs, the sharpest increase was seen among those with regional disease.

Citations

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Comparison of postoperative pain between transoral and conventional thyroidectomy: a propensity score-matched analysis
Min Kyu Park, Van Cuong Nguyen, Eugene Kim, Chang Myeon Song, Yong Bae Ji, Jin Hyeok Jeong, Kyung Tae
Surgical Endoscopy.2024; 38(3): 1512. CrossRef
Contents analysis of thyroid cancer-related information uploaded to YouTube by physicians in Korea: endorsing thyroid cancer screening, potentially leading to overdiagnosis
EunKyo Kang, HyoRim Ju, Soojeong Kim, Juyoung Choi
BMC Public Health.2024;[Epub] CrossRef
Bilateral axillo-breast approach robotic total thyroidectomy without isthmectomy: a case report
Hyeji Kim, Hyeonuk Hwang, Hyungju Kwon
The Ewha Medical Journal.2024;[Epub] CrossRef
Association between Consumption of Iodine-Rich Foods and Thyroid Cancer Prevalence: Findings from a Large Population-Based Study
Yu-Jin Kwon, Hye-Sun Lee, Sang-Wook Kang, Ji-Won Lee
Nutrients.2024; 16(7): 1041. CrossRef
Cancer and Mortality Risks of Graves’ Disease in South Korea Based on National Data from 2010 to 2019
Young Ju Choi, Kyungdo Han, Won Kyoung Cho, Min Ho Jung, Byung-Kyu Suh
Clinical Epidemiology.2023; Volume 15: 535. CrossRef
Survival Comparison of Incidentally Found versus Clinically Detected Thyroid Cancers: An Analysis of a Nationwide Cohort Study
Shinje Moon, Eun Kyung Lee, Hoonsung Choi, Sue K. Park, Young Joo Park
Endocrinology and Metabolism.2023; 38(1): 81. CrossRef
Cumulative exposure to metabolic syndrome increases thyroid cancer risk in young adults: a population-based cohort study
Jinyoung Kim, Kyungdo Han, Mee Kyoung Kim, Ki-Hyun Baek, Ki-Ho Song, Hyuk-Sang Kwon
The Korean Journal of Internal Medicine.2023; 38(4): 526. CrossRef
Cost-Effectiveness of Active Surveillance Compared to Early Surgery of Small Papillary Thyroid Cancer: A Retrospective Study on a Korean Population
Han-Sang Baek, Jeonghoon Ha, Kwangsoon Kim, Jaseong Bae, Jeong Soo Kim, Sungju Kim, Dong-Jun Lim, Chulmin Kim
Journal of Korean Medical Science.2023;[Epub] CrossRef
Long-Term Changes in the Mortality Rates of Thyroid Cancer in Korea: Analysis of Korean National Data from 1985 to 2020
Yun Mi Choi, Min-Ju Kim, Jiwoo Lee, Mi Kyung Kwak, Min Ji Jeon, Tae Yong Kim, Eun-Gyoung Hong, Won Bae Kim, Won Gu Kim
Endocrinology and Metabolism.2023; 38(5): 588. CrossRef
Age and Post-Lobectomy Recurrence after Endoscopic or Robotic Thyroid Surgery: A Retrospective Cohort Study of 2348 Papillary Thyroid Carcinoma Patients
Jin-Seong Cho, Yong-Min Na, Hee Kyung Kim
Cancers.2023; 15(23): 5506. CrossRef

Clinical Study
Short-Term Effects of Beraprost Sodium on the Markers for Cardiovascular Risk Prediction in Type 2 Diabetic Patients with Microalbuminuria: Yun Mi Choi, Hyuk-Sang Kwon, Kyung Mook Choi, Won-Young Lee, Eun-Gyoung Hong; Endocrinol Metab. 2019;34(4):398-405. Published online December 23, 2019; DOI: https://doi.org/10.3803/EnM.2019.34.4.398

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Background

To evaluate the changes in cardiovascular risk markers including pulse wave velocity (PWV), microalbuminuria, inflammatory cytokines, and adhesion molecules after treatment with beraprost sodium (BPS) in patients with diabetic nephropathy.

Methods

This was a multicenter, prospective, randomized, double-blind, placebo-controlled trial. Type 2 diabetes mellitus patients with microalbuminuria were included. The primary endpoints were changes in microalbuminuria in spot urine and PWV after BPS or placebo (PCB) treatment for 24 weeks. The secondary endpoints were changes in clinical and metabolic parameters.

Results

A total of 52 patients completed the 24-week trial. Changes in PWV were not different significantly in the BPS and PCB groups (right, P=0.16; left, P=0.11). Changes in microalbuminuria were 14.2±157.0 and 34.5±146.6 (µg/mg Cr) in the BPS and PCB groups, respectively (P=0.63). Subgroup analysis in the high blood pressure (BP) group (baseline systolic BP >120 mm Hg and diastolic BP >80 mm Hg), showed that microalbuminuria decreased by −47.6 in the BPS group compared with an increase by 116.4 (µg/mg Cr) in the PCB group (P=0.04). Also, in the large waist circumference group (>95 cm), microalbuminuria decreased significantly in the BPS group (P=0.04).

Conclusion

Short-term treatment of BPS for patients with diabetic nephropathy did not show significant improvement in various cardiovascular risk factors. However, BPS significantly decreased microalbuminuria in study subjects with higher cardiovascular risk such as high BP or large waist circumference.

Citations

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Clinical efficacy of beraprost sodium in treating chronic kidney disease: A six-month prospective study
Chen Sun, Xin Wu, Xin Zhang, Shulin Li, Ruoyu Jia, Dong Sun
Heliyon.2024; 10(2): e24156. CrossRef
Clinical efficacy and safety of beraprost sodium in the treatment of nephrotic syndrome: A meta-analysis
Peng Yan, Ben Ke, Xiangdong Fang
Medicine.2023; 102(42): e34958. CrossRef
Dysregulated coagulation system links to inflammation in diabetic kidney disease
Mengyun Xiao, Donge Tang, Shaodong Luan, Bo Hu, Wenyu Gong, Wolfgang Pommer, Yong Dai, Lianghong Yin
Frontiers in Clinical Diabetes and Healthcare.2023;[Epub] CrossRef
The effects of beraprost sodium on renal function and cardiometabolic profile in patients with diabetes mellitus: a systematic review and meta-analysis of clinical trials
Peyman Nowrouzi-Sohrabi, Reza Tabrizi, Kamran Hessami, Mojtaba Shabani-Borujeni, Mahnaz Hosseini-Bensenjan, Shahla Rezaei, Mohammad Jalali, Pedram Keshavarz, Fariba Ahmadizar
International Urology and Nephrology.2022; 54(1): 111. CrossRef
Thrombocytopenia in COVID‑19 and vaccine‑induced thrombotic thrombocytopenia
Styliani Geronikolou, Işil Takan, Athanasia Pavlopoulou, Marina Mantzourani, George Chrousos
International Journal of Molecular Medicine.2022;[Epub] CrossRef
The Role of Platelets in Diabetic Kidney Disease
Ukhti Jamil Rustiasari, Joris J. Roelofs
International Journal of Molecular Sciences.2022; 23(15): 8270. CrossRef
Comparative Efficacy of Lobeglitazone Versus Pioglitazone on Albuminuria in Patients with Type 2 Diabetes Mellitus
Kyung-Soo Kim, Sangmo Hong, Hong-Yup Ahn, Cheol-Young Park
Diabetes Therapy.2021; 12(1): 171. CrossRef

Clinical Study
Changes in Thyroid Peroxidase and Thyroglobulin Antibodies Might Be Associated with Graves' Disease Relapse after Antithyroid Drug Therapy: Yun Mi Choi, Mi Kyung Kwak, Sang Mo Hong, Eun-Gyoung Hong; Endocrinol Metab. 2019;34(3):268-274. Published online September 26, 2019; DOI: https://doi.org/10.3803/EnM.2019.34.3.268

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Background

Graves' disease (GD) is an autoimmune thyroid disorder caused by antibodies stimulating the thyrotropin (TSH) receptor. TSH receptor antibody (TRAb) measurement is useful for predicting GD relapse after antithyroid drug (ATD) treatment. However, the association of other thyroid autoantibodies with GD relapse remains obscure.

Methods

This retrospective study enrolled patients with GD who were initially treated with ATD. TRAb, thyroid peroxidase antibody (TPOAb), and thyroglobulin antibody (TgAb) were measured at the initial diagnosis and at the time of ATD discontinuation.

Results

A total of 55 patients were enrolled. The mean age was 49.7 years, and 39 patients (70.9%) were female. Antibody positivity at diagnosis was 90.9%, 69.1%, and 61.9% for TRAb, TPOAb, TgAb, respectively. Median ATD treatment period was 15.1 months. At the time of ATD withdrawal, TRAb titers decreased uniformly overall. Conversely, TPOAb and TgAb showed various changes. After withdrawal of ATD, 19 patients (34.5%) experienced relapse. No clinical features or laboratory results were significantly related to relapse in the overall patient group. However, in the TPOAb positive group at diagnosis, increasing titer of TPOAb or TgAb after ATD treatment was significantly and independently related to relapse free survival (TPOAb: hazard ratio [HR], 17.99; 95% confidence interval [CI], 1.66 to 195.43; P=0.02) (TgAb: HR, 5.73; 95% CI, 1.21 to 27.26; P=0.03).

Conclusion

Changes in TPOAb or TgAb titers during treatment might be useful for predicting relapse after ATD treatment in patients with positive TPOAb at diagnosis.

Citations

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Enhanced predictive validity of integrative models for refractory hyperthyroidism considering baseline and early therapy characteristics: a prospective cohort study
Xinpan Wang, Tiantian Li, Yue Li, Qiuyi Wang, Yun Cai, Zhixiao Wang, Yun Shi, Tao Yang, Xuqin Zheng
Journal of Translational Medicine.2024;[Epub] CrossRef
Clinical significance of thyroglobulin antibodies and thyroid peroxidase antibodies in Graves’ disease: a cross-sectional study
Masahito Katahira, Taku Tsunekawa, Akira Mizoguchi, Mariko Yamaguchi, Kahori Tsuru, Hiromi Takashima, Ryoma Terada
Hormones.2023; 22(2): 253. CrossRef
The Clinical Implications of Anti-thyroid Peroxidase Antibodies in Graves’ Disease in Basrah
Emad S Alhubaish, Nassar T Alibrahim, Abbas A Mansour
Cureus.2023;[Epub] CrossRef
Influence of Thyroid Peroxidase Antibodies Serum Levels in Graves' Disease: A Retrospective Cohort Study
Maria L Guia Lopes, Carlos Tavares Bello, José P Cidade, Clotilde Limbert, Joao Sequeira Duarte
Cureus.2023;[Epub] CrossRef
Interpretation of Thyroid Autoantibodies in Hyperthyroidism
Han-Sang Baek, Dong-Jun Lim
The Korean Journal of Medicine.2023; 98(3): 132. CrossRef
Usefulness of Real-Time Quantitative Microvascular Ultrasonography for Differentiation of Graves’ Disease from Destructive Thyroiditis in Thyrotoxic Patients
Han-Sang Baek, Ji-Yeon Park, Chai-Ho Jeong, Jeonghoon Ha, Moo Il Kang, Dong-Jun Lim
Endocrinology and Metabolism.2022; 37(2): 323. CrossRef
Differential Diagnosis of Thyrotoxicosis by Machine Learning Models with Laboratory Findings
Jinyoung Kim, Han-Sang Baek, Jeonghoon Ha, Mee Kyoung Kim, Hyuk-Sang Kwon, Ki-Ho Song, Dong-Jun Lim, Ki-Hyun Baek
Diagnostics.2022; 12(6): 1468. CrossRef
The relationship between atherosclerotic disease and relapse during ATD treatment
Xinxin Zhu, Yaguang Zhang, Xiaoyu Zhao, Xiaona Zhang, Zixuan Ru, Yanmeizhi Wu, Xu Yang, Boyu Hou, Hong Qiao
Frontiers in Cardiovascular Medicine.2022;[Epub] CrossRef
Programmed Cell Death-Ligand 1 (PD-L1) gene Single Nucleotide Polymorphism in Graves’ Disease and Hashimoto’s Thyroiditis in Korean Patients
Jee Hee Yoon, Min-ho Shin, Hee Nam Kim, Wonsuk Choi, Ji Yong Park, A Ram Hong, Hee Kyung Kim, Ho-Cheol Kang
Endocrinology and Metabolism.2021; 36(3): 599. CrossRef
Low frequency of IL-10-producing B cells and high density of ILC2s contribute to the pathological process in Graves’ disease, which may be related to elevated-TRAb levels
Xiaoyun Ji, Jie Wan, Rong Chen, Huixuan Wang, Lan Huang, Shwngjun Wang, Zhaoliang Su, Huaxi Xu
Autoimmunity.2020; 53(2): 78. CrossRef
Implication of VDR rs7975232 and FCGR2A rs1801274 gene polymorphisms in the risk and the prognosis of autoimmune thyroid diseases in the Tunisian population
S Mestiri, I Zaaber, I Nasr, H Marmouch
Balkan Journal of Medical Genetics.2020; 23(1): 69. CrossRef
Thyroid Peroxidase Antibody Positivity is Associated With Relapse-Free Survival Following Antithyroid Drug Treatment for Graves Disease
Christopher A. Muir, Graham R.D. Jones, Jerry R. Greenfield, Andrew Weissberger, Katherine Samaras
Endocrine Practice.2020; 26(9): 1026. CrossRef
Predicting the Risk of Graves Disease Relapse: Commentary on “Thyroid Peroxidase Antibody Positivity is Associated with Relapse-Free Survival Following Antithyroid Drug Treatment for Graves Disease”
D. Gallo, M.L. Tanda, E. Piantanida
Endocrine Practice.2020; 26(9): 1039. CrossRef

Bone Metabolism
Efficacy of a Once-Monthly Pill Containing Ibandronate and Cholecalciferol on the Levels of 25-Hydroxyvitamin D and Bone Markers in Postmenopausal Women with Osteoporosis: In-Jin Cho, Ho-Yeon Chung, Sung-Woon Kim, Jae-Won Lee, Tae-Won Lee, Hye-Soon Kim, Sin-Gon Kim, Han Seok Choi, Sung-Hee Choi, Chan Soo Shin, Ki-Won Oh, Yong-Ki Min, Jung-Min Koh, Yumie Rhee, Dong-Won Byun, Yoon-Sok Chung, Jeong Hyun Park, Dong Jin Chung, Minho Shong, Eun-Gyoung Hong, Chang Beom Lee, Ki Hyun Baek, Moo-Il Kang; Endocrinol Metab. 2015;30(3):272-279. Published online December 9, 2014; DOI: https://doi.org/10.3803/EnM.2015.30.3.272

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Background

The present study evaluated the efficacy of a combination of ibandronate and cholecalciferol on the restoration of the levels of 25-hydroxyvitamin D (25[OH]D) and various bone markers in postmenopausal women with osteoporosis.

Methods

This was a randomized, double-blind, active-controlled, prospective 16-week clinical trial conducted in 20 different hospitals. A total of 201 postmenopausal women with osteoporosis were assigned randomly to one of two groups: the IBN group, which received a once-monthly pill containing 150 mg ibandronate (n=99), or the IBN+ group, which received a once-monthly pill containing 150 mg ibandronate and 24,000 IU cholecalciferol (n=102). Serum levels of 25(OH)D, parathyroid hormone (PTH), and various bone markers were assessed at baseline and at the end of a 16-week treatment period.

Results

After 16 weeks of treatment, the mean serum levels of 25(OH)D significantly increased from 21.0 to 25.3 ng/mL in the IBN+ group but significantly decreased from 20.6 to 17.4 ng/mL in the IBN group. Additionally, both groups exhibited significant increases in mean serum levels of PTH but significant decreases in serum levels of bone-specific alkaline phosphatase and C-telopeptide of type 1 collagen (CTX) at 16 weeks; no significant differences were observed between the groups. However, in subjects with a vitamin D deficiency, IBN+ treatment resulted in a significant decrease in serum CTX levels compared with IBN treatment.

Conclusion

The present findings demonstrate that a once-monthly pill containing ibandronate and cholecalciferol may be useful for the amelioration of vitamin D deficiency in patients with postmenopausal osteoporosis. Moreover, this treatment combination effectively decreased serum levels of resorption markers, especially in subjects with a vitamin D deficiency, over the 16-week treatment period.

Citations

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Effect of vitamin D supplementation or fortification on bone turnover markers in women: a systematic review and meta-analysis
Nasrin Nasimi, Sanaz Jamshidi, Aida Askari, Nazanin Zolfaghari, Erfan Sadeghi, Mehran Nouri, Nick Bellissimo, Shiva Faghih
British Journal of Nutrition.2024; 131(9): 1473. CrossRef
Quality of life and patient satisfaction with raloxifene/cholecalciferol combination therapy in postmenopausal women
Dong-Yun Lee, Yoon-Sok Chung
Scientific Reports.2022;[Epub] CrossRef
Efficacy of risedronate with cholecalciferol on bone mineral density in Korean patients with osteoporosis
So Young Park, Moo-Il Kang, Hyung Moo Park, Yumie Rhee, Seong Hwan Moon, Hyun Koo Yoon, Jung-Min Koh, Jae Suk Chang, In Joo Kim, Ye Yeon Won, Ye Soo Park, Hoon Choi, Chan Soo Shin, Taek Rim Yoon, Sung-Cheol Yun, Ho-Yeon Chung
Archives of Osteoporosis.2020;[Epub] CrossRef
Efficacy and safety of vitamin D3 B.O.N intramuscular injection in Korean adults with vitamin D deficiency
Han Seok Choi, Yoon-Sok Chung, Yong Jun Choi, Da Hea Seo, Sung-Kil Lim
Osteoporosis and Sarcopenia.2016; 2(4): 228. CrossRef
Pharmacologic treatment of osteoporosis
Yong-Ki Min
Journal of the Korean Medical Association.2016; 59(11): 847. CrossRef

Case Report

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

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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
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Adult Langerhans Cell Histiocytosis Masquerading as Hidradenitis Suppurativa
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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

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