1.Beyond the Pituitary Stalk: Primary Hypothyroidism as a Rare Presentation of Multisystem Langerhans Cell Histiocytosis
Fang Chan Lim ; Shireen Siow Leng Lui
Journal of the ASEAN Federation of Endocrine Societies 2026;41(S1):102-103
Introduction:
Langerhans cell histiocytosis (LCH) is a clonal proliferative disorder of langerin-positive histiocytes. Endocrine
involvement most frequently manifests as central diabetes
insipidus or secondary hypothyroidism, caused by infiltration of the hypothalamic-pituitary axis. Conversely, direct
infiltration of the thyroid gland leading to primary hypothyroidism is an exceptionally rare clinical entity, particularly when presenting concurrently with central disease.
Case:
A 21-year-old female with known right otic and multisystem
LCH presented with septic shock secondary to a right
ear abscess accompanied by polyuria and polydipsia.
Physical examination revealed a palpable goiter. Clinical
and biochemical evaluation confirmed central diabetes
insipidus with associated anterior hypopituitarism
(low adrenocorticotropic hormone, follicle-stimulating
hormone, and luteinizing hormone). However, concurrent
thyroid function tests demonstrated overt primary
hypothyroidism, evidenced by an appropriately elevated
thyroid-stimulating hormone (26.19 mIU/L) and low free
T4 (5.47 pmol/L), rather than the anticipated secondary
hypothyroidism. Neck ultrasound showed diffuse thyroid
enlargement with heterogeneous echotexture. Crucially,
anti-thyroid peroxidase and anti-thyroglobulin antibodies
were both negative, rendering Hashimoto’s thyroiditis
highly unlikely. Although the patient declined confirmatory fine-needle aspiration, the constellation of a palpable
goiter, characteristic ultrasonographic findings, negative
autoimmunity, and active multisystem disease strongly
supported a diagnosis of direct histiocytic infiltration
of the thyroid gland. She was initiated on appropriate
glucocorticoid coverage and subsequent levothyroxine
replacement, alongside systemic intravenous cytarabine.
Conclusion
This case highlights a rare, mixed endocrine profile in
multisystem LCH, demonstrating that pituitary and
direct end-organ infiltration can coexist. Hypothyroidism
in LCH patients with established central diabetes
insipidus should not be reflexively assumed to be
central in origin. A comprehensive diagnostic workup,
including autoantibody screening, ultrasound, and ideally
histopathological confirmation, is essential to accurately
identify primary endocrine failure and guide appropriate
clinical management in these complex cases.
Histiocytosis, Langerhans-Cell
;
Hypothyroidism
;
Pituitary Gland
2.Titrating the Mind: Refractory Schizophreniform Psychosis as an Isolated “Cerebral Storm” in Graves’ Disease
Journal of the ASEAN Federation of Endocrine Societies 2026;41(S1):120-121
Introduction:
Neuropsychiatric manifestations of thyrotoxicosis range
from mild anxiety to severe psychosis. While the BurchWartofsky Point Scale (BWPS) reliably identifies systemic
thyroid storms, an isolated “cerebral storm”—where
profound thyrotoxic encephalopathy presents without
peripheral autonomic signs—remains a diagnostic
challenge. This case highlights the neuromodulatory
pathogenesis of thyrotoxic psychosis and the critical role
of biochemical control for psychiatric resolution.
Case:
A 31-year-old male with Graves’ disease and poor
medication adherence presented with a 2-week history of
aggressive behavior and auditory hallucinations. Initial
investigations revealed overt thyrotoxicosis (thyroidstimulating hormone <0.01 mIU/L; free thyroxine 4 (FT4)
59.53 pmol/L) and an unremarkable computed tomography
brain. Despite his severe neuropsychiatric decompensation,
a BWPS of 20 precluded a clinical diagnosis of systemic
thyroid storm.
His symptoms remained refractory to aggressive
psychotropic polypharmacy, including haloperidol,
olanzapine, lithium, and diazepam. Management was
therefore focused on the underlying thyrotoxicosis with
an optimized antithyroid regimen of carbimazole and
propranolol. Clinical resolution of psychotic symptoms
was achieved only after FT4 declined significantly to 20.46
pmol/L, allowing the successful tapering of all psychiatric
medications. He was discharged in a stable, clinically
euthyroid state with integrated medical and psychiatric
follow-up.
Conclusion
The failure of standard antipsychotics suggests that
thyrotoxic psychosis is driven by distinct neuromodulatory
mechanisms rather than primary dopaminergic dysfunction.
The chronic thyrotoxic state secondary to prolonged
treatment non-adherence likely upregulates and sensitizes
dopamine receptors while simultaneously suppressing
inhibitory GABAergic pathways. This neurochemical
imbalance lowers the neurological threshold, precipitating
an isolated cerebral storm. This case illustrates the limitations
of the BWPS in purely neuropsychiatric presentations and
reinforces that restoring euthyroidism is the definitive
treatment for thyrotoxic psychosis.
Graves Disease
;
Psychotic Disorders
3.Application and data analysis of the cardio-cerebrovascular events monitoring system in Yichang
Zhengchao FANG ; Jiajuan YANG ; Chi HU ; Chan WU ; Yaling DENG ; Zhiying YU ; Jie ZHU ; Ling ZHANG
Journal of Public Health and Preventive Medicine 2025;36(6):95-98
Objective To analyze the monitoring data of cardio-cerebrovascular diseases prevention and control system in Yichang in 2022, and to provide data support and experience for the precise prevention and treatment of cardio-cerebrovascular diseases. Methods Acute cardiovascular and cerebrovascular event data were collected from the Yichang Cardio-cerebrovascular Events Monitoring System from January 1, 2022 to December 31, 2022. Descriptive analysis was conducted for the data collected. Statistical analysis was performed using SPSS 20.0 software, and a chi-square test was used to analyze the count data. Results A total of 37,217 cases of cardio-cerebrovascular events were monitored in Yichang in 2022. The crude incidence and the standardized incidence were 983.84/100,000 and 541.55/100,000, respectively. The incidence in males was higher than females (554.93/100,000 vs 428.91/100,000,χ2 =464.52,P<0.05). The top three diseases were cerebral infarction, acute myocardial infarction, and cerebral hemorrhage. The incidence of events increased with age, and 79.80% of the cases were over 60 years old. The main onset time was from May to August. Conclusion The use of the cardio-cerebrovascular events monitoring system in Yichang and the implementation of “mandatory reporting card” monitoring can timely obtain the epidemic characteristics of the diseases, provide support for the precise formulation of prevention and control strategies and measures, reduce underreporting rates, and improve the monitoring system, which is worthy of reference and promotion.
4.Comparison of Embolization Coils and Patent Ductus Arteriosus Occluders for Coronary Artery Fistula Transcatheter Closure: A Single Centre Experience
Peijian WEI ; Yihang LI ; Liang XU ; Junyi WAN ; Fengwen ZHANG ; Gary TSE ; Jeffrey Shi Kai CHAN ; Shouzheng WANG ; Wenbin OUYANG ; Gejun ZHANG ; Fang FANG ; Xiangbin PAN
Korean Circulation Journal 2025;55(3):199-212
Background and Objectives:
There is no dedicated occlusive device for closing coronary artery fistulas (CAFs), and specific efficacy and safety data of various off-label occlusive devices for CAFs closure are scarce.
Methods:
Patients undergoing transcatheter closure of CAFs from January 2011 to December 2022 were included in the single-center retrospective study. The study population was divided into 2 groups: coils group (n=35) and patent ductus arteriosus (PDA) occluders group (n=66).
Results:
No significant intergroup differences were observed in demographic characteristics except age. The presence of multiple CAF origins (54.3% vs. 4.5%, p<0.001) and multiple draining sites (51.4% vs. 3.0%, p<0.001) were more common in the coils group. In contrast, the presence of aneurysm (72.7% vs. 14.3%, p<0.001), and large fistula (75.8% vs. 37.1%, p<0.001) were more prevalent in the PDA occluders group. The acute procedural success rate of the PDA occluders group was higher compared to that of the coils group (87.9% vs.62.9%, adjusted odds ratio [OR], 7.20; 95% confidence interval, 1.59–32.64; p=0.01).In addition, no significant intergroup differences were noted in both the recanalization rate (7.8% vs. 20%, p=0.107) and the reintervention rate (3.1% vs. 8.6%, p=0.342).
Conclusions
Transcatheter closure of CAFs using PDA occluders was associated with significantly higher acute procedural success rates compared to coil embolization with comparable late outcomes.
5.Comparison of Embolization Coils and Patent Ductus Arteriosus Occluders for Coronary Artery Fistula Transcatheter Closure: A Single Centre Experience
Peijian WEI ; Yihang LI ; Liang XU ; Junyi WAN ; Fengwen ZHANG ; Gary TSE ; Jeffrey Shi Kai CHAN ; Shouzheng WANG ; Wenbin OUYANG ; Gejun ZHANG ; Fang FANG ; Xiangbin PAN
Korean Circulation Journal 2025;55(3):199-212
Background and Objectives:
There is no dedicated occlusive device for closing coronary artery fistulas (CAFs), and specific efficacy and safety data of various off-label occlusive devices for CAFs closure are scarce.
Methods:
Patients undergoing transcatheter closure of CAFs from January 2011 to December 2022 were included in the single-center retrospective study. The study population was divided into 2 groups: coils group (n=35) and patent ductus arteriosus (PDA) occluders group (n=66).
Results:
No significant intergroup differences were observed in demographic characteristics except age. The presence of multiple CAF origins (54.3% vs. 4.5%, p<0.001) and multiple draining sites (51.4% vs. 3.0%, p<0.001) were more common in the coils group. In contrast, the presence of aneurysm (72.7% vs. 14.3%, p<0.001), and large fistula (75.8% vs. 37.1%, p<0.001) were more prevalent in the PDA occluders group. The acute procedural success rate of the PDA occluders group was higher compared to that of the coils group (87.9% vs.62.9%, adjusted odds ratio [OR], 7.20; 95% confidence interval, 1.59–32.64; p=0.01).In addition, no significant intergroup differences were noted in both the recanalization rate (7.8% vs. 20%, p=0.107) and the reintervention rate (3.1% vs. 8.6%, p=0.342).
Conclusions
Transcatheter closure of CAFs using PDA occluders was associated with significantly higher acute procedural success rates compared to coil embolization with comparable late outcomes.
6.Comparison of Embolization Coils and Patent Ductus Arteriosus Occluders for Coronary Artery Fistula Transcatheter Closure: A Single Centre Experience
Peijian WEI ; Yihang LI ; Liang XU ; Junyi WAN ; Fengwen ZHANG ; Gary TSE ; Jeffrey Shi Kai CHAN ; Shouzheng WANG ; Wenbin OUYANG ; Gejun ZHANG ; Fang FANG ; Xiangbin PAN
Korean Circulation Journal 2025;55(3):199-212
Background and Objectives:
There is no dedicated occlusive device for closing coronary artery fistulas (CAFs), and specific efficacy and safety data of various off-label occlusive devices for CAFs closure are scarce.
Methods:
Patients undergoing transcatheter closure of CAFs from January 2011 to December 2022 were included in the single-center retrospective study. The study population was divided into 2 groups: coils group (n=35) and patent ductus arteriosus (PDA) occluders group (n=66).
Results:
No significant intergroup differences were observed in demographic characteristics except age. The presence of multiple CAF origins (54.3% vs. 4.5%, p<0.001) and multiple draining sites (51.4% vs. 3.0%, p<0.001) were more common in the coils group. In contrast, the presence of aneurysm (72.7% vs. 14.3%, p<0.001), and large fistula (75.8% vs. 37.1%, p<0.001) were more prevalent in the PDA occluders group. The acute procedural success rate of the PDA occluders group was higher compared to that of the coils group (87.9% vs.62.9%, adjusted odds ratio [OR], 7.20; 95% confidence interval, 1.59–32.64; p=0.01).In addition, no significant intergroup differences were noted in both the recanalization rate (7.8% vs. 20%, p=0.107) and the reintervention rate (3.1% vs. 8.6%, p=0.342).
Conclusions
Transcatheter closure of CAFs using PDA occluders was associated with significantly higher acute procedural success rates compared to coil embolization with comparable late outcomes.
7.Comparison of Embolization Coils and Patent Ductus Arteriosus Occluders for Coronary Artery Fistula Transcatheter Closure: A Single Centre Experience
Peijian WEI ; Yihang LI ; Liang XU ; Junyi WAN ; Fengwen ZHANG ; Gary TSE ; Jeffrey Shi Kai CHAN ; Shouzheng WANG ; Wenbin OUYANG ; Gejun ZHANG ; Fang FANG ; Xiangbin PAN
Korean Circulation Journal 2025;55(3):199-212
Background and Objectives:
There is no dedicated occlusive device for closing coronary artery fistulas (CAFs), and specific efficacy and safety data of various off-label occlusive devices for CAFs closure are scarce.
Methods:
Patients undergoing transcatheter closure of CAFs from January 2011 to December 2022 were included in the single-center retrospective study. The study population was divided into 2 groups: coils group (n=35) and patent ductus arteriosus (PDA) occluders group (n=66).
Results:
No significant intergroup differences were observed in demographic characteristics except age. The presence of multiple CAF origins (54.3% vs. 4.5%, p<0.001) and multiple draining sites (51.4% vs. 3.0%, p<0.001) were more common in the coils group. In contrast, the presence of aneurysm (72.7% vs. 14.3%, p<0.001), and large fistula (75.8% vs. 37.1%, p<0.001) were more prevalent in the PDA occluders group. The acute procedural success rate of the PDA occluders group was higher compared to that of the coils group (87.9% vs.62.9%, adjusted odds ratio [OR], 7.20; 95% confidence interval, 1.59–32.64; p=0.01).In addition, no significant intergroup differences were noted in both the recanalization rate (7.8% vs. 20%, p=0.107) and the reintervention rate (3.1% vs. 8.6%, p=0.342).
Conclusions
Transcatheter closure of CAFs using PDA occluders was associated with significantly higher acute procedural success rates compared to coil embolization with comparable late outcomes.
8.Relationship between Klotho,IGF-1,and Irisin in the placentas of pregnant rats and the intrauterine development of fetuses
Fang ZHANG ; Linlu ZHENG ; Chan WANG ; Mengmeng LI ; Yaying CHENG
Chongqing Medicine 2025;54(6):1281-1285,1292
Objective To investigate the relationship between Klotho,insulin-like growth factor-1(IGF-1),and Irisin in the placentas of pregnant rats and the intrauterine development of fetuses.Methods Thirty SD rats were divided into the intrauterine growth restriction(IUGR)experimental animal model group(IUGR group),the macrosomia experimental animal model group(macrosomia fetal group),and the control group ac-cording to different feeding diets(low-protein diet,high-protein diet,and normal diet).All pregnant mice were subjected to laparotomy for tissue sampling on the 21st day of pregnancy.The birth weight,body length,and placental mass of fetal rats were recorded.The relative mRNA expression levels of Klotho,IGF-1,and Irisin in the placentas were detected by quantitative real-time fluorescent reverse transcription-PCR(qRT-PCR),and the correlations between various factors above were analyzed.Results Compared with the control group,the birth weight,body length and placental mass of fetal rats in the IUGR group were lower,while those in the macrosomia fetal group were higher,with statistically significant differences(P<0.05).Compared with the control group,the relative mRNA expression levels of Klotho,IGF-1,and Irisin in the IUGR group were de-creased,while those in the macrosomia fetal group were increased,with statistically significant differences(P<0.05).Correlation analysis showed that Klotho mRNA was positively correlated with the relative expres-sion levels of placental IGF-1 mRNA and Irisin mRNA,and IGF-1 mRNA were also positively correlated with the relative expression level of placental Irisin mRNA(P<0.05).Conclusion Klotho,IGF-1,and Irisin in the placenta are associated with fetal growth and possibly regulated through the Klotho/IGF-1/Irisin axis.
9.Evaluation of Malignancy Risk of Ampullary Tumors Detected by Endoscopy Using 2- 18FFDG PET/CT
Pei-Ju CHUANG ; Hsiu-Po WANG ; Yu-Wen TIEN ; Wei-Shan CHIN ; Min-Shu HSIEH ; Chieh-Chang CHEN ; Tzu-Chan HONG ; Chi-Lun KO ; Yen-Wen WU ; Mei-Fang CHENG
Korean Journal of Radiology 2024;25(3):243-256
Objective:
We aimed to investigate whether 2-[ 18F]fluoro-2-deoxy-D-glucose positron emission tomography/computed tomography (2-[ 18F]FDG PET/CT) can aid in evaluating the risk of malignancy in ampullary tumors detected by endoscopy.
Materials and Methods:
This single-center retrospective cohort study analyzed 155 patients (79 male, 76 female; mean age, 65.7 ± 12.7 years) receiving 2-[ 18F]FDG PET/CT for endoscopy-detected ampullary tumors 5–87 days (median, 7 days) after the diagnostic endoscopy between June 2007 and December 2020. The final diagnosis was made based on histopathological findings. The PET imaging parameters were compared with clinical data and endoscopic features. A model to predict the risk of malignancy, based on PET, endoscopy, and clinical findings, was generated and validated using multivariable logistic regression analysis and an additional bootstrapping method. The final model was compared with standard endoscopy for the diagnosis of ampullary cancer using the DeLong test.
Results:
The mean tumor size was 17.1 ± 7.7 mm. Sixty-four (41.3%) tumors were benign, and 91 (58.7%) were malignant. Univariable analysis found that ampullary neoplasms with a blood-pool corrected peak standardized uptake value in earlyphase scan (SUVe) ≥ 1.7 were more likely to be malignant (odds ratio [OR], 16.06; 95% confidence interval [CI], 7.13–36.18;P < 0.001). Multivariable analysis identified the presence of jaundice (adjusted OR [aOR], 4.89; 95% CI, 1.80–13.33; P = 0.002), malignant traits in endoscopy (aOR, 6.80; 95% CI, 2.41–19.20; P < 0.001), SUVe ≥ 1.7 in PET (aOR, 5.43; 95% CI, 2.00–14.72; P < 0.001), and PET-detected nodal disease (aOR, 5.03; 95% CI, 1.16–21.86; P = 0.041) as independent predictors of malignancy. The model combining these four factors predicted ampullary cancers better than endoscopic diagnosis alone (area under the curve [AUC] and 95% CI: 0.925 [0.874–0.956] vs. 0.815 [0.732–0.873], P < 0.001). The model demonstrated an AUC of 0.921 (95% CI, 0.816–0.967) in candidates for endoscopic papillectomy.
Conclusion
Adding 2-[ 18F]FDG PET/CT to endoscopy can improve the diagnosis of ampullary cancer and may help refine therapeutic decision-making, particularly when contemplating endoscopic papillectomy.
10.Asia-Pacific consensus on long-term and sequential therapy for osteoporosis
Ta-Wei TAI ; Hsuan-Yu CHEN ; Chien-An SHIH ; Chun-Feng HUANG ; Eugene MCCLOSKEY ; Joon-Kiong LEE ; Swan Sim YEAP ; Ching-Lung CHEUNG ; Natthinee CHARATCHAROENWITTHAYA ; Unnop JAISAMRARN ; Vilai KUPTNIRATSAIKUL ; Rong-Sen YANG ; Sung-Yen LIN ; Akira TAGUCHI ; Satoshi MORI ; Julie LI-YU ; Seng Bin ANG ; Ding-Cheng CHAN ; Wai Sin CHAN ; Hou NG ; Jung-Fu CHEN ; Shih-Te TU ; Hai-Hua CHUANG ; Yin-Fan CHANG ; Fang-Ping CHEN ; Keh-Sung TSAI ; Peter R. EBELING ; Fernando MARIN ; Francisco Javier Nistal RODRÍGUEZ ; Huipeng SHI ; Kyu Ri HWANG ; Kwang-Kyoun KIM ; Yoon-Sok CHUNG ; Ian R. REID ; Manju CHANDRAN ; Serge FERRARI ; E Michael LEWIECKI ; Fen Lee HEW ; Lan T. HO-PHAM ; Tuan Van NGUYEN ; Van Hy NGUYEN ; Sarath LEKAMWASAM ; Dipendra PANDEY ; Sanjay BHADADA ; Chung-Hwan CHEN ; Jawl-Shan HWANG ; Chih-Hsing WU
Osteoporosis and Sarcopenia 2024;10(1):3-10
Objectives:
This study aimed to present the Asia-Pacific consensus on long-term and sequential therapy for osteoporosis, offering evidence-based recommendations for the effective management of this chronic condition.The primary focus is on achieving optimal fracture prevention through a comprehensive, individualized approach.
Methods:
A panel of experts convened to develop consensus statements by synthesizing the current literature and leveraging clinical expertise. The review encompassed long-term anti-osteoporosis medication goals, first-line treatments for individuals at very high fracture risk, and the strategic integration of anabolic and anti resorptive agents in sequential therapy approaches.
Results:
The panelists reached a consensus on 12 statements. Key recommendations included advocating for anabolic agents as the first-line treatment for individuals at very high fracture risk and transitioning to anti resorptive agents following the completion of anabolic therapy. Anabolic therapy remains an option for in dividuals experiencing new fractures or persistent high fracture risk despite antiresorptive treatment. In cases of inadequate response, the consensus recommended considering a switch to more potent medications. The consensus also addressed the management of medication-related complications, proposing alternatives instead of discontinuation of treatment.
Conclusions
This consensus provides a comprehensive, cost-effective strategy for fracture prevention with an emphasis on shared decision-making and the incorporation of country-specific case management systems, such as fracture liaison services. It serves as a valuable guide for healthcare professionals in the Asia-Pacific region, contributing to the ongoing evolution of osteoporosis management.


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