Endocrinology, Diabetes and Metabolism Case Reports最新文献

Apparent bilateral aldosterone suppression during adrenal venous sampling associated with preferential drainage into the left inferior phrenic vein. 肾上腺静脉取样时双侧明显的醛固酮抑制与优先引流到左侧膈下静脉有关。

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Endocrinology, Diabetes and Metabolism Case Reports Pub Date : 2026-10-01 DOI: 10.1530/EDM-26-0013

Tomoko Nishigaki, Masaaki Yamamoto, Yuichi Higuchi, Kei Yoshino, Hironori Bando, Keigo Matsushiro, Tomoyuki Gentsu, Keitaro Sofue, Maki Kanzawa, Wataru Ogawa, Hidenori Fukuoka

{"title":"Apparent bilateral aldosterone suppression during adrenal venous sampling associated with preferential drainage into the left inferior phrenic vein.","authors":"Tomoko Nishigaki, Masaaki Yamamoto, Yuichi Higuchi, Kei Yoshino, Hironori Bando, Keigo Matsushiro, Tomoyuki Gentsu, Keitaro Sofue, Maki Kanzawa, Wataru Ogawa, Hidenori Fukuoka","doi":"10.1530/EDM-26-0013","DOIUrl":"10.1530/EDM-26-0013","url":null,"abstract":"Summary: A 54-year-old woman with primary aldosteronism caused by left adrenal adenoma underwent adrenal venous sampling (AVS) for subtype classification and unexpectedly exhibited bilateral aldosterone suppression (BAS) despite adequate selectivity. BAS on AVS is a recognized diagnostic pitfall that can obscure surgically curable unilateral diseases, particularly left-sided cases. In this case, venous anatomy was carefully reassessed considering the inconclusive AVS findings. Although adrenal arteriography is infrequently used in the diagnostic workup of primary aldosteronism, it was incorporated as an adjunct to assess venous anatomy. No additional drainage route was identified on the obtained angiographic images, and the cranial and medial superficial location of the tumor in close proximity to the left inferior phrenic vein raised the suspicion of preferential drainage into this vessel. Although the complete tumor-drainage pathway was not directly visualized, targeted venous sampling based on this anatomical hypothesis enabled accurate lateralization of a unilateral disease. This case underscores interpreting BAS within the context of venous anatomy, particularly on the left side, rather than as definitive evidence of bilateral primary aldosteronism.Learning points: ","PeriodicalId":37467,"journal":{"name":"Endocrinology, Diabetes and Metabolism Case Reports","volume":"2026 4","pages":""},"PeriodicalIF":0.9,"publicationDate":"2026-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13663619/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149115780","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Composite pheochromocytoma-ganglioneuroma secreting vasoactive intestinal peptide. 分泌血管活性肠肽的复合嗜铬细胞瘤-神经节神经瘤。

IF 0.9

Endocrinology, Diabetes and Metabolism Case Reports Pub Date : 2026-10-01 DOI: 10.1530/EDM-25-0184

Benjamin Brochu, Matthieu St-Jean, Yves Collin, Rabia Temmar, Frédéric Bernier, Karel Dandurand, Marie-Hélène Massicotte

{"title":"Composite pheochromocytoma-ganglioneuroma secreting vasoactive intestinal peptide.","authors":"Benjamin Brochu, Matthieu St-Jean, Yves Collin, Rabia Temmar, Frédéric Bernier, Karel Dandurand, Marie-Hélène Massicotte","doi":"10.1530/EDM-25-0184","DOIUrl":"10.1530/EDM-25-0184","url":null,"abstract":"Summary: An 80-year-old female patient presented with a 10-year history of progressively worsening diarrhea and chronic hyponatremia. An abdominal computed tomography (CT) revealed a 6.8-centimeter right adrenal mass. A Ga-68 DOTATATE positron emission tomography (PET)/CT scan subsequently demonstrated a significant expression of somatostatin receptors on the tumoral shell (maximal SUV of 14.4), associated with a hypometabolic central portion. Endocrine investigation indicated elevated levels of both urinary and plasmatic normetanephrines. Given the watery diarrhea, VIP levels were measured, which were markedly elevated. Surgical resection confirmed a composite pheochromocytoma-ganglioneuroma with VIP immunoreactivity in both tumor components. In the postoperative period, the plasma levels of VIP and normetanephrines returned to normal with resolution of diarrhea, hyponatremia, and hypertension. This case illustrates a rare instance of composite pheochromocytoma-ganglioneuroma with co-secretion of catecholamines and VIP, with evidence of VIP production in both tumor components. The finding of VIP secretion in the pheochromocytoma and the ganglioneuroma suggests that both tumors might originate from the same cell of neural crest origin.Learning points: ","PeriodicalId":37467,"journal":{"name":"Endocrinology, Diabetes and Metabolism Case Reports","volume":"2026 4","pages":""},"PeriodicalIF":0.9,"publicationDate":"2026-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13663621/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149115726","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Mitral valve regurgitation and emotional dysregulation secondary to long-standing Graves' thyrotoxicosis. 二尖瓣反流和情绪失调继发于长期格雷夫斯甲状腺毒症。

IF 0.9

Endocrinology, Diabetes and Metabolism Case Reports Pub Date : 2026-10-01 DOI: 10.1530/EDM-26-0023

Nadia Chaudhury, Brooke Puttergill, Masood Ashraf

{"title":"Mitral valve regurgitation and emotional dysregulation secondary to long-standing Graves' thyrotoxicosis.","authors":"Nadia Chaudhury, Brooke Puttergill, Masood Ashraf","doi":"10.1530/EDM-26-0023","DOIUrl":"10.1530/EDM-26-0023","url":null,"abstract":"Summary: Graves' disease is common, with an estimated prevalence of 0.2-2% and multiple associated complications. We present a case of severe mitral regurgitation secondary to poorly controlled Graves' disease for 11 years, highlighting the need for definitive treatment. A 30-year-old female attended hospital with palpitations, agitation and generalised pain. Her past medical history included Graves' disease diagnosed in 2014, with poor compliance and engagement with secondary care and medical treatment. Biochemical investigations and clinical examination confirmed thyrotoxicosis (TSH < 0.05 mIU/L, free T3 > 30.72 pmol/L and free T4 34.7 pmol/L). Carbimazole 40 mg once daily and propranolol 40 mg three times daily were restarted. She later developed worsening dyspnoea, attributed to decompensated heart failure. An echocardiogram showed severe mitral regurgitation secondary to anterior mitral valve prolapse. She also displayed psychiatric abnormalities, including anxiety and anger outbursts. Multiple conversations were thus needed with the patient, her family and the multidisciplinary team regarding treatment options, and an inpatient thyroidectomy was agreed upon. She recovered well postoperatively and is awaiting mitral valve repair. Our case highlights two crucial complications occurring secondary to long-standing uncontrolled Graves' thyrotoxicosis: mitral valve regurgitation with heart failure and neuropsychiatric complications with an impact on capacity for treatment decisions. We emphasise the complex support needed for these vulnerable patients to prevent end-stage complications of Graves' disease.Learning points: ","PeriodicalId":37467,"journal":{"name":"Endocrinology, Diabetes and Metabolism Case Reports","volume":"2026 4","pages":""},"PeriodicalIF":0.9,"publicationDate":"2026-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13663592/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149115785","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Osteoporosis associated with pregnancy and lactation (PLO): can REMS have a role in prevention and follow-up? 妊娠和哺乳期骨质疏松症（PLO）： REMS在预防和随访中是否有作用？

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Endocrinology, Diabetes and Metabolism Case Reports Pub Date : 2026-07-01 DOI: 10.1530/EDM-24-0102

Antonella Al Refaie, Caterina Mondillo, Leonardo Baldassini, Roberto Tarquini, Luigi Gennari, Stefano Gonnelli, Carla Caffarelli

引用次数: 0

Monoclonal insulin autoimmune syndrome in pregnancy and postpartum. 妊娠和产后单克隆胰岛素自身免疫性综合征。

IF 0.9

Endocrinology, Diabetes and Metabolism Case Reports Pub Date : 2026-07-01 DOI: 10.1530/EDM-26-0005

Anne Cathrine Parelius Wammer, Hanna Dis Margeirsdottir, Geir Erland Tjønnfjord, Ivar Følling

{"title":"Monoclonal insulin autoimmune syndrome in pregnancy and postpartum.","authors":"Anne Cathrine Parelius Wammer, Hanna Dis Margeirsdottir, Geir Erland Tjønnfjord, Ivar Følling","doi":"10.1530/EDM-26-0005","DOIUrl":"10.1530/EDM-26-0005","url":null,"abstract":"Summary: In insulin autoimmune syndrome (IAS), high levels of anti-insulin antibodies cause hypoglycemic episodes. Pregnancy in patients with IAS raises several concerns. The mother may experience glucose imbalances affecting the mother and fetus, transfer of antibodies may induce glucose disturbances in the fetus, and the antibodies may cause IAS in the newborn child. We present the first case of pregnancy and the postpartum period in monoclonal IAS, discuss pathogenetic mechanisms, and recommend how to handle future cases. A patient with monoclonal IAS was treated successfully with daratumumab to decrease anti-insulin antibodies from 1,840 U/mL (<0.4) to 180 U/mL. Treatment was paused to facilitate pregnancy, and after four months she conceived. Her antibody levels remained stable, and she was euglycemic during her pregnancy. The fetus grew at a normal rate. At delivery, the anti-insulin antibody level in the mother was 74 U/mL. The newborn had a level of 38 U/mL, which disappeared within 21 weeks. The child experienced no abnormal glucose fluctuations. The mother's IAS relapsed a few days postpartum, with hypoglycemia and anti-insulin antibody levels rising to 946 U/mL. Treatment with daratumumab was resumed efficaciously. The pregnancy went well for the mother, fetus, and child, probably mainly because of the relatively low levels of anti-insulin antibodies. We discuss possible explanations for the recurrence of IAS in the mother postpartum.Learning points: ","PeriodicalId":37467,"journal":{"name":"Endocrinology, Diabetes and Metabolism Case Reports","volume":"2026 3","pages":""},"PeriodicalIF":0.9,"publicationDate":"2026-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13617637/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149018139","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Amiodarone-induced thyrotoxicosis in a child with congenital heart disease. 先天性心脏病患儿胺碘酮致甲状腺毒症1例

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Endocrinology, Diabetes and Metabolism Case Reports Pub Date : 2026-07-01 DOI: 10.1530/EDM-26-0048

Pooja Karandikar, Rebecca Carter, Sudhir Vashist, Paula G Newton

引用次数: 0

Euglycaemic ketoacidosis associated with dual-acting GLP-1/GIP receptor agonist treatment following bariatric surgery. 减肥手术后双作用GLP-1/GIP受体激动剂治疗相关的血糖酮症酸中毒

IF 0.9

Endocrinology, Diabetes and Metabolism Case Reports Pub Date : 2026-07-01 DOI: 10.1530/EDM-26-0059

Deirdre Green, Nigel Glynn

{"title":"Euglycaemic ketoacidosis associated with dual-acting GLP-1/GIP receptor agonist treatment following bariatric surgery.","authors":"Deirdre Green, Nigel Glynn","doi":"10.1530/EDM-26-0059","DOIUrl":"10.1530/EDM-26-0059","url":null,"abstract":"Summary: Euglycaemic ketoacidosis is characterised by high anion gap metabolic acidosis with elevated serum ketone bodies in the absence of significant hyperglycaemia. Although traditionally associated with sodium-glucose cotransporter 2 (SGLT-2) inhibitors, incretin-based therapies have recently been implicated. Tirzepatide is a dual-acting glucagon-like peptide 1 (GLP-1)/glucose-dependent insulinotropic polypeptide (GIP) receptor agonist. It is an approved treatment for obesity in patients without diabetes mellitus. We present the case of a 51-year-old woman without diabetes, 2 years after sleeve gastrectomy, who presented with nausea, vomiting and abdominal pain after rapid dose escalation of tirzepatide. Laboratory evaluation revealed severe metabolic acidosis (pH 7.01, venous bicarbonate 9.2 mmol/L), elevated serum ketones (6 mmol/L) and normal blood glucose (5 mmol/L), consistent with euglycaemic ketoacidosis. Plasma lactate was normal (1.4 mmol/L). She was managed with intravenous fluids, insulin infusion and anti-emetics until the symptoms and acidosis resolved. Tirzepatide was discontinued. This case highlights the potential for dual-acting GLP-1/GIP receptor agonists to increase susceptibility to euglycaemic ketoacidosis in patients without diabetes, particularly in the context of prior bariatric surgery. Clinicians should consider euglycaemic ketoacidosis in patients receiving dual-acting GLP-1/GIP therapy who present unwell with metabolic acidosis.Learning points: ","PeriodicalId":37467,"journal":{"name":"Endocrinology, Diabetes and Metabolism Case Reports","volume":"2026 3","pages":""},"PeriodicalIF":0.9,"publicationDate":"2026-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13617635/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149018155","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Concurrent metformin-associated lactic acidosis and euglycemic diabetic ketoacidosis from tirzepatide starvation ketosis. 替西肽饥饿酮症并发二甲双胍相关乳酸酸中毒和血糖糖尿病酮症酸中毒。

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Endocrinology, Diabetes and Metabolism Case Reports Pub Date : 2026-07-01 DOI: 10.1530/EDM-26-0025

Frank Epitropoulos, Omar Brijawi, Khizar Ghuman, Will Van Beusekom, Inderpal Thethi

{"title":"Concurrent metformin-associated lactic acidosis and euglycemic diabetic ketoacidosis from tirzepatide starvation ketosis.","authors":"Frank Epitropoulos, Omar Brijawi, Khizar Ghuman, Will Van Beusekom, Inderpal Thethi","doi":"10.1530/EDM-26-0025","DOIUrl":"10.1530/EDM-26-0025","url":null,"abstract":"Summary: We describe a case of concurrent metformin-associated lactic acidosis (MALA) and euglycemic diabetic ketoacidosis (EDKA) in a 64-year-old woman with type 2 diabetes mellitus (T2DM) treated with metformin and tirzepatide who presented with shock, encephalopathy, and severe high anion gap metabolic acidosis after two weeks of markedly reduced oral intake following tirzepatide dose escalation. While incretin-based therapies, including dual glucose-dependent insulinotropic polypeptide/glucagon-like peptide-1 receptor agonists (GIP/GLP-1 RAs) such as tirzepatide, are highly effective for glycemic control and weight management, gastrointestinal (GI) adverse effects may lead to prolonged caloric restriction and starvation physiology. In this patient, starvation ketosis progressed to EDKA, while concurrent dehydration and acute kidney injury (AKI) impaired metformin clearance, resulting in MALA with markedly elevated lactate. The patient required endotracheal intubation, vasopressor support, and continuous renal replacement therapy (CRRT), with rapid clinical and metabolic improvement following discontinuation of metformin and tirzepatide. This case highlights the potential for severe overlapping metabolic complications involving starvation-related ketoacidosis, dehydration, AKI, and impaired metformin clearance when incretin-based therapies are continued during prolonged poor oral intake.Learning points: ","PeriodicalId":37467,"journal":{"name":"Endocrinology, Diabetes and Metabolism Case Reports","volume":"2026 3","pages":""},"PeriodicalIF":0.9,"publicationDate":"2026-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13640942/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149064973","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Significantly elevated calcium levels and gallstone disease in a patient with familial hypocalciuric hypercalcaemia. 家族性低钙性高钙血症患者钙水平显著升高与胆结石疾病

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Endocrinology, Diabetes and Metabolism Case Reports Pub Date : 2026-07-01 DOI: 10.1530/EDM-26-0022

Adam Sabo, Andrew McGovern

{"title":"Significantly elevated calcium levels and gallstone disease in a patient with familial hypocalciuric hypercalcaemia.","authors":"Adam Sabo, Andrew McGovern","doi":"10.1530/EDM-26-0022","DOIUrl":"10.1530/EDM-26-0022","url":null,"abstract":"Summary: Familial hypocalciuric hypercalcaemia (FHH) is a rare, benign genetic disorder of calcium homeostasis caused by inactivating variants in CaSR, GNA11, or AP2S1 genes. It is typically characterised by mild, asymptomatic hypercalcaemia and low urinary calcium excretion. Differentiation from primary hyperparathyroidism (PHPT) is essential, as misdiagnosis can lead to unnecessary surgery. We report a case of FHH type 1 (FHH1) in a man in his thirties presenting with a serum calcium level of 2.97 mmol/L, which is at the severe end of the FHH spectrum. Genetic testing revealed a heterozygous CaSR nonsense variant (NM_000388.4:c.1942C>T, p.Arg648Ter) consistent with FHH1. The patient had clinically stable hypercalcaemia, which was not treated. During follow-up, he developed gallstones treated with a laparoscopic cholecystectomy. A review of the literature demonstrated that calcium levels approaching 3 mmol/L, although uncommon, have been reported in other cases of FHH1. We review the literature regarding a potential association between FHH and gallstones. This case highlights that FHH can present with relatively marked hypercalcaemia, reinforcing the need for genetic evaluation before surgery, and suggests a possible but under-explored relationship between FHH and gallstone disease.Learning points: ","PeriodicalId":37467,"journal":{"name":"Endocrinology, Diabetes and Metabolism Case Reports","volume":"2026 3","pages":""},"PeriodicalIF":0.9,"publicationDate":"2026-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13588474/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148913409","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Arginine vasopressin deficiency following SARS-CoV-2 infection: a rare pituitary complication. SARS-CoV-2感染后精氨酸加压素缺乏：一种罕见的垂体并发症

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Endocrinology, Diabetes and Metabolism Case Reports Pub Date : 2026-07-01 DOI: 10.1530/EDM-26-0006

Pascoe Mannion, Edward Jude

引用次数: 0