AACE Clinical Case Reports最新文献

英文中文

Case Report: IgG4-Related Disease Presenting With Isolated Hypophysitis 病例报告：表现为孤立性肾上腺皮质功能减退症的 IgG4 相关疾病

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-09-01 DOI: 10.1016/j.aace.2024.07.004

Suhaib Radi MD , Michael Tamilia FRCPC

Background/Objective

IgG4-related disease (IgG4-RD) is an immune-mediated condition that affects multiple organs, including the pituitary gland. Here we present a patient with isolated pituitary involvement of IgG4-RD mimicking pituitary apoplexy.

Case Report

A 49-year-old woman presented to the emergency department with abdominal pain, nausea, vomiting, and weight loss. Her blood pressure was low, and she appeared euvolemic with the rest of physical examination being noncontributory. Her electrolytes showed low serum sodium of 118 mmol/L (normal 135-145). Further investigations were significant for low morning cortisol of 20 nmol/L (N:100-500) and low adrenocorticotropic hormone. Magnetic resonant imaging of the pituitary fossa showed a pituitary macroadenoma with hemorrhagic transformation. She was started on glucocorticoids and levothyroxine before undergoing surgical removal of the pituitary tumor. The pathology was positive for IgG-4-related hypophysitis (IgG4-RH) with no evidence of pituitary tumor.

Discussion

IgG4-RD is an immune-mediated condition that can affect many organs including the pituitary gland, in the form of hypophysitis. IgG4-RH can affect anterior, posterior, or both pituitary lobes. In 2011, Leporati et al developed a diagnostic criteria for IgG4-RH which includes the following: imaging, serology, histopathology, and response t glucocorticoids. The mainstay of treatment is glucocorticoids and hormone replacement therapy.

Conclusion

IgG4-RH might be underestimated and should be suspected in those with hypophysitis or unknown cause of hypopituitarism. Moreover, pituitary macroadenoma with hemorrhagic transformation and panhypopituitarism should be considered as rare and unusual presentations of IgG4-RD.

背景/目的IgG4相关疾病（IgG4-RD）是一种免疫介导的疾病，可影响包括垂体在内的多个器官。病例报告一名 49 岁女性因腹痛、恶心、呕吐和体重减轻到急诊科就诊。她的血压很低，看起来没有血容量，其他体格检查也无异常。她的电解质显示血清钠偏低，为 118 毫摩尔/升（正常值为 135-145 毫摩尔/升）。进一步检查显示，她的晨间皮质醇偏低，为 20 nmol/L（正常值：100-500），促肾上腺皮质激素也偏低。垂体窝的磁共振成像显示垂体大腺瘤伴出血性转化。她开始服用糖皮质激素和左甲状腺素，然后接受了垂体瘤手术切除。讨论IgG4-RD是一种免疫介导的疾病，可以影响包括垂体在内的许多器官，表现为垂体功能减退症。IgG4-RH 可影响垂体前叶、后叶或双侧垂体。2011 年，Leporati 等人制定了 IgG4-RH 的诊断标准，其中包括：影像学、血清学、组织病理学和对糖皮质激素的反应。结论IgG4-RH可能被低估，应怀疑患有垂体功能减退症或原因不明的垂体功能减退症。此外，垂体大腺瘤伴出血性转化和泛垂体功能减退症应被视为IgG4-RD罕见的异常表现。

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引用次数: 0

Exaggerated Increases in the Serum Cortisol Level in a Woman Following Oral Contraceptive Treatment 一名妇女口服避孕药后血清皮质醇的过度升高

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-09-01 DOI: 10.1016/j.aace.2024.07.005

Run Yu MD, PhD

Background/Objective

Extreme hypercortisolemia in an otherwise healthy patient can be due to familial dysalbuminemia, generalized glucocorticoid resistance, and estrogen-containing medications. I report a woman who appeared to have an exaggerated increase in the serum cortisol level following oral contraceptive treatment.

Case Report

A 50-year-old woman presented with extreme morning hypercortisolemia—cortisol levels of 61 and 55 mcg/dL (4 and 3 months before presentation, respectively; normal range, 8-25 mcg/dL)—found during workup of mildly increased white cell counts. The morning cortisol level had been 10 mcg/dL after administration of 1-mg dexamethasone. The 24-hour urine free cortisol level had been normal and only slightly increased after correction by creatinine. The patient was anxious about the extremely high cortisol levels but otherwise felt well. She took norgestimate-ethinyl estradiol contraceptive (0.18/0.215/0.25 mg - 35 mcg). Physical examination showed a well-appearing, lean female. The thyroid-stimulating hormone, total thyroxine, free thyroxine, total triiodothyronine, free triiodothyronine, androstenedione, dehydroepiandrosterone sulfate, aldosterone, and renin levels were normal. Morning total cortisol and cortisol-binding globulin (CBG) were tested before and after she held the oral contraceptive for 2 months. The total cortisol and CBG levels decreased from 50 to 26 mcg/dL and from 6.4 to 3.8 mg/dL (normal range, 1.7-3.1 mg/dL), respectively.

Discussion

Increases in the serum cortisol-binding proteins are a well-recognized cause for increases in the serum cortisol levels.

Conclusion

This case suggests that modern oral contraceptives with low to moderate estrogen activity can cause extreme increases in the serum cortisol levels due to marked increases in the CBG levels.

背景/目的原本健康的患者出现极度高皮质醇血症可能是由于家族性白蛋白血症、全身性糖皮质激素抵抗和含有雌激素的药物所致。病例报告一位 50 岁的女性患者在接受口服避孕药治疗后出现血清皮质醇水平的夸张性升高--皮质醇水平分别为 61 和 55 mcg/dL（发病前 4 个月和 3 个月；正常范围为 8-25 mcg/dL）--这是在白细胞计数轻度升高的检查中发现的。在服用1毫克地塞米松后，晨间皮质醇水平为10微克/分升。24 小时尿液游离皮质醇水平正常，经肌酐校正后仅略有升高。患者对皮质醇水平过高感到焦虑，但其他方面感觉良好。她服用了炔诺酮-乙炔雌二醇避孕药（0.18/0.215/0.25 毫克 - 35 微克）。体格检查显示，她是一名面容姣好的瘦弱女性。促甲状腺激素、总甲状腺素、游离甲状腺素、总三碘甲状腺原氨酸、游离三碘甲状腺原氨酸、雄烯二酮、硫酸脱氢表雄酮、醛固酮和肾素水平正常。在她口服避孕药 2 个月之前和之后，检测了晨间总皮质醇和皮质醇结合球蛋白（CBG）。总皮质醇和 CBG 水平分别从 50 毫微克/分升降至 26 毫微克/分升，从 6.4 毫克/分升降至 3.8 毫克/分升（正常范围为 1.7-3.1 毫克/分升）。讨论血清皮质醇结合蛋白的增加是导致血清皮质醇水平升高的公认原因。

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引用次数: 0

Stiff Person Syndrome and Brittle Type 1 Diabetes: Report of 2 Cases 僵人综合征和脆性 1 型糖尿病。两个病例的报告

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-09-01 DOI: 10.1016/j.aace.2024.07.003

Ismael A. Quintal-Medina MD , Francisco J. Gómez-Pérez MD , Paloma Almeda-Valdes MD, PhD

Background/Objective

Stiff person syndrome (SPS) and type 1 diabetes (T1D) are heterogeneous disorders characterized by antibodies (Abs) against glutamic acid decarboxylase (GAD).

Case Report

We describe 2 patients with T1D and autoimmune thyroid disease who presented with muscle rigidity and intermittent spasms that affected gait and with elevated circulating anti-GAD titers. Classic SPS and stiff limb syndrome were diagnosed, respectively. Muscle spasms resolved with immunotherapy and muscle relaxants in both patients, and the ability to ambulate without an assistive device was restored in 1 patient. Patients also had brittle diabetes with high glycemic variability, requiring the use of flash glucose monitoring with an insulin pump and a second-generation basal insulin analog, respectively.

Discussion

GAD Ab–associated syndromes include SPS, T1D, and other endocrinopathies. The clinical heterogeneity implies variable susceptibility of γ-aminobutyric acid-ergic neurons and pancreatic beta cells to anti-GAD or other autoantibodies.

Conclusion

Our case series represent the heterogeneity in natural history, clinical course, and response to therapy in patients with Abs against GAD-spectrum disorders.

背景/目的僵硬综合征（SPS）和1型糖尿病（T1D）是以谷氨酸脱羧酶（GAD）抗体（Abs）为特征的异质性疾病。病例报告我们描述了2例患有T1D和自身免疫性甲状腺疾病的患者，他们表现为肌肉僵硬和间歇性痉挛，影响步态，循环中抗GAD滴度升高。他们分别被诊断为典型的 SPS 和僵硬肢体综合征。通过免疫疗法和肌肉松弛剂，两名患者的肌肉痉挛都得到了缓解，其中一名患者在没有辅助设备的情况下恢复了行走能力。患者还患有血糖变异性高的脆性糖尿病，需要分别使用胰岛素泵和第二代基础胰岛素类似物进行闪光血糖监测。临床异质性意味着γ-氨基丁酸能神经元和胰岛β细胞对抗GAD或其他自身抗体的易感性各不相同。

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引用次数: 0

Type 1 Diabetes Mellitus Caused by COVID-19 mRNA Vaccination: A Case Report and Literature Review of 17 Published Cases 接种 COVID-19 mRNA 疫苗导致的 1 型糖尿病：病例报告和已发表 17 例病例的文献综述

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-09-01 DOI: 10.1016/j.aace.2024.06.001

Background/Objective

Multiple cases of postvaccination immune-related adverse events have been reported. We, hereby, present a patient who presented with new-onset type 1 diabetes mellitus (DM) after COVID-19 messenger RNA (mRNA) vaccination.

Case Report

A 38-year-old Caucasian man presented with sudden onset of polyuria, polydipsia, and blurry vision for 1 month. The patient received the second dose of the COVID-19 mRNA vaccine (Pfizer-BioNTech) 4 weeks prior to symptom onset. Initial workup revealed glucosuria and hemoglobin A1c of 9.4%. Antibodies against multiple pancreatic beta cell autoantigens were detected. The patient was then initiated on insulin.

Discussion

Hypothesized mechanisms for development of type 1 DM after COVID-19 mRNA vaccination include molecular mimicry, autoimmune/inflammatory syndrome induced by adjuvants, and possible interaction between the angiotensin-I converting enzyme-2 receptor on beta cells and viral mRNA. An initial high index of suspicion should be accompanied by early autoantibody testing and initiation of insulin, if indicated. Finally, if diagnosed with type 1 diabetes, patients must have long-term follow-up as there may be brief periods where glycemic control is maintained off insulin.

Conclusion

New-onset type 1 DM has been reported after COVID mRNA vaccination. Clinicians should maintain a high index of suspicion and pursue early testing for the same to reduce adverse outcomes and improve long-term prognosis.

背景/目的已有多例疫苗接种后免疫相关不良事件的报道。病例报告：一名 38 岁的白种男子在接种 COVID-19 信使 RNA（mRNA）疫苗后突然出现多尿、多饮和视力模糊，持续 1 个月。患者在发病前 4 周接种了第二剂 COVID-19 mRNA 疫苗（辉瑞生物技术公司）。初步检查显示患者有葡萄糖尿，血红蛋白 A1c 为 9.4%。检测到多种胰腺β细胞自身抗原抗体。讨论COVID-19 mRNA疫苗接种后发生1型糖尿病的假设机制包括分子模拟、佐剂诱导的自身免疫/炎症综合征以及β细胞上的血管紧张素-I转换酶-2受体与病毒mRNA之间可能存在的相互作用。在最初高度怀疑的同时，应尽早进行自身抗体检测，并在必要时开始使用胰岛素。最后，如果确诊为 1 型糖尿病，患者必须接受长期随访，因为可能会出现短暂的血糖控制不依赖胰岛素的情况。临床医生应保持高度怀疑，并尽早进行相关检测，以减少不良后果并改善长期预后。

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引用次数: 0

Multifocal Nonmetastatic Radioactive Iodine Avidity on Whole Body Scan After Thyroidectomy for Thyroid Cancer 甲状腺癌甲状腺切除术后全身扫描显示多灶性非转移性放射性碘惰性

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-09-01 DOI: 10.1016/j.aace.2024.07.001

Jim T.C. Chen MD , Kirun Baweja MbChB , Lurdes Tse-Agha MD , Sara Awad MBBS, MHPE, FRCPC

Background/Objective

Non-metastatic radioactive iodine (RAI) uptake can complicate the interpretation of whole-body scan (WBS) for differentiated thyroid carcinoma (DTC) post-thyroidectomy. We present a patient with DTC whose follow-up WBS showed nonmetastatic multifocal avidity in skeletal tissue, an uncommonly reported site of RAI uptake.

Case report

A 42-year-old woman underwent a right hemithyroidectomy, followed by completion thyroidectomy and RAI remnant ablation therapy, for a 4.8 cm thyroid tumor consistent with stage pT3aNxMx follicular thyroid cancer. Follow-up WBS showed intense activity in the thyroid bed, right breast, left medial subcortical acetabulum, and several vertebral bodies. Her biochemical and clinical findings were not suggestive of cancer recurrence. Further workup with SPECT/CT and MRI showed no focal vertebral lesions and identified the left femoral lesion as a benign peripheral nerve sheath. Diagnostic mammography and ultrasound showed no evidence of suspicious breast lesions. Neck ultrasound was clear with no suspicious masses or pathologic lymphadenopathy. She remained in remission on continued active surveillance.

Discussion

Nonmetastatic RAI uptake on WBS has many causes, including functional sodium-iodide symporter expression in nonthyroidal tissues, radioiodine accumulation in tissues and bodily fluids, and benign tumors. False-positive uptake can decrease the utility of post-treatment WBS in low-risk patients. Careful clinical examination, biochemical and radiologic follow-up, and close active surveillance can help distinguish false-positive uptake from metastatic or recurrent disease.

Conclusion

We describe an uncommon case of RAI uptake in skeletal tissues after thyroidectomy for DTC, and we outline the steps taken to rule out underlying metastases.

背景/目的甲状腺切除术后分化型甲状腺癌（DTC）的全身扫描（WBS）结果显示，非转移性放射性碘（RAI）摄取可能会使判读复杂化。病例报告：一名 42 岁女性因患 4.8 厘米甲状腺肿瘤（符合 pT3aNxMx 滤泡型甲状腺癌分期）接受了右半甲状腺切除术，随后进行了甲状腺全切术和 RAI 残余消融治疗。随访WBS显示，甲状腺床、右乳房、左侧皮质下髋臼内侧和多个椎体有强烈活动。她的生化和临床检查结果均未提示癌症复发。进一步的 SPECT/CT 和 MRI 检查显示没有椎体病灶，并确定左股骨病变为良性周围神经鞘。诊断性乳房 X 线照相术和超声波检查没有发现可疑的乳房病变。颈部超声检查结果清晰，未发现可疑肿块或病理性淋巴结病变。讨论WBS上的非转移性RAI摄取有多种原因，包括非甲状腺组织中钠离子-碘离子交感器的功能表达、组织和体液中的放射性碘蓄积以及良性肿瘤。假阳性摄取会降低低风险患者治疗后 WBS 的效用。仔细的临床检查、生化和放射学随访以及密切的主动监测有助于将假阳性摄取与转移性或复发性疾病区分开来。结论我们描述了一例不常见的因 DTC 而行甲状腺切除术后骨骼组织摄取 RAI 的病例，并概述了为排除潜在转移瘤而采取的措施。

{"title":"Multifocal Nonmetastatic Radioactive Iodine Avidity on Whole Body Scan After Thyroidectomy for Thyroid Cancer","authors":"Jim T.C. Chen MD , Kirun Baweja MbChB , Lurdes Tse-Agha MD , Sara Awad MBBS, MHPE, FRCPC","doi":"10.1016/j.aace.2024.07.001","DOIUrl":"10.1016/j.aace.2024.07.001","url":null,"abstract":"<div><h3>Background/Objective</h3><p>Non-metastatic radioactive iodine (RAI) uptake can complicate the interpretation of whole-body scan (WBS) for differentiated thyroid carcinoma (DTC) post-thyroidectomy. We present a patient with DTC whose follow-up WBS showed nonmetastatic multifocal avidity in skeletal tissue, an uncommonly reported site of RAI uptake.</p></div><div><h3>Case report</h3><p>A 42-year-old woman underwent a right hemithyroidectomy, followed by completion thyroidectomy and RAI remnant ablation therapy, for a 4.8 cm thyroid tumor consistent with stage pT3aNxMx follicular thyroid cancer. Follow-up WBS showed intense activity in the thyroid bed, right breast, left medial subcortical acetabulum, and several vertebral bodies. Her biochemical and clinical findings were not suggestive of cancer recurrence. Further workup with SPECT/CT and MRI showed no focal vertebral lesions and identified the left femoral lesion as a benign peripheral nerve sheath. Diagnostic mammography and ultrasound showed no evidence of suspicious breast lesions. Neck ultrasound was clear with no suspicious masses or pathologic lymphadenopathy. She remained in remission on continued active surveillance.</p></div><div><h3>Discussion</h3><p>Nonmetastatic RAI uptake on WBS has many causes, including functional sodium-iodide symporter expression in nonthyroidal tissues, radioiodine accumulation in tissues and bodily fluids, and benign tumors. False-positive uptake can decrease the utility of post-treatment WBS in low-risk patients. Careful clinical examination, biochemical and radiologic follow-up, and close active surveillance can help distinguish false-positive uptake from metastatic or recurrent disease.</p></div><div><h3>Conclusion</h3><p>We describe an uncommon case of RAI uptake in skeletal tissues after thyroidectomy for DTC, and we outline the steps taken to rule out underlying metastases.</p></div>","PeriodicalId":7051,"journal":{"name":"AACE Clinical Case Reports","volume":"10 5","pages":"Pages 188-192"},"PeriodicalIF":0.0,"publicationDate":"2024-09-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S237606052400066X/pdfft?md5=0274fd2526d98e6943328becccdd363e&pid=1-s2.0-S237606052400066X-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141694197","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

Skin Rash Appearing in a Patient with Gestational Diabetes Mellitus on Insulin Therapy 接受胰岛素治疗的妊娠糖尿病患者出现皮疹

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-09-01 DOI: 10.1016/j.aace.2024.04.004

引用次数: 0

Gender-Affirming Hormone Therapy With Estrogen Causing Gallstone Associated Acute Pancreatitis 使用雌激素的性别平权激素疗法导致胆石性急性胰腺炎

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-09-01 DOI: 10.1016/j.aace.2024.07.006

Tatiana Tselovalnikova MD, PhD, Ifrah Fatima MD, Brandon Barthel MD

Background/Objective

Although estrogen is one of the main agents used to treat transgender women, there are few reports of acute pancreatitis (AP) of this illness in this group. The objective of this report is to describe a transgender woman who developed AP in the setting of estrogen treatment and gallstone disease.

Case Report

A 38-year-old transgender woman presented with severe abdominal pain and vomiting. Her medical history included gender dysphoria managed with gender-affirming hormone therapy comprising estradiol valerate, progesterone, and spironolactone. Initial management involved supportive care, antibiotic therapy, and endoscopic retrograde cholangiopancreatography with biliary stent placement. Imaging confirmed acute interstitial edematous pancreatitis without necrosis, guiding treatment decisions toward laparoscopic cholecystectomy. Pathological examination revealed multiple gallstones, affirming the diagnosis of AP secondary to choledocholithiasis, likely associated with estrogen use. Postprocedural recovery was uneventful, with eventual removal of the biliary stent and resolution of symptoms.

Discussion

There are only 7 reported cases in literature on estrogen-induced AP in transgender individuals undergoing gender-affirming hormone therapy. Most of these were primarily linked to hypertriglyceridemia.

Conclusion

High-dose estrogen therapy in transgender women can elevate the risk of AP through the development of gallstones, underscoring the importance of thorough patient evaluation and discussion of risks assessment prior to initiating hormone therapy.

背景/目的虽然雌激素是用于治疗变性女性的主要药物之一，但很少有关于该群体急性胰腺炎（AP）的报道。病例报告一名 38 岁的变性女性因剧烈腹痛和呕吐就诊。她的病史包括性别确认激素治疗（包括戊酸雌二醇、黄体酮和螺内酯）导致的性别焦虑症。最初的治疗包括支持性护理、抗生素治疗、内镜逆行胰胆管造影术和胆道支架置入术。影像学检查证实患者患有急性间质性水肿性胰腺炎，但没有坏死，这为腹腔镜胆囊切除术的治疗提供了指导。病理检查发现多发性胆结石，确诊为继发于胆总管结石的急性间质性水肿性胰腺炎，可能与使用雌激素有关。术后恢复顺利，最终拆除了胆道支架，症状也得到了缓解。讨论目前仅有7例文献报道了接受性别确认激素治疗的变性人因雌激素引起的 AP。结论变性女性接受大剂量雌激素治疗可能会因胆结石的发生而增加患 AP 的风险，这强调了在开始激素治疗前对患者进行全面评估并讨论风险评估的重要性。

{"title":"Gender-Affirming Hormone Therapy With Estrogen Causing Gallstone Associated Acute Pancreatitis","authors":"Tatiana Tselovalnikova MD, PhD, Ifrah Fatima MD, Brandon Barthel MD","doi":"10.1016/j.aace.2024.07.006","DOIUrl":"10.1016/j.aace.2024.07.006","url":null,"abstract":"<div><h3>Background/Objective</h3><p>Although estrogen is one of the main agents used to treat transgender women, there are few reports of acute pancreatitis (AP) of this illness in this group. The objective of this report is to describe a transgender woman who developed AP in the setting of estrogen treatment and gallstone disease.</p></div><div><h3>Case Report</h3><p>A 38-year-old transgender woman presented with severe abdominal pain and vomiting. Her medical history included gender dysphoria managed with gender-affirming hormone therapy comprising estradiol valerate, progesterone, and spironolactone. Initial management involved supportive care, antibiotic therapy, and endoscopic retrograde cholangiopancreatography with biliary stent placement. Imaging confirmed acute interstitial edematous pancreatitis without necrosis, guiding treatment decisions toward laparoscopic cholecystectomy. Pathological examination revealed multiple gallstones, affirming the diagnosis of AP secondary to choledocholithiasis, likely associated with estrogen use. Postprocedural recovery was uneventful, with eventual removal of the biliary stent and resolution of symptoms.</p></div><div><h3>Discussion</h3><p>There are only 7 reported cases in literature on estrogen-induced AP in transgender individuals undergoing gender-affirming hormone therapy. Most of these were primarily linked to hypertriglyceridemia.</p></div><div><h3>Conclusion</h3><p>High-dose estrogen therapy in transgender women can elevate the risk of AP through the development of gallstones, underscoring the importance of thorough patient evaluation and discussion of risks assessment prior to initiating hormone therapy.</p></div>","PeriodicalId":7051,"journal":{"name":"AACE Clinical Case Reports","volume":"10 5","pages":"Pages 210-213"},"PeriodicalIF":0.0,"publicationDate":"2024-09-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2376060524000701/pdfft?md5=79dc08d4d7897ce11f8d27812971008d&pid=1-s2.0-S2376060524000701-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141852661","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

Editorial for September/October Issue of AACE Clinical Case Reports AACE 临床病例报告》9/10 月刊社论

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-09-01 DOI: 10.1016/j.aace.2024.08.005

Sina Jasim MD, MPH (Editor in Chief)

引用次数: 0

Parathyroid Hormone–Related Peptide Secretion From a Pancreatic Neuroendocrine Tumor: A Rare Case Report of Severe Hypercalcemia 胰腺神经内分泌肿瘤分泌甲状旁腺激素相关肽。严重高钙血症的罕见病例报告。

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-07-01 DOI: 10.1016/j.aace.2024.04.009

Erin Foley MBBS, Prashanth Hari Dass MBBS, Esther O’Sullivan MBBS, PhD

Background/Objective

Hypercalcemia is a common occurrence associated with malignancy, due to a number of causes: (1) lytic bone metastases, (2) production of 1,25-dihydroxyvitamin D from lymphoma, and (3) parathyroid hormone–related peptide (PTHrP) secretion usually from solid tumors.

Case Report

A 56-year-old woman presented with symptoms of severe hypercalcemia. Investigations determined that this was due to PTHrP secretion from a pancreatic neuroendocrine tumor (pNET), a noted complication in 1.1% of pNET cases. Although unfit for curative therapy, the patient was treated with fluid replacement, bisphosphonates, calcitonin, and denosumab. After treatment, she had recurrent severe symptomatic hypercalcemia on several occasions despite adjunctive therapy with a somatostatin analog. Ultimately, the patient died as a result of refractory hypercalcemia.

Discussion

The hypercalcemia that is rarely associated with PTHrP secretion from pNETs is aggressive and often refractory to the usual medical treatment of hypercalcemia of malignancy. Effective treatment requires cytoreduction of the causative tumor. Denosumab, a receptor activator of nuclear factor kappa beta ligand inhibitor, has proven useful in some cases.

Conclusion

This challenging case highlighted the rare but potentially fatal association of pNET with hypercalcemia. Hypercalcemia was the main cause of mortality in an otherwise relatively indolent malignancy.

背景/目的高钙血症是一种常见的恶性肿瘤并发症，其原因有很多：（1）淋巴瘤骨转移；（2）淋巴瘤产生 1,25-二羟维生素 D；（3）甲状旁腺激素相关肽（PTHrP）分泌，通常来自实体瘤。经检查确定，这是由于胰腺神经内分泌肿瘤（pNET）分泌 PTHrP 所致。虽然不适合接受根治性治疗，但患者接受了液体补充、双磷酸盐、降钙素和地诺单抗治疗。治疗后，尽管使用了体生长抑素类似物辅助治疗，她仍多次反复出现严重的症状性高钙血症。讨论pNET分泌的PTHrP很少伴有高钙血症，这种高钙血症具有侵袭性，通常对恶性肿瘤高钙血症的常规药物治疗无效。有效的治疗需要对致病肿瘤进行细胞减灭术。在某些病例中，核因子卡巴受体激活剂β配体抑制剂地诺单抗（Denosumab）被证明是有用的。高钙血症是导致这种相对不严重的恶性肿瘤死亡的主要原因。

{"title":"Parathyroid Hormone–Related Peptide Secretion From a Pancreatic Neuroendocrine Tumor: A Rare Case Report of Severe Hypercalcemia","authors":"Erin Foley MBBS, Prashanth Hari Dass MBBS, Esther O’Sullivan MBBS, PhD","doi":"10.1016/j.aace.2024.04.009","DOIUrl":"10.1016/j.aace.2024.04.009","url":null,"abstract":"<div><h3>Background/Objective</h3><p>Hypercalcemia is a common occurrence associated with malignancy, due to a number of causes: (1) lytic bone metastases, (2) production of 1,25-dihydroxyvitamin D from lymphoma, and (3) parathyroid hormone–related peptide (PTHrP) secretion usually from solid tumors.</p></div><div><h3>Case Report</h3><p>A 56-year-old woman presented with symptoms of severe hypercalcemia. Investigations determined that this was due to PTHrP secretion from a pancreatic neuroendocrine tumor (pNET), a noted complication in 1.1% of pNET cases. Although unfit for curative therapy, the patient was treated with fluid replacement, bisphosphonates, calcitonin, and denosumab. After treatment, she had recurrent severe symptomatic hypercalcemia on several occasions despite adjunctive therapy with a somatostatin analog. Ultimately, the patient died as a result of refractory hypercalcemia.</p></div><div><h3>Discussion</h3><p>The hypercalcemia that is rarely associated with PTHrP secretion from pNETs is aggressive and often refractory to the usual medical treatment of hypercalcemia of malignancy. Effective treatment requires cytoreduction of the causative tumor. Denosumab, a receptor activator of nuclear factor kappa beta ligand inhibitor, has proven useful in some cases.</p></div><div><h3>Conclusion</h3><p>This challenging case highlighted the rare but potentially fatal association of pNET with hypercalcemia. Hypercalcemia was the main cause of mortality in an otherwise relatively indolent malignancy.</p></div>","PeriodicalId":7051,"journal":{"name":"AACE Clinical Case Reports","volume":"10 4","pages":"Pages 160-163"},"PeriodicalIF":0.0,"publicationDate":"2024-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2376060524000452/pdfft?md5=11bef7d4a927d87d5ab8aa84546b1854&pid=1-s2.0-S2376060524000452-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141041108","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

Unusual Case of Adrenal Artery Aneurysm: Diagnosis and Management 肾上腺动脉瘤的罕见病例--诊断与治疗

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-07-01 DOI: 10.1016/j.aace.2024.03.004

Omolade O. Sogade MD , C. Corbin Frye MD , Daniel Picus MD , Sina Jasim MD, MPH , Taylor C. Brown MD, MHS

引用次数: 0

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