AACE Clinical Case Reports最新文献

Improvement of Glycogenic Hepatopathy With Minimal Corresponding Improvement of Glycemic Control in a Person With Type 1 Diabetes: Case Report and Literature Review 1型糖尿病患者糖原性肝病的改善与相应的血糖控制改善：病例报告和文献回顾

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2025-03-01 DOI: 10.1016/j.aace.2024.12.004

Joselyn Tessa Tonleu BS , Nicolas A. Reyes MD , Natalie D. Hillerson MD , William B. Horton MD, MSc, FACP

Background/Objective

Glycogenic hepatopathy is characterized by diffuse glycogen accumulation in hepatocytes that leads to hepatomegaly and elevated transaminases. Notably, the condition is reversible as improving glycemic control has been shown to resolve glycogenic hepatopathy and provide symptomatic relief.

Case Report

A 30-year-old female with longstanding and poorly-controlled type 1 diabetes presented to her primary care physician for a routine follow-up visit. Routine lab work demonstrated hyperglycemia and elevated liver enzymes (alkaline phosphatase of 180 U/L, aspartate aminotransferase of 111 U/L, and alanine aminotransferase of 101 U/L). At laboratory reassessment 3 weeks later, liver function tests remained elevated and hepatic ultrasound was unrevealing. She was referred to gastroenterology for further evaluation and laboratory tests for viral and autoimmune hepatitis were negative while magnetic resonance imaging of the abdomen was unremarkable. Given the nondiagnostic work-up, liver biopsy was performed and pathology was consistent with glycogenic hepatopathy. She was referred to Endocrinology for improved glycemic control; however, liver enzymes normalized over the next several months despite minimal improvement in glycemic control. She was eventually transitioned to a closed-loop automated insulin delivery system and started dulaglutide for management of obesity. Subsequent A1c values significantly improved and liver enzymes remained within normal limits.

Discussion

This case raises awareness of an under recognized complication of type 1 diabetes and challenges conventional thinking about factors leading to its resolution.

Conclusion

Further investigation into the underlying pathophysiology of glycogenic hepatopathy is needed.

背景/目的糖原性肝病的特点是肝细胞弥漫性糖原积聚，导致肝肿大和转氨酶升高。值得注意的是，这种情况是可逆的，因为改善血糖控制已被证明可以解决糖原性肝病并提供症状缓解。病例报告一名患有长期控制不良的1型糖尿病的30岁女性向她的初级保健医生进行常规随访。常规实验室检查显示高血糖和肝酶升高（碱性磷酸酶180 U/L，天冬氨酸转氨酶111 U/L，丙氨酸转氨酶101 U/L）。3周后实验室复查，肝功能检查仍升高，肝脏超声未见异常。她被转到胃肠病学做进一步的评估，病毒性肝炎和自身免疫性肝炎的实验室检查呈阴性，而腹部磁共振成像无明显异常。鉴于非诊断性检查，进行了肝活检，病理符合糖原性肝病。她被转到内分泌科以改善血糖控制；然而，在接下来的几个月里，肝酶恢复正常，尽管血糖控制的改善很小。她最终转为使用闭环自动胰岛素输送系统，并开始使用杜拉鲁肽治疗肥胖。随后的A1c值显著改善，肝酶保持在正常范围内。本病例提高了人们对1型糖尿病并发症的认识，并对导致其解决的因素的传统思维提出了挑战。结论糖原性肝病的病理生理机制有待进一步研究。

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引用次数: 0

A Woman With Persistent Abdominal Pain in the Setting of a Solitary Fibrous Adrenal Tumor 女性单发肾上腺纤维性肿瘤并发持续性腹痛1例

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2025-03-01 DOI: 10.1016/j.aace.2024.12.012

Samuel Amankwah MD , Maria Luzuriaga MD , Krista Denning MD , Logan Lawrence MS

Background/Objective

We present a case of a woman with persistent abdominal pain, leading to the discovery of a rare solitary fibrous tumor (SFT) in the adrenal gland. The rarity of adrenal SFTs and their diagnostic challenges make this case noteworthy. The objective of this report is to describe a patient with an adrenal SFT, highlighting the unique diagnostic approach and management considerations for this rare condition.

Case Report

A 35-year-old woman presented with chronic abdominal pain and was referred to the endocrinology clinic for evaluation of an adrenal incidentaloma. Computed tomography imaging revealed a 4 cm homogeneous right adrenal mass, with precontrast Hounsfield units of 1 and an absolute contrast washout of 60%. A prior computed tomography scan from 6 years earlier showed a 1.5 cm adenoma with similar characteristics. Despite normal hormonal levels, the tumor's growth and the patient's symptoms prompted surgical referral. The patient underwent robotic-assisted laparoscopic right adrenalectomy. Pathological examination identified a well-circumscribed SFT, measuring 3.7 × 3.6 × 2.9 cm.

Discussion

Adrenal SFTs are rare, typically hormonally inactive, well-circumscribed masses that often present with abdominal pain. While more common in the pleura, adrenal SFTs can mimic other benign adrenal lesions on imaging, complicating diagnosis. Histopathology is essential for accurate diagnosis, and surgical resection remains the main treatment.

Conclusion

This case highlights the diagnostic challenges of adrenal SFTs, which can mimic other benign lesions. Despite significant growth, the tumor was histologically benign with low malignancy risk. Clinicians should consider adrenal SFTs in the differential diagnosis of incidental adrenal masses with atypical imaging features.

背景/目的我们报告一位持续腹痛的女性患者，在肾上腺中发现罕见的孤立性纤维性肿瘤（SFT）。肾上腺SFTs的罕见性及其诊断挑战使本病例值得注意。本报告的目的是描述一个患者与肾上腺SFT，突出独特的诊断方法和管理的考虑，这种罕见的条件。病例报告：一名35岁的女性，因慢性腹痛被转介到内分泌科诊所评估肾上腺偶发瘤。计算机断层成像显示一个4厘米均匀的右侧肾上腺肿块，对比前霍斯菲尔德单位为1，绝对对比冲洗率为60%。6年前的计算机断层扫描显示一个1.5厘米的腺瘤，具有类似的特征。尽管激素水平正常，但肿瘤的生长和患者的症状促使手术转诊。患者接受了机器人辅助的腹腔镜右肾上腺切除术。病理检查发现范围清楚的SFT，尺寸为3.7 × 3.6 × 2.9 cm。肾上腺性SFTs是罕见的，典型的激素无活性，边界清楚的肿块，常伴有腹痛。虽然多见于胸膜，但肾上腺SFTs在影像学上可与其他良性肾上腺病变相似，使诊断复杂化。组织病理学对准确诊断至关重要，手术切除仍是主要治疗方法。结论本病例强调了肾上腺SFTs的诊断挑战，它可以模仿其他良性病变。尽管肿瘤生长明显，但组织学上为良性，恶性风险低。临床医生在鉴别诊断具有非典型影像学特征的偶发肾上腺肿块时应考虑肾上腺SFTs。

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引用次数: 0

Abnormal Antithyroglobulin Antibody in a Woman With Papillary Thyroid Carcinoma Receiving Intravenous Immunoglobulin Treatment 接受静脉免疫球蛋白治疗的女性甲状腺乳头状癌患者抗甲状腺球蛋白抗体异常

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2025-03-01 DOI: 10.1016/j.aace.2024.12.010

Verdah Ahmad MD, Sara Lubitz MD

Background

Thyroglobulin and antithyroglobulin antibodies are used in the surveillance of differentiated thyroid carcinomas. In patients with differentiated thyroid carcinoma who require intravenous immunoglobulin for autoimmune or malignant conditions, surveillance of antithyroglobulin antibodies is challenging. We present a case of a patient with papillary thyroid carcinoma who showed fluctuating antithyroglobulin antibody levels while receiving intravenous immunoglobulin.

Case Presentation

A 41-year-old woman underwent total thyroidectomy, central and left modified neck dissection, and radioactive iodine ablation for multifocal papillary thyroid carcinoma with features of oncocytic and warthin-like variant and metastasis to 1 of 28 left lateral and 1 of 10 central lymph nodes. Ten years later, she contracted COVID-19 and developed COVID-19-associated encephalopathy for which she received intravenous immunoglobulin for 7 months. Five months after intravenous immunoglobulin initiation, surveillance thyroglobulin was <0.8 ng/mL (liquid chromatography with mass spectrometry, <0.4 ng/mL) and antithyroglobulin antibodies increased from undetectable to 97 IU/mL (chemiluminescent immunoassay, ≤1 IU/mL). After the final intravenous immunoglobulin dose, antithyroglobulin antibody levels progressively decreased, reaching 1 IU/mL (chemiluminescent immunoassay, ≤1 IU/mL) 6 months posttreatment. Neck ultrasounds showed no evidence of disease recurrence.

Discussion

Intravenous immunoglobulin can affect monitoring of patients with differentiated thyroid carcinomas due to passive transfer of pooled donor antibodies, including antithyroglobulin antibodies. Newly detectable antithyroglobulin antibodies in this patient were likely due to measured donor antithyroglobulin antibodies via intravenous immunoglobulin rather than cancer recurrence.

Conclusion

In patients with differentiated thyroid carcinomas receiving intravenous immunoglobulin, the presence of donor antithyroglobulin antibodies should be considered when antithyroglobulin antibodies are newly detectable.

背景：甲状腺球蛋白和抗甲状腺球蛋白抗体用于分化型甲状腺癌的监测。分化型甲状腺癌患者需要静脉注射免疫球蛋白治疗自身免疫或恶性疾病，抗甲状腺球蛋白抗体的监测具有挑战性。我们报告一例甲状腺乳头状癌患者，在接受静脉注射免疫球蛋白时，其抗甲状腺球蛋白抗体水平波动。一例41岁女性，因多灶性甲状腺乳头状癌行甲状腺全切除术、中央及左侧改良颈部清扫术及放射性碘消融术治疗。该癌表现为嗜瘤性及warthin样变异，28例左侧淋巴结中1例及10例中央淋巴结中1例转移。十年后，她感染了COVID-19，并患上了COVID-19相关脑病，为此她接受了7个月的静脉注射免疫球蛋白。静脉注射免疫球蛋白5个月后，甲状腺球蛋白监测值为0.8 ng/mL（液相色谱-质谱联用法，0.4 ng/mL），抗甲状腺球蛋白抗体从检测不到增加到97 IU/mL（化学发光免疫法，≤1 IU/mL）。在最后一次静脉注射免疫球蛋白后，抗甲状腺球蛋白抗体水平逐渐下降，治疗6个月后达到1 IU/mL（化学发光免疫测定，≤1 IU/mL）。颈部超声检查未见疾病复发。由于供体抗体（包括抗甲状腺球蛋白抗体）的被动转移，静脉注射免疫球蛋白可影响分化型甲状腺癌患者的监测。该患者新检测到的抗甲状腺球蛋白抗体可能是由于通过静脉注射免疫球蛋白检测到供体抗甲状腺球蛋白抗体，而不是癌症复发。结论分化型甲状腺癌患者接受静脉注射免疫球蛋白治疗时，在新检出抗甲状腺球蛋白抗体时应考虑供体是否存在抗甲状腺球蛋白抗体。

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引用次数: 0

Skeletal Muscle Mass and Body Weight Fall Proportionally With Use of Dual Glucagon-Like Peptide 1/Glucose-Dependent Insulinotropic Polypeptide Receptor Agonist Tirzepatide: Case Report and Review of Literature 使用双胰高血糖素样肽1/葡萄糖依赖性胰岛素多肽受体激动剂替西肽，骨骼肌质量和体重成比例下降：病例报告和文献回顾

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2025-03-01 DOI: 10.1016/j.aace.2024.12.001

Jessica Zinn MS, RD, CDN, CDCES, Leonid Poretsky MD

Objective

Multiple studies have demonstrated that treatment with tirzepatide results in significant improvement in HgbA1c as well as meaningful weight loss. Although the loss of skeletal muscle mass has been well described with the use of glucagon-like peptide 1 receptor agonists, there is little information regarding the effect of dual glucagon-like peptide 1/glucose-dependent insulinotropic polypeptide receptor agonist tirzepatide on skeletal muscle mass.

Methods

We performed serial body composition measurements in a 68-year-old male who presented with a body mass index of 31.2 kg/m2 and hemoglobin A1c of 5.9%. Treatment with tirzepatide resulted in a reduction of weight by 28.7 lbs, a decrease in body mass index to 26.8 kg/m2, and normalization of A1c (5.3%). The patient, however, also experienced a 9.9 lb loss of skeletal muscle mass, which was proportionate to a reduction in body weight - approximately 15% from the initial value for both. Muscle mass loss constituted 34% of the total body weight loss.

Results

To our knowledge this is the first report of multiple serial body composition measurements in a patient on treatment with tirzepatide. The time course and the magnitude of the loss of body weight, fat mass and skeletal muscle mass are detailed.

Conclusion

We propose that, in addition to weight measurements, serial body composition assessments should be obtained in patients on tirzepatide. If this is not available, our findings suggest that, at least in some cases, the percent of muscle mass lost is similar to the percent reduction of total body weight.

目的多项研究表明，替西帕肽治疗可显著改善hba1c，并显著减轻体重。虽然使用胰高血糖素样肽1受体激动剂可以减少骨骼肌质量，但关于双胰高血糖素样肽1/葡萄糖依赖性胰岛素性多肽受体激动剂替西肽对骨骼肌质量的影响的信息很少。方法对一名体重指数为31.2 kg/m2、糖化血红蛋白为5.9%的68岁男性进行了一系列身体成分测量。替西肽治疗导致体重减轻28.7磅，体重指数降至26.8 kg/m2，糖化血红蛋白正常化（5.3%）。然而，患者的骨骼肌质量也减少了9.9磅，这与体重的减少成正比——两者的初始值都减少了约15%。肌肉质量损失占总体重损失的34%。结果：据我们所知，这是第一次在接受替西肽治疗的患者中进行多次连续体成分测量的报道。详细说明了体重、脂肪量和骨骼肌量减少的时间、过程和幅度。结论我们建议，除了体重测量外，还应对使用替西肽的患者进行连续体成分评估。如果不能做到这一点，我们的研究结果表明，至少在某些情况下，肌肉质量减少的百分比与体重减少的百分比相似。

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引用次数: 0

Bilateral Adrenal Nodules in the Setting of Disseminated Fungal Infection: An Important Consideration for Appropriate Management of Adrenal Pathology 播散性真菌感染的双侧肾上腺结节：肾上腺病理适当处理的重要考虑

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2025-03-01 DOI: 10.1016/j.aace.2024.12.003

Bailey N. Johnson MD , Signe Braafladt MD , John-Paul Lavik MD, PhD , Lillian Walton BS , Alexandria D. McDow MD, FACS

Background/Objective

Increased utilization of cross-sectional imaging has led to a rise in diagnosis of incidental adrenal lesions. Physicians in many clinical settings are increasingly faced with addressing these incidental lesions by initiating the correct workup, diagnosis, and long-term follow-up plan. Our objective was to demonstrate the importance of maintaining a broad differential and completing a thorough workup in determining the correct treatment plan for patients with bilateral adrenal lesions.

Case Report

We present 2 patients who recently completed chemotherapy for lymphoma, found to have new bilateral adrenal lesions on postchemotherapy imaging. Urine antigen and/or adrenal biopsy was performed to confirm the diagnosis of disseminated fungal infection. This diagnosis has major implications on the treatment plan, which includes antifungal therapy instead of surgical management or additional chemotherapy. Cross-sectional imaging after initiation of antifungal treatment demonstrated decreasing size of nodules.

Discussion

A broad differential is critical when working up and developing treatment plans for adrenal nodules, specifically considering a fungal etiology in the setting of immunosuppression or primary extra-adrenal malignancy.

Conclusion

Incidentally found adrenal lesions are becoming more common, and in turn, the obligation for appropriate management of adrenal pathology not only falls to medical and surgical endocrinologists but also to general practitioners. It is prudent to consider atypical etiologies including disseminated fungal infection prior to surgical excision or initiation of chemotherapy as those treatment strategies would not benefit select patients.

背景/目的横断成像的应用增加了偶发性肾上腺病变的诊断率。在许多临床环境中，医生越来越多地面临着通过启动正确的检查、诊断和长期随访计划来解决这些偶然病变的问题。我们的目的是证明在确定双侧肾上腺病变患者的正确治疗方案时，保持广泛的鉴别和完成彻底的检查的重要性。病例报告：我们报告了2例最近完成淋巴瘤化疗的患者，化疗后影像学发现双侧肾上腺有新的病变。尿抗原和/或肾上腺活检证实弥散性真菌感染的诊断。这一诊断对治疗计划有重要意义，包括抗真菌治疗而不是手术治疗或额外的化疗。开始抗真菌治疗后的横断面成像显示结节变小。在制定和制定肾上腺结节的治疗计划时，特别是考虑到免疫抑制或原发性肾上腺外恶性肿瘤的真菌病因，广泛的区分是至关重要的。结论偶然发现的肾上腺病变越来越多，对肾上腺病理进行适当管理的责任不仅落在内科和外科内分泌科医师身上，也落在全科医生身上。谨慎考虑非典型病因，包括手术切除或化疗开始前的播散性真菌感染，因为这些治疗策略不会使选定的患者受益。

{"title":"Bilateral Adrenal Nodules in the Setting of Disseminated Fungal Infection: An Important Consideration for Appropriate Management of Adrenal Pathology","authors":"Bailey N. Johnson MD , Signe Braafladt MD , John-Paul Lavik MD, PhD , Lillian Walton BS , Alexandria D. McDow MD, FACS","doi":"10.1016/j.aace.2024.12.003","DOIUrl":"10.1016/j.aace.2024.12.003","url":null,"abstract":"<div><h3>Background/Objective</h3><div>Increased utilization of cross-sectional imaging has led to a rise in diagnosis of incidental adrenal lesions. Physicians in many clinical settings are increasingly faced with addressing these incidental lesions by initiating the correct workup, diagnosis, and long-term follow-up plan. Our objective was to demonstrate the importance of maintaining a broad differential and completing a thorough workup in determining the correct treatment plan for patients with bilateral adrenal lesions.</div></div><div><h3>Case Report</h3><div>We present 2 patients who recently completed chemotherapy for lymphoma, found to have new bilateral adrenal lesions on postchemotherapy imaging. Urine antigen and/or adrenal biopsy was performed to confirm the diagnosis of disseminated fungal infection. This diagnosis has major implications on the treatment plan, which includes antifungal therapy instead of surgical management or additional chemotherapy. Cross-sectional imaging after initiation of antifungal treatment demonstrated decreasing size of nodules.</div></div><div><h3>Discussion</h3><div>A broad differential is critical when working up and developing treatment plans for adrenal nodules, specifically considering a fungal etiology in the setting of immunosuppression or primary extra-adrenal malignancy.</div></div><div><h3>Conclusion</h3><div>Incidentally found adrenal lesions are becoming more common, and in turn, the obligation for appropriate management of adrenal pathology not only falls to medical and surgical endocrinologists but also to general practitioners. It is prudent to consider atypical etiologies including disseminated fungal infection prior to surgical excision or initiation of chemotherapy as those treatment strategies would not benefit select patients.</div></div>","PeriodicalId":7051,"journal":{"name":"AACE Clinical Case Reports","volume":"11 2","pages":"Pages 107-112"},"PeriodicalIF":0.0,"publicationDate":"2025-03-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143644016","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

Somatostatin Receptor 2 Negative Pheochromocytoma Masked by Normal Adrenal Gland on Gallium-68 DOTATATE 生长抑素受体2阴性嗜铬细胞瘤被正常肾上腺掩盖

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2025-03-01 DOI: 10.1016/j.aace.2024.12.011

Sanghwa E. Park MD , Thanh D. Hoang DO , Derek J. Stocker MD , Mohamed K.M. Shakir MD , Andrew J. Spiro MD

Background/Objective

Gallium-68 DOTATATE (⁶⁸Ga-DOTATATE) positron emission tomography/computed tomography (CT) is a somatostatin receptor (SSTR)-based imaging with high sensitivity that can be used for detection of pheochromocytomas and paragangliomas. We report a pheochromocytoma with negative SSTR2 expression and low uptake on ⁶⁸Ga-DOTATATE, whose detection was masked by the uptake of normal adrenal tissue.

Case Report

A 50-year-old man presented with a right adrenal incidentaloma. He had mildly elevated plasma normetanephrine levels of 194 pg/mL (ref. 0-145 pg/mL). Adrenal CT scan showed a right 1.9-cm lesion with unenhanced attenuation of 38 Hounsfield units. ⁶⁸Ga-DOTATATE showed a 1.9-cm right adrenal lesion and reported diffuse uptake in the adrenal glands, with maximum standardized uptake value (SUVmax) of 17.23 on the right and SUVmax of 22.78 on the left. After a 2-year interval, plasma normetanephrine level increased to 420 pg/mL (ref. 0-136.8 pg/mL). Adrenal CT scan showed the right adrenal lesion increased in size to 2.6 cm. He underwent right adrenalectomy, and pathology reported a 2.3-cm pheochromocytoma. Subsequent review of the initial ⁶⁸Ga-DOTATATE identified the pheochromocytoma as a photopenic area in the right adrenal gland with 7.73 SUVmax. Tissue staining was negative for SSTR2 expression. Genetic testing was negative for pheochromocytoma syndromes.

Discussion

Although ⁶⁸Ga-DOTATATE has strong affinity for SSTR2, some pheochromocytomas have low expression of SSTR2. The negative SSTR2 expression, small lesion size, and background uptake of the adrenal gland can affect the detection of pheochromocytoma.

Conclusion

⁶⁸Ga-DOTATATE may have limitations when evaluating small pheochromocytomas or other neuroendocrine tumors with low SSTR2 expression.

背景/目的镓-68 DOTATATE （68Ga-DOTATATE）正电子发射断层扫描/计算机断层扫描（CT）是一种基于生长抑素受体（SSTR）的高灵敏度成像技术，可用于嗜铬细胞瘤和副节瘤的检测。我们报告了一例SSTR2表达阴性且68Ga-DOTATATE摄取低的嗜铬细胞瘤，其检测被正常肾上腺组织的摄取所掩盖。病例报告：一名50岁男性，右肾上腺偶发瘤。患者血浆去甲肾上腺素轻度升高，194 pg/mL（参考值0 ~ 145 pg/mL）。肾上腺CT扫描显示右侧1.9 cm病灶，38 Hounsfield单位衰减未增强。68Ga-DOTATATE显示右侧肾上腺病变1.9 cm，肾上腺弥漫性摄取，右侧最大标准化摄取值（SUVmax）为17.23，左侧SUVmax为22.78。间隔2年后，血浆去甲肾上腺素水平升高至420 pg/mL（参考值0-136.8 pg/mL）。肾上腺CT显示右侧肾上腺病变增大至2.6 cm。他接受了右肾上腺切除术，病理报告为2.3厘米的嗜铬细胞瘤。随后对最初的68Ga-DOTATATE进行复查，发现嗜铬细胞瘤为右侧肾上腺的一个光斑区，SUVmax值为7.73。组织染色显示SSTR2表达阴性。嗜铬细胞瘤综合征基因检测呈阴性。虽然68Ga-DOTATATE对SSTR2有很强的亲和力，但一些嗜铬细胞瘤的SSTR2表达水平较低。SSTR2的阴性表达、小的病灶大小和肾上腺的背景摄取会影响嗜铬细胞瘤的检测。结论68ga - dotatate在评估小嗜铬细胞瘤或其他低SSTR2表达的神经内分泌肿瘤时可能存在局限性。

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引用次数: 0

Recurrent Pericarditis in a Woman With Acromegaly Responding Favorably to Transsphenoidal Surgery 女性肢端肥大症复发性心包炎对经蝶窦手术反应良好

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2025-03-01 DOI: 10.1016/j.aace.2025.01.002

Samantha Jacobson MSc , Jonathan-Raphaël Stetco , Natasha Garfield MD, FRCPC

Objective

Acromegaly is associated with increased insulin-like growth factor 1 (IGF-1), promoting systemic inflammation and cardiovascular complications. We present a patient with acromegaly who developed recurrent pericarditis, resolving soon after somatotroph pituitary adenoma resection. The objective of this report is to describe a case of uncontrolled acromegaly with recurrent, unexplained pericarditis.

Case Report

A 46-year-old woman was referred after a neurologist identified a 9 mm pituitary lesion on magnetic resonance imaging. Laboratory tests showed elevated IGF-1 of 52.3 nmol/mL (12.3–32.9 nmol/L), a nonsuppressible growth hormone (GH) level of 3.8 mcg/L (<0.4 mcg/L) after a 75 g oral glucose tolerance test, confirming acromegaly. One-year postdiagnosis, the patient developed pleuritic chest pain from pericarditis with moderate-to-severe pericardial effusion. Symptoms resolved with nonsteroidal antiinflammatory drugs, colchicine and pericardiocentesis. Over 3 years she experienced multiple episodes of recurrent pericarditis. A comprehensive diagnostic workup, including rheumatologic and infectious evaluations, was negative. After transsphenoidal adenoma resection, IGF-1 normalized, and medical therapy was discontinued. Pericarditis recurred 2 months postoperatively but has not occurred again over 12 years of acromegaly remission.

Discussion

Hypersecretion of GH in acromegaly leads to elevated IGF-1 levels, which affect inflammatory responses. IGF-1 can promote systemic inflammation through proinflammatory cytokines, its effects may vary depending on tissue type. In this case, resolution of pericarditis following IGF-1 normalization suggests that elevated IGF-1 levels may mediate the inflammatory process in the pericardium.

Conclusion

The case suggests that acromegaly may predispose some patients to pericarditis, but its frequency and underlying pathogenesis remain unclear.

肢端肥大症与胰岛素样生长因子1 （IGF-1）升高有关，可促进全身炎症和心血管并发症。我们报告一位肢端肥大症患者，在切除垂体生长缺陷腺瘤后，出现复发性心包炎。本报告的目的是描述一个不受控制的肢端肥大症与复发，不明原因的心包炎的情况。病例报告一名46岁的女性在核磁共振成像上发现一个9毫米的垂体病变后被转诊。实验室检查显示IGF-1升高52.3 nmol/mL (12.3-32.9 nmol/L)， 75 g口服葡萄糖耐量试验后非抑制性生长激素（GH）水平为3.8 mcg/L (0.4 mcg/L)，证实肢端肥大症。诊断一年后，患者出现胸膜炎性胸痛，心包炎伴中重度心包积液。经非甾体类抗炎药、秋水仙碱和心包穿刺术治疗后症状消失。在3年多的时间里，她经历了多次复发性心包炎。综合诊断检查，包括风湿病学和感染性评估，结果为阴性。蝶窦腺瘤切除后，IGF-1恢复正常，停止药物治疗。心包炎术后2个月复发，但在肢端肥大症缓解后12年内未再发生。肢端肥大症患者生长激素分泌过多导致IGF-1水平升高，从而影响炎症反应。IGF-1可通过促炎因子促进全身性炎症，其作用可能因组织类型而异。在这种情况下，IGF-1正常化后心包炎的消退表明IGF-1水平升高可能介导了心包膜的炎症过程。结论本病例提示肢端肥大症可能使部分患者易患心包炎，但其发病频率和潜在发病机制尚不清楚。

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引用次数: 0

Mild Thyrotoxicosis due to Seronegative Graves’ Disease or Disseminated Thyroid Autonomy 血清阴性Graves病或弥散性甲状腺自主性所致轻度甲状腺毒症

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2025-03-01 DOI: 10.1016/j.aace.2024.12.002

Gael Charbonne MD , Fatima Hallak MD , Alma Elizabeth Rivera Cruz MD , Stephen Devitt McDonald MD

Background/Objective

Disseminated thyroid autonomy (DTA) and seronegative Graves’ disease are rare causes of hyperthyroidism with similar clinical presentations. This case report highlights the diagnostic challenges between these entities.

Case Presentation

A 35-year-old male presented with palpitations, diaphoresis, and a small goiter. His TSH was 0.249 mIU/L (reference: 0.45–4.5 mIU/L) and free T4 was 3.0 ng/dL (reference: 0.88–1.77 ng/dL). Thyroid peroxidase antibodies, TRAb, and TSI were repeatedly negative. Ultrasound showed a diffusely enlarged thyroid, and radioactive iodine uptake was 35% (reference: 10% to 35%) with thyroid scintigraphy revealing diffusely increased uptake, indicating a hyperfunctioning thyroid without nodules. Treatment with Methimazole 5 mg daily resolved symptoms within 6 months.

Discussion

Thyroid biopsy can help distinguish DTA from seronegative Graves disease. DTA is marked by nodular hyperplasia without lymphocytic infiltration, indicating a non-autoimmune nature, while seronegative Graves’ disease exhibits diffuse follicular hyperplasia with lymphocytic infiltration, just as typical Graves’ disease despite undetectable autoatibodies. The possibility of false-negative TRAb results complicates diagnosis, with up to 22% of patients initially diagnosed with DTA later testing TRAb-positive upon retesting. Some cases of DTA may involve TSH receptor gene mutations.

Conclusion

This case highlights the complexity of distinguishing DTA, seronegative Graves’ disease and typical Graves’ disease with initial false negative testing. A systematic approach with repeat testing and, when feasible, biopsy, is critical to distinguish these entities. Further studies with histologic analysis are needed to clarify outcomes and develop tailored managements, as these conditions have different remission rates and are driven by different mechanisms.

背景/目的弥散性甲状腺自主（DTA）和血清阴性Graves病是甲状腺功能亢进的罕见病因，临床表现相似。本病例报告强调了这些实体之间的诊断挑战。病例表现男性，35岁，表现为心悸、出汗、小甲状腺肿大。TSH为0.249 mIU/L（参考：0.45-4.5 mIU/L），游离T4为3.0 ng/dL（参考：0.88-1.77 ng/dL）。甲状腺过氧化物酶抗体、TRAb、TSI反复阴性。超声显示弥漫性甲状腺肿大，放射性碘摄取35%（参考文献：10% ~ 35%），甲状腺显像显示弥漫性碘摄取增加，提示甲状腺功能亢进，无结节。每日5毫克甲巯咪唑治疗6个月内症状消失。甲状腺活检有助于区分DTA和血清阴性Graves病。DTA表现为结节性增生，无淋巴细胞浸润，提示非自身免疫性，而血清阴性Graves病表现为弥漫性滤泡增生，伴淋巴细胞浸润，与典型Graves病一样，尽管自身抗体检测不到。TRAb结果假阴性的可能性使诊断复杂化，高达22%最初诊断为DTA的患者在重新检测后检测出TRAb阳性。一些DTA病例可能涉及TSH受体基因突变。结论本病例突出了鉴别DTA、血清阴性Graves病和典型Graves病与初始假阴性的复杂性。采用系统的方法进行重复检测，可行时进行活检，对于区分这些实体至关重要。由于这些疾病有不同的缓解率，并由不同的机制驱动，因此需要进一步的组织学分析研究来明确结果并制定量身定制的管理方法。

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引用次数: 0

Beyond Glucose Levels: Redefining Diabetic Ketoacidosis—A Case of Hypoglycemic Diabetic Ketoacidosis 超越葡萄糖水平：重新定义糖尿病酮症酸中毒-一例低血糖糖尿病酮症酸中毒

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2025-03-01 DOI: 10.1016/j.aace.2025.01.001

Guy I. Sydney MD

Background/Objective

Diabetic ketoacidosis (DKA) is a life-threatening condition typically diagnosed by the presence of hyperglycemia, acidemia, and ketonemia. A subset of patients may develop ketoacidosis without the traditionally increased glucose levels in a condition known as euglycemic DKA. This article describes an atypical presentation of DKA with concomitant hypoglycemia in a condition termed hypoglycemic DKA.

Case Report

A 74-year-old woman with a history of hypertension, type 2 diabetes mellitus (treated with empagliflozin), and hypothyroidism, presented from an outlying hospital due to concern for acute gallstone pancreatitis and choledocholithiasis. On arrival, laboratory evaluation revealed an anion gap of 16 mEq/L (reference range, 6-12 mEq/L), bicarbonate level of 11 mEq/L (reference range, 21-31 mEq/L), serum glucose level of 57 mg/dL (reference range, 70-105 mg/dL), beta-hydroxybutyrate level of 1.7 mmol/L (reference range, <0.6 mmol/L), and urinalysis demonstrating a ketone level of >80 mg/dL (reference range, <3.49 mg/dL). The patient was treated according to the institution DKA protocol, with resolution of her DKA.

Discussion

The case presented highlights a manifestation of DKA characterized by a concurrent state of hypoglycemia in a patient treated with a sodium-glucose cotransporter 2 inhibitor, an atypical and likely underreported phenomenon.

Conclusion

Clinicians should maintain a high level of suspicion for DKA in patients with metabolic acidosis and ketosis, irrespective of their glucose levels, in particular in those treated with sodium-glucose cotransporter 2 inhibitors. Additionally, redefining these cases as drug-induced ketoacidosis may assist in preventing delayed diagnosis and management.

背景/目的糖尿病酮症酸中毒（DKA）是一种危及生命的疾病，通常通过高血糖、酸血症和酮血症来诊断。一部分患者可能会出现酮症酸中毒，而没有传统意义上的血糖水平升高，这种情况被称为血糖正常的DKA。这篇文章描述了一个非典型的DKA与低血糖合并的条件下称为低血糖DKA。病例报告：一名74岁女性，有高血压、2型糖尿病（用恩格列净治疗）和甲状腺功能减退病史，因急性胆石性胰腺炎和胆管结石症在边远医院就诊。到达时，实验室检查显示阴离子间隙为16 mEq/L（参考范围，6-12 mEq/L），碳酸氢盐水平为11 mEq/L（参考范围，21-31 mEq/L），血清葡萄糖水平为57 mg/dL（参考范围，70-105 mg/dL）， β -羟基丁酸水平为1.7 mmol/L（参考范围，0.6 mmol/L），尿酮水平为80 mg/dL（参考范围，3.49 mg/dL）。患者按照机构DKA方案治疗，其DKA得到解决。本病例强调了在接受钠-葡萄糖共转运蛋白2抑制剂治疗的患者中以同时低血糖为特征的DKA表现，这是一种非典型且可能被低估的现象。结论：临床医生应对代谢性酸中毒和酮症患者的DKA保持高度警惕，无论其血糖水平如何，特别是那些接受钠-葡萄糖共转运蛋白2抑制剂治疗的患者。此外，将这些病例重新定义为药物性酮症酸中毒可能有助于防止延迟诊断和管理。

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引用次数: 0

Ovarian Hyperthecosis Presenting as Polycythemia 卵巢过度增生表现为红细胞增多症

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2025-03-01 DOI: 10.1016/j.aace.2024.11.008

Gayatri Jaiswal MD, Fnu Varsha MD, Patricia Bononi MD, Christina Dimech MD, Ashni Dharia MD

Background/Objective

A 58-year-old postmenopausal woman presented with polycythemia on routine laboratory examinations along with long-standing hirsutism. The objective of this report is to highlight polycythemia as a rare presenting feature of ovarian hyperthecosis (OH).

Case Report

A 58-year-old woman presented with elevated hemoglobin and hematocrit levels in the primary care setting on routine laboratory examinations. On further workup, she was found to have elevated testosterone levels. Further history was relevant for excessive facial hair, frontal hair loss, and deepened voice. Examination findings were consistent with significant hirsutism. Adrenal and ovarian imaging was negative for tumor. The patient elected to have laparoscopic bilateral oophorectomy, which revealed OH on surgical pathology. After 2 months of surgery, the total testosterone levels became normal, and polycythemia resolved.

Discussion

In a postmenopausal woman, hirsutism with virilization should generally prompt urgent investigation because these signs are associated with malignant androgen-secreting tumors of the adrenal gland and ovaries. However, these symptoms can also be the result of benign causes such as OH.

Conclusion

This case is a rare illustration of OH in a postmenopausal woman presenting with polycythemia secondary to severe hyperandrogenism. Suspicion for this condition is crucial in evaluation for rare causes of polycythemia because if left untreated, it can lead to increased morbidity and mortality.

背景/目的一名58岁绝经后妇女在常规实验室检查中表现为红细胞增多症，并伴有长期多毛症。本报告的目的是强调红细胞增多症是卵巢囊肿（OH）的一个罕见的表现特征。病例报告一名58岁女性，在初级保健机构的常规实验室检查中出现血红蛋白和红细胞压积水平升高。在进一步的检查中，发现她的睾丸激素水平升高。进一步的病史与面部毛发过多、额部脱发和声音低沉有关。检查结果与明显的多毛症一致。肾上腺及卵巢影像学未见肿瘤。患者选择腹腔镜双侧卵巢切除术，手术病理显示OH。术后2个月，总睾酮水平恢复正常，红细胞增多症消退。绝经后的女性，多毛伴男性化通常需要紧急检查，因为这些症状与肾上腺和卵巢的恶性雄激素分泌肿瘤有关。然而，这些症状也可能是良性原因的结果，如OH。结论：本病例是一例罕见的绝经后妇女因严重的高雄激素症并发红细胞增多症而患OH的病例。对这种情况的怀疑对于评估罕见的红细胞增多症病因至关重要，因为如果不及时治疗，它可能导致发病率和死亡率增加。

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引用次数: 0