Pub Date : 2024-09-01DOI: 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.
{"title":"Stiff Person Syndrome and Brittle Type 1 Diabetes: Report of 2 Cases","authors":"Ismael A. Quintal-Medina MD , Francisco J. Gómez-Pérez MD , Paloma Almeda-Valdes MD, PhD","doi":"10.1016/j.aace.2024.07.003","DOIUrl":"10.1016/j.aace.2024.07.003","url":null,"abstract":"<div><h3>Background/Objective</h3><p>Stiff person syndrome (SPS) and type 1 diabetes (T1D) are heterogeneous disorders characterized by antibodies (Abs) against glutamic acid decarboxylase (GAD).</p></div><div><h3>Case Report</h3><p>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.</p></div><div><h3>Discussion</h3><p>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.</p></div><div><h3>Conclusion</h3><p>Our case series represent the heterogeneity in natural history, clinical course, and response to therapy in patients with Abs against GAD-spectrum disorders.</p></div>","PeriodicalId":7051,"journal":{"name":"AACE Clinical Case Reports","volume":"10 5","pages":"Pages 198-201"},"PeriodicalIF":0.0,"publicationDate":"2024-09-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2376060524000671/pdfft?md5=a7c5a3f62986eb1fba7cdc95d6a54060&pid=1-s2.0-S2376060524000671-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141713720","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}
Pub Date : 2024-09-01DOI: 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.
{"title":"Type 1 Diabetes Mellitus Caused by COVID-19 mRNA Vaccination: A Case Report and Literature Review of 17 Published Cases","authors":"","doi":"10.1016/j.aace.2024.06.001","DOIUrl":"10.1016/j.aace.2024.06.001","url":null,"abstract":"<div><h3>Background/Objective</h3><p>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.</p></div><div><h3>Case Report</h3><p>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.</p></div><div><h3>Discussion</h3><p>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.</p></div><div><h3>Conclusion</h3><p>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.</p></div>","PeriodicalId":7051,"journal":{"name":"AACE Clinical Case Reports","volume":"10 5","pages":"Pages 179-183"},"PeriodicalIF":0.0,"publicationDate":"2024-09-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2376060524000567/pdfft?md5=27c7a724a04c7b3d08a5e3bec497b2b2&pid=1-s2.0-S2376060524000567-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141405785","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}
Pub Date : 2024-09-01DOI: 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}
Pub Date : 2024-09-01DOI: 10.1016/j.aace.2024.04.004
{"title":"Skin Rash Appearing in a Patient with Gestational Diabetes Mellitus on Insulin Therapy","authors":"","doi":"10.1016/j.aace.2024.04.004","DOIUrl":"10.1016/j.aace.2024.04.004","url":null,"abstract":"","PeriodicalId":7051,"journal":{"name":"AACE Clinical Case Reports","volume":"10 5","pages":"Pages 216-217"},"PeriodicalIF":0.0,"publicationDate":"2024-09-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2376060524000361/pdfft?md5=83ff849935c2dcd6f8743dcb7c2f7f52&pid=1-s2.0-S2376060524000361-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140763036","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}
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}
Pub Date : 2024-09-01DOI: 10.1016/j.aace.2024.08.005
Sina Jasim MD, MPH (Editor in Chief)
{"title":"Editorial for September/October Issue of AACE Clinical Case Reports","authors":"Sina Jasim MD, MPH (Editor in Chief)","doi":"10.1016/j.aace.2024.08.005","DOIUrl":"10.1016/j.aace.2024.08.005","url":null,"abstract":"","PeriodicalId":7051,"journal":{"name":"AACE Clinical Case Reports","volume":"10 5","pages":"Page 169"},"PeriodicalIF":0.0,"publicationDate":"2024-09-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2376060524000828/pdfft?md5=15a173bb50e0227c883624d6cf41bd7a&pid=1-s2.0-S2376060524000828-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142162301","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}
Pub Date : 2024-07-01DOI: 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.
{"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}
Pub Date : 2024-07-01DOI: 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
{"title":"Unusual Case of Adrenal Artery Aneurysm: Diagnosis and Management","authors":"Omolade O. Sogade MD , C. Corbin Frye MD , Daniel Picus MD , Sina Jasim MD, MPH , Taylor C. Brown MD, MHS","doi":"10.1016/j.aace.2024.03.004","DOIUrl":"10.1016/j.aace.2024.03.004","url":null,"abstract":"","PeriodicalId":7051,"journal":{"name":"AACE Clinical Case Reports","volume":"10 4","pages":"Pages 166-167"},"PeriodicalIF":0.0,"publicationDate":"2024-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2376060524000282/pdfft?md5=7945ae337bb1fd37a29a302f18d21cd7&pid=1-s2.0-S2376060524000282-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140279024","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}
Pub Date : 2024-07-01DOI: 10.1016/j.aace.2024.04.007
Tatiana Tselovalnikova MD, PhD , Kavita Jadhav MD, FACP , John Foxworth PharmD , Peminda K. Cabandugama MD, DABOM , Sophia Galustian MD , Betty M. Drees MD, FACP, FACE
Background/Objective
Severe hypocalcemia is common in critically ill patients. There are different mechanisms. To our knowledge, there are no data about the acute presentation of hypocalcemia at the time of diagnosis of aplastic anemia (AA). The objective of this case report was to describe the case of hypoparathyroidism with severe hypocalcemia in a critically ill patient with AA.
Case Report
A 60-year-old man presented with severe hypocalcemia with a calcium level of 6.1 mg/dL (reference range, 8.6-10.3 mg/dL) and hypoparathyroidism with a parathyroid hormone level of 11 pg/mL (reference range, 12-88 pg/mL). He developed a critical state caused by newly diagnosed AA and its complications, such as an acute decrease in the platelet value to a critically low level of 2 × 103/cmm, complicated by neutropenic fever and lower gastrointestinal bleeding. After the initiation of immunosuppressive therapy for AA, his parathyroid hormone-calcium metabolism improved and remained stable but did not normalize completely.
Discussion
In our patient, hypoparathyroidism with hypocalcemia may have been caused by cytokine-related upregulation of the calcium-sensing receptor in the setting of AA. On the other hand, given the severity of the initial hypocalcemia and only partial improvement in calcium homeostasis with residual mild hypocalcemia after treatment initiation for AA, autoimmune causes cannot be entirely ruled out, nor could a combination of cytokine-mediated and autoimmune causes.
Conclusion
It is essential to treat the underlying causes of hypocalcemia, which, in this case, were AA and hypoparathyroidism.
背景/目的严重低钙血症在重症患者中很常见。其发病机制各不相同。据我们所知，目前还没有关于再生障碍性贫血（AA）诊断时出现急性低钙血症的数据。病例报告一名60岁的男性患者出现严重低钙血症，血钙水平为6.1毫克/分升（参考范围：8.6-10.3毫克/分升），甲状旁腺功能减退，甲状旁腺激素水平为11皮克/毫升（参考范围：12-88皮克/毫升）。新确诊的 AA 及其并发症导致他病情危重，如血小板值急剧下降至 2 × 103/cmm 的极低水平，并伴有中性粒细胞减少性发热和下消化道出血。在开始对AA进行免疫抑制治疗后，他的甲状旁腺激素-钙代谢情况有所改善并保持稳定，但并未完全恢复正常。另一方面，鉴于最初低钙血症的严重程度，以及在开始治疗AA后，钙稳态仅得到部分改善，但仍存在轻度低钙血症，因此不能完全排除自身免疫原因，也不能排除细胞因子介导的原因和自身免疫原因的结合。
{"title":"Hypocalcemia and Hypoparathyroidism Associated With Critical Illness and Aplastic Anemia","authors":"Tatiana Tselovalnikova MD, PhD , Kavita Jadhav MD, FACP , John Foxworth PharmD , Peminda K. Cabandugama MD, DABOM , Sophia Galustian MD , Betty M. Drees MD, FACP, FACE","doi":"10.1016/j.aace.2024.04.007","DOIUrl":"10.1016/j.aace.2024.04.007","url":null,"abstract":"<div><h3>Background/Objective</h3><p>Severe hypocalcemia is common in critically ill patients. There are different mechanisms. To our knowledge, there are no data about the acute presentation of hypocalcemia at the time of diagnosis of aplastic anemia (AA). The objective of this case report was to describe the case of hypoparathyroidism with severe hypocalcemia in a critically ill patient with AA.</p></div><div><h3>Case Report</h3><p>A 60-year-old man presented with severe hypocalcemia with a calcium level of 6.1 mg/dL (reference range, 8.6-10.3 mg/dL) and hypoparathyroidism with a parathyroid hormone level of 11 pg/mL (reference range, 12-88 pg/mL). He developed a critical state caused by newly diagnosed AA and its complications, such as an acute decrease in the platelet value to a critically low level of 2 × 10<sup>3</sup>/cmm, complicated by neutropenic fever and lower gastrointestinal bleeding. After the initiation of immunosuppressive therapy for AA, his parathyroid hormone-calcium metabolism improved and remained stable but did not normalize completely.</p></div><div><h3>Discussion</h3><p>In our patient, hypoparathyroidism with hypocalcemia may have been caused by cytokine-related upregulation of the calcium-sensing receptor in the setting of AA. On the other hand, given the severity of the initial hypocalcemia and only partial improvement in calcium homeostasis with residual mild hypocalcemia after treatment initiation for AA, autoimmune causes cannot be entirely ruled out, nor could a combination of cytokine-mediated and autoimmune causes.</p></div><div><h3>Conclusion</h3><p>It is essential to treat the underlying causes of hypocalcemia, which, in this case, were AA and hypoparathyroidism.</p></div>","PeriodicalId":7051,"journal":{"name":"AACE Clinical Case Reports","volume":"10 4","pages":"Pages 156-159"},"PeriodicalIF":0.0,"publicationDate":"2024-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2376060524000427/pdfft?md5=32b9107dd91bea878fad51273bc7fe8f&pid=1-s2.0-S2376060524000427-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141042665","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}
Pub Date : 2024-07-01DOI: 10.1016/j.aace.2024.03.006
Sima Saberi MD , Nicholas Burris MD , Ka Kit Wong MBBS , Noah A. Brown MD , Thomas Giordano MD, PhD , Nazanene H. Esfandiari MD
Background/Objective
Follicular thyroid cancer without an intrathyroidal primary cancer is rare. We present a patient with multifocal pulmonary metastatic follicular thyroid cancer without apparent cancer within her thyroid.
Case Report
A 44-year-old woman was referred to the thyroid cancer clinic via telemedicine for evaluation of intrapulmonary thyroid tissue. Her past medical history included Roux-en-Y gastric bypass and hysterectomy with bilateral oophorectomy. Six months prior, abdominal computed tomography (CT) showed incidental bilateral lung nodules. Chest CT demonstrated 4 solid left and 1 solid right lung nodules. Lung nodule core biopsy revealed benign thyroid tissue. Thyroid ultrasound showed bilateral subcentimeter anechoic nodules. Chest CT 6 months after initial CT demonstrated stable lung nodules. The levels of thyroid-stimulating hormone, serum thyroglobulin, and thyroglobulin antibody were 1.63 mIU/L (reference range, 0.3-5.5 mIU/L), 40.9 ng/mL (reference range, 0-35 ng/mL), and <1 IU/mL (reference range, <4), respectively. Positron emission tomography/CT showed fluorodeoxyglucose-avid lung lesions measuring 1.5, 1.1, and 2.2 cm and other subcentimeter pulmonary nodules. Repeat lung core biopsy showed thyroid tissue with microfollicular architecture, favoring metastatic follicular carcinoma with neuroblastoma-RAS gene (NRAS) mutation. Total thyroidectomy performed showed multinodular hyperplasia without thyroid cancer. Her postoperative radioiodine scan demonstrated bilateral iodine-avid pulmonary nodules, a serum thyroglobulin level of 179.8 ng/mL, a thyroid-stimulating hormone level of 151.3 mIU/L, and undetectable serum thyroglobulin antibody. She received 261 mCi of radioactive iodine. Fourteen months later, chest CT revealed decreased lung nodules and a serum thyroglobulin level of 0.7 ng/mL.
Discussion
Approximately 2 cases of multifocal pulmonary follicular thyroid cancer without a primary source and no other site of metastasis have been reported.
Conclusion
Pulmonary follicular thyroid cancer without a primary source and no other site of metastasis is extremely rare.
{"title":"Pulmonary Metastatic Follicular Thyroid Carcinoma Without Intrathyroidal Primary Thyroid Cancer","authors":"Sima Saberi MD , Nicholas Burris MD , Ka Kit Wong MBBS , Noah A. Brown MD , Thomas Giordano MD, PhD , Nazanene H. Esfandiari MD","doi":"10.1016/j.aace.2024.03.006","DOIUrl":"10.1016/j.aace.2024.03.006","url":null,"abstract":"<div><h3>Background/Objective</h3><p>Follicular thyroid cancer without an intrathyroidal primary cancer is rare. We present a patient with multifocal pulmonary metastatic follicular thyroid cancer without apparent cancer within her thyroid.</p></div><div><h3>Case Report</h3><p>A 44-year-old woman was referred to the thyroid cancer clinic via telemedicine for evaluation of intrapulmonary thyroid tissue. Her past medical history included Roux-en-Y gastric bypass and hysterectomy with bilateral oophorectomy. Six months prior, abdominal computed tomography (CT) showed incidental bilateral lung nodules. Chest CT demonstrated 4 solid left and 1 solid right lung nodules. Lung nodule core biopsy revealed benign thyroid tissue. Thyroid ultrasound showed bilateral subcentimeter anechoic nodules. Chest CT 6 months after initial CT demonstrated stable lung nodules. The levels of thyroid-stimulating hormone, serum thyroglobulin, and thyroglobulin antibody were 1.63 mIU/L (reference range, 0.3-5.5 mIU/L), 40.9 ng/mL (reference range, 0-35 ng/mL), and <1 IU/mL (reference range, <4), respectively. Positron emission tomography/CT showed fluorodeoxyglucose-avid lung lesions measuring 1.5, 1.1, and 2.2 cm and other subcentimeter pulmonary nodules. Repeat lung core biopsy showed thyroid tissue with microfollicular architecture, favoring metastatic follicular carcinoma with neuroblastoma-RAS gene (<em>NRAS</em>) mutation. Total thyroidectomy performed showed multinodular hyperplasia without thyroid cancer. Her postoperative radioiodine scan demonstrated bilateral iodine-avid pulmonary nodules, a serum thyroglobulin level of 179.8 ng/mL, a thyroid-stimulating hormone level of 151.3 mIU/L, and undetectable serum thyroglobulin antibody. She received 261 mCi of radioactive iodine. Fourteen months later, chest CT revealed decreased lung nodules and a serum thyroglobulin level of 0.7 ng/mL.</p></div><div><h3>Discussion</h3><p>Approximately 2 cases of multifocal pulmonary follicular thyroid cancer without a primary source and no other site of metastasis have been reported.</p></div><div><h3>Conclusion</h3><p>Pulmonary follicular thyroid cancer without a primary source and no other site of metastasis is extremely rare.</p></div>","PeriodicalId":7051,"journal":{"name":"AACE Clinical Case Reports","volume":"10 4","pages":"Pages 123-126"},"PeriodicalIF":0.0,"publicationDate":"2024-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2376060524000300/pdfft?md5=a5282e46718123241d6426b526d7dda5&pid=1-s2.0-S2376060524000300-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140405885","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}
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