JCEM case reports最新文献_第2页

ACTH-dependent Cyclic Cushing Syndrome With Successful Pregnancy and Early Postpartum Relapse. acth依赖性周期性库欣综合征与成功妊娠和产后早期复发。

JCEM case reports Pub Date : 2025-04-11 eCollection Date: 2025-05-01 DOI: 10.1210/jcemcr/luaf079

Mohd Idris Mohamad Diah, Jin Hui Ho, Hwee Ching Tee

{"title":"ACTH-dependent Cyclic Cushing Syndrome With Successful Pregnancy and Early Postpartum Relapse.","authors":"Mohd Idris Mohamad Diah, Jin Hui Ho, Hwee Ching Tee","doi":"10.1210/jcemcr/luaf079","DOIUrl":"https://doi.org/10.1210/jcemcr/luaf079","url":null,"abstract":"Cyclic Cushing syndrome (CS) is a rare form of CS characterized by intermittent episodes of hypercortisolism. We report the case of a 30-year-old female who was diagnosed with ACTH-dependent Cushing disease, confirmed by initial biochemical tests and pituitary imaging. Although surgery was planned, she experienced spontaneous remission for several months, followed by pregnancy, and subsequently relapsed in the early postpartum period. Transsphenoidal resection of a left-sided pituitary adenoma was then performed, confirming an ACTH-secreting tumor. A review of the literature revealed that this case contributes to the increasing number of patients with cyclic CS, with particular attention to the challenges of diagnosing hypercortisolism during pregnancy. While cases of Cushing disease recurrence after pituitary surgery in the immediate postpartum period have been documented, this is the first reported case of early postpartum relapse in cyclic CS. This case highlights the importance of long-term follow-up in patients with a high index of suspicion for cyclic CS, as well as the diagnostic challenges in managing the condition during pregnancy and the peripartum period.","PeriodicalId":73540,"journal":{"name":"JCEM case reports","volume":"3 5","pages":"luaf079"},"PeriodicalIF":0.0,"publicationDate":"2025-04-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11986810/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144030205","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

A Rare Case of Pituicytoma During Pregnancy. 妊娠期罕见垂体瘤1例。

JCEM case reports Pub Date : 2025-04-11 eCollection Date: 2025-05-01 DOI: 10.1210/jcemcr/luaf071

Junchen Wu, Hao Tang, Bo Zhang, Shaojian Lin, Zhe Bao Wu

引用次数: 0

Thyroid-Stimulating Hormone/Growth Hormone Cosecreting Pituitary Adenoma With Normal Thyroid-Stimulating Hormone Level. 促甲状腺激素/生长激素共分泌垂体腺瘤促甲状腺激素水平正常。

JCEM case reports Pub Date : 2025-04-11 eCollection Date: 2025-05-01 DOI: 10.1210/jcemcr/luaf063

Feyza Erenler, Benjamin Katcher, Van Phan, Knarik Arkun, Mina G Safain

{"title":"Thyroid-Stimulating Hormone/Growth Hormone Cosecreting Pituitary Adenoma With Normal Thyroid-Stimulating Hormone Level.","authors":"Feyza Erenler, Benjamin Katcher, Van Phan, Knarik Arkun, Mina G Safain","doi":"10.1210/jcemcr/luaf063","DOIUrl":"https://doi.org/10.1210/jcemcr/luaf063","url":null,"abstract":"Thyroid-stimulating hormone (TSH; thyrotropin) adenoma is a rare pituitary tumor that can be missed due to its subtle symptoms. We are reporting a 67-year-old man with history of ventricular fibrillation on amiodarone who presented with acute headache and right third cranial nerve palsy. His computed tomography (CT) scan revealed a 2.2-cm suprasellar mass, consistent with pituitary apoplexy, and he underwent pituitary tumor resection. Preoperational hormonal workup revealed TSH 0.25 mIU/mL (0.25 IU/L) (normal reference range: 0.35-4.94 mIU/mL; 0.35-4.94 IU/L), free thyroxine (T4) 3.17 ng/dL (40.80 pmol/L) (normal reference range: 0.7-1.48 ng/dL; 9.78-19.05 pmol/L), and total triiodothyronine (T3) 91 ng/dL (140 nmol/L) (normal reference range: 58-159 ng/dL; 89-244 nmol/L). Initial differential diagnoses included TSH-producing pituitary adenoma (TSH-oma) and amiodarone-induced thyrotoxicosis. His free T4 declined significantly postoperatively, favoring a TSH-oma diagnosis. The pathology report showed a TSH and growth hormone (GH) cosecreting adenoma. Furthermore, he had a normal thyroid uptake scan, as well as negative thyroid antibodies, making primary thyroid diseases less likely. A high free T4 with normal TSH 3 years ago, prior to the start of amiodarone, suggested a long disease duration. This case demonstrates challenges in diagnosing TSH-oma, especially in patients with normal TSH and concurrent amiodarone use.","PeriodicalId":73540,"journal":{"name":"JCEM case reports","volume":"3 5","pages":"luaf063"},"PeriodicalIF":0.0,"publicationDate":"2025-04-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11986576/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144045828","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

Papillary Thyroid Carcinoma With 5 Unique Point Mutations and Typical Behavior. 5个独特的点突变和典型行为的甲状腺乳头状癌。

JCEM case reports Pub Date : 2025-04-11 eCollection Date: 2025-05-01 DOI: 10.1210/jcemcr/luaf062

Aditya Chauhan, Siddhartha Sen, Khalid Amin, Lynn A Burmeister

引用次数: 0

A Rare Cause of Acute Pancreatitis: Two Siblings With Werner Syndrome. 一个罕见的急性胰腺炎的原因：两个兄弟姐妹与维尔纳综合征。

JCEM case reports Pub Date : 2025-04-10 eCollection Date: 2025-05-01 DOI: 10.1210/jcemcr/luaf061

Kenan Sakar, Baris Akinci, Ilgin Yildirim Simsir, Tahir Atik, Gulhan Akbaba, Nese Cinar

引用次数: 0

Iododerma Following Radioactive Iodine Therapy in Thyroid Cancer: Insights From 2 Cases. 甲状腺癌放射性碘治疗后的碘皮病2例分析。

JCEM case reports Pub Date : 2025-04-10 eCollection Date: 2025-05-01 DOI: 10.1210/jcemcr/luaf056

Hawra Kamal, Julie Samantray

{"title":"Iododerma Following Radioactive Iodine Therapy in Thyroid Cancer: Insights From 2 Cases.","authors":"Hawra Kamal, Julie Samantray","doi":"10.1210/jcemcr/luaf056","DOIUrl":"https://doi.org/10.1210/jcemcr/luaf056","url":null,"abstract":"Radioactive iodine ablation is commonly used to treat thyroid diseases. However, despite its efficacy, it has inherent risks and complications. One such complication is iododerma. This rare dermatological condition triggered by iodine exposure has been infrequently documented in association with radioactive iodine therapy for thyroid diseases. Here, we present 2 cases of iododerma following radioactive iodine ablation for thyroid cancer. In the first case, a 38-year-old female developed facial swelling and red blotchy rashes accompanied by papules on the left upper eyelid. Despite initial worsening of symptoms, the patient improved after prednisone treatment. In the second case, a 71-year-old male with metastatic follicular thyroid cancer received iodine-131 therapy after levothyroxine withdrawal for pulmonary metastasis. Approximately 1 week posttherapy, he developed a nontender, nonpruritic rash on the extremities and anterior abdomen, which spontaneously resolved without intervention. Iododerma presents diagnostic challenges because of its rarity and diverse cutaneous manifestations. Although its exact pathophysiology remains unclear, it has been hypothesized to be induced by hypersensitivity reactions to and delayed clearance of iodine from the body. Physicians should be aware of this rare complication of radioactive iodine in patients with thyroid disease.","PeriodicalId":73540,"journal":{"name":"JCEM case reports","volume":"3 5","pages":"luaf056"},"PeriodicalIF":0.0,"publicationDate":"2025-04-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11982607/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144045823","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

Recurrent Hypoglycemic Coma Episodes Associated With Primary Biliary Cirrhosis. 与原发性胆汁性肝硬化相关的反复低血糖昏迷发作。

JCEM case reports Pub Date : 2025-04-10 eCollection Date: 2025-05-01 DOI: 10.1210/jcemcr/luaf030

Yang Liu, Jianbin Xu, Yimei Chen, Lingling Wang, Wen Chen, Jing Shen

引用次数: 0

A Corticotropin-Secreting Adenoma in the Setting of von Hippel-Lindau Disease. 希佩尔-林道病并发促肾上腺皮质激素分泌腺瘤。

JCEM case reports Pub Date : 2025-04-10 eCollection Date: 2025-05-01 DOI: 10.1210/jcemcr/luaf055

Xiaoxue Chen, Yue Zhou, Lin Lu, Ming Feng, Linjie Wang, Anli Tong

{"title":"A Corticotropin-Secreting Adenoma in the Setting of von Hippel-Lindau Disease.","authors":"Xiaoxue Chen, Yue Zhou, Lin Lu, Ming Feng, Linjie Wang, Anli Tong","doi":"10.1210/jcemcr/luaf055","DOIUrl":"https://doi.org/10.1210/jcemcr/luaf055","url":null,"abstract":"Von Hippel-Lindau (VHL) disease is an autosomal dominant disorder caused by germline pathogenic variants of the VHL gene, which can lead to abnormal growth of blood vessels and cause the development of benign or malignant tumors, as well as cysts in diverse organs. To date, no case reports have documented adrenocorticotropic hormone (ACTH)-secreting adenomas in individuals with VHL disease. We present the case of a 19-year-old female individual with VHL disease who developed an ACTH-secreting adenoma alongside hemangioblastomas in the central nervous system (CNS) and cystic lesions in diverse organ systems. Genetic testing and immunohistochemistry of the pituitary tumor were performed. Genetic testing revealed that the patient carried the familial germline pathogenic variant located in the first exon of the VHL gene (c.227_229del, p.76delF). Immunohistochemical staining of the pituitary tumor demonstrated positive for ACTH, chromogranin A, and synaptophysin, with Ki-67 index at 3%. In addition, tumor cells showed scattered immunoreactivity for the α subunit of hypoxia-inducible factor (HIF-1α). This case suggests that VHL disease might be associated with ACTH-secreting adenomas and broadens the tumor spectrum.","PeriodicalId":73540,"journal":{"name":"JCEM case reports","volume":"3 5","pages":"luaf055"},"PeriodicalIF":0.0,"publicationDate":"2025-04-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11982611/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144060005","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

Hypercortisolism Due to Paraganglioma Secreting Adrenocorticotropin and Catecholamines. 副神经节瘤分泌促肾上腺皮质激素和儿茶酚胺所致的高皮质醇症。

JCEM case reports Pub Date : 2025-04-10 eCollection Date: 2025-05-01 DOI: 10.1210/jcemcr/luaf052

Drew W Cox, Aleona C Zuzek, Matthew Genco, Tammy Holm, Shailendra B Patel

引用次数: 0

Insulinoma Mimic: Tramadol-induced Hypoglycemia. 胰岛素瘤模拟：曲马多诱导的低血糖。

JCEM case reports Pub Date : 2025-03-13 eCollection Date: 2025-03-01 DOI: 10.1210/jcemcr/luaf034

Shannon O'Hara, Tyler Hinshaw, Mathew McKenzie, Victoria Belcher, Ian McCoy, Adnan Haider

{"title":"Insulinoma Mimic: Tramadol-induced Hypoglycemia.","authors":"Shannon O'Hara, Tyler Hinshaw, Mathew McKenzie, Victoria Belcher, Ian McCoy, Adnan Haider","doi":"10.1210/jcemcr/luaf034","DOIUrl":"10.1210/jcemcr/luaf034","url":null,"abstract":"Endogenous hyperinsulinemia hypoglycemia has numerous etiologies. The objective of this report is to describe a patient with severe hyperinsulinemic hypoglycemia with no known history of diabetes or hypoglycemia who presented with acute altered mental status. Blood glucose was noted to be 40 mg/dL (2.22 mmol/L) (reference range 65-125 mg/dL; 3.61-6.94 mmol/L). The patient's sulfonylurea screen was negative. Following 1 mg glucagon injection, the patient's glucose did not improve, a response inconsistent with insulinoma. Imaging studies of the pancreas did not show pancreatic mass. Two weeks before the presentation, the patient started on tramadol for back pain with the dose increased 3 days prior to presentation. The patient's hypoglycemia resolved and returned to baseline 4 days after the initial presentation. Tramadol has been reported to cause hypoglycemia, especially in the elderly population. Tramadol may act on μ receptors on β cells to upregulate insulin secretion. When approaching a patient with endogenous hyperinsulinemia, one should consider tramadol or other analgesics as a possible etiology.","PeriodicalId":73540,"journal":{"name":"JCEM case reports","volume":"3 3","pages":"luaf034"},"PeriodicalIF":0.0,"publicationDate":"2025-03-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11903389/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143627023","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