JCEM case reports最新文献

Empagliflozin-induced Myopathy.

JCEM case reports Pub Date : 2024-11-28 eCollection Date: 2024-12-01 DOI: 10.1210/jcemcr/luae216

Sana Khan, Ashley Broce

引用次数: 0

Severe, Symptomatic Hypercalcemia Secondary to PTH-secreting Pancreatoblastoma.

JCEM case reports Pub Date : 2024-11-28 eCollection Date: 2024-12-01 DOI: 10.1210/jcemcr/luae217

Anand D Gandhi, James D McCallum, Jonathan S Fisher

{"title":"Severe, Symptomatic Hypercalcemia Secondary to PTH-secreting Pancreatoblastoma.","authors":"Anand D Gandhi, James D McCallum, Jonathan S Fisher","doi":"10.1210/jcemcr/luae217","DOIUrl":"10.1210/jcemcr/luae217","url":null,"abstract":"Hypercalcemia may be induced by a variety of etiologies, most commonly primary hyperparathyroidism. Although primary hyperparathyroidism represents a relatively common endocrinological disorder, ectopic PTH secretion is a rare entity that is less well described in literature. We describe the first case to our knowledge of severe, symptomatic hypercalcemia found to be secondary to a PTH-secreting pancreatoblastoma. The patient initially presented with fatigue and progressive upper extremity intermittent muscular twitching. He was found to have biochemical evidence of primary hyperparathyroidism. A computed tomography scan of the neck and a sestamibi nuclear scan failed to definitively demonstrate a parathyroid adenoma or hyperplasia and bilateral surgical parathyroid exploration was unrevealing for any pathology. Abdominal imaging via computed tomography was obtained for evaluation of progressive postoperative epigastric pain, and the patient was found to have a retroperitoneal mass that, after biopsy, was diagnostic for a pancreatoblastoma. This mass was resected resulting in a fall in intraoperative PTH values and subsequent postoperative hypocalcemia secondary to hungry bone syndrome. Upon follow-up, the patient's parathyroid function recovered and doses of supplemental calcium and vitamin D could be tapered. Ectopic PTH-secreting masses represent a rare entity but should be considered in individuals with unclear etiology of recalcitrant primary hyperparathyroidism.","PeriodicalId":73540,"journal":{"name":"JCEM case reports","volume":"2 12","pages":"luae217"},"PeriodicalIF":0.0,"publicationDate":"2024-11-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11604075/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142752499","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

Severe Hypertriglyceridemia in a Patient Treated With Sirolimus for Graft-vs-Host Disease Prophylaxis. 使用西罗莫司预防移植物抗宿主病的患者出现严重的高甘油三酯血症。

JCEM case reports Pub Date : 2024-11-04 eCollection Date: 2024-11-01 DOI: 10.1210/jcemcr/luae193

Kristin Criner, Jeffrey Student, Jacob Arkin, Julia Carp, Samantha Sokoloff

引用次数: 0

Ectopic Cervical Thymic Tissue Mimicking Parathyroid Hyperplasia: A Rare Occurrence Linked to Graves Disease. 模仿甲状旁腺增生的异位宫颈胸腺组织：与巴塞杜氏病有关的罕见病例

JCEM case reports Pub Date : 2024-11-04 eCollection Date: 2024-11-01 DOI: 10.1210/jcemcr/luae197

Waralee Chatchomchuan, Yotsapon Thewjitcharoen, Soontaree Nakasatien, Ronnarat Suvikapakornkul, Somboon Keerawat, Thep Himathongkam

{"title":"Ectopic Cervical Thymic Tissue Mimicking Parathyroid Hyperplasia: A Rare Occurrence Linked to Graves Disease.","authors":"Waralee Chatchomchuan, Yotsapon Thewjitcharoen, Soontaree Nakasatien, Ronnarat Suvikapakornkul, Somboon Keerawat, Thep Himathongkam","doi":"10.1210/jcemcr/luae197","DOIUrl":"10.1210/jcemcr/luae197","url":null,"abstract":"Thymic hyperplasia has occasionally been reported in patients with Graves disease (GD). However, ectopic cervical thymic hyperplasia in the setting of hyperthyroid GD is exceptionally rare. We describe a case of a 54-year-old Thai woman who presented with hyperthyroidism, diplopia, and pretibial myxedema. She underwent a total thyroidectomy because of diplopia caused by Graves ophthalmopathy. During the surgery, 3 macroscopically abnormal enlargements of parathyroid gland-like tissue were identified and removed. Histopathology revealed hyperplastic thymic tissue mixed with 1 normal-sized parathyroid gland at the location of the left upper parathyroid gland, and thymic tissue was found in the sample labeled as the right upper parathyroid gland. Notably, the sample labeled as the right lower parathyroid gland was actually determined to be a lymph node. Preoperative blood samples showed normal serum calcium and parathyroid hormone levels. Postoperatively, computed tomography of the chest showed thymic hyperplasia in the anterior mediastinum, which slightly regressed at the 9-month follow-up. The patient had transient hypoparathyroidism requiring oral calcium and active vitamin D supplements for 6 months postoperatively. Ectopic cervical thymic hyperplasia can be found in GD and might be indistinguishable from parathyroid hyperplasia. Biochemical evaluations are required to exclude concomitant hyperparathyroidism, and a conservative approach should be considered.","PeriodicalId":73540,"journal":{"name":"JCEM case reports","volume":"2 11","pages":"luae197"},"PeriodicalIF":0.0,"publicationDate":"2024-11-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11532649/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142577420","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

Hypoglycemia Associated With Hypermobile Ehlers-Danlos Syndrome. 与高移动性埃勒斯-丹洛斯综合征有关的低血糖症。

JCEM case reports Pub Date : 2024-11-04 eCollection Date: 2024-11-01 DOI: 10.1210/jcemcr/luae205

Hamayle Saeed, Amanda Sheehan, Mary-Elizabeth Patti

引用次数: 0

Pembrolizumab-induced Thyroiditis, Hypophysitis and Adrenalitis: A Case of Triple Endocrine Dysfunction. Pembrolizumab诱发甲状腺炎、肾上腺皮质炎和肾上腺炎：三重内分泌功能障碍病例。

JCEM case reports Pub Date : 2024-11-04 eCollection Date: 2024-11-01 DOI: 10.1210/jcemcr/luae200

Silvia Rossi, Francesca Silvetti, Monia Bordoni, Alessandro Ciarloni, Gianmaria Salvio, Giancarlo Balercia

{"title":"Pembrolizumab-induced Thyroiditis, Hypophysitis and Adrenalitis: A Case of Triple Endocrine Dysfunction.","authors":"Silvia Rossi, Francesca Silvetti, Monia Bordoni, Alessandro Ciarloni, Gianmaria Salvio, Giancarlo Balercia","doi":"10.1210/jcemcr/luae200","DOIUrl":"10.1210/jcemcr/luae200","url":null,"abstract":"Immune checkpoint inhibitor drugs can trigger autoimmune endocrine reactions as a known side effect. Several cases of immunotherapy-induced autoimmune endocrinopathies have been described, but multiple sequential endocrine toxicities are a rare occurrence. A 39-year-old patient with metastatic melanoma started adjuvant therapy with pembrolizumab. One month later he presented with asymptomatic thyrotoxicosis and, within several weeks, overt hypothyroidism, for which he started levothyroxine therapy. Subsequently the patient developed central adrenal insufficiency due to probable hypophysitis, and steroid replacement therapy was started. Pembrolizumab therapy was then discontinued. After a few months, a full recovery of pituitary function was observed, but primary adrenal insufficiency occurred, requiring additional fludrocortisone therapy. The described clinical case is a very uncommon case of triple endocrinological toxicity from immunotherapy. The clinical and biochemical manifestations of immunotherapy-induced endocrinopathies can be variable and atypical; therefore, it is necessary to pay special attention to any clue of hormonal dysfunction.","PeriodicalId":73540,"journal":{"name":"JCEM case reports","volume":"2 11","pages":"luae200"},"PeriodicalIF":0.0,"publicationDate":"2024-11-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11532647/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142577401","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

An Unusual Movement Disorder-Case of Diabetic Striatopathy. 一种不寻常的运动障碍--糖尿病纹状体病病例。

JCEM case reports Pub Date : 2024-10-25 eCollection Date: 2024-11-01 DOI: 10.1210/jcemcr/luae201

Shazia Azmat, Owais Lodhi, Harish Ashok, Hussein Harb, Mahwash Siddiqui, Janice Gilden

引用次数: 0

Bilateral Knee Effusions Secondary to Zoledronic Acid Infusion. 继发于唑来膦酸输注的双侧膝关节积液

JCEM case reports Pub Date : 2024-10-25 eCollection Date: 2024-11-01 DOI: 10.1210/jcemcr/luae199

Jenny Wei, Catherine Soo Ihn Cho

引用次数: 0

Hypercalcemia Due to Progressive Disseminated Histoplasmosis. 进行性播散性组织胞浆菌病导致的高钙血症。

JCEM case reports Pub Date : 2024-10-22 eCollection Date: 2024-11-01 DOI: 10.1210/jcemcr/luae198

Lakshmipriya Thandiyekkal Rajan, Naman Aggarwal, Jayakrishnan C Menon, Subhash Yadav, Rungmei S K Marak

{"title":"Hypercalcemia Due to Progressive Disseminated Histoplasmosis.","authors":"Lakshmipriya Thandiyekkal Rajan, Naman Aggarwal, Jayakrishnan C Menon, Subhash Yadav, Rungmei S K Marak","doi":"10.1210/jcemcr/luae198","DOIUrl":"10.1210/jcemcr/luae198","url":null,"abstract":"Hypercalcemia is a relatively common clinical problem, and evaluation for its etiology may often prove to be challenging. However, a thorough etiological workup can guide effective therapy and can often prove to be lifesaving. We describe a 61-year-old man who presented with fever, anorexia, and weight loss for 3 months, and altered sensorium for around 1 week. His evaluation revealed severe hypercalcemia, correction of which led to improvement in his symptoms. Workup for the cause revealed that he had parathyroid hormone-independent hypercalcemia with elevated levels of 1,25-dihydroxyvitamin D, suggesting a granulomatous disease. Radiological evaluation was suggestive of a multisystem disorder with bilateral adrenal enlargement, generalized lymphadenopathy, and hepatosplenomegaly. Biopsy from the adrenal gland and bone marrow clinched the diagnosis of progressive disseminated histoplasmosis, which required treatment with liposomal amphotericin B for a total duration of 4 weeks, followed by oral itraconazole. The effective treatment was associated with normalization of serum calcium and disappearance of symptoms. Histoplasmosis represents a rare cause of hypercalcemia, with only around 22 such cases having been reported worldwide.","PeriodicalId":73540,"journal":{"name":"JCEM case reports","volume":"2 11","pages":"luae198"},"PeriodicalIF":0.0,"publicationDate":"2024-10-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11495557/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142516781","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

Long-Term Effects of Incretin-Based Drugs on Glycemic Control in Permanent Neonatal Diabetes. 基于分泌素的药物对永久性新生儿糖尿病患者血糖控制的长期影响。

JCEM case reports Pub Date : 2024-10-18 eCollection Date: 2024-11-01 DOI: 10.1210/jcemcr/luae188

Ayaka Oshiro, Ryoichiro Aotani, Wakako Sakamoto, Takanari Kitazono, Toshiaki Ohkuma

{"title":"Long-Term Effects of Incretin-Based Drugs on Glycemic Control in Permanent Neonatal Diabetes.","authors":"Ayaka Oshiro, Ryoichiro Aotani, Wakako Sakamoto, Takanari Kitazono, Toshiaki Ohkuma","doi":"10.1210/jcemcr/luae188","DOIUrl":"10.1210/jcemcr/luae188","url":null,"abstract":"Permanent neonatal diabetes mellitus (PNDM) is a genetic disorder, characterized by a decrease in endogenous insulin secretion. Therefore, exogenous insulin supplementation plays a central role in controlling glycemia. Although adding a sulfonylurea can help to discontinue insulin, discontinuation is sometimes difficult when the sulfonylurea is administered at older ages. A 24-year-old woman with longstanding PNDM who had poor glycemic control using insulin (47 U/day) and high-dose glibenclamide (0.6 mg/kg/day), had successfully discontinued insulin after initiating the dipeptidyl peptidase-4 inhibitor sitagliptin (50 mg/day). Additionally, hemoglobin A1c levels decreased by 4.8%. Double dosing of sitagliptin and subsequent switching to the glucagon-like peptide-1 receptor agonist semaglutide (0.25 mg/week followed by 0.5 mg/week) further decreased hemoglobin A1c values, with graded improvements in endogenous insulin secretion. There were no episodes of hypoglycemia during which glibenclamide was titrated down from 0.6 to 0.4 mg/kg/day. Intra- and inter-day glucose variability as assessed by continuous glucose monitoring was also improved. In patients with PNDM, administration and dose escalation of incretin-based drugs, in addition to a high-dose sulfonylurea, could be a useful treatment strategy. This strategy may be helpful for discontinuing insulin, downtitrating sulfonylureas, and subsequent achievement of better glycemic control regarding long-term stability and short-term variability.","PeriodicalId":73540,"journal":{"name":"JCEM case reports","volume":"2 11","pages":"luae188"},"PeriodicalIF":0.0,"publicationDate":"2024-10-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11487289/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142482397","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