JCEM Case Reports最新文献

Recurrent Falls Due to Hypoglycemia: Case of an IGF-2-producing Fibrous Tumor of the Pleura 低血糖导致的反复跌倒：胸膜IGF-2纤维瘤病例

JCEM Case Reports Pub Date : 2024-04-23 DOI: 10.1210/jcemcr/luae061

Mathijs Cornelis Guijt, David J Heineman, Jacqueline Thérèse Jonker

{"title":"Recurrent Falls Due to Hypoglycemia: Case of an IGF-2-producing Fibrous Tumor of the Pleura","authors":"Mathijs Cornelis Guijt, David J Heineman, Jacqueline Thérèse Jonker","doi":"10.1210/jcemcr/luae061","DOIUrl":"https://doi.org/10.1210/jcemcr/luae061","url":null,"abstract":"Abstract This case report delineates the clinical presentation of a 77-year-old male who experienced falls and sustained a humerus fracture attributed to hypoglycemia. Despite the absence of insulin use and normal laboratory results for cortisol, TSH, blood count, and liver and kidney function, a fasting test revealed diminished C-peptide and insulin levels, ruling out insulinoma, exogenous insulin use, or β-cell hyperplasia. Subsequent laboratory investigations demonstrated lowered IGF-1 and elevated IGF-2 levels, indicative of an IGF-2-producing tumor as the etiology of the hypoglycemia. A positron emission tomography computed tomography scan identified a right-sided thoracic cavity tumor, prompting an open resection. Postoperatively, hypoglycemic episodes abated within 2 days, and pathology confirmed a 14.9-cm solitary fibrous tumor. Nonislet cell tumor hypoglycemia (NICTH), also known as Doege Potter syndrome, arises from aberrant production of IGF-2 or its precursors. Elevated IGF-2 levels induce hypoglycemia through heightened glucose uptake on binding to insulin receptors. The literature supports the efficacy of both surgical intervention and corticosteroids in managing NICTH. This case underscores the importance of considering NICTH as a rare etiology in unexplained hypoglycemia cases, advocating for the utility of fasting tests in diagnosis, and suggesting surgical resection as a viable treatment option when radical excision is feasible.","PeriodicalId":510224,"journal":{"name":"JCEM Case Reports","volume":"80 10","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-04-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140667717","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Adrenocortical Adenoma With Protrusion Into the Inferior Vena Cava Initially Suspected to Be Adrenocortical Carcinoma 肾上腺皮质腺瘤突入下腔静脉，最初怀疑为肾上腺皮质癌

JCEM Case Reports Pub Date : 2024-04-23 DOI: 10.1210/jcemcr/luae043

Hiroki Takizawa, Reo Higuchi, Yuki Fukumura, Muga Terasawa, Katsuhiro Sano, Hiromasa Goto

{"title":"Adrenocortical Adenoma With Protrusion Into the Inferior Vena Cava Initially Suspected to Be Adrenocortical Carcinoma","authors":"Hiroki Takizawa, Reo Higuchi, Yuki Fukumura, Muga Terasawa, Katsuhiro Sano, Hiromasa Goto","doi":"10.1210/jcemcr/luae043","DOIUrl":"https://doi.org/10.1210/jcemcr/luae043","url":null,"abstract":"Abstract Adrenal tumors with invasion into the inferior vena cava (IVC) are typically malignant. Here, we present a case of adrenocortical adenoma with protrusion into the IVC. A 70-year-old man was referred to our hospital after his magnetic resonance imaging scan of the abdomen coincidently revealed a right adrenal tumor invading the IVC. We suspected an aggressive adrenal carcinoma and tumor resection was performed. However, all 3 existing pathological criteria (Weiss, modified Weiss, and Helsinki) suggested the tumor was benign. Immunohistochemistry for CD31 showed the tumor inside the central adrenal vein (CAV), right adrenal vein (RAV), and IVC was entirely covered with CD31-positive vascular endothelial cells. The CAV is known to sometimes lack smooth muscle in its walls and normal adrenocortical cells covered by endothelial cells sometimes protrude into the CAV from this gap. These findings suggest that this tumor likely protruded into the IVC by pushing against the CAV wall, rather than by invasion into the vascular wall. In the case with adrenal tumors protruding into the IVC, the fact that the tumor surface was covered by vascular endothelial cells was considered supportive of its benign nature.","PeriodicalId":510224,"journal":{"name":"JCEM Case Reports","volume":"13 8","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-04-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140672021","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

A Large Benign Adrenocortical Adenoma Cosecreting Testosterone and Cortisol 分泌睾酮和皮质醇的巨大良性肾上腺皮质腺瘤

JCEM Case Reports Pub Date : 2024-04-23 DOI: 10.1210/jcemcr/luae045

Martha Dillon, Sara Shteyman, Samaneh Rabiehashemi, P. Madhavan, P. Luthra

{"title":"A Large Benign Adrenocortical Adenoma Cosecreting Testosterone and Cortisol","authors":"Martha Dillon, Sara Shteyman, Samaneh Rabiehashemi, P. Madhavan, P. Luthra","doi":"10.1210/jcemcr/luae045","DOIUrl":"https://doi.org/10.1210/jcemcr/luae045","url":null,"abstract":"Abstract Most adrenal incidentalomas are benign neoplasms of the adrenal cortex. While the majority are nonfunctional, many secrete cortisol. Androgen- or estrogen-secreting adenomas are rare. A 44-year-old female, with history of hypertension and prediabetes, presented with worsening acne, hirsutism, secondary amenorrhea for 2 years, and a 40-pound weight gain. Laboratory evaluation showed high 24-hour urine free cortisol, suppressed adrenocorticotropic hormone (ACTH) level, indicative of ACTH independent Cushing syndrome, and elevated testosterone and androstenedione. Abdominal computed tomography (CT) revealed a 6.3 × 5.2 × 5.6 cm left adrenal mass. Patient underwent left open adrenalectomy. Pathology revealed benign adrenocortical adenoma. Postoperatively there was a significant improvement in her blood pressure and blood sugar levels, resumption of menses, and complete resolution of hyperandrogenism and hypercortisolism. We describe a patient with an adrenal adenoma cosecreting cortisol and androgen, leading to Cushing syndrome and significant virilization. Adrenal masses secreting androgens are less common and concerning for adrenocortical carcinoma (ACC). Patients with adrenal masses cosecreting multiple hormones should undergo workup expediently since ACC confers poor outcomes.","PeriodicalId":510224,"journal":{"name":"JCEM Case Reports","volume":"43 1","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-04-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140666388","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

A Rare Prolactin-secreting Pituitary Carcinoma With Epidural and Thecal Metastases 伴有硬膜外和鳞状转移的罕见分泌催乳素垂体癌

JCEM Case Reports Pub Date : 2024-04-23 DOI: 10.1210/jcemcr/luae047

Anna Liu, Stan Van Uum, Donald Lee, Robert R. Hammond, Shereen Ezzat, Kristin K Clemens

{"title":"A Rare Prolactin-secreting Pituitary Carcinoma With Epidural and Thecal Metastases","authors":"Anna Liu, Stan Van Uum, Donald Lee, Robert R. Hammond, Shereen Ezzat, Kristin K Clemens","doi":"10.1210/jcemcr/luae047","DOIUrl":"https://doi.org/10.1210/jcemcr/luae047","url":null,"abstract":"Abstract Pituitary carcinomas are rare but associated with significant morbidity and mortality. They remain challenging to diagnose and manage. In this case, we describe a 56-year-old man who presented with erectile dysfunction and binocular vertical diplopia. He had central hypogonadism, secondary adrenal insufficiency, and central hypothyroidism on biochemical testing. His serum prolactin was 1517 mcg/L (1517 ng/mL; reference range 4-15 mcg/L), and his sellar magnetic resonance imaging showed a 2.0 × 2.2 × 3.1 cm pituitary tumor. Pathology revealed a prolactin-secreting carcinoma. Despite treatment with a high-dose dopaminergic, 2 transsphenoidal resections, and 1 course of radiation, prolactin levels continued to rise. He developed metastases to the epidural space and thecal sac from the thoracic to sacral spine, for which he received 12 cycles of temozolomide chemotherapy with initial clinical and biochemical response. This was followed by disease escape and progression. We discuss the clinical and imaging features that warrant a high index of suspicion for pituitary carcinoma and review contemporary treatment.","PeriodicalId":510224,"journal":{"name":"JCEM Case Reports","volume":"83 2","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-04-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140670284","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Persistent Gynecomastia due to Short-term Low-dose Finasteride for Androgenetic Alopecia. 短期小剂量非那雄胺治疗雄激素性脱发导致的持续性妇科肿瘤。

JCEM Case Reports Pub Date : 2024-04-23 DOI: 10.1210/jcemcr/luae050

Hal Steven Farkas, Y. Jee, V. Szymczuk, E. W. Leschek

{"title":"Persistent Gynecomastia due to Short-term Low-dose Finasteride for Androgenetic Alopecia.","authors":"Hal Steven Farkas, Y. Jee, V. Szymczuk, E. W. Leschek","doi":"10.1210/jcemcr/luae050","DOIUrl":"https://doi.org/10.1210/jcemcr/luae050","url":null,"abstract":"We report a case of persistent gynecomastia in a healthy 20-year-old man after 1 month of low-dose finasteride. Finasteride was discontinued after 2 months, and gynecomastia was unchanged 5 months after drug withdrawal. The gynecomastia regressed but did not resolve after 6 months of treatment with raloxifene, a selective estrogen receptor modulator. One year later, bilateral mammoplasty was performed to remove the remaining breast tissue. Finasteride, a 5-alpha-reductase inhibitor, is widely used for the treatment of androgenetic alopecia. Gynecomastia is an expected side effect of this therapy given its mechanism of action. However, only 8 cases of gynecomastia have been reported with low-dose (1 mg daily) finasteride treatment since its approval for androgenetic alopecia in 1997. This raises the concern that gynecomastia resulting from low-dose finasteride is significantly underreported, causing inadequately informed patients. Further, because of the risk of gynecomastia, it is important for prescribing physicians to counsel patients regarding this complication and to consider early intervention when finasteride-induced gynecomastia first arises to prevent fibrosis and thus irreversible gynecomastia.","PeriodicalId":510224,"journal":{"name":"JCEM Case Reports","volume":"88 23","pages":"luae050"},"PeriodicalIF":0.0,"publicationDate":"2024-04-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140670246","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Emerging Role of Gallium-68 DOTANOC PET/CT Guided Radiofrequency Ablation in the Treatment of Tumor-induced Osteomalacia 镓-68 DOTANOC PET/CT 引导射频消融在治疗肿瘤诱发骨软化症中的新作用

JCEM Case Reports Pub Date : 2024-04-23 DOI: 10.1210/jcemcr/luae044

Jyoti Sharma, Rajeev Kasliwal, Tarun Jain, Gaurav Kant Sharma

{"title":"Emerging Role of Gallium-68 DOTANOC PET/CT Guided Radiofrequency Ablation in the Treatment of Tumor-induced Osteomalacia","authors":"Jyoti Sharma, Rajeev Kasliwal, Tarun Jain, Gaurav Kant Sharma","doi":"10.1210/jcemcr/luae044","DOIUrl":"https://doi.org/10.1210/jcemcr/luae044","url":null,"abstract":"Abstract Tumor-induced osteomalacia (TIO) is a rare acquired form of hypophosphatemia that can be cured when the tumor responsible is completely removed. These tumors can be small and located in anatomically challenging areas, rendering surgery both risky and extensive. Radiofrequency ablation (RFA) has been explored as an effective treatment option for such tumors. We present a case of a 35-year-old man exhibiting clinical and biochemical features consistent with TIO. The culprit lesion was not detectable on the whole-body computed tomography (CT) scan. Gallium (Ga-68) DOTANOC positron emission tomography (PET)/CT showed increased uptake in the left acetabulum and magnetic resonance imaging (MRI) confirmed the location of the tumor. Given the risky anatomical location, we opted for less-invasive RFA. Following an unsuccessful attempt at CT-guided RFA of the lesion, we used real-time Ga-68 DOTANOC PET/CT guidance for precise imaging during the ablation procedure. Our patient achieved complete remission both clinically and biochemically after RFA. This response was also evident by the absence of tracer uptake in follow-up imaging. In conclusion, DOTANOC PET/CT–guided RFA can serve as a safe and effective treatment for patients with tumors causing TIO. This modality proves valuable when surgical resection is not a viable option.","PeriodicalId":510224,"journal":{"name":"JCEM Case Reports","volume":"27 11","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-04-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140667511","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Two Cases of Adrenal Cysts Lined by Thyroid Follicular Epithelium: Addressing Cellular Origin and Malignancy Concerns 两例甲状腺滤泡上皮衬里肾上腺囊肿：解决细胞起源和恶性肿瘤问题

JCEM Case Reports Pub Date : 2024-04-01 DOI: 10.1210/jcemcr/luae058

Maki Kanzawa, Tomonori Kanda, H. Fukuoka, Katsumi Shigemura, Yasuhiro Nakamura, Tomoo Itoh

{"title":"Two Cases of Adrenal Cysts Lined by Thyroid Follicular Epithelium: Addressing Cellular Origin and Malignancy Concerns","authors":"Maki Kanzawa, Tomonori Kanda, H. Fukuoka, Katsumi Shigemura, Yasuhiro Nakamura, Tomoo Itoh","doi":"10.1210/jcemcr/luae058","DOIUrl":"https://doi.org/10.1210/jcemcr/luae058","url":null,"abstract":"Abstract Adrenal cysts lined by thyroid follicular epithelium are rare, with only 14 reported cases of “ectopic thyroid tissue” to date. While the primary consideration for differential diagnosis is thyroid carcinoma metastasis, exclusion of metastases is determined based on the absence of a primary thyroid lesion, serological euthyroidism, lack of thyroglobulin elevation, and absence of epithelial atypia. Herein, we report 2 cases of adrenal cysts lined by thyroid follicular epithelium. Case 1 was a 60-year-old woman with a right adrenal cyst. Case 2 was a 51-year-old man with a left adrenal cyst. Over time, both cysts became larger, necessitating an adrenalectomy. Cystic epithelia were lined with thyroid follicular epithelium, exhibiting moderate atypia. Human bone marrow endothelial cell marker-1 and galectin-3 were focally positive; CK19 was positive in Case 1, and all 3 markers were positive in Case 2, previously reported as an immunophenotype of thyroid carcinoma. CD56 expression was positive in both cases. Targeted next-generation sequencing revealed several low-frequency mutations; however, no major driver alterations for thyroid cancer were detected. Adrenal cysts can be lined by thyroid follicular epithelium. Challenges arise in determining the malignant or benign nature of adrenal cysts.","PeriodicalId":510224,"journal":{"name":"JCEM Case Reports","volume":"417 1","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-04-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140758161","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Utility of Repeat Sampling in Bilateral Aldosterone Suppression During Adrenal Vein Sampling for Primary Aldosteronism 肾上腺静脉取样治疗原发性醛固酮增多症时，重复取样对双侧醛固酮抑制的作用

JCEM Case Reports Pub Date : 2024-04-01 DOI: 10.1210/jcemcr/luae051

Bella Halim, Eric X Z Yong, Matthew Egan, Richard J MacIsaac, David O’Neal, Nirupa Sachithanandan

引用次数: 0

Metformin-Associated Vitamin B12 Deficiency: An Underrecognized Complication 二甲双胍相关维生素 B12 缺乏症：未被充分认识的并发症

JCEM Case Reports Pub Date : 2024-04-01 DOI: 10.1210/jcemcr/luae029

Y. Thewjitcharoen, Peachaphol Chongvoranond, S. Nakasatien, S. Krittiyawong, T. Himathongkam

{"title":"Metformin-Associated Vitamin B12 Deficiency: An Underrecognized Complication","authors":"Y. Thewjitcharoen, Peachaphol Chongvoranond, S. Nakasatien, S. Krittiyawong, T. Himathongkam","doi":"10.1210/jcemcr/luae029","DOIUrl":"https://doi.org/10.1210/jcemcr/luae029","url":null,"abstract":"Abstract Metformin-associated vitamin B12 deficiency is a well-established side effect, especially in patients taking higher doses of metformin or who have existing risk factors. Severe deficiency causes a wide range of systemic disorders. Gait instability, which leads to frequent falling, is usually an underrecognized side effect. Older patients are more likely to develop chronic subdural hematoma even with minor trauma. We present a case of 84-year-old man with type 2 diabetes mellitus with acute-on-chronic subdural hematoma from frequent falls. Metformin therapy at dose of 1700 mg/day was given for more than 25 years. He had been in his usual state of health until 10 months ago when he began to have frequent fallings and fatigue. Physical examination in this admission revealed new-onset impaired vibratory sensation and proprioception in both feet and positive Romberg test. Subsequent evaluations demonstrated undetectable plasma vitamin B12 level and elevated plasma homocysteine. Improvement in neurological symptoms occurred within 1 week of vitamin B12 replacement and surgical hematoma evacuation. This case highlights the importance of awareness and periodic monitoring of vitamin B12 status among older patients taking metformin.","PeriodicalId":510224,"journal":{"name":"JCEM Case Reports","volume":"252 ","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-04-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140793578","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Attenuation of Bone Mineral Density Decline During Anemia Treatment With Methenolone Acetate in Myelodysplastic Syndrome 骨髓增生异常综合征患者使用醋酸甲地孕酮治疗贫血期间骨矿物质密度下降的减缓作用

JCEM Case Reports Pub Date : 2024-04-01 DOI: 10.1210/jcemcr/luae055

Shu Ushimaru, H. Sumi, Mea Aso, Rie Fujishima, Kazuhiro Shiizaki, Naoto Tominaga

引用次数: 0