Background: Multiple endocrine neoplasia type 1 (MEN1) is a rare autosomal dominant disorder characterized by the occurrence of combined tumors in different glands, usually the parathyroid, pancreas and pituitary, as well as in other parts of the digestive tract. The present study describes the phenotype of a Brazilian family with MEN1 caused by a previously unreported MEN1 gene mutation. Case Report: We report the case of a 41-year-old male, the proband, who presented with angiofibromas, primary hyperparathyroidism, macroprolactinoma, and pancreatic neuroendocrine tumor. Next generation sequencing analysis of the MEN1 gene in the patient's peripheral blood DNA sample revealed a deletion of 16 base pairs (c.1366-12_1369del;p) resulting in a framing error. Additional 5 members of the family (4 brothers and a first cousin) presented with clinical features of MEN1. All brothers underwent mutation screening and tested positive for the same genetic variant. Two of them were also diagnosed with papillary thyroid carcinoma. Discussion: The c.1366-12_1369del;p mutation is located between the 10th and last exon of the MEN 1 gene and it's preceding intron, encompassing the canonical sites in the splice junction. The 10th exon of MEN1, possibly lost with this variant, encodes the last 163 amino acids that compose the Menin protein's C-terminal region, which harbors nuclear localization signals essential for its internalization into the nuclear compartment and interaction with the nuclear matrix. Conclusion: Our case reports add to the literature the description of a new pathogenic variant of the MEN 1 gene.
Background/Objective: Thyrotoxic periodic paralysis (TPP) is a rare condition causing weakness of the lower extremities associated with significant hypokalemia. Likewise, agranulocytosis due to methimazole use is a rare occurrence. We present the first documented case of concomitant TPP and agranulocytosis from methimazole use. Case Report: A 48-year-old woman presented with sore throat, fevers, odynophagia, and suddenonset bilateral leg weakness. Methimazole had been started 10 weeks prior for a new diagnosis of Graves' disease. On admission, the patient was febrile, tachycardic, thyrotoxic, and neutropenic. She also experienced near-paralysis of the lower extremities. She was diagnosed with TPP and treated with beta blockade. She was admitted to the intensive care unit and started on broad-spectrum antibiotics, lithium, and propranolol for treatment of septic shock and hyperthyroidism, respectively. Given persistent hypokalemia despite 2 days of therapy, she was also diagnosed with refeeding syndrome. Discussion: TPP is a rare entity, though it should be considered on the differential for any thyrotoxic patient presenting with sudden weakness. If the associated hypokalemia does not begin to normalize within 48 h of beta blockade, other etiologies should be investigated. Lastly, alternative treatments such as lithium may be used to control hyperthyroidism in patients with methimazole-induced agranulocytosis. Conclusion: While methimazole-induced agranulocytosis and thyrotoxic periodic paralysis are independently rare diagnoses, the combination of the 2 is exceedingly rare, and our case represents the first documented case in the literature reflecting a patient suffering from both syndromes. (c) 2024 AACE. Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
Background/Objective: Papillary thyroid cancer (PTC) is considered the most common form of thyroid cancer. Papillary thyroid microcarcinomas (PTMCs) are defined as papillary carcinomas that are <1 cm and are often identified on surgical pathology but may be missed by ultrasound imaging. Case Report: Our case describes a 24-year-old woman who initially presented with a right lateral neck lump. This was thought to be a lipoma, and no workup was performed until it began to grow during pregnancy 3 to 4 years after initial presentation. The patient underwent excision with findings of a level 5B lymph node with pathology consistent with PTC, follicular variant. Neck ultrasound performed in an imaging center and in our clinic did not reveal thyroid nodules or abnormal appearing lymph nodes. Solid tumor profiling assay identified a neurotrophic tropomyosin receptor kinase (NTRK)-3 fusion: ETV6(4)-NTRK3(14) mutation. Discussion: PTMCs are identified with increased frequency due to improved ultrasound imaging and fine needle aspiration techniques. PTMCs are often associated with a benign course and are rarely metastatic. There has been growing interest in NTRK fusion mutations in PTC that has been associated with an increased risk of metastasis. Conclusion: This case is unusual in that multiple cervical nodal metastases were present even without a sizable primary thyroid tumor. Although more studies are needed to elucidate the clinical and prognostic significance of NTRK fusion mutations in PTMC, their response to tropomyosin receptor kinase inhibitors may represent a future pathway for treatment. (c) 2024 AACE. Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
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.
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 (12.332.9 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. (c) 2025 AACE. Published by Elsevier Inc. This is an open access article under the CC BY license (http:// creativecommons.org/licenses/by/4.0/).
Objective/Background Tumor-induced osteomalacia (TIO) is a rare paraneoplastic syndrome caused by excessive secretion of fibroblast growth factor 23 (FGF-23) by phosphaturic mesenchymal tumors. This leads to hypophosphatemia, vitamin D deficiency, and impaired bone metabolism. TIO is often misdiagnosed due to its rarity and nonspecific symptoms. Case Report We report a 58-year-old male presenting with multiple non-traumatic fractures, muscle weakness, and functional decline. Laboratory evaluation revealed hypophosphatemia, elevated parathyroid hormone (PTH), reduced 1,25-dihydroxyvitamin D, and markedly elevated FGF-23 levels. Imaging identified a soft tissue mass in the plantar region of the right foot, which was confirmed as a phosphaturic mesenchymal tumor upon pathological analysis. The patient underwent surgical resection, resulting in rapid normalization of biochemical abnormalities, including serum phosphorus, PTH, and 1,25-dihydroxyvitamin D, within five days. Discussion This case underscores the importance of recognizing TIO in patients with unexplained hypophosphatemia and fractures. The curative potential of tumor resection was demonstrated with rapid biochemical and clinical improvement. Diagnostic challenges often arise due to the rarity and atypical presentation of these tumors, particularly in uncommon locations such as the plantar region. Emerging therapies, such as FGF-23 inhibitors like burosumab, provide alternatives for non-localizable or unresectable tumors. Conclusion This case emphasizes the need for increased clinician awareness, multidisciplinary approaches, and advances in diagnostic imaging to reduce delays in diagnosing TIO. Further research is necessary to elucidate the pathophysiology, explore genetic associations, and improve treatment options for this debilitating condition.
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.
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.
Background/Objective Multiple endocrine neoplasia type 1 (MEN1) is a rare autosomal dominant disorder characterized by the occurrence of combined tumors in different glands, usually the parathyroid, pancreas and pituitary, as well as in other parts of the digestive tract. The present study describes the phenotype of a Brazilian family with MEN1 caused by a previously unreported MEN1 gene mutation. Case Report We report the case of a 41-year-old male, the proband, who presented with angiofibromas, primary hyperparathyroidism, macroprolactinoma, and pancreatic neuroendocrine tumor. Next generation sequencing analysis of the MEN1 gene in the patient's peripheral blood DNA sample revealed a deletion of 16 base pairs (c.1366-12_1369del;p) resulting in a framing error. Additional five members of the family (four brothers and a first cousin) presented with clinical features of MEN1. All brothers underwent mutation screening and tested positive for the same genetic variant. Two of them were also diagnosed with papillary thyroid carcinoma. Discussion The c.1366-12_1369del;p mutation is located between the 10th and last exon of the MEN 1 gene and it´s preceding intron, encompassing the canonical sites in the splice junction. The 10th exon of MEN1, possibly lost with this variant, encodes the last 163 amino acids that compose the Menin protein´s C-terminal region, which harbors nuclear localization signals essential for its internalization into the nuclear compartment and interaction with the nuclear matrix. Conclusion Our case reports adds to the literature the description of a new pathogenic variant of the MEN 1 gene.
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.
Background/Objective:The association between hypercalcemia and the risk of thromboembolic events is not clearly understood. Here, we present a unique case of a patient diagnosed with bilateral pulmonary thromboembolism in the setting of a parathyroid crisis due to primary hyperparathyroidism (PHPT). Our case may suggest a potential correlation between thromboembolism and PHPT with severe hypercalcemia. Nowadays just a few case reports provide support for this association, particularly in the settings of significant calcium and parathyroid hormone (PTH) derangement. Case Report:A 70-year-old woman presented to the hospital with a few weeks' onset of fatigue, difficulty walking, and shortness breath. Laboratory investigations revealed significantly elevated serum calcium (19.2 mg/dL) and PTH (1156 pg/mL) levels. Her past medical history was significant for PHPT with mild hypercalcemia since 2014. Computerized tomography and thyroid ultrasound of the neck showed a high suspicion of a left parathyroid adenoma. A computerized tomography angiogram of the chest revealed a bilateral lower lobe pulmonary embolism. The patient underwent medical management for hypercalcemia and pulmonary embolism, followed by parathyroidectomy. Pathology reports confirmed the diagnosis of parathyroid adenoma. Discussion:The clinical significance of hyperparathyroidism, leading to subsequent hypercalcemia and its association with the development of a procoagulable state, has been elucidated in a very limited number of case reports. Conclusion:This case suggests that parathyroid crisis with hypercalcemia could potentially provoke thromboembolic events. However, this phenomenon could be explained by an extremely high level of PTH and calcium.
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.
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: 0.881.77 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. Published by Elsevier Inc. on behalf of the AACE. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
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.
Background/Objective: Multiple endocrine neoplasia 4 (MEN4) is a rare syndrome caused by germline mutations in CKDN1B, and it shares clinical manifestations with MEN1, including primary hyperparathyroidism, pituitary adenomas, and pancreatic neuroendocrine tumors (NETs). The prevalence of MEN4 is <1 per million, whereas prevalence of MEN1 is between 1/10 000 and 1/30 000. Case Report: A 51-year-old woman presented with symptomatic hypoglycemia and incidental hypercalcemia. Workup revealed a fasting plasma glucose level of 41 mg/dL (60-99 mg/dL), proinsulin level of 84.3 pmol/L (<8.0 pmol/L), insulin level of 24 uIU/mL (3-25 uIU/mL), c-peptide level of 5.2 ng/mL (1.1-4.4 ng/mL), and b-hydroxybutyrate level of 0.34 mmol/L (0.02-0.27 mmol/L), consistent with endogenous hyperinsulinism. Computed tomography scan of the abdomen revealed a 1.5 x 1.1 x 1.0 cm pancreatic head nodule. She underwent pancreaticoduodenectomy, and pathology demonstrated a well-differentiated neuroendocrine tumor with no metastases. She became normoglycemic after surgery, and additional workup revealed primary hyperparathyroidism. Germline testing revealed a variant of unknown significance in CDKN1B (p.R93W). Discussion: Both MEN1 and MEN4 result from decreased expression of p24 and exhibit similar clinical phenotypes, but there are subtle differences in penetrance and natural history. About 10% of patients with MEN1 have insulinomas, but no insulinomas have been reported in MEN4. primary hyperparathyroidism in MEN4 exhibits a lower risk of recurrence after parathyroidectomy. This case highlights the importance of germline genetic testing when a patient presents with manifestations of MEN1. Conclusion: To our knowledge, this is the first reported case of insulinoma in MEN4. (c) 2024 AACE. Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
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. (c) 2024 AACE. Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
Adrenal incidentalomas are increasingly diagnosed with the widespread use of cross-sectional imaging modalities. Though these are often unilateral, bilateral adrenal masses uncommonly occur and can be of similar or different origins. While the majority of adrenal masses are benign, bilateral masses are more likely to be associated with hormonal disturbances. These range from syndromes of hormone excess, such as mild autonomous cortisol secretion, to primary adrenal insufficiency due to gland infiltration or destruction. Bilateral masses may herald the diagnosis of germline pathogenic variants, hereditary tumor predisposition syndromes, malignancy, or even infection. In many cases, imaging characteristics play an important role in making the diagnosis in addition to biochemical evaluation. Herein, we present various imaging findings of 10 patients with bilateral adrenal masses as evaluated with computed tomography, magnetic resonance imaging, and positron emission tomography scans.
Background/Objective: Evidence on the efficacy and safety of minimally invasive treatment for insulinoma has increased over the past decade to the point of becoming a recommendation in clinical practice guidelines for the management of this type of neuroendocrine tumor. Case Report: We describe the case of an elderly male patient with multiple comorbidities and recurrent isolated insulinoma of 3.7 x 3.5 cm involving the uncinate process of the pancreas and contacting the splenomesenteric confluent many years after first resection, in whom, after refusing surgical management, was performed as successful arterial embolization of the pancreatic tumor. Discussion: When addressing this pathology, it is common to encounter patients who are not candidates for surgical management, either due to the presence of comorbidities, the location of the tumor in relation to vascular structures, or refusal of the intervention. Therefore, it is important to be aware of the different therapeutic options in localized and metastatic disease. Conclusion: Minimally invasive procedures are positioned as an effective alternative for the treatment of the hormonal overproduction in patients with insulinoma. (c) 2024 AACE. Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
