bluuebunny/crossref_metadata_2025_split huggingface dataset:
LBODP012 A Case of Addison's Disease Presented With Rhabdomyolysis And Psychosis
Authors: Kasimoglu Ismail Hakki , Dai-Ju Jenny Qian 
Published: November 2022

Abstract

Background
Addison's disease is a disorder of deficiencies in glucocorticoids and mineralocorticoids, mostly resulting from destruction of the adrenal cortex. Rhabdomyolysis and psychosis can be symptoms presented in cases of adrenal crisis.

Clinical Case
A 42-year-old male was hospitalized for rhabdomyolysis (CK 5200 U/L), hyponatremia (Na 117 mmol/L), hyperkalemia (K 6.4 mmol/dL) and acute renal failure (Creatinine 4.4 mg/dL). Past medical history was significant for a similar episode of rhabdomyolysis: negative EMG, muscle biopsy nonconclusive, diagnosed with autoimmune hypothyroidism and started on levothyroxine 50 mcg daily five years ago. Renal function improved and electrolyte abnormality was resolved with IV hydration. ACTH was elevated (580 pg/mL), cortisol and aldosterone were undetectable; patient was discharged on hydrocortisone 10 mg bid from outside hospital. His mood and personality changed significantly after he started hydrocortisone. One week later he was admitted to a psychiatry unit due to mania and psychosis, and was started on risperidone and divalproex sodium. Cosyntropin stimulation test confirmed primary adrenal insufficiency, likely autoimmune adrenalitis with positive 21-hydroxylase antibody. Hydrocortisone was replaced with prednisone 5mg daily and fludrocortisone 0.2 mg daily. Psychiatric symptoms completely resolved in two months; patient was taken off psychiatric medications. Clinical condition was stable in follow-up visits. Electrolytes and blood pressure continued to stay in normal range. This case of Addison's disease initially presented with rhabdomyolysis as a first symptom, manifested by acute psychosis in the course of treatment with hydrocortisone. The association between adrenal insufficiency and rhabdomyolysis has been reported, but the underlying mechanism is not clear. Several theories have been proposed to explain neuropsychiatric manifestations secondary to adrenal insufficiency (1); there is no definite answer yet.

Conclusion
Adrenal insufficiency should be evaluated in patients with rhabdomyolysis. Mental disturbances triggered by adrenal crisis and treatment should be considered by both endocrinologist and psychiatrist. Reference: (1) Farah, J., et al. Severe Psychotic Disorder as the Main Manifestation of Adrenal Insufficiency. Case Rep Psychiatry. 2015; 2015: 512430.
Presentation: No date and time listed
LBODP013 Hypophysitis And Adrenalitis Associated With The Use Of Immunotherapy "clinical Case Report"
Authors: Ramos Andres Felipe Garcia , Gonzalez Juanita , Vargas Laura Valentina Estupiñan , Vargas Hernando , Buenaventura Daisy , Monsalve Claudia , Torres Jose Luis , Pompey Nestor Alfonso Lopez , Ramirez Alex , Garcia Franco Vallejo , Aguilar Carolina , Aristizabal Natalia 
Published: November 2022

Abstract

Background
we present a case of hypophysitis with gonadotropic and somatotropic axis compromise and adrenalitis secondary to the use of nivolumab and ipilimumab.

Clinical Case
A 74-year-old woman with a history of metastatic renal cell carcinoma treated with ipilimumab and nivolumab for 10 months, suspended treatment due to pneumonitis. Three months later, she consulted the emergency department due to limitations in daily activities, asthenia, and drowsiness. physical examination was unremarkable except for hypotension. Laboratory studies showed profound hyponatremia (106 mEq/l, n: 135 a 145 mEq/l), low serum osmolality (236 mmol/kg n: 275 - 295 mmol/kg) and a basal cortisol (5 μg/dL n: 3.7 - 19.4 μg/dL). Hypophysitis was suspected and a pituitary profile revealed low somatomedin C (25 ng/ml n: 54 204 ng/ml), FSH (20.6 UI/L n: 26.7 -133.4 UI/L), and strikingly elevated ACTH (91.9 pg/mL n: 4.7 - 48.5 pg/mL). Pituitary MRI had no structural anomaly. A diagnosis of hypophysitis and primary adrenal insufficiency secondary to adrenalitis due to the use of immunotherapy was made. The compromise of the gonadotropic and somatotropic axis with a normal image can occur in up to 23% of patients with this clinical entity. The patient had significant clinical improvement with glucocorticoid and mineralocorticoid replacement.

Conclusions
To our knowledge this is the first reported case of hypophysitis and adrenalitis secondary to immunotherapy. Hypophysitis has a prevalence up to 8% with the combination of ipilimumab plus nivolumab. It should be considered despite a normal pituitary MRI. However, adrenalitis associated with the use of these drugs has only been reported in 2.6% and the coexistence of both entities is even rarer
Presentation: No date and time listed
LBODP015 A Pheochromocytoma Presenting With Hypertensive Emergency With A Rare Complication Of Unilateral Adrenal Hemorrhage
Authors: Arif Aneela , Siu Alfonso , Ahmed Nazaakat , Martini Alma , Shripad Namratha 
Published: November 2022

Abstract

Introduction
Adrenal hemorrhage is an uncommon finding in patients and is estimated to be present in 0.14-1.8% of postmortem examinations. Clinical diagnosis is challenging due to its non-specific presentation and most cases are diagnosed incidentally on imaging. Here we discuss the clinical presentation, diagnostic procedures, and outcome of this rare case.

Clinical Case
A 74-year-old Asian female presented to the ED with a sudden onset of severe epigastric pain with 9/10 intensity, radiating to her upper back while walking on the street. She was found in severe distress with a blood pressure of 214/80 mmHg, heart rate of 72 bpm, respiratory rate of 20 breaths/min, and oxygen saturation of 98% on room air. The physical exam was unremarkable. Laboratory findings were significant for elevated troponin of 1. 040 ng/ml (0. 00-0. 03 ng/ml), lactate 2.36mmol/L (0.70-2. 00 mmol/L), and lipase 481U/L (23-300 U/L). Chest X-ray was within normal limits. A CT angiogram of the chest and abdomen were negative for aortic dissection, however, a retroperitoneal hemorrhage measuring 6×8.5×6.5 cm was seen but an underlying adrenal gland mass could not be excluded. The patient was started on nitroglycerine drip for blood pressure control. Hemoglobin trend showed an acute drop, requiring 2 units of red blood cell transfusion. This raised concerns of worsening hemorrhage. Repeat imaging showed no significant change in the left adrenal hematoma, but arterial phase imaging still suggested active bleeding. Interventional radiology was consulted, and embolization of the left superior and inferior renal artery was done with stabilization of the patients hemodynamics. Laboratory work for secondary causes of hypertension was done. It showed elevated normetanephrines 1281 mcg/24hr (122 - 676 mcg/24hr) and total 24-hour urine metanephrines, 114 mcg/24hr (224-832 mcg/24 hr). The patients blood pressure was controlled and therefore asked for follow-up as an outpatient.

Conclusion
The majority of adrenal hemorrhages diagnosed are found incidentally, but initial symptoms of abdominal or loin pain are very important. Early acute findings may include tachycardia, hypotension, and severe abdominal pain. Subacute symptoms include flank tenderness, diarrhea, malaise, nausea, and vomiting. Unilateral adrenal hemorrhage may be the initial presentation of an underlying adrenal mass, cyst, coagulopathy or metastasis. Elevated cortisol and catecholamines released by an expanding hematoma are often seen along with elevated blood pressure, suggesting pheochromocytoma. The Hormonal analysis includes urine metanephrine and vanillylmandelic acid to rule out active pheochromocytoma. In our case, the patient had elevated urine metanephrines and normetanephrines suggestive of pheochromocytoma.
Presentation: No date and time listed
LBSAT65 Search For A Duplicitous Pheochromocytoma
Authors: Gupta Gunjan , Riaz Aiman , Jabiev Azad , Jamal Issra 
Published: November 2022

Abstract

Introduction
Pheochromocytomas are discovered during work up of adrenal incidentalomas and can be biochemically and clinically silent. We present a case of a patient who presented with typical "adrenergic spells" suggestive of pheochromocytoma but negative biochemical testing and non-confirmatory imaging findings.

Case Report
39-year-old male presented with 4 weeks history of episodes of throbbing headaches, palpitations, sweating. He was found to be hypertensive, blood pressure of 220/110 mm Hg. Losartan was initiated, but symptoms persisted. Work-up for endocrine causes of hypertension revealed normal plasma renin activity 1.123 ng/ml/hr (0.167 to 5.380) and undetectable aldosterone level &lt;1 ng/dl (0. 0-3. 0), mildly elevated random plasma normetanephrine 270 pg/ml (0-210), normal plasma metanephrines 49 pg/ml (0-88). Low-dose dexamethasone suppression test showed unsuppressed morning cortisol of 2.1ug/dl, undetectable ACTH &lt;5 pg/ml, dexamethasone level 486 ng/dl. 24 urinary cortisol and 2 midnight salivary cortisol samples failed to confirm the diagnosis. 24-hr urine studies showed mildly elevated 24-hr normetanephrine 735 ug/24 hr (156-729), normal 24-hr metanephrines 197 ug/L (58-276), normal 24 hr epinephrine and dopamine levels. Repeat testing revealed normal plasma metanephrines levels and mildly elevated plasma normetanephrine levels 287 pg/ml (less than twice above the upper limit of normal). Dopamine and Chromogranin A levels were undetectable. Abdominal CT showed 2 cm round soft tissue density 42HU in right adrenal gland. Left adrenal gland contained 3 nodules measuring 11, 15 and 16 mm–all with density &lt;10HU. MRI findings for the 2.2 cm right adrenal nodule was without significant drop in signal intensity on the in and out of phase sequences appearing moderately T2 hyperintense and T1 hypointense. Differential included atypical/fat poor adenoma, pheochromocytoma, or metastasis. On GA 68 Dotatate PET-CT this 2.2cm adrenal nodule had minimal to low radiotracer uptake. Since a pheochromocytoma could not be ruled out, patient was started on alpha blockade. Preoperative genetic testing was negative for common mutations associated with pheochromocytoma/paraganglioma syndromes. Patient underwent laparoscopic right adrenalectomy. Histopathology report confirmed 2.8 cm pheochromocytoma with a low PASS (2/20) and low GAPP score.

Conclusion and Discussion
Patients with borderline nor metanephrine elevations represent a diagnostic and therapeutic dilemma. The patient presented with adrenergic paroxysms strongly suggestive of underlying pheochromocytoma. Labs and imaging were not typical for pheochromocytoma and were indeterminate. Given strong clinical suspicion for a pheochromocytoma, we decided to proceed with adrenalectomy after pre-treatment with alpha blockers. Final histopathologic report confirmed the diagnosis of pheochromocytoma.
Presentation: Saturday, June 11, 2022 1:00 p.m. - 3:00 p.m.
ODP303 Cushing's Syndrome Due to Ectopic Adrenocorticotropic Hormone-Secreting Metastatic Neuroendocrine Tumor
Authors: Cipriani Allison , Fernandes Jyotika , Sora Nicoleta , Tofil Kathrin 
Published: November 2022

Abstract

Introduction
Cushing's syndrome (CS) due to ectopic ACTH secretion (EAS), is an infrequent form of ACTH-dependent CS. EAS results from unregulated ACTH secretion by neuroendocrine tumors (NETs) of various locations resulting in severe hypercortisolism. The progression of CS is usually precipitous. To improve outcomes, it is imperative to recognize early so that treatments of cortisol-induced comorbidities and targeted treatments can be initiated.

Case Presentation
A 74-year-old female presented to an outside hospital with fatigue, bilateral lower extremity edema and intractable hypokalemia. She was later transferred to our facility, where workup of CS was initiated. She underwent a 1mgdexamethasone suppression test (DST)which resulted in an unsuppressed 8amcortisol of 34.7 ug/dL (&lt;2 ug/dL). This was followed by a 4mgDST, again resulting in an unsuppressed 8amcortisol of25.4 ug/dL. A 24-hour urine cortisol level was collected and resulted at 2,108.9 ug/d (&lt;45 ug/d). ACTH level was elevated at 136 pg/ml (7.2-63.3 pg/ml). CT abdomen/pelvis identified liver lesions concerning for metastatic disease. PET DOTATATE revealed multiple liver lesions and a few pulmonary nodules. Biopsy of liver lesions demonstrated strongly positive staining of synaptophysin and chromogranin confirming NET, with a weak TTF-1 positivity suggestive of pulmonary origin. Lanreotide injections were initiated for tumor control and ketoconazole for treatment of persistent hypercortisolism. Despite titration of ketoconazole, symptoms and labs showed minimal improvement. Osilodrostat was then added as a third line and examethasone was initiated in a block and replace approach. The regimen of lanreotide, osilodrostat and ketoconazole improved her24-hoururine cortisol to 15 from 2,109 ug/24hr. Subsequently patient underwent trans-arterial chemoembolization (TACE) of the liver lesions with resolution of hypercortisolism. She is currently only on Lanreotide for tumor control.

Discussion
NET with ectopic ACTH production is rare. While primary tumor location was not definitively identified, multiple lung lesions and histopathology suggested primary lung tumor with hepatic metastases. 20-40% of all NETS originate in the chest. Patient management necessitates a multidisciplinary approach not only for the diagnosis and treatment of CS but also for the specific management of neuroendocrine tumors (NET). This case highlights a rare presentation of EAS, and aggressive treatment modalities needed to treat symptoms of excess cortisol production and tumor progression.
Presentation: No date and time listed
ODP305 Differentiation Potential of Marginal Layer-Derived Adult Pituitary Stem/Progenitor Cells into Vascular Endothelial Cells and Pericytes in Mice
Authors: Higuchi Masashi , Shintani Aran 
Published: November 2022

Abstract
The anterior lobe (AL) of the pituitary gland consists five types of hormone-producing cells and fenestrated sinusoids (i. e., vascular endothelial cells and pericytes) that cooperate to maintain the physiological function of the gland. In addition, the population of cells located in pituitary stem/ progenitor cell niche, the marginal layer, which faces the residual lumen of the Rathke's pouch, serve to renew pituitary cells throughout life. We recently established a protocol for isolating marginal layer-derived adult pituitary stem/progenitor cells (MPSCs) by using differences in cell adhesion (1), and then showed differentiation potential into hormone-producing cells by subjecting MPSCs to three-dimensional culture. However, it is unclear whether MPSCs are capable of differentiating into all types of cells that constitute the pituitary gland. In this study, we performed DNA microarray analysis in MPSCs, which were proliferated by two-dimensional (2D) culture, for studying comprehensively gene expression profiles. Next, we investigated what kind of cells MPSCs would differentiate into. Comparing gene expression profiles of the AL and MPSCs, in MPSCs, 1776 genes were increased more than 4-fold and 1715 genes were decreased 4-fold compared to the AL (p&lt;0. 05, respectively). The upregulated genes included pituitary stem/progenitor cell markers (ex. Sox2, Sox9, Klf4, Prrx1), niche markers (ex. Cdh1, Krt8), and cell surface antigens (ex. Cd9, Cd34, Cd44). On the other hand, most of the downregulated genes were differentiation markers (ex. Pou1f1, Gata2, Tbx19, Kdr). Next, immunofluorescent analysis revealed that most of the MPSCs were positive for CD34, the hematopoietic stem cell marker. After further 2D culture of MPSCs for 5-7days, vascular endothelial marker PECAM1-positive cells were detected by immunofluorescent analysis, but pericyte marker CSPG4-positive cell not. When TGFβ was applied to 2D culture of MPSCs, CSPG4-positive cells were detected instead of PECAM1-positive cells. Expression levels of Pecam1and Cspg4 were also examined by qPCR analysis, which corresponded to the results of immunofluorescent analysis. On the other hand, expression levels of anterior hormone genes were almost unchanged. These results have demonstrated that MPSCs could differentiate into vascular endothelial cells and pericytes. The present study suggests that pituitary stem/progenitor cells, which derived from the marginal layer, are a source of not only hormone-producing cells but also cells that constitute blood vessels. Reference: (1) Shintani and Higuchi, Stem Cell Res. 2021 Feb 2;52: 102223.
Presentation: No date and time listed
ODP315 Hypoglycemia: Neuroendocrine Tumor of the Pancreas with Liver Metastasis and Transformation to Insulinoma
Authors: Ali Basim , Chatterjee Ritodhi , Chen Rui , La Betty , Nguyen Son , Tetlay Maryam , Vasudevan Madhuri , Villareal Dennis 
Published: November 2022

Abstract

Background
Neuroendocrine tumors (NETs) are rare, can be found in any part of the body including the gastrointestinal tract, lung, and pancreas and have potential for metastasis.

Clinical Case
A 39-year-old male with stage IV high grade neuroendocrine tumor of the pancreas with metastasis to the liver and lymph nodes whose disease progressed despite treatment with systemic therapy who presented with severe hypoglycemia. Diagnosis and management of his hypoglycemia was difficult. For the diagnosis, patient's serum blood glucose did not always correlate with his capillary blood glucose via finger sticks, which lead to a delay in his diagnosis. Also, his diagnosis was not straight forward as he did not respond adequately to the glucagon following a serum glucose of less than 55 mg/dL (normal 70-110 mg/dL) during a diagnostic fast. Once critical labs were obtained, it took days for the results. In the interim, his hypoglycemia was managed with dextrose infusions at 10%, 20% and as high as 50%, frequent meals, and high dose steroids, yet he still had daily symptomatic hypoglycemia. A diagnosis of insulinoma was made with the following critical lab results: insulin 70.4 uIU/mL (2.6-24.9), C-peptide 12.6 ng/mL (1.1-4.4), proinsulin 407.6 pmol/L (0. 0-10. 0), beta-hydroxybutyrate &lt;0. 050 mmol/L (0. 02-0.27), insulin antibodies &lt;5. 0 uU/mL, negative sulfonylurea screen, insulin-growth factor-1 (IGF-1) 77 ng/mL (90-278), and insulin-growth factor binding protein three (IGF-BP3) 2133 ug/L (2571-5982). Patient was treated with both diazoxide and hepatic artery embolization to reduce tumor burden. His dose of diazoxide was titrated to maintain normoglycemia. He had mild tumor lysis syndrome that did not require dialysis. His hypoglycemia resolved within a few days of treatment and was discharged from the hospital with a continuous glucose monitor (CGM), diazoxide and hydrocortisone. It is our belief that transformation of the liver lesions to insulinoma was the cause of his hypoglycemia.

Conclusion
We recommend that patients with metastases from their neuroendocrine tumor who develop hypoglycemia be evaluated for insulinoma and if found should be treated with diazoxide and if not surgical candidates to consider hepatic artery embolization.
Presentation: No date and time listed
ODP325 Inverse Relationship Between IL-6 and Sodium Levels in Patients With COVID-19 and Other Respiratory Tract Infections
Authors: Monnerat Sophie , Atila Cihan , Bingisser Roland , Siegemund Martin , Lampart Maurin , Rüegg Marco , Zellweger Nuria , Osswald Stefan , Rentsch Katharina , Christ-Crain Mirjam , Twerenbold Raphael 
Published: November 2022

Abstract

Background
Hyponatremia is highly prevalent in patients with COVID-19. One of the most common causes of hyponatremia in these patients is the syndrome of inadequate antidiuresis (SIAD). Interleukin 6 (IL-6) is a key mediator of inflammation in COVID-19. We hypothesized that hyponatremia in COVID-19 is due to IL-6 mediated non-osmotic arginine vasopressin (AVP) secretion, and that the inverse association between IL-6 and plasma sodium concentration is stronger in COVID-19 compared to other respiratory infections. METHODS This is a secondary analysis of a prospective, observational, cohort study including patients with COVID-19 suspicion admitted to the Emergency Department, University Hospital of Basel, Switzerland, between March and July 2020. We included patients with PCR-confirmed COVID-19 and patients without COVID-19 but similar symptoms, further subclassified in bacterial and other viral respiratory infections. The primary objective was to investigate the association between plasma sodium levels and IL-6 levels.

Results
500 patients were included, of whom 184 (37%) with COVID-19, 92 (18%) with bacterial respiratory infections, 224 (45%) with other viral respiratory infections. Hyponatremia prevalence was higher in patients with COVID-19 compared to patients with other viral respiratory infections (28% vs 12%, p&lt;0. 01), and similar to patients with bacterial respiratory infections (28% vs 30%, p&lt;0.41). In all three groups, median [IQR] IL-6 levels were significantly higher in hyponatremic compared to normonatremic patients (COVID-19: 43.4 [28.4, 59.8] vs 9.2 [2.8, 32.7] pg/ml, p&lt;0. 0001; bacterial: 122.1 [63. 0, 282. 0] vs 67.1 [24.9, 252. 0] pg/ml, p&lt;0. 05; viral: 14.1 [6.9, 84.7] vs 4.3 [2.1, 14.4] pg/ml, p&lt;0. 05). IL-6 levels were negatively correlated with plasma sodium levels in COVID-19, whereas the correlation in bacterial and other viral infections was weaker (COVID-19: ρ = − 0.52, p &lt; 0. 001; bacterial: ρ = − 0.24, p = 0. 056, viral: ρ = − 0.24, p &lt; 0. 001).

Conclusion
IL-6 levels were inversely correlated with plasma sodium levels, with a stronger correlation in patients with COVID-19 compared to patients with bacterial and other viral infections. IL-6 might stimulate AVP secretion and lead to higher rates of hyponatremia due to the syndrome of inadequate antidiuresis in these patients.
Presentation: No date and time listed