bluuebunny/crossref_metadata_2025_split huggingface dataset:
FRI323 Prolactinoma Recurrence With Testosterone Treatment In Transgender Male
Authors: Arnouk Nelly , Schroeder Pamela Rose 
Published: October 2023

Abstract
Disclosure: N. Arnouk: None. P.R. Schroeder: None.
Introduction: We present a case of recurrent hyperprolactinemia due to testosterone treatment for gender-affirming hormonal therapy in a transgender male after being controlled on cabergoline for more than a year. Case: A 27-year-old female with history of HTN and polycystic ovary syndrome was diagnosed with pituitary microadenoma after hospital admission for headache and blurry vision. MRI brain showed a mass in the sella and right parasella measuring 36 mm W x 36 mm D x 44 mm H. Hormonal workup showed prolactin (PRL) &gt;999 ng/mL in the diluted assay. The patient was treated medically with cabergoline and discharged home. PRL level improved to 22 ng/mL on treatment. Cabergoline was continued at 0.5 mg twice weekly. The patient was lost to follow-up but returned to clinic after 8 months with PRL level of 58 ng/mL. Cabergoline 0.5 mg twice weekly was continued. PRL level monitoring every ∼6 weeks showed an increase to 184 ng/mL and then 199 ng/mL. Cabergoline was increased to 1 mg twice weekly. PRL level 1 month later was 222 ng/mL. Repeat brain MRI showed markedly decreased microadenoma 4.8 X 2.6 mm. The patient was asymptomatic with no headache, blurry vision, or galactorrhea. On further questioning, the patient reported starting transgender female to male testosterone cypionate 200 mg/mL once weekly for the past 6 months. After discussion with the primary care provider who was managing testosterone treatment, he was started on 60 mg weekly, and the dose was gradually increased to 80 mg weekly to stop the menstrual period. With the 60-mg weekly cypionate dose, the testosterone level was 346 pg/mL, and PRL level was 184 ng/mL. With the cypionate dose of 80 mg weekly, PRL increased to 194 ng/mL and 222 ng/mL. The cypionate dose was decreased to 60 mg weekly, based on literature noting elevated PRL level with testosterone replacement in patients with prolactinoma and hypogonadism. Based on repeat blood work showing elevated testosterone 366 pg/mL with PRL 318 ng/mL, the plan is to start an aromatase inhibitor. Discussion: Testosterone treatment as hormonal affirming therapy in some transgender female to male patients may exacerbate pre-existing hyperprolactinemia or induce PRL secretion, thus close monitoring is required. Testosterone is aromatized to estradiol, which then stimulates the release of PRL from the pituitary gland. The serial measurement of estradiol may serve as an index of aromatization. Hyperprolactinemia in this setting is resistant to dopamine agonist therapy, as noted in multiple case reports. There are no established reference ranges for PRL levels for transgender patients on gender-affirming hormone therapy and no current guidelines regarding routine imaging or biochemical assessment during follow-up. Transgender patients with prolactinoma may require additional treatment with non-aromatizable androgens or the concomitant administration of aromatase inhibitors.
Presentation: Friday, June 16, 2023
FRI331 Autoimmune Hypophysitis Secondary To Ipilimumab And Nivolumab Combination Therapy In A Patient With Advanced Renal Cell Carcinoma
Authors: Ibrahim Eiman Y , Hulinsky Ilja , Regelmann David 
Published: October 2023

Abstract
Disclosure: E.Y. Ibrahim: None. I. Hulinsky: None. D. Regelmann: None.
Introduction: Ipilimumab and nivolumab combination therapy is an effective immune checkpoint inhibitor regimen indicated for advanced stages of renal cell carcinoma. Autoimmune hypophysitis is a rare adverse event from this therapy that occurs in only 3-6% of patients. The mechanism underlying immune checkpoint inhibitor-induced hypophysitis is not fully understood but involves immune, inflammatory, and genetic factors. Clinical Case: A 65-year-old male with a history of hypertension and type 2 diabetes mellitus, recently diagnosed with renal cell carcinoma with metastasis to the lungs presents to the emergency department with three days of generalized weakness and worsening confusion, accompanied by 2 weeks of headache. Two months prior to presentation, the patient began chemotherapy with four cycles of nivolumab and ipilimumab. The emergency department team initiated a stroke code for right facial droop and right-sided weakness seen on his physical exam. The CT of the patient’s head was unremarkable. The patient was also hypotensive (BP 90/40 mmHg) and tachycardic (140 bpm). He received 3 liters of crystalloid as well as diltiazem injection without improvement. Random cortisol on admission at 11:00 am was 1 ug/dl (3.4-22), ACTH was 7 pg/mL (6-50), Na was 135 mEq/L (137 - 145) and K was 5.1 mEq/L (3.5 - 5.0). Furthermore, TSH 0.02 μIU/ml (0.47-4.70), ILGF 39 ng/mL (41-279), and total testosterone levels &lt;3 ng/dL (139-740) were low. The free thyroxine level was measured at 1.00 ng/dL (0.8-1.9), and MRI revealed a normal sella tursica. Based on these findings, we diagnosed the patient with hypopituitarism, likely due to hypophysitis as an immune-related adverse event of immune checkpoint inhibitor therapy. Consequently, we discontinued the administration of Ipilimumab-Nivolumab combination therapy and started oral dexamethasone 8 mg and fludrocortisone 0.1 mg for mineralocorticoid supplementation. The patient’s symptoms resolved, and laboratory data showed improvements in hyponatremia, hyperkalemia, and hypoglycemia. The care team transitioned the patient’s steroid regimen to prednisone 25 mg daily maintenance dose, and the patient was discharged with stress dosing instructions. Conclusion: Diagnosis of hypophysitis as an immune-related adverse event is challenging due to its vague presentation. However, a delayed diagnosis can be life-threatening. As a result, clinicians must maintain a high index of suspicion for presentations of weakness and headache in consistent clinical contexts, in order to provide optimal management and minimize morbidity. Additionally, early management of hypophysitis can reduce the interval of immune checkpoint inhibitor therapy, which in turn affects the course of cancer.
Presentation: Friday, June 16, 2023
FRI336 A Case Of Pituitary Apoplexy In Late Pregnancy
Authors: Abdelrahman Houda , Rasiah Stephen , Braunthal Stephanie , Mahoney Lindsey , Carneiro Raquel M , Chen Kenneth K 
Published: October 2023

Abstract
Disclosure: H. Abdelrahman: None. S. Rasiah: None. S. Braunthal: None. L. Mahoney: None. R.M. Carneiro: None. K.K. Chen: None.
Case: A 20-year-old primigravida presented at 36 weeks gestation due to persistent left-sided headache of 4 weeks duration. MRI brain demonstrated pituitary enlargement (15x17x14 mm) with hemorrhage as well as suprasellar extension and mass effect upon the optic chiasm, consistent with pituitary apoplexy (PA). She has no known medical history or prior endocrinopathies. Serum prolactin was appropriately elevated for pregnancy (299 ng/mL). She was started on levothyroxine for secondary hypothyroidism (TSH 3.4 mcg/dL, free T4 0.8ng/dL (0.7-1.5 ng/dL)) and hydrocortisone for prevention of adrenal insufficiency. She was admitted for semi-urgent induction of labor following the discovery of visual field defects on formal perimetry testing by ophthalmology. After discussion with neurosurgery and maternal fetal medicine, she was permitted to have a trial of vaginal delivery with a brief period of Valsalva but she ended up requiring an emergency C-section due to failure to progress. Her delivery was complicated by significant blood loss (∼5000ml), resulting in bilateral uterine artery embolization followed by an emergency hysterectomy. Repeat MRI brain with contrast following delivery demonstrated a cystic pituitary lesion, possibly a Rathke’s cleft cyst. She underwent endoscopic transnasal exploration and resection two days following delivery. Discussion: Pituitary apoplexy is a rare cause of acute headache in pregnancy. The majority of PA occurs in the setting of undiagnosed pituitary adenomas. Rapid treatment of possible adrenal insufficiency is imperative. Conservative versus surgical treatment plans are tailored to the patient’s obstetric care and neurological status. The earliest reported PA occurrence is at 7 weeks gestation (associated with subsequent miscarriage). Coordination of care between different specialists is paramount especially in late pregnancy. It is theoretically possible that Valsalva during vaginal delivery may increase intracranial pressure and exacerbate hemorrhage. Women with pituitary adenomas are recommended to undergo standard obstetric care with close surveillance, as C-section deliveries can also be associated with increased blood loss. Timing of surgery in gestational pituitary apoplexy is variable; it may be up to one month after delivery. Dopamine agonist therapy may be started during pregnancy for confirmed prolactinomas. The European Society of Endocrinology recommends transsphenoidal surgery in the second trimester for symptomatic adenomas. In the third trimester, preterm delivery may be considered. Post-operative adverse events are not uncommon due to the high risk nature of these pregnancies. After surgery, most patients should experience some degree of vision improvement but many will continue to need hormone replacement.
Presentation: Friday, June 16, 2023
FRI341 Nothing Great Comes Without Its Risks; A Rare Case Of Pembrolizumab Induced Hypophysitis
Authors: Fernando Ashen Kavinda , Cheema Rashid 
Published: October 2023

Abstract
Disclosure: A.K. Fernando: None. R. Cheema: None.
Introduction: Immune checkpoints are proteins expressed by immune cells and are crucial for maintaining self-tolerance. Immune checkpoint inhibitors (ICIs) are antibodies that inhibit these checkpoints and enhances T-cell activation and antitumor activity. This emerged as a powerful tool in treating cancer, however its use is jeopardized by various immune related adverse events (IRAEs). Secondary hypophysitis, although rare, is associated with immunotherapy. The incidence of hypophysitis is considerably lower with PD-1 inhibitors such as pembrolizumab (0.5-1.5%) than with CTLA-4 inhibitors. We present a case of hypophysitis following initiation of pembrolizumab for NSCLC.
Case: A 64-year-old Caucasian male was seen in Endocrinology for evaluation of abnormal TFTs with nonspecific symptoms of fatigue and malaise. He was recently diagnosed with poorly differentiated lung carcinoma with metastasis to brain. After completing XRT, he was started on pembrolizumab/trastuzumab. TFTs prior to initiation of treatment were unremarkable. He developed a mild rash and fatigue post treatment but otherwise treatment was well tolerated. TFTs at week 8 after starting ICIs showed TSH 0.09 uIU/mL (0.27-4.2), T4 0.8 ng/dL (0.9-1.7) and T3 2.2 pg/mL (2.5-4.3). MRI Sella showed enlargement of the pituitary gland and stalk suggestive of hypophysitis. Pembrolizumab was discontinued. Additional studies showed low total testosterone 19.5 ng/dL (193-740) and free testosterone 4.5 pg/mL (35-130) with inappropriately normal gonadotropins suggestive of central hypogonadism. Other pituitary axis were intact. Cosyntropin stimulation test confirmed intact HP Adrenal axis. He was started on levothyroxine and hydrocortisone (20 mg AM and 10 mg PM) for hypophysitis. His symptoms improved. He is closely being monitored with plan to repeat MRI Sella in 3 months and to re-evaluate his HP adrenal prior to weaning off steroids. Discussion: IRAEs from ICIs typically present within 6 to 12 weeks but can present several months to years later. The non-specific nature of symptoms in patients on PD-1 inhibitors can delay clinical recognition with dire consequences. Treatment involves hormone replacement and stopping immunotherapy. Though benefit of GC in patients with grades 1-2 hypophysitis is unclear, low dose corticosteroids have been used to mitigate pituitary inflammation. High dose GC although commonly used in patients with critical illness (grades 3-4), its use remains controversial as they have not shown to significantly alter overall survival. Some degree of pituitary function may recover 4-12 weeks following withdrawal of ICIs. Given the increase in use of ICIs and the serious nature of IRAEs, it is critical to be aware of clinical manifestations, diagnosis, and management of this entity.
Presentation: Friday, June 16, 2023
FRI371 Serum Androstenedione As An Additive Biomarker In Ectopic Cushing’s Syndrome Evaluation
Authors: Zhang Sherry , Lo Joan C , Jaffe Marc G , Arzumanyan Hasmik 
Published: October 2023

Abstract
Disclosure: S. Zhang: None. J.C. Lo: None. M.G. Jaffe: None. H. Arzumanyan: None.
Background: Ectopic Cushing’s syndrome (ECS) results from glucocorticoid excess secondary to ectopic ACTH or CRH producing tumors. Although ECS identification can be challenging when presentation lacks classic Cushing’s features, ectopic ACTH secretion can result in a key constellation of findings that facilitate early clinical recognition. We describe a case of ECS from an ACTH-producing carcinoid tumor of the lung presenting with resistant hypertension, severe hypokalemia, and muscle weakness. In addition to significant hypercortisolism, laboratory findings were notable for high androstenedione levels without concomitant increase in DHEAS and testosterone levels. Case: A 59-year-old female athlete with history of well-controlled hypertension developed difficult to control hypertension, requiring escalation to triple antihypertensive therapy within 3 months. During this time, she had multiple emergency department visits for progressive muscle weakness, hypertension, and persistent hypokalemia despite potassium supplementation. She was hospitalized for further evaluation. Physical examination was notable for bruising, muscle wasting, thin extremities, facial fullness, and disproportionate abdominal adiposity. Workup for Cushing’s syndrome demonstrated ACTH-dependent hypercortisolism and markedly elevated androstenedione 398 ng/dL (normal 20-75 ng/dL) despite normal DHEAS 48 mcg/dL (normal &lt;430 mcg/dL) and total testosterone 11 ng/dL (normal &lt;45 ng/dL) levels. A chest CT identified a 3.8 cm right lower lobe lung mass and transbronchial biopsy confirmed atypical carcinoid tumor. She underwent a right middle and lower lobe bilobectomy with complete tumor resection. Cortisol, androstenedione, and potassium levels rapidly normalized postoperatively, and blood pressure returned to baseline, well-controlled on monotherapy. The patient only required 3 days of hydrocortisone therapy for transient steroid withdrawal symptoms. Conclusions: Our case illustrates a difficult to diagnose uncommon disease presenting with resistant hypertension, hypokalemia and marked androstenedione elevation despite normal DHEAS and testosterone levels. This steroidogenic profile has been reported in other ECS cases and attributed to differential effects of ectopic ACTH production on adrenal androgen biosynthesis (1). Although ECS is rare and the primary lesion can be difficult to identify, disproportionate elevation in androstenedione may prompt recognition of ECS and support early detection and management.
Reference: (1) Barbetta L, Dall’Asta C, Re T, Colombo P, Travaglini P, Ambrosi B. Androgen secretion in ectopic ACTH syndrome and in Cushing’s disease: modifications before and after surgery. Horm Metab Res Horm Stoffwechselforschung Horm Metab. 2001;33(10):596-601
Presentation: Friday, June 16, 2023
OR13-03 Assessment Of Skeletal Health Including Bone Density, Turnover, Microarchitecture And Osteoporosis In Patients With Sheehan’s Syndrome: A Prospective, Multicentric Study In Asian Indians
Authors: Das Liza , Laway Bashir , Sahoo Jayaprakash , Bhadada Sanjay K , Dutta Pinaki 
Published: October 2023

Abstract
Disclosure: L. Das: None. B. Laway: None. J. Sahoo: None. S.K. Bhadada: None. P. Dutta: None.
Introduction: Sheehan’s syndrome (SS) is a rare cause of hypopituitarism. Evidence on skeletal health in patients with this condition is limited, and data on bone geometry, structure and volumetric bone mineral density (BMD) are lacking. We aimed to comprehensively explore skeletal fragility in a multicentric cohort of treated SS. Patients and Methods: Patients with SS were recruited for analysis of demographic, clinico-biochemical and hormonal profile including bone turnover markers. Areal BMD, T- and Z-scores and trabecular bone score (TBS) were evaluated using DXA. In a subset of patients (n=29) as well as age-and BMI-matched controls (n=34), high-resolution peripheral quantitative computed tomography (HRpQCT, XtremeCT II, Scanco Medical AG, Switzerland) was performed to assess bone geometry and microarchitecture. Results: There were a total of 105 patients with SS with a mean age 45.2 ± 10.5 years and a lag of 7.4 ± 5.5 years from disease onset to diagnosis. Regular levothyroxine replacement was ongoing in 91.2% and glucocorticoid replacement in 86.1% of the cohort. Past HRT replacement was done in 66.7% patients. Bone turnover markers (P1NP, CTX) were not significantly different between cases and controls. The T- score [-2.3 (-3.0 to -1.6) vs -1.5 (-3.0 to -0.7)] and Z-score [-1.4 (-2.1 to -1.2) vs -0.6 (-2.1 to -0.1)] at the lumbar spine (LS) and the T- score [-1.8 (-2.3 to -1.3) vs -0.8 (-2.1 to -0.1)] and Z-score [-1.1 (-1.7 to -0.4) vs -0.4 (-1.0 to 0.2)] at the femoral neck (FN) were lower in cases as compared to controls (trend towards significance). Osteopenia or osteoporosis was prevalent in significantly greater proportion of the cases as compared to controls, at LS (96.3% vs 64.7%), FN (91.7% vs 47%) and ultradistal radius (65.2% vs 11.8%). Bone microarchitectural assessment showed no significant differences between TBS, or bone geometry. However, trabecular volumetric bone mineral density was higher at both tibia (p&lt;0.05) and radius (p&gt;0.05) in cases as opposed to age-and BMI-matched controls. There was a weak negative correlation between glucocorticoid and levothyroxine replacement duration and BMD at both LS and FN (p&lt;0.05), but not with replacement dose. Conclusion: Osteopenia-osteoporosis is highly prevalent in SS. BMD at both LS and FN is negatively correlated with duration of glucocorticoid and levothyroxine exposure. There are no significant differences in bone microarchitecture or geometry but tibial trabecular volumetric BMD is greater in treated SS.
Presentation: Friday, June 16, 2023
THU602 A Comprehensive Review Of Incidence, Demographics, Laterality And Survival Analysis Of Adrenal Malignancies.
Authors: Desai Krishna , Pereira Kristal , Iqbal Sabah , Baralo Bohdan , Vidhi Mehta , Renzu Mahvish , Rather Manzoor , Thirumaran Rajesh 
Published: October 2023

Abstract
Disclosure: K. Desai: None. K. Pereira: None. S. Iqbal: None. B. Baralo: None. M. Vidhi: None. M. Renzu: None. M. Rather: None. R. Thirumaran: None.
Background: Adrenal malignancy is extremely rare in the United States with only ∼200 new cases/per year. Adrenocortical carcinoma is the most common histologic subtype, followed by pheochromocytoma and neuroblastoma. In this study, our aim is to evaluate the incidence rate and compare the survival analysis based on race, laterality, and histologic tissue involvement. Method: The data were collected from Surveillance, Epidemiology, and End Result database 17 Registries, Nov 2021 Sub (2000-2019). We included patients above 20 years with a primary diagnosis of an adrenal malignant tumor. Neuroblastomas were excluded due to low incidence in the adult population. We analyzed incidence rates using linear regression with t-test and compared overall survival (OS) curves using the Log-rank test (GraphPad Prism). Result: 3323 cases were identified with the majority being female (54.68%). 50.29% were adrenal cortical tumors, followed by pheochromocytoma (14.44%) & neuroblastoma (0.51%). 48.03% were left-sided, 39.54% were right-sided, 3.64% were bilateral & the rest were unspecified. The incidence rate for adrenal tumors is 2.77 per 1 million. The highest incidence was among Caucasians (2.98), followed by African American (2.63, p=0.09), which is higher than Asians (2.03, p&lt;0.0001) and Hispanics (2.42, p=0.0002). A review of the incidence trends showed that only African-American patients had rising incidence rates from 1.46 in 2000 to 2.48 in 2019 (p=0.02). Patients with adrenal cortical tumors had worse survival compared to patients with pheochromocytoma (19 vs 98 months) HR 2.236 [1.988-2.232]. No significant statistical difference was noticed in the survival of left vs. right-sided tumors or racial groups when subdivided based on histology. However, there was a difference in the median survival of the women with pheochromocytoma (123m), compared to men (75m) HR 1.397 [1.064 - 1.834]. Conclusion: The incidence rate of adrenal malignancies remained stable and is higher among Caucasians compared to Hispanics and Asians. We noticed concerning rising incidence among African American patients. Patients with adrenocortical tumors had worse survival compared to patients with pheochromocytoma. Laterality of the tumor and race of the patients plays no role in survival in adrenal malignancies, unlike the colon and kidneys. However, there is a noticeably lower survival of the male population with pheochromocytoma.
Presentation: Thursday, June 15, 2023