bluuebunny/crossref_metadata_2025_split huggingface dataset:
Analysis of Treatment Outcomes of Patients With Cystic Prolactinomas: Report of Four Cases
Authors: Erdi Zuleyha Can , Ozkan Cigdem 
Published: May 2021

Abstract
Background: Prolactinomas are the most common functional pituitary adenomas. Treatment with dopamine agonists is considered as first-line therapy. Cystic prolactinoma is a variant prolactinoma. . It was assumed that since cystic area does not contain dopamine receptors, cystic prolactinomas would not respond to medical treatment. However, in recent years, it has been shown that cystic prolactinomas can also be treated with dopamine agonists, and medical treatment has become more preferred than surgical treatment. In this report, we aimed to evaluate the treatment effectiveness of patients with cystic prolactinomas who treated medically in our clinic. Materials and methods: Twenty-nine prolactinoma patients who were followed up in our hospital between 2019-2020 were evaluated retrospectively. Four patients (13%) were being followed up for cystic prolactinoma. Radiological images, laboratory findings, patients records were reviewed. Results: Four patients were being followed up for cystic prolactinoma and all were women. The average age of the patients followed up for cystic prolactinoma was 36 years. 3 patients (75%) had menstrual irregularity, 2 (50%) patients had galactorrhea, and 1 patient (25%) had headache at the time of admission. Tumor size regressed with medical treatment in two patients and the tumor disappeared completely in one patient. Although the symptomatic improvement and normalization of prolactin was achieved in one of our patients, the lesion did not shrink. We continued with medical treatment in this patient since there was no indication for surgery. Discussion: There was conflicting results in the literature regarding the management of cystic prolactinomas. Recently, dopamin agonist therapy is suggested as first- line treatment option in cystic prolactinomas. A study reported male predominance for cyst formation but we observed female predominance in our report. Faje et al. also reported female predominance in their study. Presentation of our patients were not different from solid prolactinomas. And although we evaluated very limited number of patients in our report, all of our patients respond to medical treatment which was consistent with recent literature.
Anterior Pituitary Failure in Patients with Granulomatous Inflammation
Authors: Narula Kavita , Esdaile Harriet , Martineau Marcus , Alegre Javier 
Published: May 2021

Abstract
Introduction: Langerhan cell histiocytosis (LCH) is a rare cause of eosinophilic granulomatous inflammation that can affect multiple organ systems, mainly diagnosed in children. Hypothalamic-pituitary axis involvement is well established and is the characteristic intracranial manifestation of LCH. Patients can present with several presentations including anterior pituitary axis failure; however, the most common presentation is Diabetes Insipidus (DI). Here, we present a rare case of isolated pituitary LHC in male adult. Clinical Case: 47 years old gentleman presented with 4 week history of insidious headaches associated with persistent nausea. He also noticed drooping of his left eyelid and presented to hospital with complete ptosis. His CT head did not show any abnormalities. However finding on his MRI brain were consistent with a central skull base inflammatory process centred on the pituitary, extending cranially to the chiasm and third ventricular floor as well as laterally to the cavernous sinuses. His anterior pituitary profile was as follow: LH - 0.7 U/L (1.2-8.6 U/L), FSH - 4.2 U/L (1.3-19.3 U/L), Testosterone - &lt;0.3 nmol/L (6.1 - 27.1 nmol/L), IGF-1 - 9.8 (8.8-31), Prolactin - 197 mIU/L (56-278 mIU/L), TSH - 1.18 mU/L (0.38 - 5.33 mU/L), FT4 - 4.4 pmol/L (8.4 - 19.1 pmol/L). He underwent a Short Synacthen test with a baseline cortisol of 19 nmol/L, rising to 100 nmol/L at 30 minutes and 130 nmol/l at 60 minutes. Following initiation of high dose steroid replacement (Prednisolone 15 mg daily), Levothyroxine and Testosterone, his Prednisolone was slowly weaned to 4 mg once a day in line with an improvement in his ptosis & left temporal hemianopia. His pituitary biopsy showed infiltrates of abundant histiocytes and inflammatory cells (lymphocytes). Histiocytes were organised in a granulomatous fashion with multinucleated giant cells and eosinophils (CD 68 staining positive). Langerin stain (positive) / S100 stain (some) / CD1a (few) - cells positive, confirming a diagnosis of LCH. Additional CT (chest/abdomen/pelvis) and PET imaging confirmed no additional organ involvement. Conclusion: LCH is a rare cause of anterior pituitary failure. Limited information regarding treatment for isolated pituitary LCH is available. This case suggests that a conservative approach to successful treatment with steroids can be considered.
Spexin Friend or Foe? Novel Role of Spexin During Thermogenesis of White Adipose Tissue
Authors: Gambaro Sabrina E , Zubiria Guillermina Maria , Giordano Alejandra P , Harnichar Ezequiel A , Portales Andrea E , Giovambattista Andrés 
Published: May 2021

Abstract
Spexin (SPX) is a novel adipokine playing an emerging role in metabolic diseases due to its involvement in carbohydrate homeostasis, weight loss, appetite control, gastrointestinal movement, among others. Moreover, plasma levels are reduced in obese and type II diabetic patients. In vitro, SPX favors lipolysis in adipocytes and hepatocytes and inhibits white adipogenesis. Therefore, the aim of this study was to evaluate the role of SPX in white adipose tissue (AT) thermogenesis. C57BL/6J male mice were treated or not with SPX for ten days (ip. 29 µg/kg/day; CTR and SPX). At day 3 mice were randomly divided: a group was kept at room temperature (RT) and the other at 4°C to stimulate thermogenesis (CTR-C and SPX-C). Caloric intake and body weight was daily recorded. At the end of the protocol plasma, Brown AT (BAT), abdominal AT (Epidydimal, EAT) and subcutaneous AT (Inguinal, IAT) depots were collected for several measurements. We found that caloric intake was increased when animals were exposed to cold (P&lt;0.001). Body weight change revealed a differential effect of SPX depending on temperature (interaction SPX x Cold, P&lt;0.05): SPX animals weighted less than CTR at RT, but upon cold stimulation there was no difference. No changes were observed for plasma glucose levels, however plasma triglycerides (Tg) levels decreased after cold exposure regardless SPX treatment (Cold P&lt;0.01). Liver Tg content showed a SPX x Cold interaction effect (P&lt;0.0001), where, upon cold stimulation, CTR-C animals increased their levels, but on the contrary SPX-C mice decreased it. EAT, IAT and BAT relative mass showed an interaction effect of variables (SPX x Cold P&lt;0.05). When compared upon cold, SPX-C mice had less AT mass compared to CTR-C mice. IAT and EAT mRNA expression of UCP1 and Cox8b showed SPX x Cold interaction (P&lt;0.05), with a tendency of reduction or no difference in SPX at RT, but with a significant decrease in SPX-C compared to CTR-C mice upon cold exposure. PGC1a expression was increased in EAT from cold exposed-mice and in IAT only in CTR-C mice. UCP1 protein levels showed different results depending on the AT depot. For IAT SPX x Cold interaction (P&lt;0.05) was observed, where SPX inhibited UCP1 stimulation only upon cold exposure. On the contrary, for EAT UCP1 levels decreased in SPX-treated mice, regardless cold exposure (SPX P&lt;0.05). In conclusion, SPX treatment in vivo reduced the thermogenic process in subcutaneous and abdominal AT, being more evident upon cold stimulation. PICT2017-2038, PICT2017-2314.
Are High Doses of Corticosteroids Protective in COVID-19 Patient With Panhypopituitarism?
Authors: Kurir Tina Ticinovic , Mizdrak Maja , Krnić Mladen 
Published: May 2021

Abstract
Introduction: Clinical course of COVID-19 infection is diverse and the best therapeutical guidelines are still lacking. Case Presentation: We present a case of 73 year old male COVID-19 positive patient. In 2017 transnasal hypophysectomy was performed due to prolactinoma. He receives therapy (hydrocortisone 20 + 10 mg/day, levothyroxine 75/50 µg/day, bromocriptine 2.5 mg twice/day). He suffers also from arterial hypertension. Present illness started with intensive dry cough, fever (37.5◦C), diarrhoea and loss of smell. On the first day patient was febrile up to 38.4◦C and later afebrile. Laboratory parameters showed abnormalities in several parameters: D-dimers 13.45 (RR:&lt; 0.50) mg/L, creatinine 110 (49-90) µmol/L, ALT 73 (12-28) U/L, LDH 531 (25-241) U/L, creatine kinase 549 (&lt;177) U/L, GGT 277 (9-35) U/L, CRP 38 (&lt;5 mg/L), sedimentation rate 40 (5-28) mm/3.6ks, procalcitonine 0.07 (&lt;0.05) ng/ml, sodium 128 (137-146) mmol/L, hs-TroponinT 18 (&lt; 14) ng/L, neutrophils 8.56 (2.06-6.49 x109/L), lymphocytes 0.52 (1.19-3.35 x109/L) and pO2 6.1 (11.0-14.4) kPa. At the admission X-ray showed normal presentation, while four days later on the right side pneumonia was noticed as spotty inhomogeneous shading. He was treated with azithromycin, hydrocortisone (50 mg twice/day) and oxygen supplementation. At the beginning of hospitalization he was addicted to O2 10-12 L/min. After 22 days of hospitalization he was discharged without any symptom but with still positive SARS-CoV2 swabs. After a 30 days of follow-up, his swabs are now negative with no laboratory abnormalities. In the meantime, his two brothers, born in 1945 and 1940 died due to COVID-19 infection. They did not receive substitutional corticosteroid therapy. Conclusion: The main pathophysiological mechanism of infection is explained by cytokine storm. Hypercytokinemia causes myelosuppression and vascular endothelium damage. Corticosteroids are potent anti-inflammatory agents. High-doses of corticosteroids might beneficially modulate the host immune response to SARS-CoV2 virus and have protective role in this patient.
Central Diabetes Insipidus Due to Acute Myeloid Leukemia
Authors: Boston Dakota J , Levine Steven N , Yatavelli Rajini Kanth Reddy 
Published: May 2021

Abstract
Central diabetes insipidus (CDI) is a condition characterized by decreased secretion of antidiuretic hormone (ADH) and is commonly seen with pathology involving the hypothalamic/pituitary area. Common etiologies include congenital disorders, neurosurgery or trauma, infiltrative disorders, primary or secondary cancers, and idiopathic causes. CDI associated with acute myeloid leukemia (AML) is extremely rare, with about 100 published case reports, and typically occurs in patients with chromosome 3 or 7 abnormalities.
A 49-year-old woman was admitted to the inpatient oncology service for chemotherapy with gemtuzumab with a goal of inducing remission of her AML prior to allogeneic hematopoietic cell transplantation. She had been diagnosed with AML 7 months prior to this admission and was considered high risk for poor clinical outcome due to cytogenetics demonstrating deletion of chromosome 7 and inversion of chromosome 3. She was noted to have increasing serum sodium and endocrine was consulted for evaluation of hypernatremia.
Patient had a peak sodium of 154 meq/L which improved with free water replacement. She reported excessive thirst with associated polyuria and nocturia. Laboratory tests demonstrated inappropriately dilute urine for the degree of serum osmolality, consistent with a low ADH tone. A water deprivation test was performed which resulted in plasma and urine osmolalities of 311 mosm/kg and 103 mosm/kg, respectively. After the administration of desmopressin the Uosm increased to 345 mosm/kg at 2 hours and 484 mosm/kg at 5 hours, confirming a diagnosis of Central DI.
Evaluation of other pituitary axes showed normal TSH and FT4 with a normal cortisol response on a cosyntropin stimulation test. She had low-normal LH and FSH plus a mildly elevated prolactin level (38.7 ng/mL) which were attributed to stress. An MRI of the brain done to rule out any hypothalamic/pituitary metastasis or hypophysitis was normal.
AML associated with Central DI occurs rarely, is associated with a poor prognosis, and the pathogenesis is unclear. The most commonly reported cytogenetic aberrations in patients with AML and CDI are monosomy 7 and 3q alterations. Our patient had monosomy 7 and chromosome 3 inversion. Proposed pathogenic mechanisms include leukemic infiltration of the pituitary, overexpression of ectopic virus integration (EVI-1) site interfering with hypothalamic neuroendocrine secretion, and abnormal thrombopoiesis interfering with ADH levels in blood.
This case illustrates a lesser-known cause for central diabetes insipidus. While other more common etiologies of CDI should still be considered first, recognition of this association can provide clarity to the origin of DI in select patients.
Change in Character of Headache in a Young Female Patient With Prolactin Secreting Pituitary Adenoma Complicated With Mild Hemorrhage
Authors: Soe Yuzana Myint , Ghani Marium , Siroya Pushplata 
Published: May 2021

Abstract
Background: Prolactin (PRL) secreting tumors are the most common functional neoplasms of the pituitary and are commonly subdivided into microprolactinomas (&lt;10 mm) and macroprolactinomas (≥10 mm) according to their baseline diameter. The diagnosis of macroadenomas is usually straightforward and these large tumors may be associated with mass effects such as severe headache, nerve palsies or visual changes. Hyperprolactinemia can be due to other causes like pregnancy, drugs, hypothyroidism, and pituitary stalk effect should be considered in the differential.
Clinical Case: A 30 year female having symptoms of prolactinoma showing amenorrhea of 3 years, galactorrhea, decrease libido who presented to the clinic for headache recently associated with anxiety. Headache initially frequently with 2-3 times in a week, moderate intensity lasting few hours, on one side of head and sometimes on the forehead, associated with blurry vision. Patient was on oral contraception taking on and off, but not on any medication currently. Initial work up consistent with hyperprolactinemia: elevated prolactin level 737.6 ng/ml (n 3.0-30 ng/ml), however other pituitary hormones are within normal limit, ACTH 32 pg/ml (n 6-50 pg/ml),FSH 3.2 mid/ml, LH 0.3miu/ml, total testosterone 13 ng/dl (n 2-35 ng/dl),TSH 1.6 mid/l (N 0.35-3.7 mid/l), FT4 0.92 ng/dl (N 0.76-1.46 ng/dl), human growth hormone &lt;0.1ng/ml (N &lt;=7.1 ng/dl), random cortisol 14.6 ug/dl (N 4.3-22.4 ug/dl). Pregnancy test is also negative. MRI Brain with and without contrast revealed a mass within the pituitary fossa producing smooth depression of the floor and suprasellar extension just the right of midline producing mild impingement on the undersurface of the optic chiasm, craniocaudal dimension of the tumor approximately 1.85 cm by 1.8 cm mediolateral by 1.4 cm anteroposterior without encasement or diminution of carotid arteries flow. Within the sella there is a spherical collection of T1 high signal displaying a fluid level strongly concerning for hemorrhage within the pre-existing tumor. There is no prior CT Head or MRI Brain available. Ophthalmology evaluation showed normal dilated retinal exam and no visual field defects. Medical treatment with Cabergoline 0.25 mg PO twice weekly was given on the visit, however patient did not start it yet and repeat prolactin level with 629 ng/ml. Patient endorsed that headache is better with less frequent about once in two weeks but still with absence of menstruation.
Conclusion: This is the case of prolactin secreting pituitary macroadenoma with the change in character of headache due to hemorrhage in the tumor, having gradual improvement of headache which may be due to spontaneous resolving of hemorrhage from the pre-existing pituitary mass. It is very important to inform the patient with pituitary tumor go to ED if there is change in headache or vision.​
Deep Venous Thrombosis in a Patient With Cushing’s Syndrome
Authors: Rana Sanah , Brutsaert Erika 
Published: May 2021

Abstract
Introduction: Cushing’s syndrome (CS) is associated with a prothrombotic state due to increased activation of several clotting factors. As such, patients are more susceptible to development of post-operative venous thrombosis. We present the case of a patient who underwent pituitary surgery for CS and subsequently developed acute lower extremity deep venous thrombosis (DVT) after stopping anticoagulation upon discharge. Case Report: A 57-year-old female presented with abdominal pain 1 month after gastric bypass surgery. She was found to have intra-abdominal abscesses and started on IV antibiotics. Upon further questioning, she endorsed 2-week history of coarse hair over chin, difficulty climbing stairs, and easy bruising. She had type 2 diabetes and hypertension, both diagnosed 2 years prior. Blood pressure was 169/88 on admission. On exam she had coarse hair on chin, central obesity, scattered ecchymoses, proximal muscle weakness, and no abdominal striae. Labs revealed potassium level of 1.9 mmol/L despite supplementation with 60 meq/day of potassium chloride. Workup showed random cortisol 45 mcg/dl, ACTH 114 pg/ml, elevated urinary free cortisol to 898.6 ug/24 hours, unsuppressed cortisol to 30.6 mcg/dL with low-dose dexamethasone and partial cortisol suppression to 11 mcg/dl with high-dose dexamethasone. MRI brain showed pituitary adenoma measuring 6 x 9 x 2 mm. CRH stimulation test showed rise of ACTH to 302 pg/ml and cortisol to 81.5 mcg/dl consistent with ACTH-dependent hypercortisolism from pituitary tumor. While hospitalized, the patient received thromboprophylaxis with SQ enoxaparin 40 mg daily. Patient was started on ketoconazole and subsequently underwent transsphenoidal resection to remove the pituitary tumor, which effectively reduced her serum cortisol levels. She was started on hydrocortisone replacement. She was discharged to a skilled nursing facility 2 weeks later. However, she did not receive thromboprophylaxis at the facility and was diagnosed with acute lower extremity DVT soon after hospital discharge. Discussion: CS is associated with increased risk of thromboembolic events. Although our patient received DVT prophylaxis in the hospital until discharge 2 weeks after surgery, she developed DVT soon after discharge. This emphasizes the increased susceptibility to development of venous thrombosis even after normalization of cortisol levels following pituitary surgery. A recent retrospective study showed that patients with CS are at higher risk of thromboembolism for approximately 30 to 60 days during the postoperative period. This case highlights that the clotting risk remains elevated in patients with CS for an extended period after successful surgery. In conclusion, patients with CS are at high risk of thromboembolism even after surgery and extended thromboprophylaxis should be considered.
Hypopituitarism as the Initial Presentation of Pituitary Metastasis From Lung Cancer
Authors: Ayesh Hazem , Burmeister Cameron , Abdelrahman Ahmed , Beran Azizullah 
Published: May 2021

Abstract
Introduction: The annual incidence of hypopituitarism 4.2 cases of 100,000. Causes include primary tumor, metastasis, and non-tumor causes such as radiation therapy, infiltrative lesions, infection, and traumatic brain injury. Metastatic pituitary tumors constitute about 7-9% of the cases, with lung and breast cancers are the most common primary tumors.
Case Presentation: 48-year-old Caucasian female patient with 30 pack-year smoking history presented to the emergency department with abdominal pain, diarrhea, fatigue, and headache. Review of systems was significant for loss of appetite, left eye visual field defect, anorexia, cold intolerance, and shortness of breath. Physical examination was remarkable for decreased visual acuity. Vital signs notable for hypotension BP 92/63. Notable labs include glucose of 53 mg/dL, TSH 0.50 [0.49 - 4.67 uIU/mL], low free T4 0.52 ng/dL, low FSH 1.3 mIU/ml, low LH &lt;0.2 mIU/ml, low ACTH 1.4 pg/mL, low morning cortisol 2.2 ug/dL, low DHEA-SO4 3ug/dL, low IGF-1 16 ng/ml. She was diagnosed with hypopituitarism and started on IV hydrocortisone 50 mg every 6 hours with 75 mcg levothyroxine daily. MRI brain showed interval growth of pituitary lesion into the suprasellar cistern with a mass-effect on the optic chiasm measuring 2.4 X1.6X 1.9 cm with a lesion in the right cerebellar hemisphere. Vertebral MRI showed multiple metastatic lesions in cervical/thoracic/lumbar vertebral bodies. On day two of hospital stay, she developed hypertonic hyponatremia (sodium 156 mmol/L, urine osmolarity 81 mOsm/kg, plasma osmolarity 328 mOsm/kg), and she was started on desmopressin 2 mg IV for diabetes insipidus. CT chest showed spiculated left upper lobe mass consistent with primary malignancy, and biopsy showed metastatic poorly differentiated epithelial malignancy likely from lung primary. Patient was discharged on desmopressin 100 mcg nightly, hydrocortisone 20 mg morning with 10 mg evening, levothyroxine 100 mcg daily with plans for further oncologic workup.
Discussion: Patient’s 2011 MRI brain showed a mildly enlarged pituitary gland. MRI brain two months before admission showed a pituitary gland diameter of 1.6 cm, while an MRI at presentation showed a pituitary
gland size of 2.4 cm with a new cerebellar lesion. She reported symptoms of nausea, vomiting, and weakness for more than one year ago but biochemical testing was not performed. The fact that the patient had pituitary enlargement eight years ago likely delayed the diagnosis of pituitary metastasis. Patient age precluded lung cancer screening despite smoking history and family history of lung cancer.
Conclusion: Symptomatic patients with pituitary enlargement on brain imaging may benefit from a close follow-up and biochemical testing for early diagnosis and treatment, especially if they have risk factors for malignancy.