scholarly journals SUN-285 Too Big to Be True, Too Young to Stroke!

2020 ◽  
Vol 4 (Supplement_1) ◽  
Author(s):  
Fatima Jalil ◽  
Lori De La Portilla ◽  
Joseph Anthony Lorenzo ◽  
Faryal Sardar Mirza
Keyword(s):  
Erectile Dysfunction ◽  
Case Reports ◽  
Sella Turcica ◽  
Mass Effect ◽  
Lower Extremity Weakness ◽  
Stroke Treatment ◽  
Basal Nuclei ◽  
Giant Prolactinoma ◽  
First Line Therapy ◽  
Giant Prolactinomas

Abstract BACKGROUND: Lactotroph adenomas are the most common type of pituitary adenomas and can cause infertility and menstrual irregularities in women; hypogonadism and gynecomastia in men.1 Giant prolactinomas are an unusual subset of pituitary macroadenomas with limited literature available on their management.2 We describe an unusual case of giant prolactinoma in a young man who presented with symptoms of stroke, that reversed with treatment with cabergoline. Clinical Case: 25-year old man presented with gradually progressing upper extremity weakness for evaluation of stroke. He reported stumbling into things when walking. There was a question of left sided facial droop and Bell’s palsy in recent past. He reported recent weight gain and erectile dysfunction. He was noted to have left homonymous hemianopsia on exam in addition to left upper and lower extremity weakness. MRI Brain showed an enormous mass that filled the sella turcica, invaded the sphenoid sinus and right side of the skull base, invaginating deep into the base of the right cerebral hemisphere with mass effect on the pons, right-sided midbrain, right temporal lobe and right basal nuclei, measuring 6.3 X 5.5 x 7.5 cm. Pituitary hormonal evaluation showed elevated prolactin (PRL) level with dilution at 13,580 ng/mL, with low testosterone (T) level (total T 42 ng/dL, free T 10 pg/mL, SHBG 15 nmol/L). Thyroid and adrenal axes were intact with normal IGF-1 level. In view of very high PRL level, he was started on cabergoline 0.5 mg daily initially and decreased to every other day after 2 weeks as PRL level began to decline. In 8 months, PRL levels decreased to 1293.07 ng/dl (90% reduction) and prolactinoma decreased to 6.0 x 3.7 x 4.7 cm (56% volume reduction). Total and free T improved to 134 ng/dL and 31 pg/dL respectively. He experienced marked improvement in left hemianopsia, with resolution of weakness and slurred speech. Energy level and erectile dysfunction improved. Currently he is being maintained on 0.5 mg cabergoline every other day Conclusion: Giant prolactinomas are uncommon and can present with compressive symptoms, that can be mistaken as stroke. Treatment with anticoagulation may cause hemorrhage and apoplexy with worsening of symptoms.1,2 There is limited data available regarding first line therapy for giant prolactinomas with 2 case reports where giant prolactinomas have been treated effectively with cabergoline.3,4 It is important to recognize the cause of such symptoms, and treated where possible with effective medical therapy to prevent morbidity. References: 1. Moraes A et al., Giant prolactinomas: the therapeutic approach. Clin Endo (Oxf). 2013 Oct;79(4):447-56 2. Acharya SV et al., Giant prolactinoma and effectiveness of medical management. Endocr Pract. 2010 Feb;16(1):42-6 3. Ahmed, M, et al., Large Prolactinoma. NEJM 2010; 363:177 4. Masoud, R et al., Giant prolactinoma: case report. J Diabetes Metab Disord. 2013; 12: 3

scholarly journals Giant Prolactinoma presenting with facial nerve palsy and hemiparesis

2021 ◽  
Author(s):  
Aleksandra Sliwinska ◽  
Fatima Jalil ◽  
Lori De La Portilla ◽  
Michael Baldwin ◽  
Joseph Lorenzo ◽  
...  
Keyword(s):  
Facial Nerve ◽  
Prolactin Level ◽  
Nerve Palsy ◽  
Facial Nerve Palsy ◽  
Sella Turcica ◽  
Neurological Deficits ◽  
Lower Extremity Weakness ◽  
Giant Prolactinoma ◽  
Giant Prolactinomas ◽  
The Brain

Abstract Background Giant prolactinomas are an exceedingly uncommon type of pituitary adenomas that usually occur in men, cause extremely high prolactin levels and mass related symptoms. Rarely, patients may experience neurological deficits resembling ischemic events. Methods We describe an unusual case of a young man who presented with stroke-like symptoms and was found to have a giant prolactinoma. Clinical Case 25-year-old man presented with the left facial droop and gradually progressing upper and lower extremity weakness for evaluation of stroke. He reported recent weight gain and erectile dysfunction. Physical exam revealed left homonymous hemianopsia, left VII nerve palsy, and left hemiparesis. MRI of the brain showed an enormous mass in the sella turcica, which invaded the sphenoid sinus and right side of the skull base. Prolactin level was elevated at 13580 ng/mL, and the testosterone level was low. The patient was started on cabergoline and had marked improvement in his symptoms in a few months. Fifteen months after starting treatment, he has had more than 90% reduction in tumor volume and a 93% reduction in prolactin level. Conclusion Giant prolactinomas are uncommon and present with compressive symptoms that can be mistaken for a stroke. Our case is a unique report of a facial nerve palsy and hemiparesis secondary to giant prolactinoma in the absence of stroke or pituitary apoplexy.

scholarly journals Effectiveness of long-term cabergoline treatment for giant prolactinoma: study of 12 men

2007 ◽  
Vol 156 (2) ◽  
pp. 225-231 ◽  
Author(s):  
Ilan Shimon ◽  
Carlos Benbassat ◽  
Moshe Hadani
Keyword(s):  
Visual Field ◽  
The Other ◽  
Serum Prolactin ◽  
Visual Field Defects ◽  
First Line ◽  
Giant Prolactinoma ◽  
First Line Therapy ◽  
Line Therapy ◽  
Giant Prolactinomas ◽  
Field Defects

Objective: To review our experience with cabergoline, a D2-selective dopamine agonist, for the treatment of giant prolactinomas. Design: A retrospective case series; descriptive statistics. Methods: The study group included 12 men aged 24–52 years (mean 39.2 years) treated for giant prolactinoma at our centers from 1997 to 2006. Cabergoline was started at a dose of 0.5 mg/three times a week and progressively increased as necessary to up to 7 mg/week. Patients were followed by hormone measurements, sellar magnetic resonance imaging, and visual examinations. Results: In ten patients, cabergoline served as first-line therapy. The other two patients had previously undergone transsphenoidal partial tumor resection because of visual deterioration. Mean serum prolactin level before treatment was 14 393 ± 14 579 ng/ml (range 2047–55 033 ng/ml; normal 5–17 ng/ml). Following treatment, levels normalized in ten men within 1–84 months (mean, 25.3 months) and decreased in the other two to 2–3 times of normal. Tumor diameter, which measured 40–70 mm at diagnosis, showed a mean maximal decrease of 47 ± 21%; response was first noted about 6 months after the onset of treatment. Nine patients had visual field defects at diagnosis; vision returned to normal in three of them and improved in five. Testosterone levels, initially low in all patients, normalized in eight. There were no side effects of treatment. Conclusion: Cabergoline therapy appears to be effective and safe in men with giant prolactinomas. These findings suggest that cabergoline should be the first-line therapy for aggressive prolactinomas, even in patients with visual field defects.

scholarly journals Fetus in fetu: two case reports from North African country

2021 ◽  
Vol 69 (1) ◽  
Author(s):  
Moutaz Ragab ◽  
Omar Nagy Abdelhakeem ◽  
Omar Mansour ◽  
Mai Gad ◽  
Hesham Anwar Hussein
Keyword(s):  
Histopathological Examination ◽  
Case Reports ◽  
Abdominal Mass ◽  
Mature Teratoma ◽  
Surgical Techniques ◽  
Rare Condition ◽  
Mass Effect ◽  
Fetus In Fetu ◽  
Vascular Connection ◽  
First Case

Abstract Background Fetus in fetu is a rare congenital anomaly. The exact etiology is unclear; one of the mostly accepted theories is the occurrence of an embryological insult occurring in a diamniotic monochorionic twin leading to asymmetrical division of the blastocyst mass. Commonly, they present in the infancy with clinical picture related to their mass effect. About 80% of cases are in the abdomen retroperitoneally. Case presentation We present two cases of this rare condition. The first case was for a 10-year-old girl that presented with anemia and abdominal mass, while the second case was for a 4-month-old boy that was diagnosed antenatally by ultrasound. Both cases had vertebrae, recognizable fetal organs, and skin coverage. Both had a distinct sac. The second case had a vascular connection with the host arising from the superior mesenteric artery. Both cases were intra-abdominal and showed normal levels of alpha-fetoprotein. Histopathological examination revealed elements from the three germ layers without any evidence of immature cells ruling out teratoma as a differential diagnosis. Conclusions Owing to its rarity, fetus in fetu requires a high degree of suspicion and meticulous surgical techniques to avoid either injury of the adjacent vital structures or bleeding from the main blood supply connection to the host. It should be differentiated from mature teratoma.

scholarly journals Delayed Extradural Hematoma after Evacuation of Contralateral Acute Extradural Hematoma

2020 ◽  
Author(s):  
Majid Anwer ◽  
Atique Ur Rehman ◽  
Farheen Ahmed ◽  
Satyendra Kumar ◽  
Md Masleh Uddin
Keyword(s):  
Computed Tomography ◽  
Ct Scan ◽  
Postoperative Period ◽  
Road Traffic ◽  
Case Reports ◽  
Mass Effect ◽  
Extradural Hematoma ◽  
Traumatic Head Injury ◽  
Neurologic Deterioration ◽  
The Right

Abstract Introduction Traumatic head injury with extradural hematoma (EDH) is seen in 2% of patients. Development of EDH on the contralateral side is an uncommon complication that has been reported in various case reports. Case Report We report here a case of an 18-year-old male who had a road traffic injury. He was diagnosed as a case of left-sided large frontotemporoparietal acute extradural bleed with a mass effect toward the right side. He was managed with urgent craniotomy and evacuation of hematoma. A noncontrast computed tomography (NCCT) scan performed 8 hours after postoperative period showed a large frontotemporoparietal bleed on the right side with a mass effect toward the left side. He was again taken to the operating room and right-sided craniotomy and evacuation of hematoma were performed. A postoperative NCCT scan revealed a resolved hematoma. The patient made a complete recovery in the postoperative period and is doing well. Conclusion Delayed onset epidural hematoma is diagnosed when the initial computed tomography (CT) scan is negative or is performed early and when late CT scan performed to assess clinical or ICP deterioration shows an EDH. The diagnosis of such a condition requires a high index of suspicion based on the mechanism of injury along with fracture patterns. Additionally, change in pupillary size, raised intracranial pressure, and bulging of the brain intraoperatively are additional clues for contralateral bleeding. Neurologic deterioration may or may not be associated with delayed EDH presentation. An early postoperative NCCT scan within 24 hours is recommended to detect this complication with or without any neurologic deterioration.

scholarly journals INFECTIOUS AETIOLOGY OF MARGINAL ZONE LYMPHOMA AND ROLE OF ANTI-INFECTIVE THERAPY

2016 ◽  
Vol 8 ◽  
pp. 2016006 ◽  
Author(s):  
Salvatore Perrone ◽  
Gianna Maria D'Elia ◽  
Alessandro Pulsoni
Keyword(s):  
Malt Lymphoma ◽  
Marginal Zone ◽  
Case Reports ◽  
Lymphocyte Activation ◽  
Chlamydia Psittaci ◽  
Gastric Malt Lymphoma ◽  
First Line Therapy ◽  
Causative Relationship ◽  
Chronic Hcv

Marginal zone lymphomas have been associated with several infectious agents covering both viral and bacterial pathogens and in some cases a clear aetiological role has been established. Pathogenetic mechanisms are currently not completely understood, however the role of chronic stimulation of the host immune response with persistent lymphocyte activation represents the most convincing explanation for lymphoproliferation. Gastric MALT lymphoma is strictly associated with Helicobacter pylori infection and various eradicating protocols, developed due to increasing antibiotic resistance, represent the first line therapy. The response rate to eradication is good with 80% of response at 1 year; this finding is also noteworthy because recapitulates a cancer cured only by antibacterial approach and it satisfies the Koch postulates of causation, establishing a causative relationship between Hp and gastric MALT lymphoma. Patients with chronic HCV infection have 5 times higher risk to develop MZL, in particular an association with splenic and nodal MZL has been shown in several studies. Moreover, there is evidence of lymphoma regression after antiviral therapy with interferon+ribavirin, thus rising hope that new available drugs, extremely effective against HCV replication, could improve outcome also in HCV-driven lymphomas. The rare cases of MZL localized to orbital fat and eye conjunctivas have been associated with Chlamydia psittaci infection carried by birds. Efficacy of antibacterial therapy against C. psittaci are conflicting and generally poorer thain gastric MALT. Finally some case-reports will cover the relationship between primary cutaneous B-cell Lymphomas and Borrelia Burgdorferi.

scholarly journals Treatment of Pyodermatitis-Pyostomatitis Vegetans: A Systematic Review and Meta-analysis

2021 ◽  
Vol 5 (4) ◽  
pp. 333-346
Author(s):  
Landon Kaleb Hobbs ◽  
Alina Zufall ◽  
Shadi Khalil ◽  
Richard Flowers
Keyword(s):  
Case Reports ◽  
Meta Analysis ◽  
Case Series ◽  
Calcineurin Inhibitors ◽  
Additional Patient ◽  
Aminosalicylic Acid ◽  
First Line Therapy ◽  
Pyostomatitis Vegetans ◽  
Oral Corticosteroids ◽  
Topical Medications

Background: Pyodermatitis-pyostomatitis vegetans (PDV-PSV) is a rare muco-cutaneous disorder characterized by vegetating and pustular plaques and is often associated with inflammatory bowel disease (IBD). The purpose of this study was to systematically identify and analyze reports of PDV-PSV to determine the most effective treatment. Methods: Reports of PDV-PSV were identified using the OVID-Medline database from inception through November 2019. Publications were excluded if no new patient case was included, if there was not clinical and histological evidence of PDV, PSV, or PDV-PSV, or if no treatment was discussed. Results: The final sample was comprised of 74 publications plus an additional patient from the authors’ institution, corresponding to 95 total patients. The basis of the review and analysis is limited to case reports and case series, which likely only report the cases with positive outcomes. Statistical analysis revealed that oral corticosteroids (OCS), 6-mercaptopurine/azathioprine, oral calcineurin inhibitors (OCNI), 5-aminosalicylic-acid (5-ASA), and biologics (BIO) were the most effective treatments for PDV-PSV. Topical medications, colchicine, oral dapsone, and other antibiotics were ineffective treatments, with topical medications being the least effective option. When OCS are used, they work best when used as initial treatment to induce remission. 5-ASA and BIO are most effective when used as maintenance therapies after initial remission. Conclusions: Thus, first line therapy for PDV-PSV should begin with OCS with transition to steroid-sparing agents including OCNI, BIO, and 5-ASA if indicated.  

Endovascular Treatment of an Aneurysm of a Persistent Primitive Hypoglossal Artery With Complete Resolution of Brainstem Compressive Symptoms: Case Report

Neurosurgery ◽  
2011 ◽  
Vol 68 (3) ◽  
pp. E854-E857 ◽  
Author(s):  
Ferdinand K. Hui ◽  
Albert J. Schuette ◽  
Charles M. Cawley
Keyword(s):  
Lower Extremity ◽  
Complete Resolution ◽  
Mass Effect ◽  
Neurological Deficits ◽  
Lower Extremity Weakness ◽  
Computed Tomographic ◽  
Hypoglossal Artery ◽  
Patient Reported ◽  
Persistent Primitive Hypoglossal Artery ◽  
The Right

Abstract BACKGROUND AND IMPORTANCE: Aneurysms of the posterior circulation may manifest with neurological deficits related to mass effect on the brainstem. We present an unusual case of an aneurysm resulting in selective lower-extremity weakness and gait instability. CLINICAL PRESENTATION: A 61-year-old man presents with progressively worsening gait instability over the course of several months. A magnetic resonance image and computed tomographic angiogram demonstrate a persistent hypoglossal artery associated with an aneurysm invaginating into the pontomedullary junction. The patient manifested only lower-extremity symptoms. An endovascular approach through the right internal carotid artery and persistent primitive hypoglossal artery was assayed, coiling off the aneurysm with complete angiographic occlusion. One month after the procedure, the patient reported marked improvement in symptoms with residual difficulty walking. At the 1-year postprocedure interval, he reported nearly complete resolution of symptoms. CONCLUSION: Endovascular therapy of an aneurysm invaginating into the brainstem is safe and efficacious.

scholarly journals SUN-279 A Rare Case of Atypical Rhaboid Teratoid Tumour with Germinoma Differentiation in a 59 Year Old Woman

2020 ◽  
Vol 4 (Supplement_1) ◽  
Author(s):  
Melissa-Rosina Pasqua ◽  
Huda Altoukhi ◽  
Valerie Panet-Raymond ◽  
Denis Sirhan ◽  
Jason Karamchandani ◽  
...  
Keyword(s):  
Diabetes Insipidus ◽  
Case Reports ◽  
Hormone Replacement ◽  
Sella Turcica ◽  
Proliferation Index ◽  
Pituitary Hormone ◽  
Malignant Tumours ◽  
Rhabdoid Tumour ◽  
Mixed Features ◽  
Atypical Teratoid

Abstract Atypical teratoid/rhabdoid tumours (ATRT) are a rare class of central nervous system malignant tumours which are comprised of elements of ectoderm and mesoderm germ-cell layers, but exhibit microscopic similarity to skeletal muscle. These tumours are more commonly seen in pediatric patients, with few case reports recently describing adult patients with this condition, in particular middle-aged women.1–3 We present the case of a previously healthy 59-year-old woman who was found incidentally to have a pituitary mass on CT head, with retrospective symptoms of headaches, polyuria, polydipsia, diplopia, and low blood pressure. At presentation, she was found biochemically to have pan-hypopituitarism with a left cranial nerve six deficit, with an MRI depicting a 19.5 x 22 x 11 mm suprasellar mass extending into the infundibulum and hypothalamus, with displacement of the optic chiasm; repeat imaging ruled out apoplexy. She was started on supplemental levothyroxine and hydrocortisone replacement therapy, and sent for urgent transsphenoidal resection, which was complicated afterwards by hypernatremia from diabetes insipidus. Preliminary reports were suggestive of germinoma given the diffuse presence of Oct 3/4 and C-kit, with a proliferation index of 99%; further cytology of lumbar puncture revealed no malignant cells. However, upon further pathological analysis, her tumour demonstrated loss of INI-1 expression, which is diagnostic of ATRT. Given the mixed features on immunohistochemistry, the final diagnosis was concluded as an atypical teratoid/rhabdoid tumour of the sella turcica with germinoma differentiation. A multi-disciplinary approach consisted of initial radiotherapy, with chemotherapy targeted towards a germinoma-type tumour, and pituitary hormone replacement including treatment for central diabetes insipidus. This represents a unique case of a rare tumour with germinoma differentiation in an older patient that has not been previously reported. References 1. Journal of Clinical Neuroscience 49 (2018) 16–21 2. Acta Neurochir (Wien) (2008) 150: 491–496 3. Surgical Neurology International 2014, 5:75

scholarly journals SAT-247 Use of Double Dopamine Agonists in Giant Prolactinomas: A Series of 6 Cases

2020 ◽  
Vol 4 (Supplement_1) ◽  
Author(s):  
Yueh Chien Kuan
Keyword(s):  
Visual Field ◽  
Medical Therapy ◽  
Visual Field Defect ◽  
Tolerability Profile ◽  
Residual Tumour ◽  
Careful Monitoring ◽  
Presenting Symptoms ◽  
Giant Prolactinoma ◽  
Giant Prolactinomas ◽  
Field Defect

Abstract Dopamine agonist monotherapy is first line therapy in giant prolactinomas even when visual field defect is present. The costlier cabergoline is often preferred over bromocriptine due to higher efficacy and tolerability profile. Described herein combined cabergoline and bromocriptine therapy in 6 cases of giant prolactinomas. Retrospective records review of 6 patients with giant prolactinoma (3 males: M1-M3, 3 females: F1-F3) in a single tertiary centre was performed. Mean age at diagnosis: 29 years (range 17-39). Mean duration of follow up: 7 years (range 3-11). Headache and visual field defect were the presenting symptoms in all cases. Basal prolactin concentration: 100000 to 468851 mIU/L (<300 for male, <600 for female). Three patients have hypopituitarism at presentation, one after surgery and one remained eupitary 5 years after diagnosis. One developed late onset hypopituitarism 4 years after normalisation of prolactin levels. Three patients underwent debulking at presentation because of significant mass effects with obstructive hydrocephalus. In all patients cabergoline 1-1.5 mg/wk was started at diagnosis and gradually increased to 0.5 mg daily, aiming for normoprolactinemia. From May 2017 bromocriptine were given to these patients who continued to have hyperprolactinemia despite cabergoline 3.5-4mg/wk. Bromocriptine was commenced 1.25-5mg/day and gradually increased to 10 mg/day on top of cabergoline with careful monitoring of prolactin levels and side effects. Cabergoline was tapered down to 1.5-2mg/wk if prolactin levels remained stable between 2-3x normal while maintaining dose of bromocriptine. In M1, cabergoline was tapered off while maintaining bromocriptine 10mg/day with stable prolactin levels (~1000 mIU/L). In M2, normoprolactinemia was achieved after adding on bromocriptine and is currently on cabergoline 2mg/week and bromocriptine 10mg/day. In M3, whose prolactin were 4x normal value despite cabergoline 3.5mg/week, decreased 50% with bromocriptine 5 mg/day and remained stable when cabergoline reduced to 1.5mg/week. F1 had transphenoidal section twice due to failure of medical therapy. Her prolactin remained markedly elevated 10000-20000 mIU/L despite cabergoline 3.5 mg/week and bromocriptine 10mg/day, with persistent bitemporal hemianopia. F2 developed erythema nodosum after starting bromocriptine which was stopped and continued with cabergoline 1 mg/week. F3 showed partial response with 50% reduction in prolactin to 4485 mIU/L with bromocriptine 10 mg/day and cabergoline 1.5mg/week. In patients who underwent debulking, residual tumour remained unchanged. Two patients - tumour shrank 40% (F2) and 90% (M3) with medical therapy alone. In conclusion, adding on bromocriptine can be considered when high dose cabergoline is required for treatment of giant prolactinoma with careful monitoring. This reduces cabergoline dose which saves cost.

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