Giant Prolactinoma Causing Proptosis and Stroke

Abstract Background: Although suprasellar and cavernous sinus invasion are common in giant prolactinomas, intra-orbital extension is extremely uncommon [1]. Even less reported are cases of giant prolactinomas causing cerebral ischemia or death. Clinical Case: A 51-year old woman presented to the ED with confusion, right-sided weakness and severe left eye proptosis with loss of vision. Five years prior, she underwent a partial transphenoidal resection for a macroprolactinoma due to acute vision changes with compression of the optic chiasm. Prior to surgery, prolactin level was elevated to 2,106 ng/mL (n 2.4-24.0 ng/mL). Post-operative MRI showed residual 2.7 x 3.1 x 2.6 cm mass. Thereafter she was prescribed cabergoline which she self-discontinued three years later. MRI of the brain at time of presentation demonstrated a 10.1 x 6.4 x 4.3 cm sellar/suprasellar mass extending into the left orbit causing severe proptosis and mass effect on the left frontal lobe, temporal lobe, midbrain, and basilar artery with encasement of the left cavernous internal carotid artery. A recent left striatocapsular infarct due to compression of the left middle cerebral artery was present. Prolactin level was elevated to 16,487 ng/mL. Neurosurgery was consulted and recommended medical management. Free thyroxine level was low and thyroid hormone replacement was started. Although the cosyntropin stimulation test showed an appropriate cortisol level peak of 21.5 mcg/dL, she was given stress dose glucocorticoids. Bromocriptine was initially started and titrated and later changed to cabergoline. Six weeks after discharge, she was readmitted with worsening confusion and seizure activity. On day 2 of admission, she decompensated. New hemorrhage inside the mass with increased vasogenic edema and a midline shift was discovered on a head CT. She underwent emergent craniotomy with surgical debulking of the tumor. Unfortunately, her mental status did not improve post-operatively. She was transitioned to hospice care and died 7 days after surgery. Surgical pathology showed a lactotroph adenoma with markedly elevated Ki67 proliferation index of 20-30%. Conclusion: This case demonstrates an unusually aggressive macroprolactinoma causing severe proptosis, ischemic stroke and death and adds to the very few cases previously reported [2]. References: 1. Karcioglu ZA, Aden LB, Cruz AA, Zaslow L, Saloom RJ. Orbital invasion with prolactinoma: a clinical review of four patients. Ophthalmic Plast Reconstr Surg. 2002 Jan;18(1):64-71. 2. Navarro-Bonnet J, Martínez-Anda JJ, Balderrama-Soto A, Pérez-Reyes SP, Pérez-Neri I, Portocarrero-Ortiz L. Stroke associated with pituitary apoplexy in a giant prolactinoma: a case report. Clin Neurol Neurosurg. 2014 Jan;116:101-3.

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Giant Prolactinoma presenting with facial nerve palsy and hemiparesis

Journal of the Endocrine Society ◽

10.1210/jendso/bvab069 ◽

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.

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Hyperbaric oxygen therapy in the treatment of malign edema complication after arteriovenous malformation radiosurgery

Undersea and Hyperbaric Medicine ◽

10.22462/10.12.2019.18 ◽

2019 ◽

pp. 713-717

Keyword(s):

Arteriovenous Malformation ◽

Hyperbaric Oxygen ◽

Vasogenic Edema ◽

Mass Effect ◽

Radiological Examination ◽

Temporal Region ◽

Left Frontal Lobe ◽

Steroid Resistant ◽

Alternative Treatment Option ◽

Martin Grade

A 16-year-old female patient with headache was admitted to our hospital. Radiological examination showed a Spetzler- Martin Grade III arteriovenous malformation (AVM) located at the left frontal lobe. Volume-staged stereotactic radiosurgery (SRS) treatment performed in two fractions at three-month intervals and post-procedural period were uneventful. Eight months later the patient was admitted to our hospital with headache, vomiting, right-sided facial palsy and right upper extremity paresthesia. Radiological examination demonstrated severe vasogenic edema in the left centrum semiovale and temporal region. Due to severe and steroid-resistant malign edema, hyperbaric oxygen (HBO2) therapy was performed as an alternative treatment option. Neurological symptoms resolved completely after HBO2. Radiological examination demonstrated serious improvement of brain edema and mass effect.

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Brain Abscess Masquerading as Brain Infarction

Brain Sciences ◽

10.3390/brainsci10070440 ◽

2020 ◽

Vol 10 (7) ◽

pp. 440

Author(s):

Da-Eun Jeong ◽

Jun Lee

Keyword(s):

Ischemic Stroke ◽

Acute Stroke ◽

Vasogenic Edema ◽

Brain Infarction ◽

Sudden Onset ◽

Restricted Diffusion ◽

Left Middle Cerebral Artery ◽

Magnetic Resonance Imaging Mri ◽

Radiographic Data

Occasionally, acute ischemic stroke can be difficult to differentiate from acute intracranial infection. We describe a patient who presented with sudden onset of right hemiparesis and fever. Magnetic resonance imaging (MRI) was consistent with an acute stroke, showing multiple lesions with restricted diffusion in the left middle cerebral artery territory. These lesions were not enhancing and were not associated with vasogenic edema. A diagnosis of acute stroke was made based on the clinical and radiographic data. Follow-up MRI obtained eleven days later showed interval development of ring enhancement and vasogenic edema surrounding the previously noted core of restricted diffusion. Based on these findings, the diagnosis was revised to cerebral abscesses and the patient was treated successfully with antibiotics. In retrospect, the largest diffusion-weighted lesion on baseline MRI demonstrated two characteristics that were atypical for stroke: it had an ovoid shape and a subtle T2 hypointense core. This case demonstrates that acute clinical and radiographic presentation of cerebral abscess and ischemic stroke can be strikingly similar. Follow-up imaging can be instrumental in arriving at an accurate diagnosis.

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

Journal of the Endocrine Society ◽

10.1210/jendso/bvaa046.544 ◽

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

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

Journal of the Endocrine Society ◽

10.1210/jendso/bvaa046.193 ◽

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

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

Journal of the Endocrine Society ◽

10.1210/jendso/bvaa046.1664 ◽

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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Pediatric Giant Prolactinoma Presenting With Acute Obstructive Hydrocephalus and Intracranial Hypertension

Journal of the Endocrine Society ◽

10.1210/jendso/bvab048.1434 ◽

2021 ◽

Vol 5 (Supplement_1) ◽

pp. A704-A704

Author(s):

Grace Hendrix ◽

Robert Benjamin ◽

Nancie J MacIver ◽

Daniel P Barboriak ◽

Pinar Gumus Balikcioglu

Keyword(s):

Intracranial Hypertension ◽

Tumor Volume ◽

Obstructive Hydrocephalus ◽

Brain Mri ◽

Case Series ◽

Vision Impairment ◽

Maximum Diameter ◽

Pituitary Hormone ◽

Giant Prolactinoma ◽

Giant Prolactinomas

Abstract Background: Pediatric prolactinomas (PP) are rare but represent 50% of all pediatric pituitary adenomas. Girls are affected more frequently than boys, although PP tend to be larger and more aggressive (earlier age, larger mass, and higher prolactin levels) in boys. Thus, microadenomas (tumors < 10 mm in diameter) are typical in females and macroadenomas (10–40 mm in diameter) are typical in males. Giant prolactinomas (> 40 mm in maximum diameter), an unusual subset of macroprolactinomas, are also commonly found in boys. In a large case series, the largest tumor volume reported was 93.5 cm3. Here we report a giant prolactinoma in a female requiring V/P shunt for decompression. Clinical Case: A 16-year old female presented with 2 weeks of intractable headache, nausea and vomiting, vision impairment, and changes in balance described as running into stationary household objects. Historical review revealed primary amenorrhea and short stature. On initial exam, the patient had a right eye afferent pupillary defect, concern for loss of color vision, and bilateral optic nerve edema with blurred disc margins. Brain MRI showed a large lobulated mass centered in the suprasellar cistern, measuring approximately 6.4 x 5.8 x 5.7 cm with a tumor volume of 105 cm3. There was extension superiorly, anteriorly, and laterally, with homogeneously enhancing and cystic components, and mass effect resulting in obstructive hydrocephalus. Differential diagnoses included craniopharyngioma, germinoma, and adenoma. Initial tests demonstrated prolactin of >2,000 ng/mL, with diluted result of 17,811.16 ng/mL. Morning fasting labs confirmed multiple anterior pituitary hormone deficiencies including central hypothyroidism, ACTH deficiency, GH deficiency, and hypogonadotropic hypogonadism. The patient was started on hydrocortisone and levothyroxine. Due to obstructive hydrocephalus and vision impairment, she underwent VP shunt placement for decompression. She was started on cabergoline for medical treatment of the tumor and did not require surgical resection. Repeat prolactin measurements have shown striking improvement (to 2,350 ng/ml, 824 ng/ml, and 152 ng/ml at 1 week, 1-month, and 2-month-follow-up, respectively) with central vision improved in both eyes, papilledema resolved, and resolution of headaches. Conclusion: Giant prolactinomas presenting with hydrocephalus and intracranial hypertension are very rare in pediatrics, especially in girls, and can vary greatly in mass characteristics and resulting hormone deficiencies. Our patient is unique with her large tumor volume and the extent of pituitary hormone deficiencies. Prolactin levels should be measured with all sellar masses, as this may prevent unnecessary invasive intervention and possibly provide prompt response to medical management.

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Giant Prolactinomas: Outcomes of Multimodal Treatments for 42 Cases with Long-Term Follow-Up

Experimental and Clinical Endocrinology & Diabetes ◽

10.1055/a-0597-8877 ◽

2018 ◽

Vol 127 (05) ◽

pp. 295-302 ◽

Cited By ~ 1

Author(s):

Liang Lv ◽

Yu Hu ◽

Senlin Yin ◽

Peizhi Zhou ◽

Yuan Yang ◽

...

Keyword(s):

Dopamine Agonist ◽

Early Stage ◽

Pituitary Tumors ◽

First Line ◽

Cavernous Sinus Invasion ◽

Initial Surgery ◽

Long Term Follow Up ◽

Giant Prolactinomas ◽

Almost All

AbstractGiant prolactinomas represent a rare entity of pituitary tumors so that the management of these patients is still a prevalent challenge at present. Paying special attention to the treatment strategy and outcomes, we presented a large series of 42 cases looking forward to share our understanding and experience in management of these patients. Male patients accounted for 71.4% of this series and were relatively younger (35.70±2.42 vs. 52.00±3.55 years, p=0.0011) and harbored bigger tumors (14.57 vs. 7.74 cm3, p=0.0179) compared to females. Almost all of these tumors showed suprasellar extension (97.6%) and cavernous sinus invasion (92.9%). Dopamine agonist represented an efficient method to control PRL concentrations (98.8%) and reduce tumor burdens (81.2 %). PRL normalization was detected in 13 out of the 27 patients initially treated with bromocriptine (BRC) whereas none of the 14 patients with first-line operation gained a normalization of PRL concentration after surgery. Although there was no reliable predictor of tumor response, First PRL reduction was a predictive criterion for the nadir PRL level during the long-time period of follow-up for first-line bromocriptine treatment. In conclusion, patients with giant prolactinomas did not gain more benefits from initial surgery. Dopamine agonist (BRC) should be first-line treatment for giant prolactinomas whereas operation merely served as a remedy for acute compression symptoms and dopamine agonist resistance. Consecutive monitoring of serum PRL levels in the early stage of initial BRC treatment is useful for evaluation of therapeutic effect and further therapeutic decision.

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About informative value of very high prolactin levels in patients presenting with prolactin-secreting pituitary macroadenomas (clinical observations)

Problems of Endocrinology ◽

10.14341/probl20115743-6 ◽

2011 ◽

Vol 57 (4) ◽

pp. 3-6

Author(s):

L I Astaf'eva ◽

B A Kadashev ◽

V D Tenedieva ◽

A V Moshkin ◽

M A Shifrin

Keyword(s):

Treatment Strategy ◽

Prolactin Level ◽

Plasma Samples ◽

Laboratory Studies ◽

Clinical Observations ◽

The Mean ◽

Giant Prolactinomas ◽

Very High ◽

Pituitary Macroadenomas ◽

Critical Situations

The mean blood prolactin level in patients with microprolactinomas is usually 3000-4000 mU/l compared with 10 000-20 000 mU/l in cases presenting with macroprolactinomas. It may be as high as 1 mln mU/l and more in patients with a giant tumour. The measurement of prolactin levels requires laboratory studies of strongly diluted plasma samples. Significant dilution is fraught with erroneous prolactin levels: nevertheless, this approach allows a tendency toward decreasing concentration of prolactin to be revealed which may be important for taking rapid decision concerning the choice of the treatment strategy (either surgical or medicamental) especially in critical situations. A few clinical cases of giant prolactinomas with an unusually high blood prolactin level are described with reference to its dynamics during treatment with cabergoline.

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Management of a decompensated acute-on-chronic intracranial venous sinus thrombosis

Therapeutic Advances in Neurological Disorders ◽

10.1177/1756286419895157 ◽

2019 ◽

Vol 12 ◽

pp. 175628641989515 ◽

Cited By ~ 2

Author(s):

Carmen Serna Candel ◽

Victoria Hellstern ◽

Tania Beitlich ◽

Marta Aguilar Pérez ◽

Hansjörg Bäzner ◽

...

Keyword(s):

Magnetic Resonance ◽

Endovascular Treatment ◽

Sinus Thrombosis ◽

Vasogenic Edema ◽

Factor V Leiden ◽

Venous Sinus ◽

Venous Stasis ◽

Left Frontal Lobe ◽

Factor V Leiden Mutation ◽

Venous Sinus Thrombosis

A 34-year-old female patient presented during the 10th week of her second gravidity with headache, nausea and vomiting 2 weeks before admission. Her medical history was remarkable for a heterozygous factor V Leiden mutation, elevated lipoprotein A, and a cerebral venous thrombosis (CVT) after oral contraceptive intake 15 years before. Magnetic resonance imaging (MRI) suggested acute and massive intracranial sinus thrombosis. Despite full-dose anticoagulation, the patient deteriorated clinically and eventually became comatose. Now, MRI/magnetic resonance angiography revealed vasogenic edema of both thalami, of the left frontal lobe, and of the head of the caudate nucleus, with venous stasis and frontal petechial hemorrhage. She was referred for endovascular treatment. Diagnostic angiography confirmed a complete superficial and deep venous sinus occlusion. Endovascular access to the straight and superior sagittal sinus was possible, but neither rheolysis nor balloon angioplasty resulted in recanalization of the venous sinuses. Monitored heparinization was continued and antiaggregation was initiated. The patient remained comatose for another 5 days and MRI showed progress of the cytotoxic edema. On day 6, infusion of eptifibatide at body-weight-adapted dosage was started. The following day, the patient improved and slowly regained consciousness. MRI confirmed regression of the edema. The eptifibatide infusion was continued for a total of 14 days. Thereafter two doses of 180 mg ticagrelor per os (PO) daily were started. The patient remained on acetylsalicylic acid (ASA), ticagrelor, and enoxaparin on an unchanged dosage regimen. She was discharged home 26 days after the endovascular treatment without serious neurological deficit, with the pregnancy intact. At the 30th week of pregnancy the dosage of ASA was reduced to 300 mg once PO daily. Cesarian delivery was carried out at the 38th week of pregnancy. The newborn was completely healthy. Ultima ratio therapeutic options for severe intracranial venous sinus thrombosis refractory to anticoagulation are discussed, with an emphasis on platelet-function inhibition.

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