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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 9  |  Issue : 1  |  Page : 52-55

Cerebral venous sinus thrombosis following trans-sphenoidal excision of pituitary adenoma: A case report


Department of Neurosurgery, B.Y.L. Nair Charitable Hospital, Topiwala National Medical College, Mumbai, Maharashtra, India

Date of Submission15-Jun-2021
Date of Decision21-Jun-2021
Date of Acceptance30-Jun-2021
Date of Web Publication27-Aug-2021

Correspondence Address:
Dr. Srikant Balasubramaniam
Department of Neurosurgery, B.Y.L. Nair Charitable Hospital, Topiwala National Medical College, Mumbai, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jcvs.jcvs_13_21

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  Abstract 


A 52-year-old female diagnosed to harbour a non-functioning pituitary adenoma underwent trans-sphenoidal excision of the tumour. On the 8th post-operative day, the patient developed severe headaches and had an episode of generalised convulsion. Post-ictally, the patient was drowsy, irritable, apahasic and developed right-sided hemiparesis. Computed tomography of the brain revealed a left parietal venous infarct. Magnetic resonance venography confirmed thrombosis of the straight sinus, left transverse sinus, left sigmoid sinus, left internal jugular vein and cortical veins in the left high parietal region. The patient's thrombophilia profile was positive only for heterozygous Factor V Leiden mutation. The patient was treated conservatively with anticonvulsants and low-molecular-weight heparin. The patient recovered completely within a week and was discharged. Cerebral venous sinus thrombosis (CVST) has been rarely reported in the post-operative period following trans-sphenoidal surgery for pituitary adenoma. Early diagnosis and treatment of CVST is necessary for a favourable outcome.

Keywords: Cerebral venous sinus thrombosis, Factor V Leiden mutation, pituitary adenoma, transsphenoidal surgery


How to cite this article:
Thombare PR, Nadkarni VT, Balasubramaniam S. Cerebral venous sinus thrombosis following trans-sphenoidal excision of pituitary adenoma: A case report. J Cerebrovasc Sci 2021;9:52-5

How to cite this URL:
Thombare PR, Nadkarni VT, Balasubramaniam S. Cerebral venous sinus thrombosis following trans-sphenoidal excision of pituitary adenoma: A case report. J Cerebrovasc Sci [serial online] 2021 [cited 2021 Dec 1];9:52-5. Available from: http://www.jcvs.com/text.asp?2021/9/1/52/324809




  Introduction Top


Cerebral venous sinus thrombosis (CVST) is a rare cerebrovascular disease involving the superficial and deep venous system of the brain. It accounts for about 0.5%–1% of all kinds of stroke.[1] Because of the rarity of the disease and its non-specific presentation, the diagnosis of CVST is difficult and frequently delayed.[2] Hypercoagulable state is a well-known factor for the development of CVST. CVST has not been reported following transsphenoidal excision of a non-functioning pituitary adenoma. Our literature review has yielded only three cases of the occurrence of CVST following trans-sphenoidal surgery, involving one craniopharyngioma and two cases of ACTH secreting pituitary tumours with Cushing's disease. The possible causes, diagnosis and management of CVST in the post-operative period after transsphenoidal surgery are discussed.


  Case Report Top


A 50-year-old female presented with chief complaints of headache for 15 days. She had no neurological deficit. Her vision, visual fields and fundoscopy were normal. Routine haematology, blood coagulation profile and hormonal assays were normal. Computed tomography (CT) of the brain showed an isodense mildly enhancing lesion in the sellar region with suprasellar extension. Magnetic resonance (MR) images of the brain showed a predominant sellar mass which was isointense on T1-weighted images, hyperintense on T2-weighted images, and with mild heterogeneous post-contrast enhancement with few cystic areas, suggestive of pituitary adenoma [Figure 1]a. The patient underwent a sublabial, transseptal, transsphenoidal excision of pituitary macroadenoma. The sella and sphenoid was packed with autologous fat harvested from the patient's right thigh [Figure 1]b. As there was a breach of arachnoid and intraoperative cerebrospinal fluid (CSF), a lumbar drain was inserted which was removed on the 5th post-operative day. Post-operatively, the patient was recovering well and there was no CSF leak. She had transient diabetes insipidus for 3 days which required use of subcutaneous vasopressin replacement. Serum electrolytes were in the normal range during this period. Microscopy section showed a tumour in papillae in sheets consisting of monotonous population. The tumour cells were rounded with pale eosinophilic cytoplasm, round nuclei and speckled chromatin. The histopathological features were diagnostic of a pituitary adenoma. On the 8th post-operative day, the patient had severe headaches followed by an episode of generalised tonic–clonic convulsion. Post-ictally, the patient was drowsy and irritable and progressively developed aphasia and right hemiparesis. CT scan of the brain showed a venous haemorrahgic infarct in the left parietal area [Figure 2]. MR venography (MRV) of the brain demonstrated thrombosis of left transverse sinus, left sigmoid sinus, left internal jugular vein and cortical veins in the left parietal region confirming CVST [Figure 3]. The patient's thrombophilia profile showed that serum homocysteine levels were normal. The antinuclear antibody, anti-double stranded deoxyribonucleic acid, cardiolipin antibody and Prothrombin gene mutation were absent; however, heterozygous Factor V Leiden mutation was positive. The patient was treated conservatively with anticonvulsant and low-molecular-weight heparin (LMWH). The patient was administered LMWH till oral warfarin achieved prolongation of international normalised ratio more than two. The patient showed a complete neurological recovery of aphasia and right hemiparesis in the next 7 days at discharge. The patient was advised oral warfarin till 6 months.
Figure 1: (a) Magnetic resonance T2-weighted sagittal image shows a sellar tumour. (b) Post-operative sagittal magnetic resonance image showing complete excision of the pituitary adenoma and packing of sella and sphenoid sinus with fat

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Figure 2: Plain computed tomography scan of the brain, axial image shows a left parietal, parasagittal venous infarct

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Figure 3: Magnetic resonance venography shows thrombosis of the left transverse sinus, sigmoid sinus and internal jugular vein. There is a paucity of cortical veins in the left parietal area suggestive of thrombosis

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  Discussion Top


Intracranial cerebral venous and sinus thrombosis, (CVST) was first described by Ribes in 1825.[3] The sagittal sinus has the largest diameter among intracranial vessels consequently the blood flow velocity in the sagittal sinus is slower than that in other cerebral vessels, which is the pathophysiological basis of thrombus formation. A study on a large sample of CVST patients revealed that the transverse sinus was the most common site of thrombosis (73.4%), followed by the sigmoid sinus (39.8%) and superior sagittal sinus (38.9%).[4]

In the International Study on Cerebral Vein and Dural Sinus Thrombosis, the female sex was stated to be one of the major predisposing risk factors for CVST and many of the identified causes of CVST were related to risk factors unique to women.[5] These include pregnancy, oestrogen contraception, hormonal replacement therapy and selective oestrogen receptor modulators. CVST causes can be divided into two categories, inflammatory and non-inflammatory. The former is caused by intracranial vascular inflammation, which always affects the cavernous sinus, sigmoid sinus and transverse sinus while the latter is due to a high coagulation state of blood.[6]

Dehydration, hypovolaemia, intracranial hypotension and hypernatremia associated with inadequate fluid intake and diabetes insipidus leads to venous stasis which potentiates CVST. The veins and dural sinuses distend in this setting of depleted intravascular volume and decreased cerebrospinal fluid (CSF).[5],[7] This distention and associated delayed venous transit time and increased blood viscosity may promote thrombus formation. CSF drainage through lumbar drain placed for post-trans-sphenoidal CSF leak management may aggravate the above. CVST in the post-operative period following trans-sphenoidal surgery may occur commonly due to reduced intravascular volume secondary to inadequate hydration and diabetes insipidus or secondary to depleted CSF by lumbar drainage. Parenteral use of desmopressin has been shown to increase von Willebrand factor and factor VIII levels which has been associated with thrombotic events.[8]

Our literature search has yielded three previous cases of occurrence of CVST following trans sphenoidal surgery. Of these in two cases the high cortisol state of Cushing's disease due to a functioning pituitary adenoma was the cause of thrombosis. Glucocorticoids cause overproduction of von Willebrand factor and Factor VIII leading to hypercoagulable state and CVST. Further, elevated levels of plasminogen activator inhibitor type 1, thrombin activatable fibrinolysis inhibitor and alpha 2 antiplasmin due to hypercortisolaemia contributes to hypercoagulability in Cushing's syndrome patients.[9] In the third patient undergoing trans-sphenoidal excision of a craniopharyngioma, the author's attributed intracranial hypotension due to CSF leakage as the cause of CVST.[10] In the case reported by us, CVST probably occurred due to Factor V Leiden heterozygous mutation. The homozygous mutation is associated with 9–12 fold increased risk of thrombosis and the heterozygous mutation has a 4–5 fold increased risk.[11] The contribution of lumbar drainage of CSF and consequent intracranial hypotension, diabetes insipidus and use of vasopressin in our patient remains unclear.

Headache is present in over 80% of cases and 12%–31.9% of patients with CVST manifest epilepsy as the initial presenting symptom, 44.3% of which appeared in the early stages of the disease.[12],[13] Both CT and MR scans of the brain show venous infarcts due to CVST to be confluent infarcts or haemorrhage in atypical areas, crossing arterial territories, or infarcts with cortical sparing. These are seen in parasagittal (Sagittal sinus thrombosis) or temporoparietal regions (transverse/sigmoid sinuses) or deep structures. Additional CT venogram or MRV demonstrates non-visualisation of involved sinuses and cortical veins.


  Conclusion Top


Early diagnosis and treatment of CVST is essential for a favourable prognosis. Anticonvulsants, hydration and use of LMWH followed by warfarin treatment permits resolution and recovery from CVST. Occasionally, surgical decompression may be required for large life-threatening venous infarcts. Recently, endovascular thrombolysis and mechanical thrombectomy have been used as treatment adjuncts.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Bousser MG, Ferro JM. Cerebral venous thrombosis: An update. Lancet Neurol 2007;6:162-70.  Back to cited text no. 1
    
2.
Thorell SE, Parry-Jones AR, Punter M, Hurford R, Thachil J. Cerebral venous thrombosis – A primer for the haematologist. Blood Rev 2015;29:45-50.  Back to cited text no. 2
    
3.
Ribes MF. Des recherches faites sur la phlebite. Rev Med Fr Etrang 1825;3:5-41.  Back to cited text no. 3
    
4.
Duman T, Uluduz D, Midi I, Bektas H, Kablan Y, Goksel BK, et al. A multicenter study of 1144 patients with cerebral venous thrombosis: The VENOST study. J Stroke Cerebrovasc Dis 2017;26:1848-57.  Back to cited text no. 4
    
5.
Ferro JM, Canhão P, Stam J, Bousser MG, Barinagarrementeria F, ISCVT Investigators. Prognosis of cerebral vein and dural sinus thrombosis: Results of the International Study on Cerebral Vein and Dural Sinus Thrombosis (ISCVT). Stroke 2004;35:664-70.  Back to cited text no. 5
    
6.
Miao Z, Zhang Z, Chen J, Wang J, Zhang H, Lei T. Cerebral venous sinus thrombosis following second transsphenoidal surgery: Report of a rare complication and review of literature. World Neurosurg 2018;110:101-5.  Back to cited text no. 6
    
7.
Farb RI, Forghani R, Lee SK, Mikulis DJ, Agid R. The venous distension sign: A diagnostic sign of intracranial hypotension at MR imaging of the brain. AJNR Am J Neuroradiol 2007;28:1489-93.  Back to cited text no. 7
    
8.
Waheed W, Aljerdi S, Decker B, Cushman M, Hamill RW. Cerebral venous thrombosis associated with thyrotoxicosis, the use of desmopressin and elevated factor VIII/von Willebrand factor. Case Reports. 2016;2016:bcr2016216584.  Back to cited text no. 8
    
9.
Soni P, Koech H, Silva D, Das P, Sindwani R, Dobri G, et al. Cerebral venous sinus thrombosis after transsphenoidal resection: A rare complication of cushing disease-associated hypercoagulability. World Neurosurg 2020;134:86-9.  Back to cited text no. 9
    
10.
Chang T, Yang YL, Gao L, Li LH. Cerebral venous sinus thrombosis following transsphenoidal surgery for craniopharyngioma: A case report. World J Clin Cases 2020;8:1158-63.  Back to cited text no. 10
    
11.
Gohil R, Peck G, Sharma P. The genetics of venous thromboembolism. A meta-analysis involving approximately 120,000 cases and 180,000 controls. Thromb Haemost 2009;102:360-70.  Back to cited text no. 11
    
12.
Kumral E, Polat F, Uzunköprü C, Callı C, Kitiş Ö. The clinical spectrum of intracerebral hematoma, hemorrhagic infarct, non-hemorrhagic infarct, and non-lesional venous stroke in patients with cerebral sinus-venous thrombosis. Eur J Neurol 2012;19:537-43.  Back to cited text no. 12
    
13.
Masuhr F, Busch M, Amberger N, Ortwein H, Weih M, Neumann K, et al. Risk and predictors of early epileptic seizures in acute cerebral venous and sinus thrombosis. Eur J Neurol 2006;13:852-6.  Back to cited text no. 13
    


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