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CASE REPORT Table of Contents  
Ahead of print publication
A case of puerperal cerebral venous thrombosis


 Department of Medicine, Srinivas Institute of Medical Sciences and Research Centre, Mangaluru, Karnataka, India

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Date of Submission15-Jan-2022
Date of Decision05-Feb-2022
Date of Acceptance06-Feb-2022
Date of Web Publication25-Jun-2022
 

  Abstract 


Cerebral venous thrombosis (CVT) is a relatively rare, potentially fatal neurological condition that can be frequently overlooked due to the vague nature of its clinical and radiological presentation, particularly when not associated with pregnancy and puerperium. It is a multifactorial disease, and the major forms of clinical presentation include isolated intracranial hypertension syndrome, focal neurological deficits, seizures, and altered sensorium. Magnetic resonance imaging with magnetic resonance venogram is considered the gold standard for diagnosis. Pregnancy-induced changes in coagulation result in a hypercoagulable state, which may naturally reduce the incidence of postpartum hemorrhage but may also increase the risk of CVT. Favorable outcomes have been reported in patients who receive early diagnosis and treatment.

Keywords: Cerebral venous thrombosis, pregnancy, procoagulant state, puerperium


How to cite this URL:
Konnur A, Rao G, Belle J. A case of puerperal cerebral venous thrombosis. APIK J Int Med [Epub ahead of print] [cited 2022 Oct 6]. Available from: https://www.ajim.in/preprintarticle.asp?id=348293





  Introduction Top


Cerebral venous sinus thrombosis (CVT) is a rare occurrence during pregnancy and the postpartum period.[1] In young-to-middle-aged adults, CVT is much more common in women than men, with a ratio of approximately 3:1.[2] CVT occurring in puerperium is about 10–12 times more frequent in India than in Western countries.[3] This is mostly due to predisposing factors such as anemia, increased coagulability of blood, slowing of the bloodstream, and dehydration.[3] Hypercoagulability plays an important role in the development of CVT during pregnancy and puerperium due to changes in the coagulation system, which are more marked during the third trimester. Dehydration as a result of blood loss during delivery, bad obstetric practices, and local trauma during delivery worsen the prothrombotic state. The hypercoagulability and venous stasis as a result of prolonged bed rest, instrument delivery, or cesarean section will lead to thrombosis.

Anticoagulation with heparin or low-molecular-weight heparin (LMWH) is the mainstay of treatment. Endovascular management is indicated for those cases with severe symptoms or worsening of symptoms despite anticoagulation therapy. Early diagnosis and treatment of CVT, which is potentially fatal, are quite important.

Here, we report the case of a young woman who developed CVT 10 days after cesarean section.


  Case Report Top


A 31-year-old woman who had undergone a cesarean section for delivery 10 days before presentation came with a history of low-grade fever and headache of 1-day duration. She did not have any preexisting illnesses such as diabetes, hypertension, or tuberculosis. General physical examination revealed a conscious, cooperative person with Glasgow Coma Scale (GCS) 13/15, responding to oral commands but not verbalizing. Her blood pressure was 120/80 mmHg and temperature was recorded 100°F. Systemic examination revealed right hemiparesis with a power of 2/5 in the right upper limb and 3/5 in the right lower limb. Within few hours of presentation, the patient's level of consciousness deteriorated to GCS 7/15, and hence, a plain computed tomography (CT) head was asked for evaluation.

The CT brain revealed an intraparenchymal hemorrhage measuring 40 mm × 38 mm in size in the left frontal lobe with marked surrounding edema [Figure 1]. In view of the patient having recently undergone a delivery, a provisional diagnosis of left frontal lobe venous infarct with hemorrhage was considered and hence a magnetic resonance imaging (MRI) brain with MR venography was requested. MRI brain revealed a large hemorrhagic venous infarct with surrounding edema in the left frontal region with midline shift [Figure 2]. There was also thrombosis of the superior sagittal sinus, left vein of Trolard, and left transverse sinus [Figure 3].
Figure 1: CT brain showing intraparenchymal hemorrhage (shown in arrow)

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Figure 2: MRI brain showing a large haemorrhagic venous infarct (shown in arrow)

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Figure 3: MR Angiogram showing thrombosis of the superior sagittal sinus (shown in arrow)

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A neurosurgery consultation was taken, and they advised anti-edema measures and anticoagulant therapy. She was started on therapeutic dose of low-molecular-weight heparin (enoxaparin). Her sensorium gradually improved and she regained consciousness after 3 days of therapy. However, the patient was not verbalizing. Her anticoagulant measures were continued and physiotherapy was also started for limb weakness.

After 2 weeks of admission, the patient had improved remarkably and was fully conscious, oriented, responding to oral commands, and verbalizing a few words. Her right hemiparesis had improved and she was able to walk with minimal support, with the right upper limb having 4/5 power. She was switched over from parental to oral anticoagulant therapy with warfarin, keeping a target INR of 2–2.5.


  Discussion Top


CVT is a rare entity in pregnancy and the postpartum period, with an incidence of 1:10,000–1:25,000.[4] Thrombosis of the dural sinus and/or CVT is a rare but potentially devastating type of stroke that tends to occur in young adults, especially women. In a study conducted by Umesh G et al.[5] on CVT in the postpartum period, out of 50 patients included in the study, the maximum incidence of CVT was seen in 21–31-year age group. This age group comprised 74% of the cases, with the mean age being 25.52 years. It was also found that CVT was more common in primigravida (65.6%) and in the postpartum period (90%). Our patient was a primigravida of 31 years.

The precipitating factors are pregnancy, puerperium, connective tissue disease, malignancy, contraceptive use, and infections such as sinusitis, otitis, and mastoiditis. Head injury causes sinus thrombosis in a person with a genetically increased risk. When a young, pregnant, or postpartum woman presents with new-onset, stroke-like symptoms with headache and seizure, it is significant to consider the diagnosis of CVT. During the last trimester of pregnancy and after delivery, the risk of sinus thrombosis is increased.[6] It has been reported that 39%–41% of CVT patients present with intracerebral hemorrhage, hemorrhagic venous infarcts, or isolated SAH.[7] Characteristic, but rare, is the occurrence of unilateral hemispheric symptoms such as hemiparesis or aphasia, followed within days by symptoms from the other hemisphere; these are caused by the development of cortical lesions on both sides of the superior sagittal sinus. The diagnosis is confirmed with MRI and magnetic resonance venogram (MRV). Our patient had right-sided hemiparesis and global aphasia. CT head showed intraparenchymal hemorrhage. The hemorrhage appeared to be venous origin and hence an MRI brain with MR angiogram was requested to rule out cortical venous thrombosis.

The mainstay of acute management is anticoagulation. About 40% of all patients with sinus thrombosis have a hemorrhagic infarct even before anticoagulant treatment is started. Anticoagulation therapy for CVT averts aggravation of the thrombus, and allows for improvement of the occlusion lesion. Heparin has been reported as a safe and effective treatment.[8] Heparin and warfarin have been used for more than 50 years; monitoring prothrombin time is difficult for warfarin use. In an open-label trial by Nagaraja et al. on 150 patients with CVT, 73 received low-dose heparin (2500 thrice daily) and 77 did not receive heparin. There was a reduction of death and increase in complete recovery in the group which received heparin compared to that which did not receive heparin.[9]

Newer oral anticoagulants (e.g., dabigatran, rivaroxaban, and apixaban) might offer an alternative to traditional therapies for CVT.[10]

In our patient, we started LMWH and were continued on warfarin maintenance therapy in view of insufficient data available to determine the effects of Newer Oral Anti Coagulant (NOACs) on a breastfed child or on milk production. Several case reports consistently indicate that maternal doses of rivaroxaban of 15–30 mg daily produce low levels in milk that are considerably below doses required for anticoagulation in infants.[11] Anti-seizure is also indicated for patients presenting with early seizures. Our patient was started on levetiracetam. In a study done by Anders et al., it was shown that elevated complement levels are associated with postnatal pregnancy-related venous thrombosis.[12] In our patient, complement levels C3 and C4 were not in order indicating a high risk of postpartum venous thrombosis.

CVT occurrence during pregnancy or puerperium is not a contraindication for future pregnancies. CVT occurring during these periods should be treated with LMWH and continued through 6 weeks of postpartum.[13] The outcomes after CVT in pregnancy are generally favorable, and the risk of recurrence is low. Important advances have been made in our understanding of the pathophysiology of sinus thrombosis. Sinus thrombosis remains a diagnostic challenge and a potentially disabling or lethal disease, but improved diagnosis and treatment now result in an excellent outcome for most patients.


  Conclusion Top


Cerebral venous sinus thrombosis is a challenging condition. Diagnosis is frequently overlooked or deferred due to its subacute onset and the wide spectrum of clinical symptoms. CVT is an uncommon complication with a favorable outcome after delivery. Timely diagnosis and treatment with anticoagulants seem to be important in preventing deterioration of neurological function.

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.
Borum SE, Naul LG, McLeskey CH. Postpartum dural venous sinus thrombosis after postdural puncture headache and epidural blood patch. Anesthesiology 1997;86:487-90.  Back to cited text no. 1
    
2.
Bushnell C, Saposnik G. Evaluation and management of cerebral venous thrombosis. Continuum (Minneap Minn) 2014;20:335-51.  Back to cited text no. 2
    
3.
Srinivasan K. Cerebral venous and arterial thrombosis in pregnancy and puerperium. A study of 135 patients. Angiology 1983;34:731-46.  Back to cited text no. 3
    
4.
McCaulley JA, Pates JA. Postpartum cerebral venous thrombosis. Obstet Gynecol 2011;118:423-5.  Back to cited text no. 4
    
5.
Rajoor UG, Seema BN. Clinical profile of postpartum cerebral venous thrombosis. Int J Reprod Contracept Obstet Gynecol 2017;6:1192-5.  Back to cited text no. 5
    
6.
Cantú C, Barinagarrementeria F. Cerebral venous thrombosis associated with pregnancy and puerperium. Review of 67 cases. Stroke 1993;24:1880-4.  Back to cited text no. 6
    
7.
Ghandehari K, Riasi HR, Noureddine A, Masoudinezhad S, Yazdani S, Mirzae MM, et al. Safety assessment of anticoagulation therapy in patients with hemorrhagic cerebral venous thrombosis. Iran J Neurol 2013;12:87-91.  Back to cited text no. 7
    
8.
Coutinho JM, Ferro JM, Canhão P, Barinagarrementeria F, Bousser MG, Stam J, et al. Unfractionated or low-molecular weight heparin for the treatment of cerebral venous thrombosis. Stroke 2010;41:2575-80.  Back to cited text no. 8
    
9.
Nagaraja D, Haridas T, Taly AB, Veerendrakumar M, SubbuKrishna DK. Puerperal cerebral venous thrombosis: Therapeutic benefit of low dose heparin. Neurol India 1999;47:43-6.  Back to cited text no. 9
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10.
Gross PL, Weitz JI. New anticoagulants for treatment of venous thromboembolism. Arterioscler Thromb Vasc Biol 2008;28:380-6.  Back to cited text no. 10
    
11.
Anderson P. Drugs and Lactation Database (LactMed) [Internet]. Bethesda (MD): National Library of Medicine (US); 2006-. Rivaroxaban. [Updated 2021 May 17]. Available from: https://www.ncbi.nlm.nih.gov/books/NBK500742/.  Back to cited text no. 11
    
12.
Dahm AE, Jacobsen EM, Wik HS, Jacobsen AF, Mollnes TE, Kanse SM, et al. Elevated complement C3 and C4 levels are associated with postnatal pregnancy-related venous thrombosis. Thromb Haemost 2019;119:1481-8.  Back to cited text no. 12
    
13.
Idiculla PS, Gurala D, Palanisamy M, Vijayakumar R, Dhandapani S, Nagarajan E. Cerebral venous thrombosis: A comprehensive review. Eur Neurol 2020;83:369-79.  Back to cited text no. 13
    

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Correspondence Address:
Gurukanth Rao,
Department of Medicine, Srinivas Institute of Medical Sciences and Research Centre, Mangaluru, Karnataka
India
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ajim.ajim_14_22



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