The Journal of International Medical Research 2005; 33: 711 – 714 Cerebral Venous Thrombosis Initially Considered as a Complication of Spinal–Epidural Anaesthesia A KARCI1, F BOYACI1, E YAKA2, R ÇAKMUR2, S MEN3 AND Z ELAR1 1 Department of Anaesthesiology and Reanimation, 2Department of Neurology, and 3 Department of Radiology, Dokuz Eylül University Medical School, Izmir, Turkey Cerebral venous thrombosis is an unusual condition characterized by headache, nausea, vomiting, focal deficits and epileptic seizures. In this case report we describe a patient who presented with headache and focal motor deficits after an uneventful Caesarean section, performed using combined spinal–epidural anaesthesia. Magnetic resonance imaging was performed because of the pronounced neurological symptoms, and a diagnosis of cerebral venous thrombosis was made. The patient was treated with anticoagulant agents and made a complete recovery. This case emphasizes the importance of considering cerebral venous thrombosis in the differential diagnosis of headache in the post-partum period prior to instituting conventional therapy. KEY WORDS: CEREBRAL VENOUS THROMBOSIS; POST-DURAL PUNCTURE HEADACHE; SPINAL–EPIDURAL ANAESTHESIA Introduction Cerebral venous thrombosis (CVT) is an unusual condition characterized by headache, nausea, vomiting, focal deficits and epileptic seizures. It is associated with the use of oral contraceptives, pregnancy and the puerperium, obesity, Behçet’s syndrome and other diseases.1,2 CVT has been recognized with increasing frequency in recent years as a result of heightened clinical awareness and advances in diagnostic technology, in particular the advent of magnetic resonance imaging (MRI).3 Despite increasing clinical awareness, the broad spectrum of CVT continues to pose a diagnostic challenge to physicians. We describe here a patient who developed signs of post-dural puncture headache and focal motor deficits after an uneventful Caesarean section performed using combined spinal–epidural anaesthesia. Case report A 34-year-old woman, gravida 2, para 1 and weight 75 kg, was admitted at 38 weeks’ gestation with painful contractions. She had undergone a Caesarean section in her previous pregnancy due to cephalopelvic disproportion. She had had an uncomplicated pre-natal course. An emergency Caesarean section using combined spinal– epidural anaesthesia was recommended. An 18-gauge Tuohy needle was inserted, using the loss of resistance to saline Downloaded from imr.sagepub.com at WAYNE STATE UNIVERSITY on March 26, 2015 711 A Karci, F Boyaci, E Yaka et al. CVT following spinal–epidural anaesthesia technique to identify the epidural space. A 27-gauge spinal needle was then passed through the epidural needle, beyond its tip and through the dura. Following intrathecal injection of fentanyl 25 µg with 1 ml bupivacaine 0.25%, the spinal needle was removed. An epidural catheter was placed in the epidural space at the first attempt to be used for post-operative analgesia. A solution containing fentanyl 2 µg/ml and bupivacaine 0.125% was prepared for patient-controlled epidural analgesia. The Caesarean section was performed without incident and the epidural catheter was removed 24 h following the operation. On the third day following delivery, the patient experienced headache involving the frontal region and the neck and shoulder area, but she was not referred to the anaesthesiologists. Six days following delivery she presented to the hospital with loss of strength in the right arm and right leg. On neurological examination, the patient’s muscle strength was 3/5 in the right upper limb and 0 – 1/5 in the right distal lower limb. She had hypoaesthesia on the right side of her body and Babinski’s sign was positive. An MRI scan was ordered immediately. The scan showed venous infarctions in the superior frontal, pre-central and post-central gyri on the left, and loss of the signal void in the anterior half of the superior sagittal sinus (Fig. 1). A diagnosis of CVT was established. Magnetic resonance venography confirmed absence of flow in the anterior part of the sinus (Fig. 2). These findings were consistent with a superior sagittal sinus thrombosis. The patient was admitted to the Neurology Department, and anti-coagulation with intravenous heparin and oral warfarin was started. Her headache, which was not severe at the time of admission, resolved without any specific medication. Whole blood count, FIGURE 1: Axial, fluid-attenuated, inversion recovery magnetic resonance image through the vertex level in a patient with cerebral venous thrombosis showing an abnormally high signal in the swollen superior frontal, pre-central and post-central gyri on the left (arrows) FIGURE 2: Magnetic resonance venography image in a patient with cerebral venous thrombosis confirming the absence of flow in the anterior part of the sinus (arrows) electrolytes, alkaline phosphatase, γ-glutamyl transferase, plasminogen, anti-cardiolipin G, lupus anti-coagulant and activated protein C levels were within normal limits. There was a Downloaded from imr.sagepub.com at WAYNE STATE UNIVERSITY on March 26, 2015 712 A Karci, F Boyaci, E Yaka et al. CVT following spinal–epidural anaesthesia small decrease in the protein S level to 40% (normal range, 55 – 160%) and a small increase in the anti-thrombin III level to 135% (normal range, 75 – 125%). On the second day of hospitalization, the patient developed generalized tonic–clonic convulsions. Computed tomography revealed no pathological findings additional to the venous infarcts present on MRI. An electroencephalogram was performed and was unremarkable. Anti-oedema and antiepileptic agents were added to her current medications. Eighteen days later, a control MRI was performed and the patient was discharged on oral warfarin and anti-epileptic therapy with no neurological sequelae. Discussion Headache is a known complication of dural puncture, and the concomitant presence of intra-cranial pathology in patients with symptoms of post-dural puncture headache (PDPH) is rare. Since symptoms are not always specific for either PDPH or CVT, and because both conditions may occur in the early post-partum period, distinguishing between them can be difficult. This raises the possibility of both misdiagnosis and mistreatment if headache after dural puncture is actually caused by CVT.4 The classic description of PDPH is a frontal or occipital headache that is aggravated by assuming the upright position and essentially disappears when returning to the supine position. The onset and duration of PDPH can be extremely variable, but most occur within 48 h of dural puncture and are self-limiting, lasting only days.5 Aidi et al.6 have proposed that when a typical PDPH loses its postural component, investigations should be performed to rule out CVT, particularly in the presence of risk factors. Lateral or superior sagittal venous sinus thromboses are rare in the absence of infection or trauma, but pregnancy and especially the puerperium are predisposing factors. In postpartum superior sagittal sinus thrombosis, the thrombosis is thought to be secondary to damage to the venous sinuses from fluctuations in intra-cranial pressure during labour and delivery, and the hypercoagulable state associated with pregnancy.4,7,8 It is not possible to ascertain the exact mechanisms responsible for the development of CVT in the patient reported here. In our case report, an emergency Caesarean section was required based on a history of Caesarean section and the onset of painful contractions. The patient had no contraindications for regional anaesthesia, and combined spinal–epidural anaesthesia was preferred to provide early-onset anaesthesia. The epidural catheter was placed at the first attempt and no untoward events were encountered during the perioperative period. Magnetic resonance imaging is the technique of choice for the immediate evaluation of CVT, but the decision concerning whether or not to perform MRI remains problematic in patients presenting with headache as their major symptom.7 An MRI scan is inadvisable in every patient with a typical, recent-onset headache after dural puncture, but should be considered in patients in whom intracranial pathology is suspected. The main reason for performing an immediate MRI in our patient was the presence of obvious neurological signs accompanying a severe headache that was not postural. The presence of neurological deficits pointed to the possibility of a serious complication of the puerperium or of spinal–epidural anaesthesia. Infectious complications were excluded from the differential diagnosis because of the absence of fever or meningeal features, including rigidity. In the light of this, the patient did not receive treatment for PDPH before the results of the MRI scan were available. Downloaded from imr.sagepub.com at WAYNE STATE UNIVERSITY on March 26, 2015 713 A Karci, F Boyaci, E Yaka et al. CVT following spinal–epidural anaesthesia Cerebral venous thrombosis has been reported in patients with anti-coagulant abnormalities such as deficiencies of protein C or S, anti-thrombin III or plasminogen, as well as lupus anti-coagulant or antiphospholipid antibodies.9 While protein S and anti-thrombin III values were not within the normal range in our patient, the deviation of both parameters was not considered to be sufficient to cause hypercoagulability and venous thrombosis. There is currently no consensus on the specific therapy for CVT. Anti-coagulation with intravenous heparin is generally considered to be first-line treatment and is continued until an appropriate partial thromboplastin time is obtained. Oral anti-coagulants are continued for 3 – 6 months or longer.4 The case presented here shows the importance of the differential diagnosis in patients presenting with symptoms resembling PDPH after delivery. A thorough history is important in order to exclude other possible diagnoses before assuming that the headache is of spinal origin. CVT should be considered if the characteristics of the headache are changing, or if neurological signs are present. The long-term prognosis for patients with CVT from all causes is good, with better outcomes in cases occurring during pregnancy or the puerperium. Early diagnosis and treatment will promote optimal medical care and are associated with excellent functional recovery. Conflicts of interest No conflicts of interest were declared in relation to this article. • Received for publication 3 May 2005 • Accepted subject to revision 17 May 2005 • Revised accepted 22 July 2005 Copyright © 2005 Cambridge Medical Publications References 1 Vandenberghe N, Debouverie M, Anxionnat R, Clavelou P, Bouly S, Weber M: Cerebral venous thrombosis in four patients with multiple sclerosis. Eur J Neurol 2003; 10: 63 – 66. 2 Akdal G, Donmez B, Cakmakci H, Yener GG: A case with cerebral thrombosis receiving tamoxifen treatment. Eur J Neurol 2001; 8: 723 – 724. 3 Fink JN, McAuley DL: Cerebral venous sinus thrombosis: a diagnostic challenge. Intern Med J 2001; 31: 384 – 388. 4 Borum SE, Naul LG, McLeskey CH: Postpartum dural venous sinus thrombosis after postdural puncture headache and epidural blood patch. Anesthesiology 1997; 86: 487 – 490. 5 Davignon KR, Dennehy KC: Update on postdural puncture headache. Int Anesthesiol Clin 2002; 40: 89 – 102. 6 Aidi S, Chaunu MP, Biousse V, Bousser MG: Changing pattern of headache pointing to cerebral venous thrombosis after lumbar puncture and intravenous high-dose corticosteroids. Headache 1999; 39: 559 – 564. 7 Benzon HT, Iqbal M, Tallman MS, Boehlke L, Russell EJ: Superior sagittal sinus thrombosis in a patient with postdural puncture headache. Reg Anesth Pain Med 2003; 28: 64 – 67. 8 Chisholm ME, Campbell DC: Postpartum postural headache due to superior sagittal sinus thrombosis mistaken for spontaneous intracranial hypotension. Can J Anaesth 2001; 48: 302 – 304. 9 Lee JH, Kim SW, Sung Kim J: Sagittal sinus thrombosis associated with transient free protein S deficiency after L-asparaginase treatment: case report and review of the literature. Clin Neurol Neurosurg 2000; 102: 33 – 36. Address for correspondence Dr A Karci Huzur Mah. Açelya Sok. No. 1/1 D:4, PK: 35320 Narlidere, Izmir, Turkey. E-mail: ayse.karci@deu.edu.tr or ayse.karci@isbank.net.tr Downloaded from imr.sagepub.com at WAYNE STATE UNIVERSITY on March 26, 2015 714