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Obstetric Anesthesia for a Pregnant Woman With Brainstem Cavernous Malformations: A Case Report

Hayashi, Misuzu MD; Kakinohana, Manabu MD, PhD

doi: 10.1213/XAA.0000000000000525
Case Reports: Case Report
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There are no well-defined guidelines for obstetric and anesthetic management of pregnant patients with cerebral cavernous malformations (CMs). We need to pay careful attention to the attendant risks of both general and neuraxial anesthesia in this population. Here, we describe the anesthetic management of a pregnant woman with brainstem CMs who underwent cesarean delivery. We selected a general anesthetic technique because of concerns of undesirable effects of neuraxial anesthesia in the presence of cerebral CMs.

From the Department of Anesthesiology, Ryukyu University Hospital, University of the Ryukyus, Nishihara, Okinawa, Japan.

Accepted for publication January 23, 2017.

Funding: None.

The authors declare no conflicts of interest.

Address correspondence to Misuzu Hayashi, MD, Department of Anesthesiology, Ryukyu University Hospital, University of the Ryukyus, 207, Uehara, Nishihara, Okinawa, Japan, 9030125. Address e-mail to koba_shu1982@yahoo.co.jp.

Cavernous malformation (CM) is an important cause of intracranial hemorrhage. CM is a vascular malformation consisting of thin-walled vascular sinusoids lined by a thin endothelium lacking smooth muscle and elastin.1 CMs occur mostly in the brain and sometimes exist in the spinal cord, retina, and skin. With availability of magnetic resonance imaging (MRI) in the early 1990s, CMs were discovered incidentally in increasingly significant numbers, and the overall incidence of cerebral CMs is reported to be approximately 0.4%.2 According to a meta-analysis study on the natural history of CMs including a total of 837 patients, the male-to-female ratio is 1:1 and the mean age at presentation is 30.6 years.3 Cerebral CMs are not uncommon in pregnant women. The peripartum risk of cerebral hemorrhage and the optimal peripartum management, however, remain to be determined. In addition, reports about the anesthetic management of patients with such pathology are limited.

In the following report, we discuss the anesthetic management of a pregnant woman with brainstem CMs who underwent cesarean delivery under general anesthesia. In addition, we review the literature addressing the related anesthetic and obstetric management principles of pregnant women with cerebral CMs.

The patient provided written consent for publication of this case report.

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CASE REPORT

A 22-year-old woman presented for elective cesarean delivery at 38 weeks’ gestation. She had experienced sudden-onset right-sided facial numbness, facial palsy, and hemiplegia at the age of 18 years. MRI of the brain revealed a hemorrhage from brainstem CMs located in the ventral mesencephalon and pons (Figure), and multiple CMs in the cerebral hemispheres and cerebellum. She was managed conservatively and recovered almost completely.

Figure.

Figure.

After her pregnancy was confirmed, neurosurgeons considered her at high risk of recurrent hemorrhage from the CMs during the peripartum period because the lesion was located in the brainstem and previously had caused intracerebral hemorrhage. It was agreed between neurosurgeons and obstetricians that cesarean delivery would be the child delivery method of choice. We abstained from neuraxial anesthesia and opted for general anesthesia because the concurrent presence of spinal cord CMs was a possibility that had not been ruled out. The primary anesthetic management goal was prevention of a hypertensive episode during induction of and emergence from anesthesia and avoidance of straining, coughing, and breath-holding, which might have caused increased intracranial pressure.

Preanesthetic blood pressure was 110/75 mm Hg, and heart rate was 84 bpm. After insertion of a catheter into the radial artery for continuous blood pressure measurement, anesthesia was induced with sodium thiopental 250 mg and remifentanil 0.5 μg/kg/min. After endotracheal intubation facilitated by rocuronium 60 mg, anesthesia was maintained with end-tidal sevoflurane 1.5% to 3.0%.

Immediately after delivery of the child, sevoflurane was discontinued and an intravenous infusion of propofol at 300 to 400 mg/h was started to maintain the depth of anesthesia at a bispectral index value of below 60. Systolic blood pressure increased briefly to 135 mm Hg during endotracheal intubation and at the start of surgery, but it returned to baseline values almost immediately. We inserted a nasogastric tube and confirmed that the patient’s stomach was empty. The intraoperative course was uneventful.

After completion of surgery and before discontinuation of anesthesia, we performed an ultrasound-guided transversus abdominal-plane block with catheter placement for postoperative analgesia. Although the patient was still under deep anesthesia, the endotracheal tube was removed, and the supraglottic airway device i-gel (Intersurgical Ltd, Wokingham, Berkshire, United Kingdom) was inserted. The neuromuscular blockade was reversed by intravenous sugammadex 130 mg. The propofol infusion was then stopped. The patient emerged from anesthesia and was able to follow simple commands without coughing about 10 minutes after the propofol infusion was stopped. After sufficient spontaneous breathing had been established, the i-gel was removed gently.

Subsequent physical examination revealed no detectable neurologic symptoms. The postoperative pain regimen included intravenous patient-controlled analgesia with fentanyl and intermittent boluses of local anesthetic (0.25% levobupivacaine) administered via the indwelling transversus abdominal-plane catheter. Diclofenac sodium was added because the patient complained of pain at rest (pain score of 2–4 on a numerical rating scale 0–10). The postoperative course was uneventful. In accordance with local practice, the patient was discharged home at postoperative day 16 without any neurological deficit.

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DISCUSSION

Numerous publications have addressed the natural history of cerebral CMs. Although various risk factors for hemorrhage are documented, such as previous hemorrhage,4 female sex,4,5 pregnancy,2 and deep location of the lesion,2,5 these remain controversial. Brainstem CMs (such as in this case) are identified as a risk factor for hemorrhage in many publications, and these lesions can cause life-threatening hemorrhages.3,5,6 Although pregnancy has been considered to increase the risk of hemorrhage from cerebral CMs,2 this has been disputed recently.7 Those authors do not consider a history of cerebral CMs a contraindication to pregnancy or vaginal delivery.

Based on the past publications and our own experience with this case, the key management issues of these patients concern cardiorespiratory management and choice of anesthetic technique. There is little information about hemodynamic characteristics of CMs. Little et al8 intraoperatively inserted a 25-gauge needle into one of the large chambers of a cerebral CM and measured pressure in the CM. They reported the following findings: (1) mean pressure within the lesion in the supine position was 38.5 ± 0.5 mm Hg; (2) jugular compression resulted in a 9 mm Hg rise in CM pressure; and (3) changes in mean systemic blood pressure and Paco2 resulted in a slight change in CM pressure. Of interest, Lekovic et al9 reported intraoperative rupture of CMs in a patient with thrombosis in the venous system and suggested that alterations in venous hemodynamics caused by thrombus formation and resultant obstruction of venous drainage might precipitate rupture of fragile CMs. Thus, factors interrupting venous return—such as increased intrathoracic pressure or intra-abdominal pressure—seem likely to increase CM pressure, potentially leading to rupture of CMs. We, therefore, have to avoid risk factors during the perioperative period, such as coughing, breath-holding, straining, high positive end-expiratory pressure, and high intrathoracic pressure.

A previous report on spinal anesthesia for cesarean delivery in patients with cerebral CM recommended MRI examination of the brain and spinal cord within a year of delivery.10 CMs in the central nervous system can present with multiple lesions in the supraspinal area and sometimes in the spinal cord.11 Spinal cord CMs can be extradural, intradural extramedullary, or intramedullary. Compared with cerebral CMs, it is more difficult to estimate the prevalence of spinal CMs. Because of the increased availability of MRI, the prevalence of spinal CMs reported in the literature has increased exponentially.12 Spinal CMs account for approximately 5% of intramedullary lesions in adults. Coexistence of CMs in the brain and spinal cord typically occurs in the familial form of CM but is also observed sporadically.13 In a series of 100 patients with brainstem CMs, 24 of them had multiple lesions including one in the spinal cord.5 It is, therefore, considered necessary to rule out a lesion at the intended spinal/epidural puncture site.

With preoperative MRI screening for CMs, this case might have been managed well under neuraxial anesthesia in preference to general anesthesia. General anesthesia entails the risk of unexpected difficult airway and pulmonary aspiration, and it may increase the risk of CM rupture by increases in arterial and venous pressures during induction of and emergence from anesthesia. In contrast, persistent cerebrospinal fluid (CSF) leakage after spinal anesthesia or accidental dural puncture may produce fatal brain herniation in patients with poor intracranial compliance,14 and a compensatory increase in cerebral blood volume is theoretically caused by CSF leakage-induced intracranial hypotension.15 This may potentially trigger increased transmural pressure across the fragile vessel wall in CMs and precipitate rupture. In addition, even if an epidural catheter is placed successfully without any event, epidural fluid injection may increase intracranial pressure, probably because of compression of the lumbar dural sac with displacement of CSF upward to the intracranial space. Therefore, we could not guarantee that neuraxial anesthesia would not affect the brainstem CMs, even if there was no lesion at the puncture site.

Because we were confident that adverse cardiovascular responses during the induction of anesthesia could be suppressed by the use of high-dose opioid anesthesia with remifentanil, and that the supraglottic airway device would inhibit coughing and straining during the emergence from anesthesia, we selected general anesthesia in this case. As a result, the cardiovascular response was sufficiently stable, and neither coughing nor straining was observed during the emergence from anesthesia.

In conclusion, we have described successful general anesthesia for cesarean delivery in a patient with brainstem CMs. However, neither the present report nor any previous literature describing anesthetic management for these cases could conclude which anesthetic management technique is the best. Additional case reports presenting anesthetic management techniques are required.

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DISCLOSURES

Name: Misuzu Hayashi, MD.

Contribution: This author helped manage the case and write the manuscript.

Name: Manabu Kakinohana, MD, PhD.

Contribution: This author helped revise the manuscript.

This manuscript was handled by: Hans-Joachim Priebe, MD, FRCA, FCAI.

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