Trigeminocardiac reflex during supratentorial craniotomy for intraventricular meningioma: illustrative case

Abstract

BACKGROUND

Trigeminocardiac reflex (TCR) is a vagal reflex produced by trigeminal nerve stimulation that causes bradycardia, hypotension, and, occasionally, cardiac arrest. TCR usually occurs during surgical manipulation of the posterior fossa. In this report, the authors describe an extremely rare case of TCR that developed when a burr hole was drilled on the temporal bone during surgery for an intraventricular meningioma.

OBSERVATIONS

The patient was referred to the authors’ department after brain imaging revealed a mass lesion in the right trigone of the lateral ventricle. The surgery, which was performed using a transcortical approach via the right temporal lobe, was conducted under general anesthesia with the patient in the supine lateral position. During craniotomy, the patient experienced 17 seconds of cardiac arrest during the drilling of a burr hole in the temporal bone. TCR could not be diagnosed at that time because it occurred during the manipulation of supratentorial craniotomy with a burr hole. The operation was aborted immediately. The second surgery was performed with the patient in the lateral position, and total removal of the tumor was achieved without causing TCR.

LESSONS

In this report, the authors describe a rare TCR caused by supratentorial manipulation.

https://thejns.org/doi/10.3171/CASE257

Trigeminocardiac reflex (TCR), a response produced by stimulation of the trigeminal sensory branches, occurs primarily during surgery around the cerebellar pontine angle, basilar apex, orbit, and trigeminal ganglia. TCR has been recently reported to occur during endovascular treatment.¹ In TCR, central or peripheral trigeminal stimulation causes sudden bradycardia, cardiac arrest, hypotension, respiratory arrest, and increased intestinal peristalsis.² The exact cause of TCR is not yet fully understood, but it is thought to involve the release of neurotransmitters, including acetylcholine, and the integration of central nerve cells.²,³ In this report, we describe a rare case of cardiac arrest caused by TCR induced by supratentorial craniotomy for intraventricular meningioma and provide a literature review of related cases.

Illustrative Case

A 74-year-old right-handed man without any relevant medical history was referred to our department after he was diagnosed with a tumor on the right trigone of the lateral ventricle. MRI revealed a homonymous enhancing lesion in the right trigone with perifocal edema, indicating meningioma (Fig. 1A–C).

FIG. 1.

Pre- and postoperative MRI findings for intraventricular meningioma in the right trigone. Preoperative axial (A and B) and coronal (C) T2-weighted and enhanced MR images showing a mass in the right trigone. Postoperative axial enhanced MR image showing gross-total removal (D).

Because of the tumor size and perifocal edema, resection was recommended. We planned surgical removal using a transcortical approach via the right temporal lobe. The patient underwent a temporo-occipital craniotomy under general anesthesia in a supine lateral position with the neck rotated to the left side, vertex down, and the head fixed with three-point fixation. After skin incision and retraction of the temporal muscle, burr holes were drilled (Fig. 2A and B). When the first two points on the cranial side were drilled, no TCR occurred. Cardiac arrest occurred while drilling a burr hole on the cranial side near the asterion. The drill tip had not completely penetrated the bone when TCR occurred. Therefore, the occurrence of TCR was not related to tension to the dura. When the procedure was interrupted, the patient’s heartbeat resumed (Fig. 3). The patient had received general anesthesia with remifentanil and propofol, and the bispectral index at the time of the cardiac arrest was 46, with an adequate anesthetic depth. TCR was not identified at the time of the cardiac arrest because the craniotomy was performed on a supratentorial lesion and only a burr hole was drilled. Therefore, after consultation with the anesthesiologist and cardiologist, the operation was terminated. Subsequently, since no cardiac disease was identified after the initial surgery, a diagnosis of TCR was made. A second surgery was planned a month later. The second surgery was performed with the patient in the lateral position; the neck was placed in the neutral position, and temporal cardiac pacing was also implemented. During the second operation, no TCR occurred from craniotomy to tumor removal, and total removal of the tumor was achieved (Fig. 1D). The patient showed a good postoperative course and was discharged from the hospital. MRI 12 months after surgery revealed no recurrence.

FIG. 2.

Intraoperative images indicating skin incision (A) and the site of TCR (B; white arrow).

FIG. 3.

Intraoperative electrocardiograms obtained before TCR (A), during TCR (B and C), and at the restart of the heartbeat (D), showing 17 seconds of cardiac arrest (top row: electrocardiogram, middle row: oxygen saturation, bottom row: arterial pressure).

Informed Consent

The necessary informed consent was obtained in this study.

Discussion

TCR is defined as hypotension with a mean arterial blood pressure reduction of at least 20% and bradycardia with a heart rate reduction of at least 20% from baseline. In the field of neurosurgery, TCR has been reported to occur during surgeries around the cerebellar pontine angle, basilar apex, orbit, and trigeminal ganglia. The occurrence of TCR during surgical procedures in the anterior, middle, and posterior skull base is directly related to the intra- and extracranial course of the trigeminal nerve.⁴ TCR can also be elicited when stimulating the maxillary and mandibular divisions of the trigeminal nerve in craniomaxillofacial operations.

The mechanism underlying TCR involves stimulation conducted from the afferent pathway to the trigeminal spinal tract nucleus, which stimulates the cardiovascular system through the dorsal nucleus of the vagus nerve via interneurons to cause cardiovascular effects such as bradycardia and cardiac arrest.⁵ The risk factors for the development of TCR include hypercapnia, hypoxemia, light general anesthesia, pediatric surgery, the use of narcotics and potent narcotic agents such as sufentanil or alfentanil, and preoperative use of beta-blockers and calcium channel blockers.⁴ However, none of these risk factors were observed during the initial surgery in our case.

TCR is generally caused by manipulation during surgery for infratentorial craniotomy, but a review of the literature for TCR during supratentorial craniotomy revealed two cases. In the first case, during clipping of an unruptured anterior communicating aneurysm by a pterional approach, cardiac arrest occurred three times when the authors manipulated the dura attached to the sphenoid ridge. The TCR was considered to be caused by the concomitant course of the second and third branches of the trigeminal nerve near the middle dural artery.⁶ In another case, TCR was observed during surgery for a supratentorial parafalcine meningioma. In that case, TCR occurred during coagulation and detachment of the meningioma and dura. The TCR was considered to have been caused by stimulation of the dural branch of the trigeminal nerve on the tentorium.⁷ In both cases, TCR occurred due to direct dural manipulation after craniotomy. However, no previous report has described the occurrence of TCR during craniotomy and burr hole drilling, as in our case.

Observations

In our case, TCR occurred in the first surgery and did not occur in the second surgery. Although it is not possible to definitively determine the cause of the TCR, the following text discusses some possible causes. The TCR could have been caused by tensions to the periosteum and direct stimulation of the afferent sensory branches of the trigeminal nerve that had developed within the skull. The occurrence of TCR due to retraction of the temporalis muscle has been reported,⁸ but there have been no previous reports of direct retraction of the periosteum. Another possible factor could be that while the two surgeries showed no obvious differences in the depth of anesthesia or the choice of anesthetics, they did differ in terms of the patient’s position. Because the first surgery was performed in the supine position, neck extension was greater than we had expected. The second surgery was performed in the lateral position, precluding neck extension. Therefore, neck extension could have played a role in the development of TCR in our case.

Lessons

We report a rare case of TCR-induced cardiac arrest during supratentorial craniotomy, which was thought to have been induced by the combination of neck extension and stimulation to the sensory branch of the trigeminal nerve. TCR can occur during the creation of a burr hole using a perforator; thus, the possibility of TCR should be considered throughout craniotomy. Moreover, since TCR can occur in all head and neck surgeries wherein trigeminal sensory branches are distributed, it requires attention even in surgeries for supratentorial brain tumors.

Disclosures

The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this paper.

Author Contributions

Conception and design: Matsuda, Kawano. Acquisition of data: Matsuda, Kawano, Ko. Analysis and interpretation of data: Matsuda, Kawano. Drafting the article: Matsuda, Kawano. Critically revising the article: Matsuda, Nakagawa. Reviewed submitted version of manuscript: Matsuda, Abe. Approved the final version of the manuscript on behalf of all authors: Matsuda. Study supervision: Matsuda, Morisaki, Yamada, Nakagawa.

Supplemental Information

Previous Presentations

This case was presented orally at the 84th Kansai Regional Meeting of the Japanese Neurosurgical Society, Osaka, Japan, September 2, 2023.

Correspondence

Ryosuke Matsuda: Nara Medical University, Kashihara, Japan. rmatsuda@naramed-u.ac.jp.