Delayed Cerebral Infarction Following Trans-Sylvian Surgery for Craniopharyngioma Presenting as Status Epilepticus
Correspondence Address: Source of Support: None, Conflict of Interest: None DOI: 10.4103/0028-3886.304097
Source of Support: None, Conflict of Interest: None
The postoperative period after craniopharyngioma surgery has a high likelihood of complications and its management can be challenging. We present the case of a 10-year-old boy who was operated for craniopharyngioma. In the postoperative period he developed lacunar infarct, endocrine disturbances, delayed vasospasm leading to cerebral infarction and status epilepticus. The likely cause of status epilepticus was likely delayed cerebral infarction compounded with dyselectrolytemia. We discuss the possible etiology of delayed cerebral infarction.
Keywords: Craniopharyngioma, delayed vasospasm, endocrine complications, postoperative complications, status epilepticus
Delayed cerebral ischemia and infarction following surgery in the suprasellar cisterns are relatively rare. The cause is mostly vasospasm of the arteries of circle of Willis. The diagnosis is often missed unless the possibility is kept in mind. It leads to high morbidity and mortality. We present a case of delayed cerebral infarction following trans- sylvian surgery for craniopharyngioma manifesting as status epilepticus.
A 10-year-old boy presented with history of gradual diminution of vision of 2 months duration. Examination revealed complete loss of vision in right eye with absent PL and PR and dilated pupil. Left eye had normal vision. His hormone levels were within normal limits. MRI revealed a cystic supra sellar tumor with compression of optic chiasma [Figure 1]a & [Figure 1]b. He underwent left pterional craniotomy and trans-sylvian approach for excision of tumor. The tumor was cystic with thick calcified wall. Dissection was carried out along the arachnoid plane, but the latter was lost towards the end. The tumor did not have much adhesion. It was ruptured to ease dissection – Machine oil type of fluid oozed out. Two bleeding perforators were coagulated. The whole tumor was removed. There was no obvious injury to major intracranial vessels.
The patient was conscious in the immediate postoperative period. On the first postoperative day the patient developed aphasia and right hemiplegia. Urgent CT scan showed a small infarct in the left thalamus. [Figure 1]c. There was no blood in the operated area or in the subarachnoid space. The speech and right hemiplegia improved over a few days with conservative management.
By postoperative day 4, patient developed polyuria and dyselectrolytemia. Endocrinology consultation was taken and diabetes insipidus was confirmed. The patient was started on injection vasopressin. He had wide fluctuation in serum sodium and fluid levels (serum sodium ranging from 125 to 165 meq/L, daily urine output ranging from 900 mL to 5800 mL) over the next two months) in spite of strict monitoring. Repeat NCCT head on postop day 10 revealed subacute infarct in left thalamus [Figure 1]d.
On postoperative day 12, the patient developed left focal seizures with secondary generalization which evolved into status epilepticus. Patient was managed with multiple antiepileptic drugs (fosphenytoin 100 mg twice daily, valproate 300 mg thrice daily, levetiracetam 400 mg twice daily, midazolam infusion, thiopentone infusion) with mechanical ventilation. Active convulsions had subsided, but EEG showed continuous epileptiform discharges suggestive of non-convulsive status epilepticus. Due to hemodynamic instability and recurrent seizures patient could not be shifted for brain imaging for the first 48 hours after seizure. On postoperative day 14, NCCT head could be done which revealed a large right middle cerebral artery (MCA) infarct with midline shift [Figure 1]e. CT angiogram could not be done. Urgent decompressive hemicraniectomy was performed [Figure 1]f. Over the next few days the anesthetic agents were gradually tapered with EEG monitoring. However, the patient remained comatose. He could not be weaned off the ventilator and succumbed four months after the surgery.
Postoperative management of craniopharyngioma is often turbulent and fraught with many complications like fluid and electrolyte imbalance, seizures, infection, vascular complications etc., According to a previous review by Suero Molina et al. 34 cases of documented cerebral vasospasm following trans sphenoidal surgery have been reported from 1980 till November 2018. After that two more cases of delayed cerebral vasospasm following pituitary surgery have been reported till November 2019.[2.3] Delayed cerebral vasospasm presenting as hemiparesis, altered sensorium, vision loss has been previously described.,,,, One previous case report by Kasliwal et al. describes the onset of delayed cerebral infarction causing hemiparesis with focal seizures. To the best of our knowledge, delayed cerebral infarction following vasospasm manifesting as status epilepticus has not been reported.
Our patient suffered from two ischemic events. The initial thalamic infarct could be due to perforator injury during surgery. The second right MCA infarct was possibly due to spasm of right MCA artery. The second stroke was clinically missed during the window period due to multiple confounders. The patient had more than one causes for decreased sensorium viz. dyselectrolytemia, status epilepticus, antiepileptic drugs, anesthetic agents and the stroke itself.
A previous review of literature by Alotaibi et al. found 40 cases of cerebral vasospam following tumor resection. They concluded that pituitary tumours and surgery involving sellar area were most commonly associated with cerebral vasospasm. The cause of vasospasm has been ascribed to sub-arachnoid hemorrhage in the basal cisterns mechanical trauma to vessels during surgery, spillage of craniopharyngioma fluid and hypothalamic involvement. In our patient the last two factors were present. Also, there was fluid and electrolyte imbalance which must have aggravated the ischemia. The second stroke along with dyselectrolytemia was the likely cause of seizures which evolved into status epilepticus.
The unfortunate outcome could have been prevented by an earlier suspicion of cerebral infarction followed by imaging including angiography. This could have led to a timely diagnosis followed by intravascular intervention viz. intra-arterial papaverine or milrinone or balloon angioplasty. This was not possible in our patient since the imaging was delayed due to hemodynamic instability.
Postoperative management of craniopharyngioma is crucial and one should be vigilant for complications like endocrine insufficiency, fluid and electrolyte imbalance, seizures and ischemic events. Delayed cerebral infarction may present as hemiparesis, dip in sensorium, seizures or any new neurological deficit. Symptoms of new stroke may be missed in patients who are deeply comatose. There should be a high index of suspicion for delayed cerebral infarction in such cases with a low threshold for repeat brain imaging.
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