Keywords
craniopharyngioma - basilar artery - thrombosis - posterior circulation stroke - vascular
complication
Key Messages
A posterior circulation stroke can also be a rare but possible postoperative complication
of craniopharyngioma surgery, and one should be on the vigil for prodromal symptoms
of a posterior circulation stroke in the immediate and the early postoperative periods.
Introduction
Craniopharyngiomas are rare tumors arising from remnants of the Rathke pouch (craniopharyngeal
duct) and are associated with significant morbidity and mortality. It is a rare type
of brain tumor of uncertain behavior that occurs at a rate of 1.3 per million person
years.[1] The most common complications are due hormonal imbalances caused by damage to the
pituitary or hypothalamus. Neurovascular complications after surgery are very rare,
with only a few documented cases being reported in literature including fusiform dilatation
of the carotid artery,[2] carotid artery laceration,[3] or delayed vasospasm secondary to vessel handling or cyst rupture.[4] No case of thrombosis of posterior circulation vessels has been reported in literature.
Here the authors describe a case of acute posterior circulation stroke due to basilar
artery thrombosis as an early postoperative complication.
Case Presentation
A 13-year-old obese boy presented with complaints of progressive deterioration of
vision and progressive intermittent holocranial headache for the past few months.
Contrast-enhanced magnetic resonance imaging (CEMRI) brain ([Fig. 1]) was done suggestive of solid cystic craniopharyngioma in the sellar–suprasellar
region with a small cystic component extending in the posterior fossa in the prepontine
cistern. The child underwent right frontotemporal craniotomy and complete excision
of lesion via interoptic and opticocarotid routes. Intraoperatively, there was handling
of only right internal carotid artery (ICA) with sparing of all other vessels. Perforators
were preserved. The prepontine cystic component of tumor along with rest of the tumor
with no manipulation needed in posterior fossa. Basilar artery was visualized and
was intact. There was no evidence of vasospasm of vessels intraoperatively. There
was no significant blood loss during surgery and pituitary stalk was undamaged. Postoperatively,
after a night of elective ventilation, the child was extubated and was neurologically
intact with Glasgow coma scale of 15 of 15. Patient developed transient type of diabetes
insipidus (DI) on postoperative day (POD) 2 for which correction was done using desmopressin
spray and adequate hydration. The next day, the child was found to be drowsy with
decreasing sensorium. Electrolyte and hormonal profile was suggestive of hypopituitarism
for which hormone replacement therapy was initiated with oral thyroxine and corticosteroids.
Postoperative noncontrast computed tomography (NCCT) head scan done which showed satisfactory
postoperative changes ([Fig. 2]). DI was stabilized and the child was doing well and was planned for discharge.
Fig. 1 Preoperative contrast enhanced magnetic resonance images showing sellar–suprasellar
lesion (A) extending into posterior fossa (B) suggestive of craniopharyngioma.
Fig. 2 Postoperative computed tomography scan of the head showing complete excision of the
lesion (A).
On POD-9, patient complained of vertigo with no cerebellar signs. His vertigo was
controlled by supportive medications, and there were no other complaints, so we chose
to observe. However, child developed tonic–clonic seizures along with deterioration
in sensorium at night. The seizures were persistent despite multiple intravenous (IV)
antiepileptics. CEMRI brain was done which revealed multiple acute cerebellar, pontine,
and midbrain infarcts with thrombosis of basilar artery ([Fig. 3]). Magnetic resonance (MR) angiogram of the neck and brain was obtained which revealed
thrombosis of the basilar artery with poor flow in the right posterior cerebral artery
with normal neck vessels ([Fig. 4]). There was no evidence of any vasospasm or vessel dissection. Anticoagulant therapy
was started immediately in the form of subcutaneous low molecular weight heparin.
Laboratory reports of coagulation profile were within normal limits and echocardiography
also did not reveal any abnormality. No evident cause was found for the basilar artery
thrombosis. The child did not respond to therapy and passed away.
Fig. 3 Magnetic resonance images of the brain showing acute multiple cerebellar, pontine,
and midbrain infarcts.
Fig. 4 Magnetic resonance angiogram of the brain showing basilar artery thrombosis with
poor flow in right posterior cerebral artery (A).
Discussion
Vascular complications are very rare postoperative complications for craniopharyngioma.
Of these, most commonly reported are those that occur due to trauma during surgery
or vessel handling, such as fusiform dilatation of the ICA or vasospasm of vessels
of the anterior circulation. Thrombosis of the vessels of posterior circulation is
unheard of. Pediatric arterial ischemic stroke (PAIS) has an incidence of 3.3 cases
per 100,000 children/year (with the vertebrobasilar territory involvement seen in
up to 36% of cases); however, the incidence of isolated childhood basilar artery occlusion
(BAO) and stroke (BAS) is unknown.[5] The most common cause of acute BAO in children is vertebral artery dissection caused
due to trauma.[6] In our patient, there was no evidence of arterial dissection in the neck or brain
vessels. Coagulation profile, echocardiography, and thrombophilia panel were also
within the normal range. There was no evidence of any vasospasm. The authors conclude
that if prodromal symptoms such as vertigo and nausea were taken into consideration
at an earlier stage and had posterior circulation involvement been suspected, proper
imaging could have been done sooner and appropriate measures could have been taken
to save the child. If the posterior circulation stroke was diagnosed in time, then
mechanical thrombectomy could have been done. With timely intervention, it may have
been possible to save the child.
