Endovascular Therapy in Paediatric Dissecting Intracranial Aneurysm: A Case Report
Correspondence Address: Source of Support: None, Conflict of Interest: None DOI: 10.4103/0028-3886.317236
Source of Support: None, Conflict of Interest: None
Keywords: CT angiography, dissection, intracranial aneurysm, paediatric, subarachnoid haemorrhage, traumaKey Message: The incidence of cerebral aneurysms is rare in children, and it has to be definitively ruled out in all cases of intracranial bleed even if there is associated history of trauma. High clinical suspicion and complete history taking are the most important factors in the correct initial diagnostic workup.
An 11-month-old female child with a history of fall from bed resulting in cheek laceration. She presented to the casualty department where the patient's laceration was sutured by a plastic surgeon. On admission to the hospital, she was active. On day 2, the patient became drowsy and rapidly progressed into the coma with eye examination showing anisocoria, prompting an emergency Computerized Tomography Scan (CT scan) demonstrating left temporoparietal hematoma with left sylvian fissure subarachnoid hemorrhage and subdural hematoma [Figure 1]a and [Figure 1]b. At this time emergency surgical intervention was done to instantly relieve the mass effect. She recovered well post decompression craniotomy. A repeat CT scan demonstrated resolving intraparenchymal bleed [Figure 1]c, and hence discharge was planned. However, due to excessive crying that day, a repeat CT scan demonstrated small rebleed in the hematoma, which was managed conservatively. The next day, the patient suddenly developed loss of consciousness and was intubated emergently with repeat CT scan demonstrating large temporal clot with subarachnoid hemorrhage and intraventricular extension [Figure 1]d. CT angiography was then done demonstrating a dissecting middle cerebral artery aneurysm [Figure 1]e and [Figure 1]f. Reference was then given for endovascular management. Digital subtraction angiography with emergency endovascular coiling of a dissecting aneurysm was done [Figure 2]. Post coiling the patient gradually improved. At one month follow-up, the craniotomy was closed, the patient's general exam showed appropriate weight gain and neurological examination showed significant neurological recovery. At 3 months post procedure, the child was walking with a hemiplegic gait and had resolved facial asymmetry.
Childhood intracranial aneurysms are exceedingly uncommon. Diagnosis of intracranial aneurysms in infancy is difficult because of their infrequency and confusing clinical presentation, but it must be unequivocally excluded. Cerebral angiography is the criterion standard for the evaluation of an aneurysm. In rare cases, identifying ruptured cerebral aneurysms may also prove a challenging task because a delay in diagnosis and subsequent cerebrovascular spasm can present with a stroke. Here, we present an unusual case of an 18-month-old infant with a dissecting middle cerebral artery aneurysm that was confirmed on the basis of CT angiography and the challenges encountered in the management of aneurysms.
Intracranial aneurysms in children are very rare, and they are rarer in infants, in spite of that pediatric aneurysms form a different pathophysiological entity. The location and morphology as well as the clinical and radiological features of pediatric cases differ from those of adult cases. Posterior circulation and internal carotid artery bifurcation aneurysms are more common. However, one study revealed that many aneurysms in children are found in the anterior circulation (76%), especially in the middle cerebral artery (MCA). Moreover, the ratio of dissecting aneurysms is high (16-45%) in children and Lasjaunias et al. demonstrated the tendency for the rate of dissecting aneurysms to increase with younger age. We present a rare case of a cerebral aneurysm in an infant, where the history of trauma in the patient distracted and delayed the diagnosis of a cerebral aneurysm. The recurrent, repeated intracerebral bleeds alerted to the possibility of an underlying aneurysm. Traumatic intracranial aneurysms in children comprise less than 1% of all cerebral aneurysms.
Dissecting aneurysms can generally be detected by computed tomographic angiography (CTA), MRA, and cerebral angiography. Findings that suggest dissection are irregular stenosis, segmental stenosis, and aneurysmal formation (pearl-and-string sign), irregular fusiform or aneurysmal dilation, double lumen, and occlusion. CT angiogram is mandatory in cases of intracerebral bleed with atypical presentation. The presence of subarachnoid, intraventricular, intraparenchymal, or subdural hemorrhage with or without hydrocephalus should be an important pointer towards any possibility of underlying vascular cause even with a history of trauma.
Detailed account of the events by the parents and particularly significant findings like history such as thunderclap headache, nausea and vomiting, inconsolable crying, bulging fontanel, focal neurological deficits, or seizures maybe helpful.
Intracranial aneurysms in patients younger than 18 years are reported to account for only 0.5-4.6% of all diagnosed aneurysms. The incidence appears to be particularly low in the neonate (younger than 4 weeks) and infant (younger than 2 years) populations. 72% of those 0-18 years old with intracranial aneurysm presented with SAH, which is less than the 89% reported for the adult population. Although the most frequent cause of subarachnoid hemorrhage in children is trauma or non-accidental injury.
Only 5-10% of childhood aneurysms are related to head trauma and majority of them are due to non-traumatic causes. Infectious aetiology of intracranial aneurysms is more common in children as compared to adults and account for about 15% of pediatric aneurysms, the most common infectious agents being bacterial, and the most common organisms to be isolated are Staphylococcus Aureus and Streptococcus Viridians.
The most common location for large pediatric intracranial aneurysm is the middle cerebral artery. Large (> 1 cm) or giant (>2.5 cm) aneurysms are more common in children and are associated with mass effect and seizures. Children with higher grade aneurysms present in a better condition as compared to adults with the similar grade. Although vasospasm seems to be more severe initially, children also seem to be less susceptible to delayed ischemic deficits.
Our patient had a remarkable recovery, despite the extensive bleed and delayed diagnosis which shows the extensive re-learning which can occur in the brain.
Although the incidence of intracranial aneurysm is very low in the infant age group, we propose that in all case of intracranial hemorrhage without significant trauma a CT angiography should be done to rule out intracranial aneurysm as a suspected cause. Endovascular treatment with detachable platinum coils is now considered to be the first line treatment for adult patients with ruptured intracranial aneurysms. The International Subarachnoid Aneurysm Trial showed the chances of independent survival at 1 year were significantly better following endovascular treatment than after neurosurgical intervention. Advances in techniques and equipment mean that endovascular interventions is now possible even in infants and newborn patients.
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The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
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[Figure 1], [Figure 2]