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Table of Contents    
CASE REPORT
Year : 2021  |  Volume : 69  |  Issue : 3  |  Page : 748-750

Endovascular Therapy in Paediatric Dissecting Intracranial Aneurysm: A Case Report


1 Division of Interventional Neuroradiology, Department of Radiology, KEM Hospital, Mumbai, Maharashtra, India
2 Division of Interventional Neuroradiology, Medical Trust Hospital, Kochi, Kerala, India

Date of Submission14-Jan-2019
Date of Decision15-Nov-2019
Date of Acceptance09-Jul-2020
Date of Web Publication31-May-2021

Correspondence Address:
Dr. Rashmi Saraf
Division of Interventional Neuroradiology, Department of Radiology, KEM Hospital, Mumbai, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.317236

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 » Abstract 


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. We report a case of 11-month-old girl who presented with intracranial bleed after a history of minor trauma whose diagnosis of an intracranial aneurysm was initially missed which later led to a rebleed. It was managed emergently with endovascular coiling and the patient showed incredible recovery in the post-operative period.


Keywords: CT angiography, dissection, intracranial aneurysm, paediatric, subarachnoid haemorrhage, trauma
Key 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.


How to cite this article:
Saraf R, Garg T, Parvathi S. Endovascular Therapy in Paediatric Dissecting Intracranial Aneurysm: A Case Report. Neurol India 2021;69:748-50

How to cite this URL:
Saraf R, Garg T, Parvathi S. Endovascular Therapy in Paediatric Dissecting Intracranial Aneurysm: A Case Report. Neurol India [serial online] 2021 [cited 2021 Sep 26];69:748-50. Available from: https://www.neurologyindia.com/text.asp?2021/69/3/748/317236




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.
Figure 1: (a and b) Plain CT brain showing left temporal hematoma (arrowhead), SAH (long thin arrow), SDH (thick arrow). (c) Repeat Plain CT brain at the time of discharge shows rebleed in the same region. (d) Plain CT brain at the time of sudden deterioration shows third episode of hemorrhage. (e and f) CTA shows multilobulated aneurysm (arrow). CTA shows multilobulated left MCA dissecting aneurysm embedded deep within the clot (arrow)

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Figure 2: Digital subtraction cerebral angiography was done, Towne view (a) and lateral view (b) shows bilobed dissecting aneurysm at the origin of superior trunk. (c) Delayed phase shows stasis within the superior lobule of the aneurysm sac. (d) Microcatheter position within the aneurysm sac. (e and f) Post coiling left ICA angiogram shows complete exclusion of the aneurysm from the circulation with slow flow in the superior trunk. (g) Plain skull radiograph shows large craniotomy defect (arrowhead) and the coil mass (arrow)

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 » Introduction Top


Childhood intracranial aneurysms are exceedingly uncommon.[1] 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.[2] 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.[3] 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.


 » Discussion Top


Intracranial aneurysms in children are very rare, and they are rarer in infants, in spite of that pediatric aneurysms form a different pathophysiological entity.[1] 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).[4] 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.[5] 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.[6]

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.[5] CT angiogram is mandatory in cases of intracerebral bleed with atypical presentation.[7] 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.[8]

Intracranial aneurysms in patients younger than 18 years are reported to account for only 0.5-4.6% of all diagnosed aneurysms.[9] 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.[10] Although the most frequent cause of subarachnoid hemorrhage in children is trauma or non-accidental injury.[11]

Only 5-10% of childhood aneurysms are related to head trauma and majority of them are due to non-traumatic causes.[12] 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.[4]

The most common location for large pediatric intracranial aneurysm is the middle cerebral artery.[13] Large (> 1 cm) or giant (>2.5 cm) aneurysms are more common in children and are associated with mass effect and seizures.[11] 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.[14]

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.[15]

Endovascular treatment

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.[16]

Declaration of patient consent

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.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
 » References Top

1.
Saraf R, Shrivastava M Siddhartha W, Limaye U. Intracranial pediatric aneurysms: Endovascular treatment and its outcome. J Neurosurg Pediatr 2012;10:230-40.  Back to cited text no. 1
    
2.
Young BJ, Seigerman MH, Hurst RW. Subarachnoid hemorrhage and aneurysms. Semin Ultrasound CT MR 1996;17:265-77.  Back to cited text no. 2
    
3.
Hulsmann S, Moskopp D, Wassmann H. Management of a ruptured cerebral aneurysm in infancy. Report of a case of a ten-month-old boy. Neurosurg Rev 1998;21:161-6.  Back to cited text no. 3
    
4.
Choux M, Lena G, Genitori L. Intracranial aneurysms in children. In: Raimoni A, Choux M, Di Rocco C, editors. Cerebrovascular Disease in Children. Springer Vienna; 1992. pp. 123-31.  Back to cited text no. 4
    
5.
Yatomi K, Oishi H, Yamamoto M, Suga Y, Nonaka S, Yoshida K, et al. Radiological changes in infantile dissecting anterior communicating artery aneurysm treated endovascularly. A case report and five-year follow-up. Interv Neuroradiol 2014;20;796-803.  Back to cited text no. 5
    
6.
Tan TC, Chan CM, Chiu HM. Traumatic intracranial aneurysm in infancy. Br J Neurosurg 2001;15:137-9.  Back to cited text no. 6
    
7.
Hotta K, Sorimachi T, Osada T, Baba T, Inoue G, Atsumi H, et al. Risks and benefits of CT angiography in spontaneous intracerebral hemorrhage. Acta Neurochir (Wien) 2014;156:911-7.  Back to cited text no. 7
    
8.
Garg K, Singh PK, Sharma BS, Chandra PS, Suri A, Singh M, et al. Pediatric intracranial aneurysms--our experience and review of literature. Childs Nerv Syst 2014;30:873-83.  Back to cited text no. 8
    
9.
Gemmete JJ, Toma AK, Davagnanam I, Robertson F, Brew S. Pediatric cerebral aneurysms. Neuroimaging Clin N Am 2013;23:771-9.  Back to cited text no. 9
    
10.
Sorteberg A, Dahlberg D. Intracranial non-traumatic aneurysms in children and adolescents. Curr Pediatr Rev 2013;9:343-52.  Back to cited text no. 10
    
11.
Norris JS, Wallace MC. Pediatric intracranial aneurysms. Neurosurg Clin N Am 1998;9:557-63.  Back to cited text no. 11
    
12.
Krings T, Geibprasert S, terBrugge KG. Pathomechanisms and treatment of pediatric aneurysms. Childs Nerv Syst 2010;26:1309-18.  Back to cited text no. 12
    
13.
Pruvot AS, Curey S, Derrey S, Castel H, Proust F. Giant intracranial aneurysms in the pediatric population. Neurochirurgie 2016;62:20-4.  Back to cited text no. 13
    
14.
Herman JM, Rekate HL, Spetzler RF. Paediatric intracranial aneurysms: Simple and complex cases. Pediatr Neurosurg 1992;17:66-73.  Back to cited text no. 14
    
15.
Johnston MV. Plasticity in the developing brain: Implications for rehabilitation. Dev Disabil Res Rev 2009;15:94-101.  Back to cited text no. 15
    
16.
Tai YP, Chou IC, Yang MS, Lin HC, Chiu HY, Kuo HT, et al. Neonatal intracranial aneurysm rupture treated by endovascular management: A case report. Pediatr Neonatol 2010;51;249-51.  Back to cited text no. 16
    


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  [Figure 1], [Figure 2]



 

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