Pediatric Intracranial Aneurysms. Experience of 47 Cases.: Corresponding Author

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Pediatric Intracranial Aneurysms. Experience of 47 cases.

Alexandru Vlad Ciurea MD, PhD1,2 , Aurel Mohan MD,PhD3,4 Andrei Voicu MD5, Vicentiu Saceleanu
MD,PhD6,7, Mihai-Stelian Moreanu8, Horia Ples, MD, PhD9,10

1. Chief of Neurosurgery Department, Sanador Clinical Hospital, Bucharest, Romania.


2. Professor of Neurosurgery at “Carol Davila” University of Medicine and Pharmacy, Bucharest,
Romania.
3. Department of Neurosurgery, Bihor County Emergency Hospital, Oradea, Romania.
4. Associate Professor of Neurosurgery at University of Oradea, Faculty of Medicine and Pharmacy,
Oradea, Romania
5. Resident of Neurosurgery at Bucharest Emergency Teaching Hospital, Bucharest, Romania.
6. Chief of Neurosurgery Department at Sibiu County Emergency Hospital, Sibiu Romania.
7. Lecturer at „Lucian Blaga” University of Medicine and Pharmacy, Sibiu, Romania.
8. Medical Student at “Carol Davila” University of Medicine and Pharmacy, Bucharest, Romania.
9. Professor Habil of Neurosurgery at Timis County Emergency Hospital, Head of Neurosurgical
Department
10. Professor Habil of Neurosurgery at “Victor Babes” University of Medicine and Pharmacy,
Timisoara, Romania

Corresponding author:

Mihai-Stelian Moreanu - [email protected]

Abstract:

Purpose: Pediatric aneurysms are rare, under 5% of cases, and their clinical presentation, rupture
probability, location and outcome are different from those in adults. In older pediatric patients’
symptoms are more similar to those in adults. Materials & Methods: The authors present 47 cases
of children operated between January 1999 and January 2019 - 20 years – multicenter study. Our
retrospective operated cases study is elaborated based on medical reports and imaging. Treatment
approach consists of open microsurgical dissection – clipping and endovascular embolization –
coiling. Results: The mean age is 14.3 years – ranging from 5 months to 16 years, with 28 boys
(59.5%) and 19 girls (40.4%). In our series, pediatric aneurysms represent 6.1% of all intracranial
aneurysms (771 cases). Clinical features are dominated by headache (45 cases- 95.7%), neck stiffness
(43 cases – 91.4%) and vomiting (42 cases – 89.3%). Most frequent location are anterior
communicating artery (17 cases, 36.1%), middle cerebral artery (12 cases, 25.5%) and internal
carotid artery bifurcation (9 cases, 19.1%). Glasgow outcome scale at six months shows good
recovery in 36 cases (76.5%), moderate disability in 9 cases (19.1%), severe disability in 1 patient
(2.1%) and (preoperative) death in 1 patient (2.1%). Conclusion: IA in children is a very rare
pathologic entity. Early neuro-imagistic diagnosis and microsurgical approach is mandatory and has
excellent results.

Abbreviations:

IA – Intracranial Aneurysm; H&H - Hunt & Hess Scale; ICA – Internal Carotid Artery; MCA – Middle
Cerebral Artery; ACoA – Anterior Communicating Artery; PCoA – Posterior Communicating Artery;

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VA – Vertebral Artery; GOS – Glasgow Outcome Scale; SAH – Subarachnoid hemorrhage; GCS –
Glasgow Coma Scale.

Introduction We report 47 consecutive cases of pediatric (≤


16 years old) aneurysms operated on a period
Intracranial aneurysms (IA) usually form at the
of 20 years from 1999 to 2019 at three
bifurcation of an artery where the
medical centers in Romania: “Bagdasar –
hemodynamic stress is high. In order to grow
Arseni” Teaching Hospital, Sanador Medical
and be distinguished, aneurysms require time.
Center Hospital and Timis County Emergency
Pediatric aneurysms are rare - under 5% of
Hospital. Most of the cases (40) were
cases, and their clinical presentation, rupture
operated in Bagdasar Arseni – Pediatric
risk, location and outcome are different from
Neurosurgical Department.
those in adults[1]. In older pediatric patients
symptoms are more similar to those in adults. Admission clinical status was assessed using
However, in neonates or younger children H&H scale. Our retrospective consecutive
symptoms are non-specific or even lacking. operated cases study was elaborated based
on medical reports and Imagistics. Regarding
The proportion of ruptured aneurysms
the primary investigations, we consider Digital
decrease with the age, being under 1% in
subtraction angiography (DSA) to be the main
patients under 15 years. Yet, children present
tool in diagnosing aneurysms. CT or Angio CT
better clinical aspects than adults after IA
were also used in diagnostic of SAH and
rupture. Male to female ratio of 2:1 contrast
aneurysm location and in the postoperative
with the female predominance found in adult
control assessment.
IA[2].
Treatment approach consisted of open
The purpose of our study is to analyze the
microsurgical dissection – clipping and
etiology, clinical aspects and surgical
endovascular embolization – coiling. Our
outcomes in children taking in consideration
policy for patient management includes
Table 1. Clinical status at admission follow-up at every 6 months with neurological
and ophthalmological examination with a
Symptoms No. Cases (%)
follow-up CT scan. Follow-ups were assessed
Headaches 45 (96%) ranging from 6 months to 8 years.
Neck Stiffness 43 (91%)
Table 2. Hunt & Hess Scale
Vomiting 42 (89%) H&H GCS
Neurological deficits 21 (45%) 2 25 (53%) 13 - 14 (w/o
focal deficit
Impaired 17 (36%) 3 9 (17%) 13 – 14 (with
consciousness focal deficit)
4 3 (8%) 7-12
Comatose status 9 (23%) 5 0 (0%) 3-6
Seizures 21 (45%) Total 47 (100)

Fever 16 (34%) Results

Patients age ranges from neonates – the


the primary factors that lead to long-term
youngest case was 5 months, to 16 years with
good results.
the mean age of 14.3 years. There were 28
Materials & Methods boys (58.7%) and 19 girls (41.2%). 78%
patients admitted presented SAH.

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experience in endovascular approach. In our
case series we reported 37 patients (78.5%)
We report 47 cases of pediatric IAs with
with good recovery, 8 patients (17.2%) with
headache (45 cases - 96%), neck stiffness (43
moderate recovery, 1 patient (2.1%) with
cases - 91%) and vomiting (42 cases - 89%).
severe disability and 1 death (2.1%). No
Table 3. Aneurysms Location vegetative state has been reported (Table 4).
Artery No. Cases (%)
ICA 9 (19.5%)
MCA 12 (25%) Discussions
ACoA 17 (36%)
Pediatric aneurysms differ from those
PCoA 6 (17.5%)
encountered in adults having male
Basilary Top 1 (2.5%)
predominance[3], supported by the findings of
VA 1 (2.5%)
this study. The female predominance in adults
Multiple 1 (2.5%)
may be associated with low levels of
Total 47 (100%)
estrogens that lead to low vessel resistance
Most common neurological deficits were causing aneurysm formation.
hemiparesis and speech disorders, The most common risk factors in adults are
hemianopia with oculomotor paresis (21 cases systemic hypertension, smoking,
– 45%) and seizures (21 cases – 45%) (Table hypercholesterolemia, diabetes mellitus and
1). 75% of patients (35 cases) were in good cardiopathy[4]. However, in children aneurysm
clinical status at admission and 10 cases (22%) etiology is more often associated with
presented with grade 1 H&H and 25 (53%) congenital malformations of blood vessel wall
patients with grade 2 H&H (Table 2). (if the aneurysms appear in the first 2 years of
life) or with a vascular syndrome, heart and
aortic malformation (if it appears later)[4].
Anterior Communicating Artery (ACoA) was
the most common location – 17 cases (36%), Development of aneurysms may be caused by
then Middle Cerebral Artery (MCA) – 12 cases trauma, infection or it can be associated with
(25%), followed by Internal Carotid Artery a vasculopathy or an alteration of vessel wall
(ICA) – 9 cases (19.5%) (Table 3). structure [5]. Genetics may play an important
role in aneurysms development when
Open microsurgical dissection under associated with heritable disorders (Ehlers–
Table 4. Postoperative GOS at 6 months Danlos Types IV, Marfan syndrome, Osler-
Weber-Rendu Syndrome, fibromuscular
Status No. Cases (%)
dysplasia and polycystic kidney disease) or
Good recovery 37 (78.5) congenital malformations[6][7]. Systemic
arterial malformations of the aorta could lead
Moderate Recovery 8 (17.2)
to high cerebral pressure that will cause a
Severe disability 1 (2.1) focal degeneration of the internal lamina of
Vegetative state 0 (0) the cerebral vessels. In such situations to
prevent the endovascular manipulation of the
Death 1 (2.1) systemic malformation and to remove the
Total 47 (100) possible hemorrhage open clipping is the
elected treatment[8].
intraoperative monitoring was performed in
Most commonly, pediatric IA are found on the
46 cases (97%) and endovascular embolization
imagistic examination – SAH, especially in
in 1 case (3%). Authors do not have enough

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younger patients where headache is difficult
to be objectifiied[9], in accordance with our
study findings. SAH is better tolerated in
children than in adults, associating a low-rate
of vasospasm, lower postoperative
administration of anti-ischemic medication
and better long-term outcomes – Table 5.
Children also associate better preoperative
grades, fewer comorbidities, higher brain
plasticity and lower levels of
atherosclerosis[10]. An important role in
decreasing the effect of vasospasm is also
attributable to the high-potential
leptomeningeal circulation[11]. Aneurysm
rebleeding after SAH appears in 52-60% cases
and divides opinions on outcome [12][13]. One
study presents it as the primary causes of
mortality - 76% of total deaths [12], while in
other case series study approximatively 80%
of rebleeding were associated with good
postoperative outcomes[14].

Higher incidence in posterior circulation was


also reported in children than in adults [15]. In
our series, most of the aneurysms belonged to
the anterior circulation. This feature was
similar with adult series where ACoA is the
most common aneurysm site[16]. Studies have
reported a direct relationship between the
age of the patients and the location of the
aneurysms showing up that aneurysms in the
young patient tend to arise more often in the
MCA. The early embryological development
with associated vascular pathology may play
an important role in aneurysm development
after birth [17].

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Surgical approaches vary – clipping, proximal Although endovascular approach showed
vessel occlusion, wrapping, trapping, bypass good results, the rate of re-intervention is
alone, endovascular treatment. The selected high especially in complex or giant aneurysm.
treatment however depends also on location, Hence, bypass surgery combined with artery
aneurysm size and patient preoperative clipping reported good results and therefore
status. We consider clipping to be the elective should be used[21].
choice when dealing with ruptured aneurysm
Moreover, anatomical variations in the Willis
or associated systemic pathology that lead to
polygon - aneurysms associated with arterial
hypertension. Depending on location,
aplasia or asymmetric vessel organization,
vertebral tip aneurysm is preferred to be
tend to recur more often[22].
treated by coiling[18] while MCA aneurysms is
treated more often with clipping because of Long-term outcome depends on the
the arterial branches that may arise directly preoperative status of the patients. Those
from the MCA aneurysm. admitted with good GCS showed a better
recovery. In addition, absence of vasospasm
Studies are not in consensus regarding
and complete obliteration of aneurysm play
recurrence rate. Some reported high-
an important role in patient recovery [23]. Both
recurrence when ICAs were treated with
clipping and coiling were associated with good

Figure 1,2. : Preoperative and postoperative angiographies of a 12-years-old girl admitted in our
department

coiling[19], but some stated that endovascular outcome in 69-91% of cases [3][22][23]. Findings
treatment alone showed good long-term of our study supports the aforementioned
outcomes[19]. Immediate higher postoperative statement, reporting low postoperative
mortality and longer hospital stay is more vegetative state and mortality. Moreover, no
frequently associated with clipping than perioperative mortality and recurrence after
embolization[20]. Recurrence rate is between clipping was reported.
12-36%[16] and depends on aneurysm size.

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Treatment of pediatric cases faces big A 12-years-old girl with headaches and altered
challenges. Newly formation of aneurysms general status was admitted the neurosurgical
after surgery is greater in children than in department. At presentation she had SAH
adults[24]. Thus, our policy recommend that with H&H Score 1. Other symptoms were neck
the elective treatment should be chosen stiffness, vomiting and impaired
separately according to the complexity of the consciousness. The first Angiography showed
case and neurosurgeon experience. a large MCA Aneurysms (Figure 1). The case
was microsurgically approached. A clip was
Table 5. Pediatric patients series published placed at the neck of the aneurysms. At the
starting from year 2000
postoperative control angiography, general
Age SAH status was improved and symptoms were
Author Year No.
(yrs) (%) ameliorated (Figure 2).
Current
47 ≤16 78 Conclusions
Study
Proust et Al. IA is a rare pathology in pediatric
[25] 2001 22 ≤16 95
neurosurgery. Pediatric aneurysms are usually
Lasjaunias et
2005 59 ≤15 51 large and singular in pediatric population,
al. [11]
frequently located on carotid bifurcation. Our
Agid et al. [26] 2005 33 ≤17 36 series of 47 consecutive operated cases
Krishna et al. illustrates the clinical aspects, most frequent
[27] 2005 22 ≤18 91 location - ACoA and surgical outcomes in
Huang et al. children taking in consideration the primary
[28] 2005 19 ≤18 58 factors that lead to long-term good results.
Sharma et al. Clinical presentation on admission, especially
[29] 2007 55 ≤18 78
in patients younger than 3 years represents a
Stiefel et al. major challenge for physicians. A
[18] 2008 12 ≤16 100
multidisciplinary approach is needed to
Vaid et al. [4] 2008 27 ≤18 100 effectively overcome this pathology.

Liang et al. [30] 2009 24 ≤14 46


Disclaimer
Jordan et al.
[31] 2009 15 ≤19 100
No conflict of interests. Original study. No
Songsaeng et financial support.
2009 8 <16 25
al. [22]
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