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Child’s Nerv Syst (2001) 17:37–41

© Springer-Verlag 2001 O R I G I N A L A RT I C L E

M. Wojtacha Cerebral aneurysms in childhood


P. Bażowski
M. Mandera
I. Krawczyk
A. Rudnik

Received: 12 July 1999 Abstract Objects: The aim of this nance in our population. In children
Revised: 7 July 2000 work was to ascertain any clinical we found only 1 case of middle cere-
and anatomical factors allowing dif- bral aneurysm and 1 case of multiple
ferentiation between aneurysms of aneurysms. We also found a high
childhood and those occurring in rate of rebleeding in the paediatric
adults by comparing both groups. group. Conclusions: We suggest that
Methods: Results obtained in a total the very good outcome (100% very
of 17 children and adolescents aged good results in patients operated on
up to 18 who had been operated on early) obtained and the high risk of
for cerebral aneurysm in our depart- rebleeding in children with cerebral
ment from 1989 to 1997 (3% of all aneurysm allow the recommendation
patients treated for subarachnoid of early surgery in children with rup-
haemorrhage resulting from ruptured tured cerebral aneurysms.
M. Wojtacha (✉) · P. Bażowski cerebral aneurysm in this period)
M. Mandera · I. Krawczyk · A. Rudnik were compared with those in the Keywords Intracranial aneurysm ·
Department of Neurosurgery,
Silesian University School of Medicine, adult group operated on in our de- Subarachnoid haemorrhage ·
40-752 Katowice, Poland partment. In contrast to the situation Children
Fax: +48-32-2525812 in adults, there was a male predomi-

Introduction dence of traumatic and infectious lesions was reported


[1, 6].
Intracranial aneurysms are rare in childhood, constituting The aim of this work was to determine clinical and
approximately 0.5–4.6% of all patients admitted to neu- anatomical factors that make it possible to distinguish
rosurgical departments with a diagnosis of cerebral aneu- the aneurysms of childhood from those in adults by com-
rysm [7, 10, 18, 19, 20]. In the paediatric population rup- paring the two groups.
ture of the aneurysm is a cause of subarachnoid haemor-
rhage (SAH) in 18–52.1% [7, 12). Aneurysms before the
age of 15 are extremely rare, accounting for approxi- Materials and methods
mately 1% of all aneurysms [20]. In the literature, how-
ever, we can find reports of cases of aneurysms even in Throughout the period 1989–1997, a total of 561 patients with
infants [8, 18]. Analysis of previously reported cases in- confirmed saccular aneurysm were treated surgically in the De-
partment of Neurosurgery at the Silesian University School of
dicates some distinct characteristics that distinguish an- Medicine in Katowice. Of this total population, 17 patients were
eurysms in childhood from those occurring in adults. 18 years old or younger. The aneurysms with traumatic or mycotic
Many authors have noted the male predominance in aetiology were excluded from this study a priori. We compared the
childhood, in contrast to the female predominance in paediatric group with 358 adults operated on for ruptured aneu-
rysm in our department between January 1989 to June 1995.
adults [7, 12, 18, 19]. Some authors have also noted a On admission, the neurological and consciousness status of all
high incidence of posterior circulation aneurysms [18] patients was estimated according to the Hunt-Hess scale [13].
and giant aneurysms [18]. In other studies a higher inci- General clinical state was also evaluated. The incidence and the
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timing of rebleeding were also analysed. In all patients cerebral


angiography was performed. We took into consideration location,
size, and occurrence of multiple aneurysms and the presence of
narrowing of the vessels. Computed tomography (CT) on admis-
sion was performed in all patients admitted early (before 72 h after
initial bleeding) and in several patients admitted after this time. In
CT scans we evaluated the amount of extravasated blood accord-
ing to Fisher’s scale [4], widening of cerebral ventricles using
both Evans’ [2] and Hahn’s [9] indexes, and the presence of intra-
ventricular haemorrhage (IVH). The measurements were estimat-
ed as abnormal if the Evans index score exceeded 0.3 [2] and the
score on Hahn’s index exceeded 36% [9].
All patients were treated surgically, and in all cases the aneu-
rysms were successfully clipped.
The outcome according to the Glasgow Outcome Scale [14]
and the presence of seizures were evaluated in both groups at least Fig. 1 Location of aneurysms in children
1 year after operation.

Results
These 17 children with aneurysm constituted about 3%
of the population with intracranial aneurysms treated
surgically at our department. The youngest patient was
10 years old, and the average age was about 15 years
old. In the adult group the average age was 45.2 years
(SD=11.682). There were 4 girls and 13 boys among
the operated children. The male-to-female ratio was
3.25 to 1 (77.5% of males) in this group. In the adult
group there were 169 men (47.2%) and 189 women
(57.8%).
In 7 cases of surgically treated children we ascer-
tained rebleeding before surgery. Taking into consider- Fig. 2 Location of aneurysms in adults
ation that 12 of our patients were treated on late surgery
(later then 3 days after initial bleeding) we could expect
rebleeding in 60% of cases. In adults rebleeding oc- ple aneurysms. The child with multiple aneurysms also
curred in 27%. suffered from systemic arterial hypertension.
On admission the Hunt and Hess score in 8 children In the paediatric group we found no cases of giant an-
after SAH (50%) was evaluated as 1. We classed 3 chil- eurysm. In the adult group we found giant aneurysms in
dren (18.8%) as grade 2, 1 patient (6.3%) as grade 3, and 4.74% of cases.
four patients (25%) as grade 4. In the adult group grade In the paediatric population we found aneurysms aris-
1 was observed in 89 patients (28.8%), grade 2 in 125 ing from the ACoA in 9 cases (50%), while in 8 cases
(40.5%), grade 3 in 62 (20.1%), grade 4 in 32 (10.4%) (44.44 %) the aneurysm was located on the ICA. Only in
and grade 5 in 1 patient (0.3%). 1 case (5.56%) was the aneurysm located on the MCA.
In the children’s group we found systemic arterial hy- We did not find any cases of posterior circulation aneu-
pertension only in 1 case (5.9%), whereas in the adult rysm (Fig. 1). In adults we found aneurysms arising from
patients it was found in 132 cases (36.9%). In the child the ACoA in 148 cases (34.66%), from the MCA in 155
with arterial hypertension this was associated with adi- cases (36.30%) and from the ICA in 100 cases (23.42%).
pose-genital syndrome. The case of anterior communi- Occasionally, aneurysms were found on the posterior cir-
cating artery aneurysm (ACoA) became manifest with culation arteries (13 cases; 3.04%), and on the perical-
the syndrome of anorexia. In the paediatric population losal artery (11 cases; 2.58%; Fig. 2).
only 1 case without SAH in a patient showing the neuro- Hydrocephalus was observed in 1 case (5.9%), and
compressive symptoms on admission was found. Poly- this patient required a shunting operation. In the adult
cystic kidney was also diagnosed in 1 case. We did not group widening of the ventricular system was present in
find a positive family history or coexisting aortal coarc- 79 cases (22%), 18 of them requiring shunt surgery.
tation in any case. In children a good outcome (grade I and II according
In 1 case (5.9%) we found two aneurysms, one locat- to GOS) was obtained in 12 cases (70.6%), and in the
ed on the MCA and the other on the ICA, both on the adult group a good outcome was noted in 196 cases
right side. In the adult group 17% of patients had multi- (64%). Two children died (11.8%). In the adult group the
39

large area, with a catchment population of 7 million in-


habitants.
The male-to-female ratio (3.25 : 1) in our patients
confirms the observations of other authors. In the study
of Meyer et al. [18] the ratio was 2.8 : 1. In many other
studies this predominance was also evident [7, 10, 12,
19]. The female predominance in adult patients with an-
eurysm may be connected to the decreased level of oes-
trogen in this group. The decreased level of oestrogen re-
sults in a decrease of collagen, which plays an important
part in maintaining the strength of the vessel wall [16].
We noted the coexistence of polycystic kidney in only
1 case. Ostergaard et al. also found polycystic kidney in
1 case out of 43 analysed [19]. Other authors did not re-
port polycystic kidney [7, 18, 20].
Fig. 3 Surgical results by time of surgery in children Systemic arterial hypertension was noted in 1 (5.9%)
of our patients, in whom we also diagnosed adipose-gen-
ital syndrome. In the series reported by other authors the
incidence of arterial hypertension was very low [19].
On analysis of our patient population we were also
unable to confirm the more common occurrence of posi-
tive familial history and aortal coarctation in cases with
cerebral aneurysm. This fact was also noted by other au-
thors [7, 18, 19, 20, 22]. Heiskanen [10] reported the co-
arctation of the aorta associated with cerebral aneurysm
only in 1 case in the paediatric population.
In 1 case we found no SAH, but only the symptoms
of anorexia. We did not find the same symptomatology
in the patients of other authors. Gerosa et al. [7] reported
1 case of ACoA aneurysm with concomitant pituitary
dysfunction and amenorrhoea.
More adults than children were in a poor neurological
Fig. 4 Surgical results by time of surgery in adults
condition at the time of admission. In our material 25%
of the children admitted were classed as grade IV and V
according to the Hunt-Hess scale and about 69% of pa-
tients were in a good neurological state (grades I and II
mortality rate was 19% (58 patients). Poor outcomes on the Hunt-Hess scale). In the adult group the corre-
were observed in 3 (17.6%) children (grades III and IV sponding proportions were, respectively, 68% and about
according to GOS), a similar proportion to that in adults 11%. In the study of Ostergaard et al. [19] only 58% of
(52 patients – 17%). children admitted were classed as grade I and II accord-
Four children were operated on early (up to 72 h after ing to the GCS, that is in a good clinical condition.
SAH). The most important cause of delayed surgery was Gerosa et al. found only 2 patients (about 15%) scored as
too-late transfer from the general paediatric unit. In all grade IV on the Hunt-Hess scale on admission, and an
cases we obtained a very good outcome (Fig. 3). The re- intracerebral haematoma was present in both these cases
sults of treatment in correlation with the time of surgery [7].
in adults are presented in Fig. 4. In children we found that most of the aneurysms arose
from the ACoA and the ICA, and only in 1 case from the
MCA. These localisations are very different from those
Discussion found in adults. The common localisation of aneurysms
on the ACoA and on the bifurcation of the ICA has also
The incidence of aneurysms is rather low in children been observed by other authors [7, 10, 12, 19, 22]. How-
compared with adults and constitutes 0.5–4.6% of all ever, in some series an appreciable number of MCA an-
aneurysm cases [7, 10, 18, 19, 20]. The relatively high eurysms was noted [7, 12, 19].
incidence rate of paediatric aneurysms in our patient We did not find posterior circulation aneurysms.
population can be explained by the fact that our depart- Gerosa et al. also found no posterior circulation aneu-
ment is the only paediatric neurosurgical centre in a rysms [7]. In the large series of Pasqualin et al. posterior
40

circulation aneurysm was found only in 3% of cases It is difficult to compare our results with those in oth-
[20]. Similarly, in the material of Heiskanen only 1 case er reports, because the periods of observation have been
of posterior circulation aneurysm was observed [10]. The longer in the other works. The techniques of surgery
incidence of posterior circulation aneurysm in the large have changed radically since the 1960s and 1970s. The
series of Hourihan and al. was also very low [12]. In the surgical mortality reported from the premicrosurgical era
study of Ostergaard the posterior circulation aneurysms was extremely high. The timing of surgery has also
make up 6% of all aneurysms [19]. In the series of other changed in the case of aneurysmal SAH.
authors the number of posterior circulation aneurysms is In our series the outcome was better in children than
very high, found in 35–46% [18, 22]. in adults. The clinical outcome also appeared to be sig-
In contrast to our study, giant aneurysms were reported nificantly better in children than in adults in the studies
in the literature quite often. The proportion of giant aneu- of others, especially in the case of patients who were in
rysms in other series approached 20–31% [7, 17, 22]. In the good neurological condition on admission [10, 11, 19,
series of Meyer giant aneurysms made up as much as 54% 20]. The reversibility of neural injury in young patients
of all cases [18]. Conversely, Heiskanen [10] did not find and the low incidence of vasospasm in this age group
any giant aneurysms at all, and Ostergaard et al. [19] report- could be the explanation for this [10, 19, 20]. The low
ed only 2 cases of such aneurysms in his population (4.5%). incidence of hydrocephalus in the paediatric group could
The giant aneurysms commonly occur in infants [18]. also have some effect on outcome. This has also been re-
In the report of Lena and Choux, two of four giant aneu- ported in other studies [19, 20].
rysms were found in infants [17]. As surgery of giant an- Our results seem to confirm that early surgery is indi-
eurysms is still very dangerous, the endovascular tech- cated in the cases of aneurysmal bleeding in childhood.
nique seems to be the method of choice in such cases The number of patients operated on early in the current
and has been performed with good results [15, 17]. study is very low, but the results of surgery obtained are
We found only 1 case of multiple aneurysm. The mal- very good. Early surgery is recommended because of
formation was associated with hypertension and adipose- better tolerance of surgery and the greater risk of re-
genital syndrome in this case. Compared with adults the bleeding in children than in adults [19, 20].
incidence of multiple aneurysm is very low in children. Epilepsy was occasionally found in children after sur-
In the patient group of Heiskanen there were no cases of gery in our series. In other paediatric series the occur-
multiple aneurysm [10]. In the study of Hourihan et al. rence of epilepsy was infrequent [10]. However, in the
only 9.2% of children had multiple aneurysm [12]. series of Ostergaard et al. the incidence of postoperative
Ostergaard et al. reported that only 4 children had diag- epilepsy was very high. In this material 7 patients [24%]
nosed arterial hypertension before admission. Three of [19] developed epilepsy in spite of receiving preventive
them had multiple aneurysm [19]. Our study also con- anticonvulsive therapy.
firms that hypertension in children coexists with multiple
aneurysm. The rare incidence of MCA aneurysms, multi-
ple aneurysms and their coexistence with arterial hyper- Conclusions
tension suggests a different pathogenesis of aneurysm in
childhood. In the congenital theory suggested by Forbus 1. In contrast to the definitive female predominance
the developmental deficits in internal elastic membrane found in adults, in paediatric cases of intracranial saccu-
and muscular layer were to be responsible for the devel- lar aneurysms a male predominance is confirmed. We
opment of aneurysm [5]. On the other hand, Stehbens did not confirm any predominance of posterior cerebral
claimed that the acquired nature of this lesion was con- circulation aneurysm and giant aneurysm in children.
nected rather with hypertension than with developmental The low incidence of aneurysms arising from the MCA
defects of the vessel wall [21]. Fergusson confirmed this and of multiple aneurysms was also shown in our study.
theory in his excellent experimental work [3].
2. The great risk of rebleeding in children and the fact
In our material the rebleeding rate was very high
that children tolerate the surgery very well inclines us to
(60%) in the paediatric group compared with adults
operate on these patients as soon as possible.
(27%). Ostergaard et al. [19] showed a 13% rebleeding
rate in his study of a paediatric population.

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