J Neurosurg Pediatrics 12:367–373, 2013

©AANS, 2013

Resection of supratentorial lobar cavernous malformations in

children
Clinical article

Bradley A. Gross, M.D., Edward R. Smith, M.D., Liliana Goumnerova, M.D.,

Mark R. Proctor, M.D., Joseph R. Madsen, M.D., and R. Michael Scott, M.D.
Department of Neurosurgery, Boston Children’s Hospital, Harvard Medical School, Boston, Massachusetts

Object. The authors present a series of children with supratentorial lobar cavernous malformations (CMs). Cur-
rent imaging and operative techniques along with long-term follow-up were incorporated to characterize the response
to surgical treatment in this pediatric population.
Methods. The senior author’s operative experience was reviewed retrospectively along with a review of the
Boston Children’s Hospital database from 1997 to 2011 for children with supratentorial lobar CMs. Lobar CM was
defined as having a supratentorial location but not involving the thalamus/hypothalamus or basal ganglia. Baseline
patient demographics, pertinent radiographic findings, operative outcomes, and long-term results were evaluated and
compared between patients managed operatively and those who were managed nonoperatively.
Results. Of 238 CMs identified, 181 (76%) were lobar. Compared with patients managed with observation only,
those selected for surgery were older (p = 0.03), more likely to have symptomatic lesions (p < 0.001), and had larger
lesions (p < 0.001). Of the 83 CMs selected for surgery, 98% were completely resected. Over a total of 384.5 patient-
years of follow-up after surgery (mean 4.6 years; median 2.7 years; range 0.1–22.3 years), there were no subsequent
hemorrhages in any patient undergoing complete resection; 1 of the 2 incompletely resected lesions rebled during
the follow-up period. Radiographically, there was 1 recurrence (1.2%) in a child with multiple CMs; there were no
recurrences of completely resected single lesions. Of the 48 patients who presented with seizures (acute or chronic),
46 (96%) were seizure free at follow-up. The permanent neurological complication rate of surgery was 5%; these
complications were limited to those patients whose lesions were in eloquent locations.
Conclusions. Pediatric patients with symptomatic supratentorial lobar CMs are ideal candidates for surgery, for
which there are high complete resection rates, rewarding long-term seizure outcomes, and low operative morbidity.
Observation may be warranted in smaller asymptomatic lesions located within eloquent cortex.
(http://thejns.org/doi/abs/10.3171/2013.7.PEDS13126)

Key Words • cavernous malformation • cavernoma • lobar • pediatric •

children • surgery • seizure • epilepsy

C
avernous malformations (CMs) are blood vessel
malformations of the brain composed of sinusoi-
dal vessels lined by a single layer of endothelium
usually without interdigitated brain tissue. They are typi-
cally well circumscribed but may be intimately associ-
ated with other vascular anomalies of the brain, including
developmental venous anomalies (DVAs) and venous or
capillary telangiectasias. Cavernous malformations usu-
ally present because of seizures, headaches, or neuro-
logical deficits due to hemorrhage or focal mass effect,
and they frequently come to neurosurgical attention for
evaluation of surgical excision.7,18,28,30
We undertook this study to review data obtained in
a large cohort of children with lobar CMs, comparing
Abbreviations used in this paper: CM = cavernous malformation;
DVA = developmental venous anomaly.
those selected for surgery with those managed nonop-
eratively. After comparing demographics and angioar-
chitectural features between groups, we evaluated long-
term outcomes and complications for each management,
particularly in terms of freedom from seizures and late
hemorrhage.
Methods
Population and Inclusion Criteria
With the approval of the Boston Children’s Hospital
Institutional Review Board, we undertook a retrospective
review of departmental and hospital databases to find all
This article contains some figures that are displayed in color
on­line but in black-and-white in the print edition.

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patients less than 22 years of age, surgically and nonsur-
gically treated, who were diagnosed with cerebral CMs
from 1997 through 2011. We required that all lesions be
at least 4 mm in size and visible on T2-weighted MRI.
We added a retrospective analysis of the senior author’s
operative experience with these lesions spanning 4 dec­
ades. This combined search identified 183 children with
238 CMs.30 Of the 238 CMs, 181 (76%) were supratento-
rial lobar lesions, which we defined as located above the
tentorium but not involving the thalamus/hypothalamus
or basal ganglia.
Data Collection and Analysis
We reviewed these patients’ hospital and office charts
to determine the following information: patient age at
presentation, sex, modality of presentation, family his-
tory of CMs, history of cranial radiation therapy, CM size
and location, associated DVAs, CM treatment modality
(resection or observation), initial results, and long-term
neurological follow-up. We performed statistical analysis
(STATA 12.0) of categorical variables using the Fisher
exact test and of mean values (patient age and CM size)
with a 2-tailed t-test.
Surgical Technique
Surgical technique was as previously described,30
with the usage of intraoperative frameless stereotaxy in
all cases since 2003. Intraoperative MRI has been used
for select cases since 2005, and intraoperative ultrasound
has been employed almost universally since 2005. Fol-
lowing craniotomy, the CM was identified (on the surface
or after a small corticectomy), and its margins were identi-
fied and dissected from adjacent normal brain tissue with
microdissectors and bipolar cautery under the operating
microscope, frequently employing suction for traction on
the lesion. The lesion was grasped and manipulated with
microforceps as its feeders were coagulated and divided
to deliver it from the operative bed. Care was taken to
preserve any DVA, if present. No effort was made in any
patient to resect hemosiderin-laden tissue around the le-
sion proper.
Results
Background and Demographics for Surgically Treated CMs
Of the 181 supratentorial lobar CMs identified, 83
(46%) were selected for resection, and 98 (54%) were
observed without operation (Table 1). The mean patient
age in the surgical cohort was 11.8 years (median 12.1
years; range 0.7–22 years) and 57% were male. Fourteen
percent of children had a family history of CM, and 8%
had undergone prior cranial radiation therapy (6 children
for acute lymphocytic leukemia and 1 for medulloblas-
toma). The majority (75%) presented with symptomatic
hemorrhagic lesions, 58% had experienced at least 1 sei-
zure, 40% had headaches, and 12% had focal neurologi-
cal deficits. Four (5%) were asymptomatic, undergoing
surgery due to either an increase in CM size or strong
family preference. Lesions were predominantly (52%) in
the frontal lobe; 25% were temporal, 18% parietal, and
368
B. A. Gross et al.
5% occipital. Thirty percent of lesions were in eloquent
cortex (motor, sensory, speech, or calcarine cortex). Mean
lesion size was 2.2 cm, and 13% had a radiographically
apparent DVA seen on post-Gd T1-weighted MRI.
Surgical Treatment
Overall, 81 (98%) of 83 CMs were completely resect-
ed. In 1 case, a hemorrhagic CM confirmed by intraop-
erative motor stimulation to be entirely embedded in the
motor cortex without a safe corridor of access was left in
place (Fig. 1). After 15 years of follow-up, the patient has
not had a recurrent event and remains off medications.
Remarkably, the lesion appears on follow-up MRI to have
spontaneously thrombosed. In another case, the CM was
associated with an extensive venous telangiectasia that
precluded complete resection (Fig. 2).
Follow-Up, Complications, and Long-Term Outcome After
Surgery
The mean postoperative follow-up period was 4.6
years (median 2.7 years; range 0.1–22.3 years) for the
full cohort, representing a total of 384.5 patient-years of
follow-up. There were no hemorrhages in any cases fol-
lowing a complete resection. One of the 2 incompletely
resected lesions bled 7.5 years postoperatively; this lesion
was associated with an extensive venous telangiectasia
(Fig. 2). At the 9-year follow-up, the patient had an im-
proving left upper-extremity paresis and medically con-
trolled seizures.
Radiographically, there was 1 recurrence (1.2%) in
a child with multiple CMs; there were no recurrences in
completely resected single lesions. This recurrence repre-
sents an unusual case of a child with von Willebrand dis-
ease who also developed multiple other de novo lesions
(Fig. 3).
Symptomatically, 46 (96%) of 48 children were
seizure-free at the last follow-up. Seizure outcomes are
further summarized in Table 2. The operative permanent
complication rate was 5%, with complications occurring
only in patients with eloquent lesions (16% vs 0%, respec-
tively, of noneloquent CM). Complication rates did not
significantly differ for lesions associated with a DVA (p =
0.45), in patients with a family history of CM (p = 1.0), or
in those with radiation-induced lesions (p = 1.0).
Only 1 child (1.2%) developed a delayed de novo sei-
zure condition after resection of a temporal CM, now con-
trolled with antiepileptic medications. Six children devel-
oped new or worsening neurological deficits after surgery:
5 with frontal lesions had new or worsening hemiparesis,
and 1 with an occipital lesion had a new visual field deficit.
The deficits were permanent in 3 cases (all hemiparesis).
Including the 1 child with de novo seizures, the permanent
complication rate was 5% (4 of 83 patients).
Comparative Analysis to Nonoperative Cases
Compared with patients managed with observation
only, patients selected for surgery were older (mean age
11.8 vs 9.8 years, respectively, p = 0.03), were more likely
to have symptomatic lesions (p < 0.0001), and had larger
lesions (mean lesion size 2.2 vs 1.0 cm, respectively, p
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Lobar cavernous malformations
TABLE 1: Demographics, presentation modality, and CM characteristics in surgical and observed cohorts

Characteristic Surgical Cohort (n = 83) Observed Cohort (n = 98) p Value

mean patient age 11.8 9.8 0.03
male 57% (42/74)* 60% (59/98) 0.75
family history 14% (12/83) 23% (23/98) 0.14
prior radiation 8% (7/83) 14% (14/98) 0.25
presentation
  asymptomatic 5% (4/83) 61% (60/98) <0.0001
  seizures 58% (48/83) 20% (20/98) <0.0001
  hemorrhage 75% (62/83) 15% (15/98) <0.0001
location
  frontal 52% (43/83) 45% (44/98) 0.37
  temporal 25% (21/83) 20% (20/98) 0.48
  parietal 18% (15/83) 19% (19/98) 0.85
  occipital 5% (4/83) 15% (15/98) 0.03
eloquent 30% (25/83) 20% (20/98) 0.17
mean lesion size (cm) 2.2 1.0 <0.0001
associated DVA 13% (11/83) 13% (13/98) 1.0

* The denominator is 74 for the surgical cohort because sex for the first 9 patients was not readily available in this retrospective
review.

< 0.0001). A slightly greater percentage of nonoperative Discussion

lesions were occipital (p = 0.03). There were otherwise
no significant differences in sex, family history, prior ra-
diation, lesion eloquence, or the rate of associated DVAs
between the two cohorts (Table 1).
Although 20 patients in the nonoperative cohort
had medically controlled seizures, of 75 patients in the
nonoperative group who had not initially presented with
seizures, 5 patients developed new seizures (7%); in the
operative cohort, of 35 patients who did not have seizures
preoperatively, only 1 (3%) developed new seizures after
surgery. Over a total of 561.2 patient-years of follow-up for
98 cases of nonoperative CM, 5 hemorrhages occurred,
corresponding to an overall hemorrhage rate of 0.9% per
lesion-year. This rate was 0.5% per lesion-year for asymp-
tomatic CM, 2.3% per lesion-year for CM associated with
seizures, and 3.0% per lesion-year for hemorrhagic CM.
Over a total of 360.8 patient-years of follow-up for 78 cas-
es of completely resected CM, no hemorrhages occurred.
Cerebral CMs are clusters of dilated sinusoidal ves-
sels lined by endothelium without intervening neural
parenchyma.7,18,28,30 They are the most common cerebral
angiographically occult vascular malformation, with an
estimated prevalence ranging from 0.4% to 0.6% across
both adult and pediatric populations.3,7,18,28,30 The most
common location for intracranial CMs is lobar, defined as
supratentorial but not involving the thalamus/hypothala-
mus or basal ganglia.3,18,23,25,28,30 These lesions can serve as
a source of neurological morbidity as a result of seizures
and/or hemorrhage. It has been repeatedly demonstrated
that hemorrhagic CMs are more likely to rebleed,4,14,18,23
adding further neurological morbidity with time.
Although the management of CMs in eloquent or
deep locations, such as the basal ganglia or brainstem,
accounts for some of the most intriguing reports in the
literature,1,7–9,11,16,17,29 it is important to segregate their re-

Fig. 1. A: Sagittal T1-weighted MR image. This 12-year-old girl originally presented with a seizure and weakness from hemor-
rhage of her CM. She was taken to the operating room, where intraoperative mapping and stimulation confirmed the lesion to be
embedded entirely in the motor cortex without a corridor of access. B: The lesion was thus left in place. C: Axial T2-weighted
MR image. After 15 years of clinical and imaging follow-up, she has had no further events and her lesion has thrombosed.

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 B. A. Gross et al.

Fig. 2. A and B: Axial T2-weighted and T1-weighted post-Gd MR images. C: Axial MR image of a complex right frontal CM
in association with an extensive venous telangiectasia. This 3-year-old male presented with seizures from this CM. Partial resec-
tion was performed; however, the lesion rebled after surgery.

sults from those of managing lobar CM in order to more
clearly delineate the morbidity and long-term outcomes
in the more common lobar lesions. Surgical management
of lobar CMs has been described, although the prepon-
derance of the data comes from adult patients.6,8,12 Some
excellent pediatric series have been reported, although
larger cohorts that include current operative techniques,
recent imaging technologies, and lengthy follow-up are
more limited in number.2,5–7,11,12,20,24,25,30 This report pre­
sents a large surgical cohort of children exclusively with
lobar supratentorial CM, focusing on surgical results and
complications as well as long-term outcomes.
Descriptions of operative treatment of cerebral CMs
in children have existed for decades, with the earliest sur-
gical series reported by Pozzati et al. in 1980.27 As noted
then, the relatively safe access to supratentorial lobar
lesions marks them as appealing surgical targets, with
resection predicated on reducing seizure frequency and

Fig. 3. A: Axial T2-weighted MR image. This 2-year-old child with von Willebrand disease had undergone partial resection of
a left frontal CM at an outside institution. The lesion was then resected. B: Postoperative axial T2-weighted MR image. C:
Axial T2-weighted MR image at 6-year follow-up. Black arrow designates recurrent lesion, which was first seen at 1.5-year follow-
up. D: Axial T2-weighted MR image at 6-year follow-up. Black arrow indicates recurrent lesion. E: Axial T2-weighted MR
image at 6-year follow-up. Black arrow designates 2 de novo lesions.

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Lobar cavernous malformations
TABLE 2: Seizure outcomes stratified by Engel class

Engel Class Description Seizure Cohort (n = 48) Entire Cohort (n = 83)

I seizure free 46 (96%) 80 (96%)
II rare seizures 2 (4%) 2 (2%)
III improvement in seizure frequency 0 0
IV no improvement/worsening in seizures 0 1 (1%)

eliminating future hemorrhages and resultant neurologi-
cal deficits. However, a small risk of complications ex-
ists with any cranial surgery, and there is ongoing debate
regarding how best to find equipoise between the risk of
observation and the risk of surgery in this population.2,5,12
Our series serves to reinforce the findings from
larger adult series and smaller pediatric series that sug-
gest that operative management of lobar CMs can be
achieved with a high rate of complete resection. Extract-
ing patient data from previous reports (9 series with 189
patients) demonstrates consistent, exceedingly high com-
plete resection rates for these lesions (99% overall) (Table
3).2,5,10,12,13,15,19,27 Improved intraoperative navigation, in-
traoperative ultrasound, and now intraoperative MRI are
key components to achieving this statistic. Our 2 cases
of incomplete resection represent exceptional cases—one
with a lesion entirely embedded in motor cortex without a
safe corridor of access (Fig. 1) and another in association
with a complex venous telangiectasia (Fig. 2). These cases
underscore the need for careful assessment of operability
and risk, including the potential role for functional MRI
and diffusion tensor imaging, coupled with intraoperative
neurophysiological monitoring for select cases. In partic-
ular, subcortical lesions contained within eloquent cortex
or intimately associated with critical vascular structures
may be less amenable to complete resection.
Surgical treatment of lobar CMs has a low compli-
cation rate, with new permanent neurological complica-
tions at approximately 4% across pediatric series (Table
3).2,5,12 Of particular note is the marked increased risk for
complications (not unexpectedly) for lesions in eloquent
cortex. Only 2 patients (1%) across all reviewed series had
worsening seizures after treatment, while 94% of patients
TABLE 3: Compendium of extracted data from surgical series of pediatric CM detailing supratentorial lobar lesions
were seizure free at follow-up. In our own cohort, 96%
of patients presenting with seizures were seizure free at
follow-up, although none of our patients had intractable
seizures prior to surgery. Nevertheless, this high rate of
symptom resolution, coupled with low morbidity, sup-
ports the role of resection in this cohort.
One of the important aspects of our series is the rela-
tively long-term follow-up in a pediatric population (total
of 384.5 patient-years of follow-up; mean 4.6 years). Ra-
diographic cure rates for completely resected lesions were
excellent, with only 1 recurrence (1.2%) found in a child
who ultimately went on to develop multiple lesions, dem-
onstrating a likely underlying genetic propensity to grow
new CMs. Clinically, perioperative morbidity commonly
resolves and symptom relief is substantial and durable.
Risk Stratification and Case Selection
Attempts to risk-stratify cerebral CMs in grading
schemes analogous to those utilized for arteriovenous
malformations have been recently attempted;22 however,
the inclusion of lesions in all locations creates a focus on
the challenge of ganglionic, thalamic, and brainstem le-
sions. These CMs have a significantly greater associated
surgical risk,16,17,30 and including results of their treatment
among supratentorial lobar lesions potentially dilutes the
accuracy of predicting outcomes for specific locations. As
we demonstrate, a focus on supratentorial lobar lesions,
generally ideal surgical targets, reveals exceedingly high
resection and seizure control rates with very low morbid-
ity. However, our series also underscores the importance
of caution when considering lesions in eloquent cortex for
resection.
Based on our results, surgical risk stratification is

Seizure Outcome
No. of Permanent
Authors & Year Cases Full Resection Complications Engel I Engel II Engel III Engel IV
Acciari et al., 2009 35 35/35 3/35 29 5 1
Alexiou et al., 2009 14 14/14 0/14 11 3
Buckingham et al., 1989 7 0/7 7
Consales et al., 2010 18 18/18 0/18 18
Di Rocco et al., 1996 17 17/17 0/17 17
Fortuna et al., 1989 5 5/5 0/5 5
Herter et al., 1988 5 0/5 5
Pozzati et al., 1980 5 1/5 5
current series 83 81/83 4/83 80 2 1
pooled (%) 189 170/172 (99) 8/189 (4) 177 (94) 10 (5) 2 (1)

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exclusively predicated on lesion eloquence, particularly
if involving the motor cortex. Noneloquent symptomatic
lesions are ideal surgical targets, while eloquent lesions,
particularly if involving the motor cortex, may be con-
sidered for observation in the case of only 1 hemorrhagic
event or medically manageable seizures. Asymptomatic
nonhemorrhagic lesions are also generally observed. We
have not used radiosurgery as a treatment modality in any
of our pediatric patients since several patients referred to
us after prior radiosurgery have suffered marked brain
edema and even additional CM growth following radio-
surgery. Without proven efficacy in the management of
CM, particularly supratentorial lobar lesions, the known
high complication rates associated with general CM ra-
diosurgery contraindicates its usage.21,26,31
Summary
Our series supports the literature advocating a role
for the resection of lobar CMs in children. The high rate
of complete resection, low frequency of recurrence, and
small number of complications reported in other series is
further corroborated by our experience. The long follow-
up in our pediatric population suggests that the beneficial
effects of surgery are durable, information hopefully of
use to clinicians treating affected children. Factors such
as lesion size, hemorrhage at presentation, and specific
location in the hemispheres are intuitively based patient
treatment selection factors that are born out by this series.
The quantification of their effect in this patient popula-
tion will improve the ability of the neurosurgeon to make
an informed therapeutic decision in the pediatric patient
with a lobar CM.
Conclusions
The resection of symptomatic supratentorial lobar
CMs is a safe and effective procedure with high com-
plete resection rates, rewarding long-term seizure out-
comes, and low complication rates. Asymptomatic lesions
or those in eloquent cortex may be better candidates for
observation, although individualized assessment of risks
and benefits remains critical. Following complete exci-
sion of lobar CMs in the pediatric patient, there appears
to be both excellent symptom relief and durable radio-
graphic cure rates.
Disclosures
The authors report no conflict of interest concerning the mate-
rials or methods used in this study or the findings specified in this
paper.
Author contributions to the study and manuscript prepara-
tion include the following. Conception and design: Scott, Gross.
Acquisition of data: Scott, Gross, Goumnerova. Analysis and
interpretation of data: all authors. Drafting the article: Gross, Smith,
Scott. Critically revising the article: all authors. Reviewed submit-
ted version of manuscript: all authors. Approved the final version
of the manuscript on behalf of all authors: Scott. Statistical analy-
sis: Gross. Administrative/technical/material support: Scott, Smith,
Goumnerova, Proctor, Madsen. Study supervision: Scott.
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surg 16:385–391, 2002
Manuscript submitted March 14, 2013.
Accepted July 1, 2013.
Please include this information when citing this paper: pub-
lished online August 23, 2013; DOI: 10.3171/2013.7.PEDS13126.
Address correspondence to: R. Michael Scott, M.D., Department
of Neurological Surgery, Boston Children’s Hospital, 300 Long-
wood Ave., Boston, MA 02115. email: michael.scott@childrens.
harvard.edu.
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