Decompressive craniectomy in paediatric traumatic brain injury: a systematic review of current evidence

Abstract

Introduction
Paediatric traumatic brain injury (pTBI) is one of the most frequent neurological presentations encountered in emergency departments worldwide. Every year, more than 200,000 American children suffer pTBIs, many of which lead to long-term damage.

Objectives
We aim to review the existing evidence on the efficacy of the decompressive craniectomy (DC) in controlling intracranial pressure (ICP) and improving long-term outcomes in children with pTBI.

Methods
A comprehensive search of the MEDLINE and EMBASE databases led to the screening of 212 studies, 12 of which satisfied inclusion criteria. Data extracted included the number and ages of patients, Glasgow Coma Scale scores at presentation, treatment protocols and short- and long-term outcomes.

Results
Each of the nine studies including ICP as an outcome reported that it was successfully controlled by DC. The 6–12 month outcome scores of patients undergoing DC were positive, or superior to those of medically treated groups in nine of 11 studies. Mortality was compared in only two studies, and was lower in the DC group in both.Very few studies are currently available investigating short- and long-term outcomes in children with TBI undergoing DC.

Conclusion
The currently available evidence may support a beneficial role of DC in controlling ICP and improving long-term outcomes.

Full text

REVIEW PAPER
Decompressive craniectomy in paediatric traumatic brain
injury: a systematic review of current evidence
Maddalena Ardissino
1
&Alice Tang
1
&Elisabetta Muttoni
2
&Kevin Tsang
3
Received: 17 July 2018 /Accepted: 5 September 2018 /Published online: 13 September 2018
Abstract
Introduction Paediatric traumatic brain injury (pTBI) is one of the most frequent neurological presentations encountered in
emergency departments worldwide. Every year, more than 200,000 American children suffer pTBIs, many of which lead to
long-term damage.
Objectives We aim to review the existing evidence on the efficacy of the decompressive craniectomy (DC) in controlling
intracranial pressure (ICP) and improving long-term outcomes in children with pTBI.
Methods A comprehensive search of the MEDLINE and EMBASE databases led to the screening of 212 studies, 12 of which
satisfied inclusion criteria. Data extracted included the number and ages of patients, Glasgow Coma Scale scores at presentation,
treatment protocols and short- and long-term outcomes.
Results Each of the nine studies including ICP as an outcome reported that it was successfully controlled by DC. The 6–12 month
outcome scores of patients undergoing DC were positive, or superior to those of medically treated groups in nine of 11 studies.
Mortality was compared in only two studies, and was lower in the DC group in both.Very few studies are currently available
investigating short- and long-term outcomes in children with TBI undergoing DC.
Conclusion The currently available evidence may support a beneficial role of DC in controlling ICP and improving long-term
outcomes.
Keywords Paediatric traumatic brain injury, TBI .Decompressive craniectomy .Surgery .Intracranial pressure, ICP .Outcomes .
Management
Introduction
Background
Paediatric traumatic brain injury (pTBI) is one of the most
frequent neurological emergencies affecting children through-
out the world: ten million injuries lead to hospitalisation or
death every year [1]. In the USA alone, approximately
230,000 children suffer a TBI every year, and these lead
to even more severe and long-lasting neurological disabilities
than those occurring in adolescents or adults [2]. Despite this,
there is little evidence on which to base protocols for the man-
agement of pTBI as most of the treatment algorithms are
founded on experience gained in adults and adapted on the basis
of subtle differences in physiology and anatomy. However, since
the introduction of the 2012 guidelines, a number of studies have
been published concerning especially the surgical management
of adult and pTBI, the results of which can be considered for
future modifications of management protocols.
Aims
The aim of this review is to summarise and assess current
methods of surgically managing pTBI, concentrating on
theuseofdecompressivecraniectomy(DC)asameans
of reducing intracranial pressure (ICP) in the short term,
and improving rehabilitative outcomes in the long term.
Although it is still a subject considerable debate, it has
*Maddalena Ardissino
ma5713@imperial.ac.uk
1
Imperial College School of Medicine, Imperial College London,
London SW7 2AZ, UK
2
St. Helens and Knowsley Teaching Hospitals, Whiston Hospital
NHS Trust, Liverpool, UK
3
Department of Neurosurgery, Imperial College Healthcare NHS
Trust, Charing Cross Hospital, London, UK
Child's Nervous System (2019) 35:209–216
https://doi.org/10.1007/s00381-018-3977-5
#The Author(s) 2018
Content courtesy of Springer Nature, terms of use apply. Rights reserved.

been shown that DC effectively decreases ICP and its fluc-
tuations, and may increase cerebral perfusion pressure [3],
it has also been shown to be more economically effective
than medical management approaches such as the use of
barbiturate-induced coma [4]. However, its impact on the
clinical outcomes of TBI patients has yet to be fully
ascertained. A systematic review was carried out with the
aim of summarising the currently available evidence on the
effect of the DC on ICP reduction in the short term, and
rehabilitation outcomes in the long term.
This review considers the published evidence concerning
the short- and long-term outcomes of DC in children pTBI, as
well as the most important recent studies of its therapeutic role
in adult patients.
Methods
Literature search
This systematic review was made following the guidelines pro-
posed in the PRISMA statement [5]. The MEDLINE and
EMBASE databases were searched using the terms ‘(paediatric
traumatic brain injury) AND (decompressive craniectomy)’.
Only studies published in English were considered.
Of the 617 studies published up to October 2017, we se-
lected the randomised clinical trials (RCTs), case series or
two-arm studies that involved patients aged < 18 years, in-
cluded TBI patients who underwent DC to control ICP, and
measured long-term (> 4 weeks) outcomes. Individual case
reports were excluded, as were studies that did not provide
quantitative data or were designed to answer different ques-
tions (e.g. those investigating complications or outcome
predictions).
Twelve of the 212 screened studies satisfied our selection
criteria (one RCT, and 11 case series involving a total of 260
patients), and were read in full and analysed by all of the authors
of this review. The selection process is outlined in Fig. 1.
Data analysis
The extracted data included the names of the authors and
the year of study publication, the number and age ranges of
the patients involved, Glasgow Coma Scale (GCS) scores
at the time of presentation, treatment protocols, and short-
and long-term outcomes and mortality. This information
was summarised and reviewed by three reviewers indepen-
dently (MA, EM and AT), and further evaluated by the
senior author (KT). Two of the reviewers independently
assessed the quality of the data using the GRADE scoring
system of the British Medical Journal (BMJ) [6], and the
risk of bias was analysed using the Cochrane risk of bias
tool [7].
Results
Quality assessment
The only RCT that met the inclusion criteria [8] was judged to
be at risk of bias on the basis of the Cochrane risk of bias tool
because of incomplete information regarding the blinding of
the outcome measurer. A number of studies enrolled only a
few patients, and some did not fully describe outcomes, or
assess and control for confounding factors. Overall, the num-
ber of patients included in the 12 studies analysed is extremely
low, varying from 5 to 53 (median 17) per study. Table 1
shows the level of the quality of evidence for each study on
the basis of the BMJ’s GRADE scoring system, and the risk of
bias was assessed using RevMan [20].
Effect of DC on ICP
Of the 12 studies reviewed, nine included ICP as a short-term
outcome. All nine reported that ICP was successfully reduced
by DC and that the patients required fewer ICP control inter-
ventions. Two studies directly compared ICP control in pa-
tients receiving medical treatment (MT) and those undergoing
DC. Cho et al. [9] found an 80% reduction in ICP in DC
patients, which was greater than that observed in the MT
group (p< 0.05), and the RCT by Taylor et al. [8]alsofound
Fig. 1 PRISMA flowchart of study selection
210 Childs Nerv Syst (2019) 35:209–216
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Table 1 Quality of evidence assessment for the included studies
Author/year Design No. of patients/age Limitations Consistency Directness Other factors Quality
Cho 1995 [9] Case series n=17
2–12 months
No serious limitations No important inconsistency Direct None Moderate
Thomale 2010 [10] Case series (retrospective) n=53
<16 years
No serious limitations No important inconsistency Direct None Moderate
Taylor 2001 [8] RCT n=27
1–18 years
No serious limitations No important inconsistency Direct None High
Hejazi 2002 [11] Case series n=7
1–18 years
No serious limitations No important inconsistency Direct Few data Low
Figaji 2003 [12] Case series n=5
5–12 years
Limitations No important inconsistency Direct Few data Low
Ruf 2003 [13] Case series n=6
5–11 year s
No serious limitations No important inconsistency Direct None Moderate
Josan 2006 [14] Case series (retrospective) n=12
2–16 years
No serious limitations No important inconsistency Direct Limited number of participants Moderate
Kan 2006 [15] Case series n=51
4 months–14 years
No serious limitations No important inconsistency Direct Imprecise data Low
Rutigliano 2006 [16] Case series n=6
12–15 years
No serious limitations No important inconsistency Direct Limited number of participants Moderate
Skoglund 2006 [17] Case series (retrospective) n=19
7–16 years
No serious limitations No important inconsistency Some uncertainty about
directness
High risk of reporting bias Low
Jagannathan 2007 [18] Case series (retrospective) n=23
2–19 years
No serious limitations No important inconsistency Direct High risk of reporting bias Low
Guresir 2012 [19] Case series n=34
0–18 years
No serious limitations No important inconsistency Some uncertainty about
directness
Imprecise data Very low
Childs Nerv Syst (2019) 35:209–216 211
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Table 2 Characteristics and outcomes of included studies
Author /year Study No. of patients/age Treatment Outcome measures ICP results Functional outcome
Cho 1995 [9]Caseseriesn=17
2–12 months
ICP > 30 mmHg treated with DC,
n= 10 (unilateral or bilateral)
MT, n=7
ICP
Mortality
6-month 6-year COS
80% reduction with DC
(p< 0.05)
Mortality lower in DC group
(0/10 vs 3/7; p< 0.05)
Better 6-month 6-year COS in
DC group (p< 0.05)
Hearing preservation higher in
DC group (p< 0.05)
Thomale 2010 [10]Caseseries
(retrospective)
n=53
<16 years
Median GCS = 6.5 in
DC group and 3 in
MT group
Severe TBI patients presenting at
the author’s centre, 14 DC
and39MT
ICU stay
ICP
GOS
ICP control achieved in all
DC patients. No report
on ICP in MT patients
No significant difference in
12-month and long-term GOS
Taylor 2001 [8] Retrospective
controlled trial
n=27
1–18 years
If high ICP, randomised to early
DC (bitemporal craniectomy)
or MT
ICP
CPP
ICU stay
6-month GOS
Better control with DC
(p= 0.057)
6-month GOS ‘favourable’in
14% of MT group vs 54% of
DC group (p= 0.048)
Hejazi 2002 [11]Caseseries n=7
1–18 years
DC if herniation or decorticate
posturing (unilateral DC)
5-week GCS N/A All had GCS 15 after 5 weeks
Figaji 2003 [12]Caseseriesn=5
5–12 years
Mean GCS 4.6 (3–9)
DC if GCS < 8 (unilateral with
duraplasty floating flap)
ICPGOS FullICPcontrolin2and
moderate reduction of
ICP in 2
All patients had GOS 4–5 at time
of follow-up (14–40 months)
Ruf 2003 [13]Caseseriesn=6
5–11 years
DC performed in patients with
ICP > 20 mmHg for > 30 min
ICP
6-month survival and
6-month neuro-logical
follow-up
ICP normalised immediately
in all cases
4 had no disability and 2 mild/
moderate disability
Josan 2006 [14]Caseseries
(retrospective)
n=12
Mean GCS 6.8
6DC,6MT
DC or MT in patients with refractory
high ICP post-TBI
Mean time between TBI and
DC 7 h
ICP
12-month GOS
Mean ICP after intervention
12.33 mmHg
100% survival in DC group,
66% survival in MT group
6 months, 100% favourable
GOS in DC group vs 50%
in MT group
Early intervention (may improve
outcome)
Kan 2006 [15]Caseseriesn=51
4 months −14 years
Mean GCS 4.6
DCs performed at the author’s
institution between 1996
and 2005
± mass lesion evacuation
ICP
Mortality
KOSCHI
69.4% had normal ICP
after surgical intervention
31% died
Mortality highest (5/6) in patients
who underwent DC for ICP
alone (no mass lesion)
Mean follow-up KOSCHI 4.5
Rutigliano 2006 [16]Caseseries n=6
12–15 years
DC if refractory high ICP ICP
FIM
ICP normalised in 5/6 6/6 had FIM indicating independence
or minimal assistance at discharge
Skoglund 2006 [17]Caseseries n=19
7–16 years
DC if GCS deterioration, herniation
and refractory ICP
12-month GOS N/A 3 patients GOS 5
1patient GOS 4
212 Childs Nerv Syst (2019) 35:209–216
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that DC led to better ICP control than MT, although the dif-
ference was not statistically significant (p=0.057).The rates
of ICP control achieved in all of the other studies [10,12–16,
18] ranged from 69.4 to 100% (Table 2).
Effect of DC on mortality
Only two of the studies compared mortality among the pa-
tients undergoing DC and those receiving MT. Cho et al. [9]
found that mortality was significantly lower in the patients
who underwent DC (0/10 vs 3/7; p< 0.05), and Josan et al.
[14] recorded higher survival rates (100% vs 66%) (Table 2).
Effect of DC on long-term outcomes
The method of assessing long-term outcomes varied: the most
widely used scoring system was the original Glasgow
Outcomes Scale (GOS) or the Extended Glasgow Outcomes
Scale (GOS-E), and several studies included other systems
(the Functional Independence Measure, Children’s Outcome
Score, King’s Outcome Scale for Closed Head Injury and
quality of life scales). These systems generally assess func-
tional ability and independence, return to school and perfor-
mance in doing everyday activities but, given their heteroge-
neity, we qualitatively compared the results by dividing them
into positive or negative functional outcomes. Complete re-
covery, or a mild disability that does not interfere with inde-
pendence or activities, was regarded as a positive outcome,
and severe disability, dependency, vegetative state and mor-
tality as negative outcomes.
Ten studies reported positive outcomes in the patients who
underwent DC [8,9,11–19]. Four directly compared the
follow-up GOS scores of the patients who underwent DC with
those of the patients receiving MT [8–10,14](Table2). Cho
et al. [9] found that the scores assigned between 6 months and
6 years after a TBI were significantly better in the patients who
underwent DC (p< 0.05). This is in line with the results of the
retrospective study of Josan et al [14]: 6 months after their
TBIs, all of the DC patients were assigned a favourable
GOS score as against 50% of those who received MT.
Taylor et al. [8] also found that the DC group had a higher
incidence of favourable GOS scores after 6 months (54% vs
14% in the MT group), but Thomale et al. [10]didnotfindany
significant difference in long-term GOS scores between the
two groups.
Discussion
The use of DC to treat high ICP in paediatric and adult TBI
patients has long been a subject of debate. The recommenda-
tions for surgery in children are even less clear than those
included in the adult guidelines because of the severe lack of
Tab le 2 (continued)
Author /year Study No. of patients/age Treatment Outcome measures ICP results Functional outcome
1patientGOS3
1 patient died
Jagannathan 2007 [18] Case series
(retrospective)
n=23
2–19 years
DCs performed at the centre
between 1995 and 2006
ICP
GOS
Likert QOL scale
83% ICP controlled
with DC
7died
83% of the survivors returned
to school
Mean follow-up GOS 4.5, median 5
Guresir 2012 [19] Case series n=34(23TBI)
0–18 years
DC performed in 23 TBI patients,
2 SAH, 3 ICH, 5 infarction and
3other
Modified Rankin Score
(favourable 0–2)
Return to school
N/A Favourable outcome in 40% of
TBI patients
30% did not return to school due to
disability
9th grade, 1
10th grade, 5
13th grade, 1
DC decompressive craniectomy, MT medical therapy, ICP intracranial pressure, ICU intensive care unit, GOS Glasgow Outcome Score, COS Children’s Outcome Score, CPP cerebral perfusion pressure,
FIM Functional Independence Measure, QOL quality of life, KOSCHI King’s Outcome Scale for Closed Head Injury, SAH sub-arachnoid haemorrhage, ICH intra-cerebral haemorrhage
Childs Nerv Syst (2019) 35:209–216 213
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clinical evidence [21]. As highlighted in this review, it has
been consistently found that ICP is well controlled by DC in
children with TBI in the short term, but the correlation be-
tween successful ICP control and long-term clinical outcomes
is more questionable. Furthermore, the existing evidence is
derived from studies with high risk of bias, and containing
low patient numbers. The current guidelines [21]recommend
DC when performed together with other surgical procedures
such as haemorrhage evacuation, or if there is strong suspicion
of herniation, but its use as a stand-alone procedure to relieve
ICP in patients without herniation is limited to those with
intracranial hypertension (> 25 mmHg) showing signs of neu-
rological deterioration, or high ICP refractory to optimal MT.
The findings of the retrospective case series and the RCT
reviewed generally indicate that DC has a positive effect in
controlling ICP, though the quality of evidence is generally
low. Hejazi’s[11] study reported a full recovery in five out of
six patients who underwent the procedure, Figaji et al. [12]
similarly reported notable improvements in neurological func-
tion in a cohort of patients on whom DC was performed fol-
lowing neurological deterioration, and similar findings were
reported by Ruf et al. [13] and others. The only RCT was
conducted by Taylor et al. [8], who compared the outcomes
of DC and MT in respectively 27 children with refractory an
ICP of > 30 mmHg and found that they were much worse in
the children receiving MT. However, only two studies com-
pare ICP control directly between patients receiving MT and
those undergoing DC. Mortality was only analysed in two
studies with low patient numbers [9,14], of 12 and 17 respec-
tively. More studies looked at long-term outcomes; however,
outcome scales used to compare these were variable; it is
therefore hard to draw general conclusions from them.
Although adult studies do not provide direct evidence
concerning the paediatricuse of DC, it is important to consider
Table 3 Characteristics and outcomes of recent important studies in the adult population
Author/year Study type no.
Patients
Age
Treatment Outcome
measures
Results, outcomes Study quality
and bias
Cooper
2011 [22]
Randomised
clinical trial
n=155
Patients with ICP
> 20 mmHg
for > 15 min
Randomly allocated
to DC or MT
ICP
6-month GOS-E
ICP
DC group had fewer hours with
high ICP than MT group
(p<0.001)
DC group had fewer days in ICU
DC group had fewer ICP control
interventions
Outcomes
DC group had worse GOS-E
(OR 1.84, CI 1.05–3.24;
p=0.03)
DC group at greater risk of
unfavourable outcomes (death,
severe disability and vegetative
state), OR 2.21, CI 1.14–4.26;
p=0.02
ICP threshold does not
reflect clinical
guidelines for DC
Mismatch in severity of
TBI between DC and
MT group
Timofeev
2006 [25]
Retrospective
observational
study
n=49
Age 9–67 years
DC, bilateral or unilateral
in patients with
persistently high ICP
6-month GOS
and SF-36 QOL
questionnaire
Outcomes at 6 months, 30 (61.2%)
had good outcomes, 10 (20.4%)
had severe disability and
9 (18.4%) died
No comparison with
untreated patients
No randomisation or
control for
confounders
Hutchinson
2016 [24]
Randomised
clinical trial
n=408
Age 10–65 years
Refractory ICP > 25 mmHg
Randomly allocated to
DC or MT
Mortality
6-month GOS
ICP
DC group had fewer hours with
high ICP than MT group
(p<0.001)
Outcomes
DC group had lower mortality
rate
MT group had lower severe
disability rate
Rates of good recovery and
moderate disability were the
same
DC decompressive craniectomy, MT medical therapy, ICP intracranial pressure, GOS Glasgow Outcome Score
214 Childs Nerv Syst (2019) 35:209–216
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their results as supplementary information not least because
there are more high-quality studies referring to adults. The
DECRA study (decompressive craniectomy in diffuse trau-
matic brain injury) found that patients with refractory intracra-
nial hypertension (> 20 mmHg) who underwent DC required
shorter ICU stays and fewer interventions to control ICP, but
experienced worse long-term clinical outcomes [22].
However, questions have been raised about the potential bias
of this study mainly because the randomisation process led to
unbalanced cohorts with discrepancies in the severity of TBI
(greater in the DC group) and GCS scores upon admission;
furthermore, it has been pointed out that the definition of re-
fractory raised ICP (> 20 mmHg for > 15 min) does not reflect
clinical practice [23]. On the other hand, the RESCUE-ICP
(trial of decompressive craniectomy for traumatic intracranial
hypertension) trial found that the use of DC in adult patients
with an ICP of > 25 mmHg was associated with fewer deaths
and cases of severe disability than medical management, al-
though it was also associated with a higher incidence of pa-
tients experiencing a vegetative state. There was no difference
in the incidence of ‘good outcomes’between the two groups
[24]. Table 3summarises the results of the most important
recent studies of adults undergoing DC.
Conclusions
This review aims to summarise the presently available evi-
dence in the treatment of paediatric traumatic brain injury
using decompressive craniectomy versus medical treatment.
The evidence considered in this review indicates a possible
benefit in use of DC in patients with pTBI for reducing high
ICP (> 25 mmHg) that is refractory to medical treatment.
However, the quality of evidence remains extremely low,
and there is very little evidence from RCTs to indicate whether
this correlates with long-term benefits in the paediatric popu-
lation. The findings of retrospective studies generally indicate
a beneficial effect with improved long-term neurological re-
covery, but they are sometimes inconsistent and their quality
varies because of differences in the patient age, the criteria for
and timing of surgery, injury factors, rating scales used and the
use of concomitant medical treatment. Overall, though avail-
able evidence unanimously indicates a short-term benefit in
using DC to reduce ICP and mortality, and possible long-term
rehabilitative improvement, the assessment of evidence qual-
ity carried out highlights the lack of evidence in the field, and
further high-quality studies on larger patient numbers are cer-
tainly required.
Compliance with ethical standards
Conflict of interest On behalf of all the authors, the corresponding au-
thor states that there is no conflict of interest.
Open Access This article is distributed under the terms of the Creative
Commons Attribution 4.0 International License (http://
creativecommons.org/licenses/by/4.0/), which permits unrestricted use,
distribution, and reproduction in any medium, provided you give appro-
priate credit to the original author(s) and the source, provide a link to the
Creative Commons license, and indicate if changes were made.
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