Childs Nerv Syst (2006) 22: 368374

DOI 10.1007/s00381-005-1233-2 ORIGINAL PAPER

Juan F. Martnez-Lage
Antonio M. Ruz-Espejo
Amparo Gilabert
Miguel A. Prez-Espejo
Encarna Guilln-Navarro
Received: 21 September 2004
Published online: 20 October 2005
# Springer-Verlag 2005
Positional skull deformities in children:
skull deformation without synostosis
Abstract Background: Patients with
craniosynostosis are readily diag-
nosed by clinical and neuroimaging
findings. Surgical treatment is indi-
cated for preventing neurological
deficits and for correcting esthetically
unacceptable head deformities. In
recent years, we have witnessed a
progressive number of neurosurgical
consultations for abnormal head
shapes unrelated to premature fusion
of the cranial sutures, especially of
positional plagiocephaly. There have
been descriptions of abnormalities in
the cerebrospinal fluid (CSF) spaces
in children with craniosynostosis.
Objectives: The aim of the present
study was to investigate the role of the
changes of the CSF spaces in the
development of positional skull de-
formities in children. Patients and
methods: The authors reviewed de-
mographic, clinical, and neuroimag-
ing data of 23 patients assessed for
some form of nonsynostotic skull
deformity (group A). The results were
compared with those of a simulta-
neous group of nine infants diagnosed
with benign extracerebral collections
of fluid (group B). Results: The
study group was composed of 11 boys
and 12 girls, aged 3 years or younger.
Seventeen children had plagiocepha-
ly, four scaphocephaly, and two
brachycephaly. Sixteen children
(15 with plagiocephaly and 1 with
brachycephaly) exhibited enlarged
subarachnoid CSF spaces. In group B,
the boy/girl ratio was of 7:2. Infants
in group A presented at an older age
(mean 12.7 months) than group B
(mean age at presentation of 7.17
months). Children with benign extra-
cerebral fluid collections were born
with a head circumference (HC)
greater than infants with positional
skull deformities (p=0.005). The per-
centile of the childrens HC at con-
sultation was also larger for children
of group B (p=0.03). The form of
clinical presentation differed between
the two groups. Most infants of group
Awere seen because of a type of head
deformity, and children in group B
were studied for macrocephaly. Long-
term follow-up assessment showed
better outcomes for patients in group
B than for children of group A in
regard to regression of initial symp-
toms (p=0.03). Conclusions: Most
positional head deformities appear to
be related with the childrens posi-
tioning for sleeping. We have not
confirmed macrocephaly as a contrib-
uting factor for positional deformities.
The distribution of extracerebral
CSF and the presence of abnormal
collections of fluid in children with
positional head deformities do not
seem to be related with the findings of
pericerebral CSF encountered in
children with benign extracerebral
collections of fluid. In our view, brain
pulsations, transmitted to these accu-
mulations of CSF, play an important
part in the development of the infants
skull deformities (p=0.02). The find-
ings of enlarged CSF spaces in
children with nonsynostotic skull
deformation constituted an age-
related event, as these collections
tended to disappear as the children
grew older (p=0.04).
Keywords Benign pericerebral CSF
collections
.
Craniosynostosis
.
Deformational plagiocephaly
.
Positional plagiocephaly
.
Positional
skull deformity
J. F. Martnez-Lage (*)
.
A. M. Ruz-Espejo
.
M. A. Prez-Espejo
Regional Service of Neurosurgery,
Virgen de la Arrixaca
University Hospital,
30120 Murcia, Spain
e-mail: juanf.martinezlage@carm.es
Tel.: +34-968-369489
Fax: +34-968-369678
J. F. Martnez-Lage
.
M. A. Prez-Espejo
.
E. Guilln-Navarro
Laboratorio de Neurologa y
Neurociruga Experimental
(Grupo NYNE), School of Medicine,
University of Murcia,
Murcia, Spain
A. Gilabert
Unit of Pediatric Radiology,
Virgen de la Arrixaca
University Hospital,
30120 Murcia, Spain
E. Guilln-Navarro
Unit of Clinical Genetics,
Service of Pediatrics,
Virgen de la Arrixaca
University Hospital,
30120 Murcia, Spain
Introduction
Surgery for craniosynostosis has evolved rapidly over the
last decades. Earlier recognition of head deformities has led
to an increase in the referrals to neurosurgical and cranio-
facial centers seeking treatment [8, 9, 12, 14]. The es-
timated incidence for all forms of craniosynostosis is of
approximately 1 per 1,000 live births. The sagittal suture is
the most frequently affected, accounting for 60% of cases,
while the lambdoid suture is rarely involved. The recent
increase in referrals of children with occipital plagiocephaly
has been associated with a change of the infants position in
bed following the recommendations of the American Acad-
emy of Pediatrics for preventing the sudden infant death
syndrome [1, 8, 9, 12, 14]. Several papers have dealt with
the complications arising from the supine position for
sleeping in children [14]. The incidence of synostotic pos-
terior plagiocephaly, due to the premature closure of the
lambdoid suture, has been estimated in 1 per 1,000 live
births, while the incidence of positional occipital plagio-
cephaly has been reported to be as high as 1 in 300 live
births [12, 14]. The back to sleep campaign has been
adopted in many countries, including Spain [8]. However,
the supine position alone seems to be an unsatisfactory ex-
planation for the observed increase in the incidence of
posterior plagiocephaly [6]. Some authors have ascribed
some prenatal factors such as fetal lie, multiple pregnancies,
and uterine constraint to the occurrence of positional
plagiocephaly [6]. These antenatal factors imply that head
deformity must be present at birth [3, 6, 9, 10]. Other
researchers claim that positional plagiocephaly must be
attributed to postnatal events, such as changes in the infants
position in bed due to macrocephaly, torticollis, spinal ab-
normalities, and even to the situation of the crib in regard
to the parents bed [6, 12, 14].
From the year 1992, we are conducting a prospective
study on the brain and the cerebrospinal fluid (CSF) spaces
in cases of true craniosynostosis. Our preliminary results
indicate that most forms of craniosynostosis are associated
with abnormal CSF, extracerebral accumulations of CSF,
and with abnormal CSF hydrodynamics [10]. Similar to the
findings of Sawin et al. [15] in positional plagiocephaly, we
observed collections of enlarged extracerebral fluid spaces
not only in cases of positional plagiocephaly, but also in
other instances of position-related head deformities. We
hypothesize that the existence of abnormal collections of
CSF in nonsynostotic skull distortion constitutes a main
etiologic factor for position-induced skull deformation.
Anomalous CSF collections might cause an increase in the
elasticity of the infants calvarial bones that, together with
the childrens position in bed, would contribute to the de-
velopment of the diverse forms of acquired skull deformity.
Our report was conducted for ascertaining the part played
by extracerebral CSF redistribution in the development of
positional skull deformities.
Patients and methods
We investigated the medical records of 110 consecutive
children seen at our hospital for evaluation of an abnormal
skull shape. Eighty patients were diagnosed with true cra-
niosynostosis, 65 of them were submitted to surgery. Twen-
ty patients were excluded from the survey because they
refused surgery (n=15) or were treated in other centers
(n=5). The study group (group A) comprised 23 children
with head deformities other than true craniosynostosis. We
investigated demographic and clinical features of these
children, including duration and incidences of pregnancy,
type of delivery, condition of the children at birth [weight,
Apgar score at 1 min, head circumference (HC), age at
consultation, and type of main complaint (head deformity,
developmental delay, or others], and HC and neurological
examination at presentation. As the childrens age varied at
the time of consultation, we analyzed their HC by means of
the corresponding percentile instead of by annotating their
HC absolute value, which allowed the comparisons among
cases. Group B comprised nine children assessed for mac-
rocephaly and who were diagnosed after work-up with
benign extracerebral collections of CSF (external hydro-
cephalus). Demographic and clinical data of this group were
recorded in a similar way to those of group A.
Group Awas studied by skull radiographs (n=19), com-
puterized tomography (CT) head scan (n=18), 3-dimension-
al CT (3D CT) (n=12), and magnetic resonance imaging
(MRI) (n=3). Infants of group B were studied mainly with
MRI (n=7) or CT (n=2). 3D CT of the head was performed
in only one case.
The results of the neuroimaging studies, the coexistence
of other cerebral and extracerebral anomalies, the treat-
ments given, and the outcomes were also evaluated for both
groups. The outcomes were obtained from the outpatient
clinical records and completed, when necessary, by a tele-
phone call or a revision visit. The data thus collected were
introduced in an Excel spreadsheet and evaluated with the
program SPSS for Windows (version 10.0.1) for statistical
significance.
Results
Demographic and clinical data
Group AThe study group consisted of 23 children, 11 boys
and 12 girls, whose ages at presentation ranged from1 to 36
months (median 8) (Fig. 1). The duration of their gestations
averaged 38.5 weeks (range 29 to 42). Fourteen pregnancies
were uneventful, two coursed with arterial hypertension,
two with diabetes mellitus, and two with an abnormal fetal
lie (transverse or breech). The children were delivered by
vaginal route in 17 instances and by a C-section in 6. The
Apgar score was 8 or higher in 21 children and lower than
369
8 in 2. Birth weight ranged from 1,125 to 4,100 g and
averaged at 3,110 g. Mean HC at birth measured 34 cm
(range 3036 cm). In two children, there was a family
history of craniosynostosis. Most children (n=21) were
brought to consultation because of the head deformity. In
addition to the misshapen head, two children were noted to
have some developmental delay. In two children, the head
deformity was noted incidentally, one after a severe cra-
niocerebral trauma and another during the assessment of an
occipital intraosseous dermoid ipsilateral to the plagio-
cephaly.
The head deformity was noted either at birth (n=13) or
at variable intervals from 1 to 27 months of age (n=10).
The cranial shape observed at examination was plagio-
cephaly (n=16, including 1 case of anterior plagiocephaly),
brachycephaly (bilateral occipital, n=4), and scaphoceph-
aly (n=3). Table 1 shows the type of skull deformity. In 18
children, the results of the neurological examination were
normal; in two, there was a mild developmental delay; and
in the remaining, the findings of the examination corre-
sponded to that of the craniocerebral trauma suffered by
the child. In the cases of plagiocephaly, the right side was
involved in seven occasions, and the left was involved in
nine. The deformity was bilateral in four children, three
presented with a flat occiput, and one with a plane forehead.
There was an antecedent of torticollis in three children,
of birth-related fracture of clavicle in one, and of multiple
vertebral anomalies in another, factors that have been
implicated in the sustained position of the childrens head
in bed. The percentiles of the childrens HC at the time of
consultation ranged from <2 to 80%, being the mean per-
centile of 40 (Fig. 2). No child showed macrocephaly
features. Seven children had associated, cerebral, or ex-
tracerebral malformations that are listed in Table 2.
Group B The group with benign pericerebral collections of
CSF consisted of seven boys and two girls, aged 1 to 14
months (mean 7.2) at the time of consultation (Fig. 1).
Group B infants pregnancies had been of normal duration
(mean 38.7 weeks), and their delivery was vaginal in seven,
while two were born by C-section. There were some in-
cidences in two gestations, one case of arterial hyperten-
sion and another that coursed with significant autoimmune
anemia. These infants mean birth weight was of 3,638 g
and ranged from 3,000 to 4,400. Their average HC at birth
was of 36.5 cm (range 3538.5). Their Apgar score at
1 min ranged from 9 to 10 (mean 9.5). There were three
instances with associated malformations: one with cerebral
anomalies, another with severe congenital cardiopathy, and
the third exhibited features of Sotos syndrome. There was a
child with a family history of macrocephaly. Eight children
0
1
2
3
4
1 2 3 4 5 6 7 8 9 10 11 12 14 18 22 24 27 29 30 36
Age (mos)
No.
Group A
Group B
Fig. 1 Patients ages at consultation (group A, gray; group B black)
Table 1 Type of skull deformity in 23 children with nonsynostotic
skull deformity and in 9 with benign extracerebral collections of
CSF
Skull shape Group A Group B
Plagiocephaly, posterior 15
Plagiocephaly, anterior 1
Brachycephaly 4
Scaphocephaly 3 1
Normal but macrocephalic 8
0
1
2
3
4
5
6
7
8
9
10
2 5 10 25 40 50 60 75 80 98 110
percentile
No.
Group B
Group A
Fig. 2 Percentile of head circumference at the time of consultation
in 23 children with positional skull deformation (group A) and in 9
infants with pericerebral CSF collections (group B)
Table 2 Associated anomalies in 23 children with nonsynostotic
skull deformity and in 9 with benign extracerebral collections of
CSF
Anomaly Group A Group B
Cardiac 2 1
Renal 2
Cerebral 1 1
Skeletal 1
Dysmorphic features 1 1
Cataract 1
Mild delay 2
370
were seen for assessment of macrocephaly and showed a
rather rounded and broad head (brachycephaly), and the
remaining was assessed for dolicocephaly. The percentiles
of the HC in the children of group B at consultation (Fig. 2)
ranged from 75 to >100 (mean 10011.38). No child was
found to have an abnormal neurological examination.
Table 3 compares the data for the groups A and B.
Neuroimaging studies
Group A Skull radiographs, performed for 19 children,
identified an abnormal head shape and showed the patency
of the cranial sutures. CT scan (Figs. 3 and 4) and MRI
(Fig. 5) showed striking anterior collections of fluid and
enlarged sylvian fissures in 14 cases of positional pla-
giocephaly and in one of brachycephaly. The existence of
these enlarged extracerebral collections of CSF was the
only factor that was statistically significant in regard to the
type of head shape (p=0.02). The ventricles were normal
in 17 children. There was ventricular asymmetry in one
child and mild ventricular enlargement in another. The
study with a 3D CT head scan has recently been introduced
in our hospital as part of the evaluation of children with
skull deformities and was performed for 12 patients. A 3D
CT scan was very helpful in children whose plagiocephaly
had been noted early after birth, to rule out true lambdoid
synostosis, as neonates with posterior plagiocephaly are at
increased risk for having true craniosynostosis (Fig. 6).
Group B Plain skull radiographs were obtained in 5/9
children that demonstrated an enlarged head size. CT
(n=5) and MRI (n=7) showed dilated CSF pericerebral
spaces in all children, which constitutes the main diag-
nostic criteria for patients in this group. There were two
instances of mildly enlarged ventricles, although they were
considered as being in the upper limit of normal.
Treatment and outcomes
The three children of group A with torticollis and the one
with the fractured clavicle were submitted to rehabilitation.
The parents of the remaining children of this group were
instructed to introduce changes in the childrens position-
Fig. 3 CT scan of a 2-month-old boy with left positional
plagiocephaly. Note the appearance of occipital collections of CSF
seen in early stages. The right lateral ventricle is diminished, and
there is also effacement of the ipsilateral occipital horn
Table 3 Demographic, clinical,
and outcome data in infants with
positional skull deformation and
in infants with benign pericer-
ebral CSF collections
HC Head circumference, NA not
applicable
Feature Group p value
Group A, positional skull
deformation
Group B, benign
extracerebral
fluid collection
No. of infants 23 9
Sex (boy/girl) 11:12 7:2
Age (months) 12.710.53 7.174.39 0.01
Duration pregnancy (months) 38.302.69 38.671.12
Delivery (vaginal/C-section) 17:6 7:2
Apgar at 1 min 8.610.84 9.560.53
Birth weight (g) 3,108631 3,638420
HC at birth (cm) 341.56 36.51.3 0.005
HC at consultation
(percentile)
38.225.09 10011.38 0.03
Clinical presentation
(deformity/others)
21:2 1:8 0.003
Plagiocephaly/other shapes 16:7 0:9 NA
Age symptoms noted 3.266.98 2.892.52
Increased/normal cisterns 15:4 9:0 NA
Result equal/better 10:13 0:9 0.03
Follow-up (months) 19.619.45 32.2215.08
371
ing in bed, especially in children younger than 6 months
and in those with developmental delay. We did not use
helmets or other types of orthosis. Seven children with
positional head deformity seen after the age of 12 months
or later were not considered susceptible of treatment. Sur-
gery was refused by the parents in two instances of severe
deformity as they considered that their childrens deformity
was unimportant, after that, the possibility of brain com-
pression was ruled out. Several children are being observed
in the outpatient clinic. The time of follow-up for group A
ranged from 3 to 36 months (mean 19 months). Regarding
outcomes, 10 children were considered as being un-
changed, and 13 had clearly improved their skull deformity.
Almost all patients pertaining to group B did not require
treatment and were observed for a mean period of 32
months (range 6 to 54). Interestingly, one child of this
group later on developed sagittal craniosynostosis and was
submitted to surgery.
Discussion
Synostotic vs positional skull deformities
Several authors have reported a recent increase in the
number of referrals to Neurosurgical departments and Cra-
nio-facial units, especially for the so-called nonsynostotic
positional posterior plagiocephaly [3, 9, 12, 14]. The in-
crease has been attributed to the supine sleeping position
for infants recommended by the American Academy of
Pediatrics [1], a measure that has proved to decrease the
incidence of sudden infant death syndrome. These rec-
ommendations were also followed in several countries [8].
Several authors have commented on the difficulties en-
countered for differentiating positional plagiocephaly from
lambdoid synostosis [6, 7, 13].
In positional occipital flattening, the head shape reminds
that of a parallelogram. There is unilateral flattening of the
Fig. 4 CT corresponding to a 4-month-old boy with right de-
formational plagiocephaly. Note the enlarged subarachnoid spaces
and dilated left sylvian fissure
Fig. 5 MRI of a 10-month-old
girl, with spinal malformation,
depicting a in the axial cut, left
occipital flattening, collections
of CSF over the convexity, and
in the Sylvian fissure and
b enlargement of CSF spaces
over the convexities and dilated
sylvian fissure and basal cisterns
on the coronal view
Fig. 6 3D CT scan of a child with right posterior occipital flattening
depicting patent sutures
372
occipital region, with ipsilateral frontal and parietal boss-
ing, being the ear ipsilateral to the flattened occiput dis-
placed anteriorly [7, 12, 14]. Usually, the head is normal at
birth, and the deformity appears after a few weeks or
months. The head distortion does not progress, and, on the
contrary, it usually improves after a few months. Some
children require treatment that can be positional, orthotic,
or even surgical [3, 11, 13, 14]. A picture similar to that of
positional plagiocephaly has been reported in relation to
the sagittal suture and has been designated sticky sagittal
suture. In deformational scaphocephaly, there is no anatom-
ical fusion, but the sticky sagittal suture may result in func-
tional craniosynostosis [2].
In true lambdoid synostosis, the deformity is usually
present at birth, tends to advance, and, in most cases,
requires surgery. In synostotic plagiocephaly, there is con-
tralateral frontal bossing, and the ear is displaced back-
wards and inferiorly when compared with the contralateral
ear [6, 13]. In children with plagiocephaly noticed at (or
soon after) birth, there always exist concerns on the
possibility of being faced with true lambdoid synostosis. It
is in these cases where we found very useful to perform a
head 3D CT that permits to distinguish a deformational
from a synostotic deformity. With the same objectives, we
applied this technique in children of group A that presented
with bilateral occipital flattening.
Etiology of deformational abnormal skull shapes
The mechanisms for explaining abnormal suture growth
remain poorly understood. The cranial vault develops as a
result of the interaction between the brain, dura mater, su-
tures, and skull bones. Obviously, the brain push is re-
quired for maintaining skull growth [10]. An excessive and
prolonged force applied to a part of an infants head over a
long period may deform the skull [7, 14]. Both prenatal and
postnatal factors have been reported to explain the head
distortion. Some conditions may predispose a child to
suffer a positional skull deformity [14]. Oligohydramnios,
multiple pregnancies, abnormal fetal lie, and diseases of the
pelvis or of the uterus may produce antenatal constraint of
the fetus [12, 14]. In deformational plagiocephaly, a right to
left preference of 2:1 has been observed, which probably
reflects the predominance of fetal lie [12, 14]. Some
external causes have also been noted as contributing factors
for positional head deformities such as congenital muscular
torticollis, fracture of the clavicle, severe spinal deformi-
ties, and brain injuries or developmental anomalies. In
these cases the infant tends to lean its head repeatedly on a
certain side. These exogenous factors were also present in
our series: three neonates had torticollis, one a fractured
clavicle, another a severe spinal deformity, and two had
developmental brain diseases. Macrocephaly has been also
reported as a contributing factor due to larger weight of the
childrens head. However, in our series, no child in group A
presented with macrocephaly. On the contrary, a large head
was, by definition, found in all the children of group B.
Most authors report a male predominance in cases of
positional plagiocephaly, that was not confirmed in our
study. In our survey, children with benign pericerebral ac-
cumulations of CSF were seen earlier than patients with
positional cranial deformities (p=0.01). We found no sta-
tistical relationship between the childrens gestations, de-
livery or condition at birth, and positional skull deformities.
However, children of group Ain our study had a smaller HC
at birth than those of group B(p=0.005). HCat consultation,
recorded as percentile, was also lower in patients with
positional head deformities (p=0.03), a fact that is in con-
tradiction with the reported high incidence of macrocephaly
in positional occipital flattening [14, 15].
CSF spaces in synostotic and nonsynostotic
skull deformities
In 1982, Carmel et al. [4] analyzed the CT findings of 24
children with craniosynostosis and reported prominent
collections of CSF at the anterior and posterior poles of the
calvarium and in the interhemispheric fissure in cases of
sagittal craniosynostosis. They also found a redistribution
of CSF in cases of coronal synostosis, mainly consisting of
compression on the adjacent ventricle. In patients with
lambdoid synostosis, they found a decrease of the ipsilat-
eral ventricle together with effacement of the involved
occipital horn [4]. Chadduck et al. [5] described the CTand
MRI findings of 75 patients diagnosed with various forms
of craniosynostosis. These authors studied 14 patients with
posterior plagiocephaly, which included nine cases of po-
sitional occipital flattening. The neuroimaging findings for
5/9 children with nonsynostotic plagiocephaly consisted of
dilatation of the subarachnoid space beneath the contralat-
eral frontal bossing [5]. However, in five of six, there was
generalized dilatation of the subarachnoid spaces, that
these authors attributed to some kind of brain atrophy with
motor retardation resulting in a position-induced deformity
[5].
Sawin et al. [15] compared CT findings of 31 children
with nonsynostotic posterior plagiocephaly with those of
20normal controls. Twenty-nineof 31infantswithpositional
plagiocephaly showed a generalized enlargement of extra-
axial CSF spaces, similar to the findings usually described
for external hydrocephalus or benign enlargement of the
subarachnoid spaces. These infants exhibited macrocephaly
that would predispose to intrauterine head constraint [15].
The authors suggest that the excess of extracerebral CSF
predispose these children to skull deformation since the
shifting fluid increases the plasticity of the skull bones [15].
In our series, there were no cases of macrocephaly, but 15 of
19 children with positional head deformation, studied by
CT or MRI, exhibited a marked expansion of the CSF
spaces (14 infants with plagiocephaly and 1 with brachy-
373
cephaly). On the contrary, no case of positional scapho-
cephaly had any of these abnormal collections of fluid. The
three cases with scaphocephaly corresponded one to a
premature child, and two instances sent to consultation at a
late age (34 and 36 months, respectively). In the statistical
analysis of our cases, the presence of dilated CSF spaces
was the only factor that was associated with positional head
deformation (p=0.02). The increase in fluid occurred in
three locations: the frontal subarachnoid spaces, the sylvian
fissures, and the basal cisterns (Fig. 5).
The development of enlarged subarachnoid spaces in our
series of children with positional vault deformation was
more pronounced in neonates and in children younger than
1 year and tended to disappear with the childrens growth
(p=0.04). These findings are in agreement with those
reported by Chadduck et al. [5] for children with cranio-
synostosis.
In our view, there are two possible explanations for the
occurrence of these abnormal accumulations of fluid [15].
First, the abnormal collections of CSF would be an etiologic
agent, as they would contribute to the development of the
deformation by debilitating the structure of the skull vault
bones. Transmission of brain pulse through the fluid-filled
spaces might constitute the hydrodynamic factor that pro-
duces the compensatory skull distortion. Second, the en-
larged fluid-filled spaces encountered in positional skull
deformities would merely reflect the displacements of
CSF toward areas of brain atrophy caused by the skull
deformation.
The inclusion in the study of children of group B is
justified as we wanted to ascertain the prevalence of
calvarial deformity in children with benign enlargement of
the subarachnoid spaces.
Conclusions
1. Patients with positional skull-vault deformities usually
have dilated subarachnoid spaces over the regions of
compensatory cranial growth.
2. These abnormal collections of fluid beneath areas of
compensatory skull growth supposes the existence of a
hydrodynamic mechanism that contributes to the
formation and progression of the skull distortion.
3. The enlarged subarachnoid spaces constitute an age-
dependent event, as they tend to disappear as the
children grow older.
4. These findings seem to be applicable not only to
positional plagiocephaly but also to other types of
positional cranial vault deformities.
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