Childs Nerv Syst (2017) 33:1727–1733
DOI 10.1007/s00381-017-3517-8
SPECIAL ANNUAL ISSUE
Shaken baby syndrome: what certainty do we have?
Matthieu Vinchon 1
Received: 27 June 2017 / Accepted: 29 June 2017
# Springer-Verlag GmbH Germany 2017
Abstract
Background Shaken baby syndrome is a common and devas-
tating disease in infants. In spite of its frequency, many con-
troversies persist, regarding the pathophysiology, diagnosis,
and management.
Aim of the study We reviewed several salient and challenging
issues related to SBS, like its pathogenesis, predisposing fac-
tors, differential diagnosis, and prevention programs.
Material and methods We derive arguments from the litera-
ture and from our prospective registry of accidental and non-
accidental traumas in infants.
Conclusions Much remains to be understood in SBS, and pre-
vention programs for this entirely man-made disaster are still in
their infancy. Pediatric neurosurgeons should be involved ac-
tively in the medical management and research on SBS.
Keywords Child abuse . Infantile subdural hematoma .
Pathophysiology . Prevention program
Introduction
Shaken baby syndrome (SBS) is a devastating illness, causing
high morbidity and mortality in infants. Its cardinal features
are subdural hematoma (SDH), cerebral edema, and retinal
hemorrhage (RH). Because SBS entails complex legal prob-
lems regarding the responsibility of caretakers, this topic is
highly controversial, regarding the pathophysiology, mecha-
nism of initial bleeding and subsequent brain lesions and
* Matthieu Vinchon
mvinchon@yahoo.fr
1
Department of Pediatric Neurosurgery, University Hospital, Hôpital
Roger Salengro, 59037 Lille Cedex, France
pericerebral collections, and also the eventuality of spontane-
ous bleeding.
The pediatric neurosurgeon is at the forefront in the man-
agement of these patients because the SDH often requires
evacuation or drainage. Prior to this, the diagnostic phase re-
quires careful evaluation in order to differentiate traumatic
from non-traumatic bleeding, then to gather arguments in fa-
vor of accidental versus non-accidental trauma. Subdural col-
lections (SDC) are common occurrences in children, occur-
ring in contexts as diverse as trauma, meningeal infection,
arachnoid cysts, or after surgery for hydrocephalus or craniot-
omy. All these SDC are well known to the pediatric neurosur-
geon, but often little heard of by other specialists.
The consequences of SBS are all the more nail biting that it
happens in previously healthy children and is an entirely man-
made disaster. This emphasizes the importance of prevention
and information of the public and raises the question of the
identification of an at-risk group of newborns requiring
targeted information and follow-up.
Material and methods
We have been collecting prospectively all cases of infantile
head trauma in a monocentric registry opened in 2001. Early
data from this registry were initially published in 2005 [1].
Our department is the only pediatric neurosurgical department
with a virtual monopoly in our population catchment area of
around four million people, so that our series roughly repre-
sents a population-based study of cases requiring neurosurgi-
cal care. Among this registry, cases of accidental trauma hav-
ing occurred in public spaces under the eyes of independent
witnesses (like traffic accidents) and cases of confessed abuse
were labeled as corroborated. Data regarding confession were
collected from legal hearings via the legists involved in the
1728
proceedings; we reported the comparative findings in corrob-
orated accidental vs non-accidental trauma in a paper pub-
lished in 2010 [2]. For the present review, we drew data from
the same registry, now numbering 1138 cases, regarding cor-
roborated traumas following the same selection criteria.
What do we know about the pathophysiology
of SBS?
Pathophysiology of cerebral lesions in SBS
Cerebral edema is one of the cardinal features of SBS and the
hallmark of its severity. Conversely, it may be absent and the
child’s status may be deceptively good; the risk is then to miss
the diagnosis, because recurrent SBS has a peculiarly poor
outcome [3].
Autopsy findings have shown that cerebral lesions leading
to death are mostly ischemic and not related to energy transfer
[4, 5]. Cerebral ischemia results from intracranial hyperten-
sion, with a drop in perfusion pressure, apneas caused by
initial fainting, or by spinal cord insult [6], and seizures which
are especially devastating in infants under 3 months, whose
brain is undermyelinated. These different mechanisms are of-
ten compounded and potentiate each other with devastating
results. This solves the paradox between the apparently milder
trauma in SBS, compared with accidental trauma, and the
damage it causes.
Pathophysiology of retinal hemorrhage
Retinal hemorrhages are an important hallmark of SBS.
However, it is important to stress that RH are also found in
non-traumatic bleeding like aneurysm rupture (the original
Terson’s syndrome) [7] and also in accidental head trauma.
However, most cases of RH caused by accidental trauma pub-
lished in literature were mild and caused by a major trauma like
traffic accident or defenestration [8]. It is thus of high impor-
tance to evaluate the specificity of RH. In our team, Dr. S De
Foort-Dhellemmes has proposed a four-tier grading of RH,
with grade 0 (absent), grade 1 (flame shaped, non-specific of
trauma), grade 2 (pearl shaped: traumatic but not specific of
SBS), and grade 3 (diffuse, extending to the periphery of the
retina, specific of SBS), itself divided in 3A (diffuse dots) and
3B (dome shaped with retinoschisis) [2]. These types are illus-
trated in Fig. 1. On the other hand, RH may be lacking in child
abuse, because beating does not cause RH or the fundoscopy is
performed with delay. In our experience, RH can clear almost
totally in 7 to 10 days (Fig. 2), which stresses the need for early
and repeated fundoscopy by an experienced ophthalmologist
and good-quality photographs.
Because RH are so important for the diagnosis of SBS,
these have become almost synonymous with legal
Childs Nerv Syst (2017) 33:1727–1733
Fig. 1 Grading of retinal hemorrhages as proposed by S De Foort-
Dhellemmes [2]. a Grade 1 flame shaped: non-specific of trauma. b
Grade 2 pearl shaped: traumatic but not specific of SBS. c Grade 3A
diffuse dots. d Grade 3B dome shaped with retinoschisis. We found that
grades 3A and 3B were specific of SBS with a high positive predictive
value
implications. However, the evaluation of the diagnostic value
of RH is fraught with of a circularity bias. We performed a
study of corroborated trauma, accidental or not, obviating the
circularity bias since the criterion for abuse or accident was
not based on medical findings. We could thus study the diag-
nostic value of grade 3 RH: the positive predictive value was
0.961, the negative predictive value was 0.665 [2]. This con-
firms the very high diagnostic value of RH, when present, and
also that RH can be falsely negative.
Pericerebral collections in SBS
Subdural hematomas happen in SBS as well as accidental
trauma and are of high importance because of their diagnos-
tic value and also because the intracranial hypertension they
cause can be easily corrected by simple measures. SDH
must be separated from benign arachnoidomegaly, a
condition which is common in the same age group and
with which they are often associated. The diagnosis of
SDH is mostly based on imaging showing compressed
cerebral sulci, displaced cortico-dural veins, and/or pres-
ence of the subdural membrane at the deeper aspect of the
SDH. SDH are generally mostly composed of hemorrhagic
cerebrospinal fluid (CSF) and appear within hours or days
after the trauma [9]. Chronic SDH are rare in infants,
resulting from scarring processes, and are often associated
with brain atrophy. Importantly, chronic SDH are associated
with spontaneous rebleeding, caused by fragile vessels as-
sociated with scarring, which should not be mistaken for
repeated trauma.
Childs Nerv Syst (2017) 33:1727–1733
Fig. 2 Eight-month male, admitted in emergency for malaise, hypotonia,
and left-sided deficit; CT showed a recent subdural hematoma which
eventually required drainage. a Fundoscopy performed 2 days after
clinical onset, showing diffuse retinal hemorrhage (RH) grade 3A. b, c
Role of CSF in SDH
A particular feature of traumatic SDH in infants is the predom-
inance of fluid over blood, in contrast with findings in adults.
Seriated studies show that the fluid in SDH gets more CSF-
like with time [10], as illustrated in our Fig. 3. In a previous
study, we also noted that the distribution of blood clots in the
subdural space evolved rapidly in the days following the trau-
ma, following gravity to the posterior regions, but also toward
the upper midline [11]; these midline regions have been
shown to be site of Bdural hole,^ likely to be responsible for
CSF absorption prior to the development of arachnoid villi
[12]. We interpreted the kinetics of meningeal blood as the
cause of clogging of the dural holes, causing engorgement
up flow with constitution of subdural collections composed
of hemorrhagic CSF.
Age and sex bias
The prevalence of SDH in males is verified in all series of
SBS, generally in the range of two males for one female. We
also noted the same sex ratio in subdural collections caused by
meningitis in infants [13] and also in Sylvian arachnoid cysts
[14]. The age peak occurrence of traumatic SDH is also very
much centered on 3- to 6-month babies, as exemplified in our
Figs. 4 and 5. Some authors have pointed out that the age and
gender biases in SDH coincided with the population at risk of
developing benign expansion of the arachnoid spaces [15],
more conveniently named as arachnoidomegaly. The latter
has also been linked with the development of arachnoid cysts
[16]. All these converging data suggest that subdural collec-
tions in infants, whatever their cause, are a consequence of
hydrodynamics fragility related with age and male gender.
How can we be sure that lesions are not spontaneous?
Establishing a firm diagnosis of SBS is among the clinician’s
chief duties, because the law in most countries mandates that
cases of suspected child abuse must be referred to the judicia-
ry. This implies ruling out, first, non-traumatic bleeding and,
1729
Fundoscopy performed at day 4 and day 7, respectively, showing rapid
disappearance of RH. This stresses the need for early and repeated
fundoscopy in SBS
second, accidental trauma. Among non-traumatic causes of
SDH are vascular diseases (arteriovenous malformations, an-
eurysms, thrombophlebitis) and such systemic causes are he-
mophilia and other clotting diseases, type 2 glutaric acidosis,
and Lobstein disease. Most of these very rare conditions can
be guessed from the context, imaging, complete clotting anal-
ysis, and assay of urinary organic acids. Accidental SDH are
most commonly identified for the presence of signs of impact,
coherent with the alleged history and the child’s developmen-
tal age, and the absence of RH.
Having ruled out all these occurrences, the question re-
mains of whether an isolated infantile SDH can develop with-
out any discernible trauma. Most pediatric neurosurgeons
have experienced the dilemma of an isolated SDH, without
systemic cause, and for which a thorough investigation fails to
Fig. 3 Two-month male victim of Silverman syndrome, presenting with
coma, status epilepticus, and hypothermia. a The CT on the day of onset
shows a partly hypodense subdural collection with ischemic brain. b The
subdural fluid punctured on the same day shows thin, incoagulable
hemorrhagic fluid. c The CT scanner performed the following day
shows a more diffuse and hypodense collection with more pronounced
brain hypodensity, while subdural puncture yields a more diluted
fluid (d). These findings illustrate the role of CSF in the early
constitution of subdural collections
1730
Fig. 4 Gender distribution of children with traumatic head injuries,
accidental or not. NS not statistically significant; **p < 0.01. Extradural
lesions (subcutaneous hematomas, fracture, epidural hematoma) were
slightly more frequent in boys, whereas intradural lesions (mostly
subdural collections) were significantly more frequent in boys. This
suggests that although traumas are not significantly more frequent in
boys, they result more often in intradural lesions because of gender-
specific hydrodynamic fragility
disclose any hint of concealed trauma. A normal fundoscopy
does not solve the problem since, as stated above, fundoscopy
can be falsely negative. The problem may appear insoluble,
because it is impossible to prove the absence of trauma, unless
positive elements in favor of spontaneous occurrence of a
SDH can be identified. In a previous study reviewing 16 cases
of infantile SDH for whom all medical and social investiga-
tions were negative, we found that potential predisposing or
precipitating factors such as macrocrania, arachnoidomegaly,
lumbar puncture, or dehydration could be identified in many
of these cases [11]. Interestingly, the male/female ratio in this
small subgroup was even more tilted toward males (7/1) than
in traumatic SDH. As mentioned above, arachnoidomegaly is
often considered as a predisposing factor for SDH [15]. The
link between arachnoidomegaly and SDH is unclear; the hy-
pothesis of stretched corticodural veins has been falsified by
computer models [17]. We can hypothesize that hydrodynam-
ic fragility in infancy, especially in boys between 3 and
6 months, could be the common source of arachnoidomegaly,
arachnoid cysts, and SDH caused by trauma or other causes.
The cause of this hypothetical fragility remains to be under-
stood and, until better substantiated by hard data, highly
speculative.
Whatsoever, considering the gravity of missing a diagnosis
of SBS, caution should be exercised before declaring a SDH
Childs Nerv Syst (2017) 33:1727–1733
Fig. 5 Prevalence of subdural CSF accumulation requiring drainage after
head trauma in children admitted consecutively in pediatric neurosurgery.
The high prevalence of CSF accumulation in children between 3 and
6 months suggest a high hydrodynamic fragility around that age. This
correlates well with the age of onset of idiopathic macrocrania caused by
subarachnoid fluid
Bspontaneous^ and a systematic and multidisciplinary ap-
proach is warranted [18].
Can we date lesions in SBS?
The dating of trauma in SBS is another major issue, raised
mostly by the judiciary in order to establish penal responsibil-
ity. The general consensus is that SBS manifests clinically
very early after trauma in the majority of patients. However,
since in many cases lesions are the result of repeated shaking
[19], it is likely that clinical manifestations of the initial epi-
sodes are often mild enough to be unnoticed by caretakers and
caregivers. Careful analysis of seriated imaging can help. In a
previous study, we analyzed the changes in the distribution of
blood clots in the subdural space, finding that blood clots were
initially located at the convexity of the brain, then migrated
toward the falx and tentorium [11]; rapid changes on seriated
imaging suggest that the first CT was performed early after the
trauma (Fig. 6).
Careful examination of fundoscopy can also help in dating
the abuse. In our experience, early fundoscopy finds RH of all
types (flame shaped, pearl shaped, and diffuse with or without
retinoschisis); since dome-shaped hemorrhages are much lon-
ger lasting than the other hemorrhagic lesions, isolated reti-
noschisis suggests a trauma at least several days earlier
(Fig. 2).
Whatever the method used, because of the rarity of reliably
dated material and a high rate of inter-individual variations,
the dating from imaging is grossly inaccurate and is generally
Childs Nerv Syst (2017) 33:1727–1733
Fig. 6 Three-month male,
confessed shaking by his father,
presenting with respiratory arrest,
seizures, epilepsy, and hypotonia.
CT scanners on day 0 (a–c), day 1
(d–f), and day 4 (g–i) show that
initially (c), blood clots were
found mostly at the convexity on
the right side, then thickened
along the falx (e, f, h, i) and
tentorium (d, g), while decreasing
at the convexity (i). Rapid
modifications over a few days
confirm the early date of the first
CT scanner
well below the expectations of the state attorney, who would
need precise dating by the hour.
Can we identify at-risk groups for SBS?
The general consensus is that SBS can happen in any social
group [20], and that beating attests of more intentional abuse
than shaking. Many authors consider that the whole popula-
tion is at risk because unbearable crying of the infant, the
triggering event of SBS, is universal. The objective of
targeting the whole population for information and prevention
[21] is probably correct but involves huge medico-social re-
sources. However, many studies on socio-familial background
of child abuse point at the same risk factors: perpetrators are
more frequently younger, less educated, and unmarried, com-
pared with the overall population [22]. Certainly, some adults
are less well equipped to cope with unbearable crying and
would benefit most from help before committing the
irreparable.
From our registry, we made a comparison between cor-
roborated accidental trauma (with independent witnesses,
like fall in public spaces or traffic accidents) and confessed
1731
abuse; the latter group was then split in beaten and shaken,
according to medical findings (presence or not of impact
lesions). We studied variables pertaining to the child’s peri-
natal history, and socio-familial factors, and compared the
prevalence of these variables in the accidental vs abusive
trauma groups and in the shaken vs beaten subgroups. The
results are shown in Fig. 7. Whereas the profiles showed
statistically significant differences between accident and
abuse, the profiles of SBS and beaten child syndrome were
similar, which suggests that the background for shaking is
not different from beating. Above all, these results allow the
definition of an at-risk group: infants with a prenatal history
or stay in intensive care and dysfunctional families with a
history of psychiatric disorder or substance abuse were sig-
nificantly more frequently associated with abusive than
with accidental head trauma. Our results corroborate the
findings of the Canadian study [23] and allow comparison
with accidental trauma. At-risk situations can be spotted in
prenatal consultation and monitored after discharge from
maternity wards and, especially, from neonatal intensive
care. Our findings confirm that efforts to Bidentify vulnera-
ble children^ [24] would allow targeted prevention.
1732
Fig. 7 Comparison between the profiles of infants victim of witnessed
accidental vs confessed abusive trauma (a, b) and socio-familial profile of
their families (c, d). *p < 0.05; **p < 0.01. Overall, the comparison
between accidental trauma and abusive trauma (a, c) shows significant
How can we organize the prevention of SBS?
From the Japanese experience, other authors advised preven-
tion in the prenatal period, through interview with pregnant
women, allowing the diagnosis of maternal stress and at-risk
situations [25]. Certainly, information and prevention through
networking with the perinatal care services is most promising.
One of the main caveats is that, since the overwhelming ma-
jority of perpetrators of child abuse are male, the larger part of
potential culprits would evade attention. One of the most in-
tegrative efforts to prepare parents to cope with unbearable
crying (the so-called Bcrying plan^) has been developed in
Canada [26]. It is difficult to evaluate the impact of these
programs, since the incidence of SBS may falsely increase
due to greater awareness among caregivers and in the greater
public, but certainly this is the right direction to go.
Conclusions
The SBS is a severe, complex, and multidisciplinary disease.
Pediatric neurosurgeons should not limit their implication in
the surgical management of SDH, but should be proactive in
the clinical management of these patients and in research. The
basic and clinical science that neurosurgeons master through
the management of different diseases in children, in particular
Childs Nerv Syst (2017) 33:1727–1733
differences in the profiles, both of the child and of the familial context. By
contrast, no difference was found between shaken and beaten patients,
both regarding the child and his familial context. These results suggest
that shaking and beating occur in the same context
the knowledge on accidental trauma and CSF pathophysiolo-
gy, have far-reaching applications in the field of SBS.
Compliance with ethical standards
Conflict of interest None.
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