Abstract
Abusive head trauma (AHT) is complex to define for establishing proper diagnosis criteria. This observation complicates greatly the diagnosis process when faced with an infant in consultation.
Objective
This study aims at clarifying the main criteria to be considered during the medical examination and interview with the social worker.
Method
An exhaustive literature survey was conducted to examine carefully the symptoms and signs as risk factors proposed to physicians.
Results
An analysis of the data available in the literature was quite difficult due to the various methods and the lack of real controlled studies. However, this analysis did show that the initial clinical signs and symptoms that are most frequently encountered are not very specific and can lead to a large differential diagnosis where AHT is often omitted.
Conclusion
The presence or combination of symptoms, even not highly specific, associated to a lack of a precise alternative diagnosis, especially in children under the age of one, should bring specialist to ask for additional brain imaging exams. The risk factors should not be taken into account for the diagnosis.
Résumé
Le traumatisme crânien non-accidentel (TCNA) est une entité complexe tant par sa définition que par les critères diagnostiques qui le caractérise. Ce constat complique grandement le processus diagnostique quand vient le temps de rencontrer un nourrisson en consultation.
Objectif
L’objectif de l’étude est de clarifier les principaux critères à considérer dans le cadre de l’examen médical et de l’entretien social.
Méthode
Une revue de littérature exhaustive est proposée afin de d’examiner de façon détaillée et critique les signes et symptômes de même que les facteurs de risques proposés au clinicien.
Résultats
L’analyse des données de la littérature rendue difficile par la variété de la méthodologie et l’absence de véritables études contrôles montre bien quand même que les signes et symptômes cliniques initiaux les plus fréquemment rencontrés sont peu spécifiques et conduisent à un large diagnostic différentiel où celui du TCNA est souvent omis.
Conclusion
La présence de combinaisons de signes, mêmes apparemment peu spécifiques, et l’absence d’un diagnostic alternatif étayé, particulièrement chez les enfants de moins d’un an, demeurent des plus éloquentes et devraient inciter le spécialiste à demander des examens paracliniques en imagerie cérébrale. Les facteurs de risques ne devraient pas être pris en compte dans l’émission du diagnostic.
1
English version
1.1
Introduction
Shaken baby syndrome (SBS) is a clinical entity with multiple facets. Even though hundreds and even thousands of articles were written on the subject starting with Guthkelch in 1971 and Caffey in 1972, there is no commonly accepted definition for this clinical entity. The term itself is the object of controversy. In North America, the term Shaken Baby Syndrome is being challenged. The American Academy of Pediatrics (AAP) suggests using the term Abusive Head Trauma. In their opinion, Shaken Baby Syndrome designates one of the mechanisms leading to skull and brain injuries in infants when in fact several mechanisms are involved. Nowadays, it is a well-known fact that impact signs might not be clinically visible yet will be revealed at the autopsy, when the infant dies or with the advances in medical imaging.
Starling et al., 2004 , after analyzing the confessions of several abusers, did report impact signs in infants, even though the abusers confessed to only shaking the baby and not hitting him or her. Conversely, when abusers confessed to hitting the infant, no impact signs were found. Thus, it would be more judicious to use Abusive Head Trauma (AHT) rather than SBS in order to include all types of head, brain and skull injuries.
Reece 2008 underlines the importance of trying to establish a proper definition. Rightly so, he proved that the lack of definition makes it complex and almost impossible to conduct scientific studies on this phenomenon even on an epidemiological level. After revising the various definitions proposed by several authors, he tried to suggest one, but it was rather heavy and difficult to use in the context of scientific studies. In fact, SBS is more of a phenomenon than a real nosological entity. For Minns 2005 , it is more than a syndrome because on top of clinical evidence, symptoms and signs, lab results and imaging exams, there will be circumstantial elements that will point towards physical abuse rather than an accident or disease. In fact, signs and symptoms are not truly specific of physical abuse if the circumstances of the injuries are not taken into consideration.
1.2
Method
An exhaustive and critical review of the literature was conducted, in French and English going back to 1975 focused on abusive head trauma to look for factors to be considered during differential diagnosis. The following databases were used Medline, Eric, Sociological abstract and ProQuest (Essays and Theses). Several keywords in French and English were entered. All the works relevant to the objective of this study were considered.
1.3
Signs and symptoms
The goal was to compile all the clinical signs and symptoms that should alert us to a possible case of AHT. The review of literature regarding AHT was quite complicated due to the diversity and disparity of the various studies. It was impossible to conduct a true meta-analysis. In fact, the subjects’ inclusion criteria were too heterogeneous meaning that the groups were disparate and different. Some authors limited their admission criteria according to age; others exclude any infant showing external marks of physical abuse or impact. The studies were often focused on small groups of subjects, less than 40 and were most often based on retrospective analyses of medical charts. Finally, almost all authors agree that the eye exam belongs to a specific diagnosis examination not included in the basic infant evaluation, unless head trauma is suspected. This means that ophthalmic evaluation is not listed in the initial signs and symptoms.
1.3.1
Lack of initial complaint
Shaking or any kind of infant abuse is rarely the cause triggering the initial medical consultation of the infant. In fact, it is only in less than 5% of cases that a person will finally admit, during the investigation, to shaking the infant to resuscitate him or her . Furthermore, in 70 to 97% of the cases, any history of trauma or injury, even minor ones, will be denied . The sociolegal pediatric clinic of the Sainte-Justine University Hospital in Canada has more than 20 years of experience and has performed over 400 clinical exploratory examinations for potential AHT. In less than 10% of the cases, clinical evaluation was initiated following alleged shaking episode usually because one of the parents was accusing the other of abuse and shaking the baby. These exploratory examinations were always negative and no validation or invalidation was possible. In two cases, the shaking was observed in a hospital setting by a healthcare professional, in one case the ongoing investigation had already documented evidence of previous abuse (shaken baby), whereas in the other case the investigation post-incident was negative.
1.3.2
Age and sex
The majority of studies indicate the age and sex of their subjects. Regarding age, Table 1 suggests that most abused children are younger than 12 months and the mean age is under 6 months. The median is between 3 and 5 months. There are more male victims than female, for obscure reasons, for this kind of physical abuse. Several hypotheses were brought up to try and explain this element, but none has been scientifically validated to this day.
Authors | # cases | Mean age (months) | Median age (months) | Less than 6 months (%) | Less than 12 months (%) | Boys (%) |
---|---|---|---|---|---|---|
Ludwig 1984 | 20 | 5.8 | 3 | 75 | – | 70 |
Duhaime et al., 1987 | 48 | 7.85 | – | – | 65 | |
King 2000 | 364 | – | 4.6 | – | – | 56 |
Ettaro et al., 2004 | 89 | – | – | – | 87 | 53 |
Mireau 2005 | 404 | 5.4 | 62.8 | 94.7 | 72.7 | |
Talvick 2006 | 26 | 3.9 | – | – | 100 | 77 |
CPSP 2008 | 220 | – | 5 | 58 | 75 | 58 |
1.3.3
Initial signs and symptoms
Very few studies analyzed the initial signs and symptoms, e.g., the ones that bring the infant to a medical consultation. In regards to this, four studies are interesting, the ones from Ludwig and Warman, 1984 , King 2000 , Mireau 2005 and PCSP 2008 . The first three ones are retrospective studies, whereas the last one is more prospective in its nature.
The study from Ludwig and Warman, 1984 was based on children who, from 1977 to 1982, were the object of a report for suspected child abuse from the Philadelphia children hospital to Child Protective Services. Out of the 1250 medical files revised, only the cases with “shaken-only symptoms”, e.g. 20 cases, were kept; all children with other types of abuse such as burns, bruises or fractures were excluded. Most times the parents came to the hospital because their infant was lethargic (80% of cases) or had difficulty breathing (60% of cases).
In his retrospective study, King 2000 compiled 364 reported cases between 1988 and 1998. These cases came from 11 university hospitals spread all over Canada and offering advance pediatric care. This study included all children under the age of 5 presenting head traumas with suspicion of SBS, with or without impact, that were reported by healthcare teams to the child protective services. The most frequently encountered clinical symptoms were seizures (45% of the cases) and decreased alertness (43% of the cases).
Mireau 2005, in his thesis , focused on all cases of subdural hematoma (SDH) in children under the age of 3 hospitalized in the neurosurgery ward of the Necker Hospital for Sick Children in Paris (Hôpital Necker–Enfants-Malades de Paris) between 1994 and 2004. However, all cases of SDH caused by a validated accidental head trauma or a well-documented pathology were excluded. Finally, 404 cases were kept where SDH seemed to have been the result of abusive head trauma. The most common initial clinical symptoms were seizures (73.3% of cases) and hypotonia (65% of cases).
The Canadian Pediatric Surveillance Program (CPSP) from the Canadian Pediatric Society (CPS) compiled 220 cases of abusive head trauma between 2005 and 2008. This study sent each month to each Canadian pediatrician, member of the CPS, a form to be filled out to indicate if he or she had in the last month treated a child under the age of 14 for head or brain trauma with a suspicion of physical abuse or neglect and had reported that case to Child Protective Services. In Canada, according to local laws, any professional suspecting child abuse has the obligation to report the case to the Child Protective Services of the county or province. Once the case is reported, in a confidential manner, the CPSP sent a detailed questionnaire that the pediatrician had to fill out. Thus 394 cases were reported for the study. Ninety-two were reported twice, meaning by more than one pediatrician simultaneously, 51 cases did not meet the inclusion criteria and in 31 cases the additional information was too tenuous to be included in the final data analysis. In these series also, the most common initial symptoms were decreased alertness (36% of cases), traumatic injuries of the soft tissues (30% of cases), followed by seizures (29% of cases).
Table 2 sums up the main signs and symptoms reported in each of the four studies listed. Obviously, we cannot be sure that the signs and symptoms meant the same to all authors. However, in average, validated or suspected seizures as well as decreased alertness seemed to be the most frequently encountered symptoms without being present in all cases. Even if they are non-specific, they point towards a potential neurological lesion. Still, in most cases, more than 50% of children seemed to initially show non-neurological symptoms such as respiratory dysfunction, hypotonia, vomiting, apnea or irritability.
Ludwig 1984 (%) | King 1998 (%) | Mireau 2005 (%) | CPSP 2008 (%) | Mean total (%) | |
---|---|---|---|---|---|
Decreased alertness | – | 43 | 55.30 | 36.00 | 44.77 |
Seizures | 30 | 45 | 73.30 | 29.00 | 44.33 |
Lethargy | 80 | 23 | 36.00 | 28.00 | 41.75 |
Respiratory distress | 60 | 34 | – | 14.00 | 36.00 |
Skin lesions of abuse | 35 | 46 | 23.70 | 30.00 | 33.68 |
Hypotonia | 15 | 45 | 65.00 | – | 31.25 |
Vomiting | 15 | 22 | 50.40 | 24.00 | 27.85 |
Apnea | 45 | 21 | 22.00 | 15.00 | 25.75 |
Irritability | 25 | 25 | – | 25.00 | 25.00 |
Curiously, only Ludwig and Warman and Mireau reported the state of the fontanel which isn’t closed in most children because of their young age (68% of cases according to Mireau). The two authors found a bulging or over tense fontanel in 55% of cases for Ludwig and Warman and 68% for Mireau . This telltale sign is very relevant since it directly points to a potential brain injury and should require additional imaging exams. Being aware of this sign will completely change the differential diagnosis and orientation of the clinical examination when faced with an infant presenting other non-specific symptoms such as vomiting, irritability or lethargy and other symptoms that can be commonly found in the ER or in a walk-in consultation.
It is also very important to underline that in more than one third of the cases, trauma-related skin lesions were found at the first medical evaluation of these infants. Several authors, such as Labbé and Caouette 2001 and Wedgwood 1990 , correlated the significance of bruises to age and reported that, in non-walking children they most often are sign of child abuse. According to Atwal 1998 , who studied 24 cases of death by inflicted brain trauma, 70% of the children had bruises on their skin. Most often, the bruises were found on the cheeks, forehead, back, buttocks and more rarely on the limbs. This same author also reported that the aspect of the lesions were more symptomatic of hitting than grabbing. Greenes and Schutzman 1999 , in a study on head trauma in infants, noticed that 93% of newborns that had bruises on the scalp also had associated brain injuries.
The signs and symptoms are rarely isolated, but only Mireau 2005 studied their concomitant association to other symptoms as presented in Table 3 . These associations, even if there are non-specific, do orientate towards a neurological origin rather than a respiratory or gastrointestinal one. It seems here quite clear that in AHT cases vomiting, which is an extremely frequent primary complaint in newborns, is associated to other signs and symptoms that should guide the physician towards additional neurological exams and thus, lead eventually to the discovery of suspicious brain injuries.
According to Newton 2006 , death can also be added to the list of telltale clinical conditions for AHT. In fact, a certain proportion of children with shaken baby syndrome are brought to the hospital dead or agonizing as reported by Carty and Pierce 2002 . Just like in Sudden Infant Death Syndrome (SIDS), many AHT infants do not show any external signs of injuries, this is why an autopsy and a complete investigation surrounding the circumstances of death are necessary to correctly identify the cause of death, AAP 2008 . It is difficult to estimate the number and percentage of cases since often, they are not included in the retrospective studies and the diagnosis was based on data from various authorities as well as coroners, outside of a clinical setting ( ).
Axial hypotonia and seizures | 53% |
Seizures and tense fontanel | 46% |
Vomiting and tense fontanel | 38% |
Vomiting and axial hypotonia | 34% |
Abnormal head circumference for age and tense fontanel | 31% |
Vomiting, seizures and tense fontanel | 25% |
Vomiting and decreased alertness | 24% |
Vomiting, hypotonia and tense fontanel | 23% |
Vomiting, decreased alertness and tense fontanel | 19% |
The initial telltale signs and symptoms, i.e. the ones for which infants, later identified as AHT victims, are brought to the medical consultation can be grouped into four categories:
- •
non-specific of head or brain injury: loss of appetite, vomiting, irritability, sleepiness and death;
- •
suggesting head or brain lesions: decreased alertness, seizures or seizure-like episodes, tense or bulging fontanel;
- •
suggesting head or brain traumatic injuries: bruise or signs of head contusion, known history of minor head trauma;
- •
suggesting physical abuse or violence: numerous bruises on the body of a non-walking infant.
In fact, as reported by Jenny et al. 1999 and Case et al. 2001 when known history of trauma, even minor ones, is unavailable and first signs or symptoms are non specific, some aspects of an exhaustive clinical examination should point to AHT, such as fontanel examination, looking for bruises on the skin, especially on the head or face or anywhere on the body of a non-walking infants. These last two elements, looking for bruises and palpation of the fontanel should systematically be included in any basic clinical examination of an infant brought to the medical consultation, regardless of the other alleged signs or symptoms. All suspicious cases should get brain imaging especially simple (without infusion) CT-scan which seems to be the imaging exam of choice.
1.3.4
Symptoms that could negatively impact the diagnosis
Several authors such as Carty and Pierce 2002, Jenny et al. 1999 and Oral et al. 2008 , reported that a certain number of their validated AHT cases could have been diagnosed earlier during a previous medical consultation than the one where their condition was accurately diagnosed. Only Jenny et al. 1999 presented a systematic study. The author studied retrospectively all the cases of AHT in infants under the age of 3 that were referred to their pediatric unit within the University Hospital from 1990 to 1995. Out of the 232 cases studied, 173 were included in the final study. In three cases, the children were older than 3, 38 cases were accidental and 13 could not be included due to the non-availability of relevant medical charts. Fifty-four cases (31.2%) were not correctly diagnosed during the first medical evaluation. The diagnosis was made after a mean of 2.8 medical consultations and a 7-day lapse. The authors were able to establish that errors in diagnosis were more common in young newborns (mean age 180 days) than in older ones (mean age 278 days). The diagnosis was missed in 37% of children of Caucasian origin versus 19% in African-American children and in 40% of cases in married couples opposed to 19% of separated parents.
In regards to the signs and symptoms, 65% of the children who were not properly diagnosed were alert and awake during the first consultation, 82% did not present any respiratory dysfunctions, 56% had vomiting symptoms essentially and 65% were irritable. The statistical analysis tends to show that four factors or variables have a significant predictive value, i.e. the presence of:
- •
respiratory disorders;
- •
seizures;
- •
traumatic injury of the face or scalp;
- •
separated parents.
The authors estimated that the lack of one of these four variables meant that the baby had only 20% chances for AHT to be diagnosed at the first consultation.
An exhaustive study of each medical chart showed that four out of five deaths could have been prevented in the group of 54 children where the diagnosis was missed, whereas for the 15 other children (28%), they would probably not have been submitted to another shaking episode.
In 14 and 10 cases respectively, the reported wrong diagnoses were viral gastroenteritis and accidental head trauma and in another 20 cases, it was infectious pathology. In two cases, a spinal tap with blood was erroneously attributed to a traumatic procedure.
In order to reduce the rate of missed diagnoses, the authors recommend, as mentioned above, that for all infants a physician should systematically look for skin bruises, evaluate the state of the fontanel and assess the head perimeter. They also recommend an eye funduscopic examination with pharmacological pupil dilation if necessary.
To conclude, the signs and symptoms most likely to distract the physician from the proper diagnosis are the ones belonging to the “non specific of head or brain injury” group: e.g. loss of appetite, vomiting, irritability and sleepiness which are so frequently the main reason for medical consultation. Most often, they result from benign infectious pathologies rather than traumas. Faced with the lack of family or social risk factors or the notion of a benign traumatic event, they can distract the physician from a neurological diagnosis.
1.4
Data from the parents’ interview
When the parent come to walk-in clinic or arrive to the ER, they talk about signs and symptoms that worry them and do not know how to explain them, except when children present obvious signs of trauma such as pain, bruises, burns, etc. Often, especially for young children or newborns, parents rush to the consultation after an accidental asymptomatic injury or trauma because they are afraid of potential undetected internal lesions. In the cases of AHT, or other kind of physical abuse, often the contrary happens. In fact in spite of clear and obvious traumatic signs, the parents deny that anything happened and they are unable to explain the symptoms. The absence of history of previous trauma faced with obvious signs of traumatic lesions is an important element in favor of a non-accidental origin or inflected abusive injuries. Mireau 2005 reports that in almost 72% of the cases, no history of trauma was admitted to by the parents. Reece and Sege 2000 studied the medical charts of 297 cases, representing all children under the age of 6 showing signs of head trauma and admitted in a pediatric hospital in Cleveland, OH, USA over a 5-year period. They were able to analyze 287 cases and differentiate them between non-accidental (54 cases) and accidental (233 cases). In 30% (56%) of the 54 non-accidental cases, the parents gave no explanation regarding the circumstances surrounding the head trauma. This fact is weakened by one of the criterion to classify the case as abusive, i.e. the lack of a valid explanation for severe head trauma.
The lack of a valid explanation for the objective injuries is one of the universally recognized clinical criteria for abusive head trauma or other physical abuse observed in children. However, in practice, it can be difficult sometimes to establish if the alleged circumstances can explain or not the nature of the observed head injuries. It is sometimes impossible for all professionals involved to reach a consensus. The issue of falls is often at the heart of the controversies.
In 1991, Chadwick et al. published an interesting study related to this issue. They did a retrospective analyses of the medical charts of all children (317) admitted to a children’s trauma center between 1984 and 1988 with a history from the parents or caretakers that the child had fallen. They analyzed the first story data as reported by the parents without evaluating the veracity or credibility in light of the diagnosis and outcomes. This group of 317 children included 67% of boys. Thirty children (9.5%) under the age of 1 and 145 (46%) were between the age of 1 and 3. Tables 4 and 5 of their study are reported below and are quite eloquent.
Height | Number of deaths | Number of falls | Death rate % |
---|---|---|---|
1–4 feet (1 m) | 7 | 100 | 7.0 |
5–9 feet (3 m) | 0 | 65 | 0.0 |
10–45 (12 m) | 1 | 118 | 0.8 |
Total | 8 | 283 | 2.8 |
Child fell from his/her own height | 2 |
Child fell from bed or table | 2 |
Child fell down the stairs | 1 |
Child fell from the arms of an adult | 2 |
For these seven children who died from a short-distance fall, intracranial injuries were identified as the cause of death. However, age of the victims is not available, but it is completely outrageous to see that 7% of children falling from less than 4-feet high die from the consequences of these falls, whereas no child died after falling from a height between 5- and 9-feet and only one out of 118 children (0.8%) died from a 30-feet fall.
Some studies brought reliable data on the immediate outcomes of falls occurring in well-documented circumstances with multiple or objective witnesses. Helfer et al., 1977 conducted a study on 85 children under the age of five who fell from a maximum height of three feet while they were hospitalized. Fifty-seven (67%) of them did not have any signs of trauma, 17 had superficial and benign skin abrasions or cuts, 20 presented some superficial signs of bruises and only one out of 40 children who had X-rays showed a simple skull fracture. None had apparent or serious sequels. In the same study, during a visit to the pediatrician, parents were asked to complete a questionnaire when they were able to remember an incident of fall at home for one of their children under the age of 5. The parents reported 176 fall incidents from a height lower than six feet. Even though the stories of the parents could not be controlled, no children had symptoms of intracranial trauma.
Nimityonskul and Anderson 1987 studied the charts of 76 children under the age of 16, 75% of them were younger than 5 and almost a third were under the age of 1. They fell during the course of their hospital stay in a pediatric unit. The maximal height for these falls was between three and four feet. Sixty-three percent of children only had a slight bruise or nothing at all, whereas 30% showed signs of superficial edema of the soft tissues. Only one simple skull fracture was reported that had no consequences, but only nine out of the 76 children had X-rays. Williams 1991 conducted a prospective evaluation on 106 children for whom an independent witness (outside of parents) could provide reliable information regarding the fall. Only children who fell from five to 40 feet had major head trauma such has intracranial hemorrhages, brain edema or comminuted or depressed skull fractures.
Conversely, Plunkett 2001 commented and analyzed the medical charts and legal investigation files for 18 children aged 1 to 13 years who died in falls from short distances (0.6 to 3 m) in playgrounds. Twelve out of these 18 accidents occurred while the children were swinging, adding additional speed to gravity and in another case, the autopsy unveiled a dissection/thrombosis of the external carotid. In four other cases, the death was attributed to an acute massive brain edema, yet there was no autopsy and the clinical data on hydration and electrolytic levels were not available. For the five remaining cases, only two falls were witnessed by an independent observer. Finally, no data were available on the total number of children who would have had similar kinds of playground accidents over the same period of time. This study is quite controversial, but it brings up the possibility of major trauma from a low height, justifying an attentive and thorough analysis of the surrounding circumstances in each case.
These studies show clearly that a short-distance fall (four feet or under) does not cause any injury in most cases. However, 30 to 45% of children can show signs of superficial bruises without any significant consequences. A simple skull fracture, with no sequels, can be found in 1% of the cases. However, the children from these studies are quite older than the populations of infants’ victims of AHT.
Reece and Sege 2000 suggested that intracranial hemorrhages or edemas in young children, with no indications of traumatic incident or fall from a significant height, should bring up a strong suspicion of physical abuse.
Just like in the other types of physical abuse, the delay for consulting is often a telltale sign of abuse. In fact, most parents and caretakers, especially with young children, rush to the emergency room or to their general practitioner when their child gets hurt or into an accident that could lead to some kind of damage.
Physicians are not responsible for validating the circumstances of onset of a traumatic incident. The physician’s role is to evaluate if the reported circumstances fit with the injuries or wounds observed. In terms of head lesions, it is quite easy to differentiate the pathological from the traumatic origin. However, within those of traumatic origin, it is more difficult to distinguish those of accidental from inflicted causes and often requires additional investigation by non-medical professionals. There is no lesion which is exclusively of accidental origin, nor that is exclusively of inflicted origin. However, it is essential to keep in mind that a short-distance fall (less than nine feet) does not usually cause severe skull or brain damage.
1.5
Risk factors
Several studies tried to link the phenomenon of child abuse to different risk factors, some related to the parents, e.g. the mother’s age, unplanned pregnancy, mental health, physical health, education; others to the child, such as birth weight, health status, behavior and finally, the last ones related to social and family environment such as the lack of social support and various stress factors. Furthermore, whether faced with abuse or neglect, these studies showed that there is also an interaction between these three categories of risk factors. Before going any further in the studying of these risk factors, it is quite essential to be reminded that a risk factor is a variable statistically association to a phenomenon, a pathology or a syndrome without being the cause. The analysis of the risk factors correlated to AHT seems quite complex especially due to the methodological problems already mentioned, i.e. the small-size samples, heterogeneity of the inclusion criteria, the lack of a common definition of the syndrome itself and the specific confidentiality rules. This limits the array of studied variables and reliability of the data, meaning that studies are most often incomplete with contradictory conclusions. The risk factors usually found in a context of abuse or neglect are thus rarely presented or analyzed in SBS or AHT studies. The reader must examine the characteristics of the studied samples to carefully extrapolate some elements that could suggest a specific profile for the actors involved or their life and sociocultural environment.
1.5.1
Risk factors related to the abuser
Most times, the abuser is a man (68,5% Starling, 1995; 70% Starling, 2000; 72% King, 2000 under the age of 30 (mean: 22 years [14–46 years], Showers, 1994; median: 22 years [16–46 years], Starling, 2008) having a privileged relationship with the victim, either being the father of the child or the mother’s boyfriend (44% biological father, 23% boyfriend: Showers, 1994; 37% biological father, 20.5% boyfriend: Starling et al. 1995; 44% biological father, 20% boyfriend: Sinal, 2000; 33% biological father, 13% boyfriend: King, 2000) . Caretakers are also potential abusers. In fact, in the series of 127 cases examined by the team of Starling 1995 , they amounted for more than 20% of the abusers. Similar results were found in the retrospective study of the medical files conducted by Sinal and his team in 2000 . This category of unrelated abusers is rarely discriminated in the studies on social and family risk factors, which could really have created a bias in their analysis.
1.5.2
Risk factors related to the child
In most studies, the victim is most often a boy. This data has not been clearly explained yet but should be mentioned since it contradicts the most recent Canadian data on the incidence of physical abuse for the 0–3 years of age range . According to this large Canadian study, more girls (57%) would be the object of physical abuse corroborated after a report to Child Protection Services.
Even though AHT is often seen in children under the age of 2 and especially under the age of 1, most of the victims are under the age of 6 months (62.8% Mireau 2005; 65% Goldberg 2000; King, 2000) . Ethnicity was suggested for the first time as a risk factor by Guthkelch in 1971. He suggested that white populations (in the United Kingdom) were more prone to shake their babies as a disciplinary method, probably under the assumption that shaking is more socially acceptable than slapping. This hypothesis was also studied by Sinal and Ball as well as Brenner and his team in 1987. In fact, their results suggested that there was a higher occurrence of Caucasian victims (17/24 Sinal 1987; 8/12 p < 0.008 Brenner 1987) . Due to some methodological biases, mainly on the type of population going to the targeted hospitals, Sinal and his team (2000) focused on this controversial issue by using a more rigorous methodology and this time, did not find a significant difference ( p = 0.0895) between a group of South Carolina Caucasian children and another group with South Carolina children from different racial origins. In the series of 80 cases of abusive head trauma in North Carolina reported by Keenan et al. in 2003 , race/ethnicity (non–European American) was considered a significant risk factor for inflicted traumatic brain injury (TBI) in regards to the general population. However, race or ethnicity was not significantly correlated to AHT compared to accidental brain trauma. Data suggest that the variable “race or ethnicity” is a risk factor stemming from other social markers that put the child at a higher risk of injuries. On the other hand, in the analysis of 173 cases of false negative diagnosis of AHT Jenny in his 1999 study reported that the cases that were less likely to be recognized by medical professionals were young Caucasian infants. In the same line, in 2002, Lane and his team concluded that more children from African American or Hispanic ethnicity were more likely to be evaluated and reported for suspicion of child abuse. This observation, if true, could challenge the lack of significant difference between ethnic groups and could show also the bias linked to racial preconceptions.
Several studies show higher AHT rates for some pregnancy-related conditions or delivery-related conditions than those usually found in the general population. Mireau 2005 reported an 11% rate of pathological pregnancies and an 11% rate of premature births. Concordet and Bonnaure 2000 unveiled 55% of complicated pregnancies and 17% of premature births. A similar rate of premature births (15.6%) was shown by Keenan et al. 2003 . Minns and Brown 2005 found a 29% rate of premature births < 37 weeks in a sample of 129 Scottish medical files, amounting to the higher rate found in the literature. King 2000 reported a rate of 16% for pathological pregnancies and 21% for premature births and Matsuura 2002 showed a rate of 13.8% for premature births and 22% of pregnancy disorders on 72 medical files classified as SBS in the state of Hawaii. Multiple births were also found to be more frequent than in the general population: Minns and Brown 2005 found 9%, Concordet and Bonnaure 2000 7%, Keenan et al. 2003 5.3%, whereas Mireau 2005 found a 5% rate in his sample of 404 cases of children treated at the Necker Hospital Center in Paris.
Some studies have also identified the birth rank of the child in the family to show that in at least half of the cases, the victim was the mother’s first born (51% Mireau 2005; 58% Concordet and Bonnaure 2000; > 50% Goldberg 2000; 92% Bonnier 1995, Keenan et al., 2003 5.3%) .
1.5.3
Risk factors related to parents
The mean age of parents at the time of the physical abuse seems to be much lower than the general population at the birth of their first child. According to a sample group in Minns and Brown’s study in 2005 , mothers had a mean age of 22 years (SD 5 years) and fathers/partners had a mean age of 26 years (SD 5.8 years) whereas in the series from Mireau in 2005 , mothers and fathers had a respective mean age of 28 and 32. By comparing the age of mothers at the birth of their child, Keenan et al. 2003 and his team unearthed a significant difference between the age of mothers in the general North Carolina population ( p < 0.001) and the ones of mothers of children victims of AHT, the latter being significantly younger. Unfortunately, most studies on parents’ age only offer diffuse data not allowing for a precise comparison. However, the young age of parents is often mentioned by authors in the literature and noted as a potential risk factor.
Several incomplete and heterogeneous data do show that parents or abusers are in most cases involved with social issues or emotional disorders. The Scottish series of 129 victims reports 25% of cases with a history of drug or alcohol abuse. Starling 2008 mentions the history of family violence in almost half (40.7%) of the identified abusers. Minns and Brown 2005 only report 25% of family violence in the victims’ parents, but a 41% rate of violence inflicted at a young age. In a Canadian study on 364 cases, King 2000 found lower rates, i.e. 12% for a history of family violence, 7% of violence at young age and 9% of substance abuse and addictions in the abusers. Minns and Brown 2005 reported 32% of present or past mental disorders whereas Concordet and Bonnaure reported a 16% in their 2000 study , so did Goldberg and Maurey-Forguy 2000 and a 4% rate for King’s study in 2000 .
The psychosocial analyses on two series of French infants by Concordet and Bonnaure 2000 and Goldberg and Maurey-Forguy 2000 underline the major distress in the parent-child relationship, lack of family, friends or social support and presence of stress factors (e.g. problems in the parenting couple, work problems, moving house). These studies were essentially descriptive and it was quite complicated to extract precise data. However, the presence of this type of factors seem more important in the first series ( n = 69) than in the second one ( n = 106) for no apparent reason. King, in his 2000 study on Canadian parents, highlighted several factors suggesting that social isolations and lack of parenting skills were involved. Mireau 2005 noted that parents did not have a proper understanding of the needs and normal behaviors of their child.
1.5.4
Risk factors related to the social context
The recent study by Minns in 2008 offers a detailed description of the socioeconomic context, over a 10-year period, for 25 cases of Abusive Head Trauma (AHT) from the Scottish region of Lothian. This series represents probably all cases from this region over the given period. The authors demonstrated that most cases (64%) came from the most impoverished neighborhoods. For their evaluation, they used the Scottish Index of Multiple Deprivation (SIMD), which unveiled that 76% of cases came from the poorest areas as far as education, skills and social abilities were concerned, 72% in areas with the highest rates of crimes, 68% in areas with the poorest health, 60% of cases in areas with the lowest incomes, 52% in area with the poorest quality of housing, 48% in areas with high rates of unemployment. But paradoxically, 76% of these 25 cases who lived in these poor and socially challenged neighborhoods had access to many more services, e.g. transportation, healthcare, schools, shops or postal services. King’s study in 2000 reported that poverty affected 28% of the cases, ranking this factor second in the list of risk factors related to family and social context.
These studies contradict the results and hypotheses from the previous study that showed that AHT did not have a higher incidence in poorer populations. For example, Concordet and Bonnaure 2000 reported that most parents had a stable job, 81% of mothers were working. For Goldberg and Maurey-Forguy 2000 ), most parents had a secondary or even higher level of education. The middle classes were the most represented with a good number of upper classes as well. Mireau 2005 showed that the parents in his study most often did not have any major difficulties or social issues.
To sum up, the data available to this day are incomplete and sometimes contradictory, but suggest that the most likely victims of AHT are boys, firstborns, under the age of 6 months, born prematurely after a difficult or multiple pregnancy, living with parents with a past or present history of abuse (alcohol, drugs, family violence) that are unaware of how to manage the needs of their child. Abuse can also have no correlation to the socioeconomic, cultural or ethnic context. Furthermore, if it is safe to assume that a child victim of AHT has more chances to belong to that group, it is wrong to think that most children with these characteristics are victim of this type of abuse. Thus, to establish the proper diagnosis, it is essential to focus more on the clinical, biological and imaging data.
1.6
Immediate cause
This review of risk factors would not be complete without talking about cries that seem to be the triggering factor of abusive behavior on infants. In the Netherlands, Reijneveld et al. 2004 and his team tried to evaluated the parents’ reactions faced with their crying infant at the age of 1, 3 and finally 6 months. They asked 3259 parents to answer an anonymous and confidential questionnaire regarding their own behavior with their crying infant. Six percent of the parents revealed that they had some kind of abusive behavior such as muffling, suffocating, hitting or shaking their baby. This was most likely to happen when parents did not understand why their child was crying and judged that it had to stop. According to the different studies, this behavior became more frequent when the baby was older, more parents admitted to having this abusive behavior when their child was 6 months old rather than 1 month old. Furthermore, this behavior was more common in family where both parents were not living together, or had different ethnic origins, or came from third-world countries or finally were unemployed.
Barr et al. 2006 studied the crying curve in young babies. The duration and occurrence of daily cries are spread out according to a specific frequency; they usually start at the 2nd week of life to reach their peak around week 5 or 6. According to the data published on age upon admission for AHT during the first year of life, they found a similar curve as regards to shape and peak incidence of cries but transposed a few weeks later. In fact, the crying peak was established between weeks 5–6 whereas for the AHT curve, the peak was at week 10–13. They suggested various hypotheses to explain that element such as the fact that often during the first admission for AHT, there is evidence of previous abuse or that some children have longer crying periods than others.
Lee et al. 2007 , conducted a study based on the AHT database compiled between January 2003 and August 2004 by the National Center on Shaken Baby Syndrome in the State of Utah, USA. They collected 591 cases of AHT in children under the age of 18 months. Among these cases, cries were reported as being the triggering element in 166 cases. They compared the age distribution curve upon admission in the two groups, the one where cries where identified as the triggering element and the group where it wasn’t. The curves are similar in both groups and match the ones reported by Bar et al. 2006 . The authors see it as an indication of the relevance of cries on the genesis of AHT, at least in the first month of life.
Talvik et al. 2008 reported a cohort of 26 children victims of AHT, 21 of them were included in a prospective study. All children were under the age of 1. The age distribution curve upon diagnosis matches the ones found by the authors previously listed. In 23 of the 26 victims (88.5%), the medical charts showed that parents had gone to a doctor for excessive crying or irritability previous to the AHT hospital admission.
It is essential to recognize this element as a potential risk factor and take into account for setting up AHT prevention strategies.
1.7
Conclusion
The diagnosis of AHT is a difficult one due to the lack of specific clinical symptoms. It has been clearly demonstrated that in only two thirds of the cases was the diagnosis made during the first medical consultation, even in a university teaching pediatric hospital setting . The analysis based on the data from the literature can be argued due to the various methodologies used and obviously the lack of real controlled studies. However, it does show that the initial signs and symptoms are not very specific and can lead to a wide differential diagnosis where AHT is often omitted or discarded.
But in fact, some non-specific symptoms such as vomiting, irritability, sleepiness rarely occur alone and are most often associated to other signs or symptoms such as decreased alertness, seizures or seizure-like episode, tense of bulging fontanel. This association should trigger additional exams to look for a neurological disorder. Unless a well-documented alternate diagnosis is suggested, these infants (especially in the first months of life) should all have additional imaging exams and a simple (without infusion) head CT-scan is the exam of choice in that case.
It is also essential to point out those most young victims of AHT show external signs of trauma such as bruises, abrasions, superficial hematomas. A systematic search of these signs during a routine infant examination could lead to an early diagnosis of AHT and avoid recurrent abuse or significant neurological damage. Finally, taking into account the risk factors should not be part of the diagnostic analysis. It could lead to false negatives as well as false positives.
2
Version française
2.1
Introduction
Le syndrome de l’enfant secoué (SES) est une entité clinique au visage très polymorphe. Bien que depuis les premières descriptions qu’en faisaient Guthkelch en 1971 et Caffey en 1972, des centaines, voire des milliers d’écrits scientifiques sur le sujet aient vu le jour, il n’existe pas de définition commune généralement acceptée de cette entité. Son appellation même ne fait pas l’unanimité. L’usage du vocable de SES, dont l’équivalent dans la littérature nord américaine serait SBS pour Shaken Baby Syndrome ou Syndrome du bébé secoué, est de plus en plus contesté. L’American Academy of Pediatrics (AAP) suggère que l’on utilise plutôt le terme de Abusive Head Trauma ou Trauma crânien abusif. De leur avis, SES ou syndrome du bébé secoué désigne plutôt l’un des mécanismes susceptibles d’entraîner des lésions crâniocérébrales à l’enfant, alors qu’il serait de plus en plus clair qu’un ensemble de mécanismes ou de forces, et non uniquement le secouement, sont le plus souvent en cause. Il est en effet maintenant bien reconnu que des signes d’impact puissent être cliniquement indécelables alors qu’ils seront indiscutablement révélés par l’autopsie, dans l’éventualité d’un décès, ou l’évolution des examens d’imagerie. Starling et al. 2004 , après avoir analysé les confessions de différents agresseurs, constatent également que des signes d’impact sont retrouvés chez des enfants où, de l’aveu de ceux-ci, il n’y aurait pas eu de coups portés à l’enfant mais uniquement des secouements. Inversement, des traces d’impact apparaissent absentes chez des enfants qui, encore de l’aveu des agresseurs, en auraient été victimes. Il serait donc préférable d’utiliser le terme de Traumatisme crânien infligés ou encore de Traumatisme crânien non accidentel (TCNA) plutôt que celui de SES ou SBS de façon à y inclure toute forme de sévices crâniocérébraux.
Quant à la définition, Reece 2008 souligne l’importance d’arriver à en établir une. À juste titre, il affirme que cette absence rend complexe, voire impraticable, l’étude scientifique de ce phénomène, ne serait-ce qu’au plan épidémiologique. Après avoir révisé celles proposées par divers auteurs, il tente d’en formuler une qui, de notre avis, s’avère lourde et difficilement utilisable pour l’étude scientifique du phénomène. De fait, le SES est davantage un phénomène qu’une entité nosologique propre. Pour Minns et Brown 2005 , c’est plus qu’un syndrome, car aux évidences cliniques, symptômes et signes, données de laboratoire ou d’imagerie, s’ajoutent nécessairement des éléments circonstanciels qui le démarqueront de l’accident ou de la maladie, en faisant des sévices à l’enfant. Nous verrons en effet que, au niveau des signes et symptômes à tout le moins, aucun n’est véritablement spécifique de sévices si les circonstances de la survenue des atteintes, dont ils témoignent, ne sont pas prises en compte.
2.2
Méthode
L’examen des facteurs à prendre en considération dans l’analyse menant à un éventuel diagnostic de TCNA se base sur une révision exhaustive et critique des écrits scientifiques francophones et anglophones produits depuis 1975 portant sur le trauma crânien non-accidentel de l’enfant. Les bases de données bibliographiques Medline, Eric, Sociological abstract et ProQuest (Dissertations and Theses) ont été interrogées en utilisant de multiples mots clés tant français qu’anglais. Tous les travaux répertoriés et jugés pertinents à l’objectif poursuivi par les auteurs ont été considérés.
2.3
Signes et symptômes
Nous réviserons, dans un premier temps, l’ensemble des signes et symptômes cliniques qui devraient nous alerter et nous amener à considérer la possibilité d’un SES. La revue de la littérature concernant le SES est un exercice difficile à cause de la diversité et disparité des études. Il est impossible de réaliser une véritable méta-analyse. En effet, les critères d’inclusion des sujets sont trop variables de sorte que les groupes de sujets sont disparates et dissemblables. Certains auteurs limitent l’admissibilité des sujets à leurs études en fonction de l’âge, d’autres excluent tout enfant montrant des signes externes de violence physique ou d’impact. Les études portent souvent sur de petits nombres de sujets, moins d’une quarantaine et sont le plus souvent basées sur des revues rétrospectives de dossiers médicaux. Finalement, précisons que pour pratiquement tous les auteurs, l’examen du fond d’œil semble être considéré comme un examen diagnostic particulier qui ne fait pas partie de l’évaluation de base du nourrisson, à moins que déjà un problème intracrânien ne soit suspecté, de telle sorte qu’il n’apparaît pas dans la liste des signes et symptômes initiaux.
2.3.1
Absence de plainte initiale
Le secouement, ou toute autre forme de sévices à l’enfant, est rarement le motif qui amène initialement l’enfant en consultation médicale. Effectivement, ce n’est que dans moins de 5 % des cas, qu’une personne reconnaîtra finalement en cours d’investigation avoir secoué l’enfant dans le but de le réanimer . De même dans 70 à 97 % des cas, toute histoire d’un traumatisme quelconque, même mineur, demeurera niée . À la clinique de pédiatrie sociojuridique du CHU Sainte-Justine, en 20 ans, après plus de 400 explorations cliniques pour possibilité de traumatisme crânien non accidentel de l’enfant, dans moins de 10 % des cas, l’exploration fut initiée en raison d’une histoire de secouement rapportée par un des deux parents de l’enfant, ce dernier accusant l’autre d’en être l’auteur. Ces explorations se sont toujours avérées négatives et n’ont pas permis de confirmer ou infirmer les faits. En deux occasions, le secouement a été observé en milieu hospitalier par un professionnel de santé, dans un cas l’investigation, déjà en cours, avait documenté des évidences de secouement antérieur, alors que dans l’autre, l’investigation post-événement demeura négative.
2.3.2
Âge et sexe
La majorité des études indiquent l’âge et le sexe des sujets répertoriés ( Tableau 1 ).
Auteurs | Nombre de cas | Âge moyen (mois) | Âge médian (mois) | Moins de 6 mois (%) | Moins de 12 mois (%) | Garçons (%) |
---|---|---|---|---|---|---|
Ludwig 1984 | 20 | 5,8 | 3 | 75 | – | 70 |
Duhaime et al., 1987 | 48 | 7,85 | – | – | 65 | |
King 2000 | 364 | – | 4,6 | – | – | 56 |
Ettaro et al., 2004 | 89 | – | – | – | 87 | 53 |
Mireau 2005 | 404 | 5,4 | 62,8 | 94,7 | 72,7 | |
Talvick 2006 | 26 | 3,9 | – | – | 100 | 77 |
CPSP 2008 | 220 | 5 | 58 | 75 | 58 |