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The shaken baby syndrome

A clinical, pathological, and biomechanical study

Ann-Christine Duhaime, Thomas A. Gennarelli, Lawrence E. Thibault, Derek A. Bruce, Susan S. Margulies and Randall Wiser

✓ Because a history of shaking is often lacking in the so-called “shaken baby syndrome,” diagnosis is usually based on a constellation of clinical and radiographic findings. Forty-eight cases of infants and young children with this diagnosis seen between 1978 and 1985 at the Children's Hospital of Philadelphia were reviewed. All patients had a presenting history thought to be suspicious for child abuse, and either retinal hemorrhages with subdural or subarachnoid hemorrhages or a computerized tomography scan showing subdural or subarachnoid hemorrhages with interhemispheric blood. The physical examination and presence of associated trauma were analyzed; autopsy findings for the 13 fatalities were reviewed. All fatal cases had signs of blunt impact to the head, although in more than half of them these findings were noted only at autopsy. All deaths were associated with uncontrollably increased intracranial pressure.

Models of 1-month-old infants with various neck and skull parameters were instrumented with accelerometers and shaken and impacted against padded or unpadded surfaces. Angular accelerations for shakes were smaller than those for impacts by a factor of 50. All shakes fell below injury thresholds established for subhuman primates scaled for the same brain mass, while impacts spanned concussion, subdural hematoma, and diffuse axonal injury ranges. It was concluded that severe head injuries commonly diagnosed as shaking injuries require impact to occur and that shaking alone in an otherwise normal baby is unlikely to cause the shaken baby syndrome.

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Roger J. Packer, Leslie N. Sutton, Kantilal M. Patel, Ann-Christine Duhaime, Steven Schiff, Steven R. Weinstein, William D. Gaillard, Joan A. Conry and Luis Schut

✓ Detailed preoperative electroencephalographic (EEG) studies are now recommended for children with seizures and cortical tumors to define seizure foci prior to surgery. To develop a historical perspective for better evaluation of results from series reporting tumor removal combined with resection of seizure foci, the authors reviewed seizure outcome in 60 children with seizures and low-grade neoplasms treated consecutively since 1981 by surgical resection without concomitant EEG monitoring or electrocortical mapping. Forty-seven of the 60 tumors were totally or near-totally resected; 45 patients were seizure-free and two were significantly improved 1 year following surgery. Of the 50 children in this series with more than five seizures prior to surgery, 36 were seizure-free, two were significantly improved, and 12 were not improved. Factors associated with poor seizure control included a parietal tumor location, a partial tumor resection, and a history of seizures for more than 1 year prior to surgery. The children at highest risk for poor seizure control at 2 years had experienced seizures for more than 1 year prior to surgery and had undergone partial resection of their parietal low-grade glial tumors or gangliogliomas. In contradistinction, the best seizure control was seen in patients with totally resected low-grade gliomas or gangliogliomas who had experienced seizures for less than 1 year (concordance rates for being seizure-free ranged from 78% to 86%). Long-term seizure control remained excellent. These results suggest that seizure control can be obtained 2 years following tumor surgery in the majority of children with presumed tumors after extensive tumor resection without concomitant EEG monitoring or electrocortical mapping.

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Ann-Christine Duhaime, Susan S. Margulies, Susan R. Durham, Maureen M. O'Rourke, Jeffrey A. Golden, Sunil Marwaha and Ramesh Raghupathi

Object. The goal of this study was to investigate the relationship between maturational stage and the brain's response to mechanical trauma in a gyrencephalic model of focal brain injury. Age-dependent differences in injury response might explain certain unique clinical syndromes seen in infants and young children and would determine whether specific therapies might be particularly effective or even counterproductive at different ages.

Methods. To deliver proportionally identical injury inputs to animals of different ages, the authors have developed a piglet model of focal contusion injury by using specific volumes of rapid cortical displacement that are precisely scaled to changes in size and dimensions of the growing brain. Using this model, the histological response to a scaled focal cortical impact was compared at 7 days after injury in piglets that were 5 days, 1 month, and 4 months of age at the time of trauma. Despite comparable injury inputs and stable physiological parameters, the percentage of hemisphere injured differed significantly among ages, with the youngest animals sustaining the smallest lesions (0.8%, 8.4%, and 21.5%, for 5-day-, 1-month-, and 4-month-old animals, respectively, p = 0.0018).

Conclusions. These results demonstrate that, for this particular focal injury type and severity, vulnerability to mechanical trauma increases progressively during maturation. Because of its developmental and morphological similarity to the human brain, the piglet brain provides distinct advantages in modeling age-specific responses to mechanical trauma. Differences in pathways leading to cell death or repair may be relevant to designing therapies appropriate for patients of different ages.

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Michael T. Prange, Brittany Coats, Ann-Christine Duhaime and Susan S. Margulies

Object. Rotational loading conditions have been shown to produce subdural hemorrhage and diffuse axonal injury. No experimental data are available with which to compare the rotational response of the head of an infant during accidental and inflicted head injuries. The authors sought to compare rotational deceleration sustained by the head among free falls, from different heights onto different surfaces, with those sustained during shaking and inflicted impact.

Methods. An anthropomorphic surrogate of a 1.5-month-old human infant was constructed and used to simulate falls from 0.3 m (1 ft), 0.9 m (3 ft), and 1.5 m (5 ft), as well as vigorous shaking and inflicted head impact. During falls, the surrogate experienced occipital contact against a concrete surface, carpet pad, or foam mattress. For shakes, investigators repeatedly shook the surrogate in an anteroposterior plane; inflicted impact was defined as the terminal portion of a vigorous shake, in which the surrogate's occiput made contact with a rigid or padded surface. Rotational velocity was recorded directly and the maximum (peak—peak) change in angular velocity and the peak angular acceleration were calculated.

Analysis of variance revealed significant increases in the and associated with falls onto harder surfaces and from higher heights. During inflicted impacts against rigid surfaces, the and were significantly greater than those measured under all other conditions.

Conclusions. Vigorous shakes of this infant model produced rotational responses similar to those resulting from minor falls, but inflicted impacts produced responses that were significantly higher than even a 1.5-m fall onto concrete. Because larger accelerations are associated with an increasing likelihood of injury, the findings indicate that inflicted impacts against hard surfaces are more likely to be associated with inertial brain injuries than falls from a height less than 1.5 m or from shaking.

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Ann-Christine Duhaime, Jill V. Hunter, Loretta L. Grate, Anje Kim, Jeffrey Golden, Eugene Demidenko and Christine Harris

Object. Whether the brain differs in its response to traumatic injury as a function of age remains unclear. To further investigate the age-dependent response of the brain to mechanical trauma, a cortical contusion model scaled for brain growth during maturation was used to study the evolution of injury over time as demonstrated on serial magnetic resonance (MR) imaging studies in piglets of different ages.

Methods. Sixteen Yorkshire piglets received scaled cortical contusions. Animals were either 5 days (six animals), 1 month (five animals), or 4 months (five animals) of age at injury. These ages correspond developmentally to human infants, toddlers, and early adolescents, respectively. Serial MR imaging examinations, including fluid-attenuated inversion-recovery and T1-, T2-, and diffusion-weighted sequences were performed at 24 hours, 1 week, and 1 month after injury. Lesions were quantified and expressed as a ratio of the lesion volume divided by the volume of the uninjured hemisphere for each animal and each MR sequencing. Differences in relative lesion volume among the varied ages at a single time point and in lesion volume over time at each age were compared. In addition, the relationship between age and evolution of injury were analyzed using a two-compartment mathematical model. Histological features were examined at 1 month postinjury. Despite comparable injury inputs, the youngest animals had lesions whose volumes peaked earlier and resolved more quickly than those in older animals. The intermediate-age piglets (toddler) had the most pronounced swelling of any age group, and the oldest piglets (adolescent) had the latest peak in lesion volume.

Conclusions. Scaled cortical contusions in piglets demonstrated age-dependent differences in injury response, both in magnitude and time course. These observations may shed light on development-related trauma response in the gyrencephalic brain.

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Patricia B. Quebada and Ann-Christine Duhaime

✓The authors present the case of an 11-year-old girl who fell from standing height and experienced sudden cardiorespiratory arrest. A Chiari malformation Type I and a dolichoodontoid process were identified, and a brainstem contusion was found that correlated with the patient's clinical signs and symptoms. The authors hypothesize that the dolichoodontoid in a compromised craniocervical space created a contusion in the apposing brainstem at the time of head impact. The patient was left with permanent vocal cord paresis and spastic quadriparesis. Awareness of this rare occurrence is important in managing these congenital abnormalities and in assessing the risk of deterioration.

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S. Scott Lollis, Eugen B. Hug, David J. Gladstone, Sara Chaffee and Ann-Christine Duhaime

✓The authors present the case of a 20-month-old boy who underwent fractionated radiation therapy to the paranasal sinuses and anterior skull base during treatment for nasopharyngeal parameningeal rhabdomyosarcoma. Subsequent magnetic resonance imaging demonstrated progressive development of a Chiari malformation Type I (CM-I) and partial hypoplasia of the posterior fossa. Since the tonsillar herniation was discovered, the child, now 3 years old, remains asymptomatic except for mild, intermittent neck discomfort. For the time being, his family has elected for him to undergo clinical and neuroimaging follow up. The authors believe this is the first report of a progressive acquired CM-I after cranial irradiation in the pediatric population.