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Andrew T. Hale, David P. Stonko, Jaims Lim, Oscar D. Guillamondegui, Chevis N. Shannon, and Mayur B. Patel

OBJECTIVE

Pediatric traumatic brain injury (TBI) is common, but not all injuries require hospitalization. A computational tool for ruling in patients who will have a clinically relevant TBI (CRTBI) would be valuable, providing an evidence-based way to safely discharge children who are at low risk for a CRTBI. The authors hypothesized that an artificial neural network (ANN) trained on clinical and radiologist-interpreted imaging metrics could provide a tool for identifying patients likely to suffer from a CRTBI.

METHODS

The authors used the prospectively collected, publicly available, multicenter Pediatric Emergency Care Applied Research Network (PECARN) TBI data set. All patients under the age of 18 years with TBI and admission head CT imaging data were included. The authors constructed an ANN using clinical and radiologist-interpreted imaging metrics in order to predict a CRTBI, as previously defined by PECARN: 1) neurosurgical procedure, 2) intubation > 24 hours as direct result of the head trauma, 3) hospitalization ≥ 48 hours and evidence of TBI on a CT scan, or 4) death due to TBI.

RESULTS

Among 12,902 patients included in this study, 480 were diagnosed with CRTBI. The authors’ ANN had a sensitivity of 99.73% with precision of 98.19%, accuracy of 97.98%, negative predictive value of 91.23%, false-negative rate of 0.0027%, and specificity for CRTBI of 60.47%. The area under the receiver operating characteristic curve was 0.9907.

CONCLUSIONS

The authors are the first to utilize artificial intelligence to predict a CRTBI in a clinically meaningful manner, using radiologist-interpreted CT information, in order to identify pediatric patients likely to suffer from a CRTBI. This proof-of-concept study lays the groundwork for future studies incorporating iterations of this algorithm directly into the electronic medical record for real-time, data-driven predictive assistance to physicians.

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Kathryn V. Isaac, John G. Meara, and Mark R. Proctor

The authors compared the effectiveness of two main surgical techniques used for treating sagittal craniosynostosis (SC): endoscopic suturectomy (ES) and cranial vault remodeling (CVR). The safety, head growth, and aesthetic results following ES and CVR were compared by reviewing the charts of more than 200 patients. By comparing the effectiveness of these two treatments, this study will help guide selection of the optimal surgical treatment for patients with SC.

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Michael D. Cearns, Samantha Hettige, Paolo De Coppi, and Dominic N. P. Thompson

OBJECTIVE

It is well established that Currarino syndrome (CS) may be associated with spinal dysraphism. Here, the authors report on 10 CS patients with dysraphic anomalies who had undergone a combined neurosurgical and general surgical approach to repair the dysraphic anomalies and resect the presacral mass in a single operation. They discuss the spectrum of spinal dysraphism that may coexist in CS in the context of its developmental etiology.

METHODS

Children with a confirmed CS diagnosis who had undergone the combined operative approach were identified from a departmental database. Presenting features were recorded and preoperative imaging was analyzed to record features of the presacral mass and the dysraphic anomalies. The histopathological nature of the resected presacral mass and the outcomes postoperatively and at the last follow-up were reviewed.

RESULTS

Between 2008 and 2015, 10 patients presented with CS, 9 with constipation. Median age at the time of surgery was 1.3 years. Six of the 10 patients had anorectal malformation consisting of anal stenosis, rectal stenosis, or imperforate anus. Spinal anomalies included anterior meningocele (5 cases), low-lying conus (8), terminal syrinx (4), fatty filum (5), caudal lipoma (3), and intraspinal cyst (1). In all cases, the lumbosacral spinal canal was accessed via a midline approach with laminoplasty, allowing spinal cord untethering and repair of the dysraphic anomalies. Following dural closure, the incision was extended inferiorly to incorporate a posterior sagittal approach to resect the presacral mass. The histopathological nature of the mass was mature teratoma (8 cases), complex hamartomatous malformation (1), or neurenteric cyst (1). There were no new instances of neurological deterioration, with most instances of persisting morbidity related to constipation (6 cases) or neurogenic bladder dysfunction (8). There were no infective complications, no instances of cerebrospinal fluid fistula, no recurrences of the presacral mass, and no cases of retethering of the spinal cord.

CONCLUSIONS

Although not part of the original triad, spinal dysraphic anomalies are common in CS and in keeping with a disorder of secondary neurulation. Lumbosacral MRI is an essential investigation when CS is suspected. Children are at risk of sphincter impairment due to the anorectal malformation; however, both spinal cord tethering and compression from the presacral mass may further compromise long-term continence. A combined operative approach to repair the dysraphic anomalies and resect the presacral mass is described with good postoperative and long-term outcomes.

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Allison Strickland, Cordell M. Baker, R. Michael Siatkowski, and Timothy B. Mapstone

The authors present a case of Chiari type 1.5 malformation with the uncommon presenting symptoms of esotropia and diplopia due to divergence insufficiency in a 12-year-old girl. Imaging at initial diagnosis revealed cerebellar herniation with extension of the tonsils to the C2 vertebral body, a retroflexed odontoid, and a small cervical syrinx. The patient was initially treated with an uncomplicated Chiari malformation decompression without dural opening. Repeat imaging revealed an adequate decompression. Three months postoperatively the patient’s diplopia recurred and she underwent repeat posterior fossa decompression with dural opening and duraplasty. Following repeat decompression with dural opening and duraplasty, the patient’s diplopia had not recurred by the 2-year follow-up.

https://thejns.org/doi/abs/10.3171/2018.5.PEDS1886

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Andrew F. Alalade, Giovanni Briganti, Jo-Lyn Mckenzie, Mitesh Gandhi, Damian Amato, Benedict J. Panizza, and James Bowman

The fossa navicularis is an anatomical variant of the skull base thought to be a rare finding. It represents a bony depression in the skull base. The authors here report the case of a fossa navicularis magna in a 9-year-old female who had been treated for recurrent episodes of meningitis.

A literature review was also done to highlight the unique features and clinical importance of this distinctive radiological skull base finding. The literature search covered papers from the 19th century up to 2018. Earlier authors described “fossa navicularis” as a very rare skull base finding. So far, only three cases of fossa navicularis with associated clival or intracranial infection have been reported in the literature. This is the fourth reported case, and the defect was closed endoscopically via a transnasal route. This morphological skull base anomaly should be considered in the differential diagnoses for an unexplained skull base infective pathology.

Skull base surgeons should be aware of the existence of the fossa navicularis because of its clinical importance in rendering a prompt diagnosis and appropriate treatment.

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Takuro Sakai, Masanori Sasaki, Yuko Kataoka-Sasaki, Shinichi Oka, Masahito Nakazaki, Shinobu Fukumura, Masaki Kobayashi, Hiroyuki Tsutsumi, Jeffery D. Kocsis, and Osamu Honmou

The authors intravenously infused mesenchymal stem cells (MSCs) into a rat model of neonatal hypoxia-ischemia and found improvements in functional outcome, increased brain volume, and enhanced synaptogenesis. The results of this animal study suggest that the intravenous administration of MSCs should be further explored as a potential treatment for patients suffering from cerebral palsy after hypoxic-ischemic encephalopathy.

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Jun T. Park, Guadalupe Fernandez Baca Vaca, Rachel Tangen, and Jonathan Miller

Resection of the hippocampus ipsilateral to the verbal memory–dominant hemisphere frequently results in severe memory deficits. In adults with epilepsy, multiple hippocampal transections (MHTs) have resulted in excellent seizure outcome with preservation of verbal memory. The authors report the first detailed case of a child undergoing MHTs for mesial temporal lobe epilepsy. A 13-year-old right-handed boy had intractable seizures characterized by epigastric discomfort evolving to unresponsiveness and chewing automatisms, lasting 1 minute and occurring 2–3 times weekly, sometimes ending in a generalized tonic-clonic seizure. He had no seizure risk factors and nonfocal examination results. Interictal electroencephalography (EEG) showed frequent left temporal epileptiform discharges (maximum FT9) and intermittent slowing. Video EEG, FDG-PET, and 1.5-T MRI were nonlocalizing. Neuropsychological evaluation suggested left temporal lobe dysfunction. A stereo-EEG investigation using 8 electrodes localized the seizure onset zone to the anterior mesial temporal region, immediately involving the hippocampus. The temporal pole and amygdala were resected en bloc with 3 MHTs. Comparison of neuropsychological tests 4 months before and 6 months after the surgery showed a significant decline only in confrontational naming and no significant change in verbal memory. Six and a half years later, the patient remains seizure free with no antiepileptic drugs. In children with established hemispheric dominance suffering from mesial temporal lobe epilepsy, MHTs may be an option.

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Cecilia L. Dalle Ore, Robert C. Rennert, Alexander J. Schupper, Brandon C. Gabel, David Gonda, Bradley Peterson, Lawrence F. Marshall, Michael Levy, and Hal S. Meltzer

OBJECTIVE

Pediatric traumatic subarachnoid hemorrhage (tSAH) often results in intensive care unit (ICU) admission, the performance of additional diagnostic studies, and ICU-level therapeutic interventions to identify and prevent episodes of neuroworsening.

METHODS

Data prospectively collected in an institutionally specific trauma registry between 2006 and 2015 were supplemented with a retrospective chart review of children admitted with isolated traumatic subarachnoid hemorrhage (tSAH) and an admission Glasgow Coma Scale (GCS) score of 13–15. Risk of blunt cerebrovascular injury (BCVI) was calculated using the BCVI clinical prediction score.

RESULTS

Three hundred seventeen of 10,395 pediatric trauma patients were admitted with tSAH. Of the 317 patients with tSAH, 51 children (16%, 23 female, 28 male) were identified with isolated tSAH without midline shift on neuroimaging and a GCS score of 13–15 at presentation. The median patient age was 4 years (range 18 days to 15 years). Seven had modified Fisher grade 3 tSAH; the remainder had grade 1 tSAH. Twenty-six patients (51%) had associated skull fractures; 4 involved the petrous temporal bone and 1 the carotid canal. Thirty-nine (76.5%) were admitted to the ICU and 12 (23.5%) to the surgical ward. Four had an elevated BCVI score. Eight underwent CT angiography; no vascular injuries were identified. Nine patients received an imaging-associated general anesthetic. Five received hypertonic saline in the ICU. Patients with a modified Fisher grade 1 tSAH had a significantly shorter ICU stay as compared to modified Fisher grade 3 tSAH (1.1 vs 2.5 days, p = 0.029). Neuroworsening was not observed in any child.

CONCLUSIONS

Children with isolated tSAH without midline shift and a GCS score of 13–15 at presentation appear to have minimal risk of neuroworsening despite the findings in some children of skull fractures, elevated modified Fisher grade, and elevated BCVI score. In this subgroup of children with tSAH, routine ICU-level care and additional diagnostic imaging may not be necessary for all patients. Children with modified Fisher grade 1 tSAH may be particularly unlikely to require ICU-level admission. Benefits to identifying a subgroup of children at low risk of neuroworsening include improvement in healthcare efficiency as well as decreased utilization of unnecessary and potentially morbid interventions, including exposure to ionizing radiation and general anesthesia.

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James L. West, Madison Arnel, Atilio E. Palma, John Frino, Alexander K. Powers, and Daniel E. Couture

OBJECTIVE

Spine surgery is less common in children than adults. These surgeries, like all others, are subject to complications such as bleeding, infection, and CSF leak. The rate of incidental durotomy in the pediatric population, and its associated complications, has scarcely been reported in the literature.

METHODS

This is a retrospective chart review of all pediatric patients operated on at Wake Forest Baptist Health from 2012 to 2017 who underwent spine surgeries. The authors excluded any procedures with intended durotomy, such as tethered cord release or spinal cord tumor resection.

RESULTS

From 2012 to 2017, 318 pediatric patients underwent surgery for a variety of indications, including adolescent idiopathic scoliosis (51.9%), neuromuscular scoliosis (27.4%), thoracolumbar fracture (2.83%), and other non–fusion-related indications (3.77%). Of these patients, the average age was 14.1 years, and 71.0% were female. There were 6 total incidental durotomies, resulting in an overall incidence of 1.9%. The incidence was 18.5% in revision operations, compared to 0.34% for index surgeries. Comparison of the revision cohort to the durotomy cohort revealed a trend toward increased length of stay, operative time, and blood loss; however, the trends were not statistically significant. The pedicle probe was implicated in 3 cases and the exact cause was not ascertained in the remaining 3 cases. The 3 durotomies caused by pedicle probe were treated with bone wax; 1 was treated with dry Gelfoam application and 2 were treated with primary repair. Only 1 patient had a persistent leak postoperatively that eventually required wound revision.

CONCLUSIONS

Incidental durotomy is an uncommon occurrence in the pediatric spinal surgery population. The majority occurred during placement of pedicle screws, and they were easily treated with bone wax at the time of surgery. Awareness of the incidence, predisposing factors, and treatment options is important in preventing complications and disability.

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Nisha Gadgil, Sandi Lam, Monika Pyarali, Michael Paldino, I-Wen Pan, and Robert C. Dauser

OBJECTIVE

Numerous surgical procedures facilitate revascularization of the ischemic brain in patients with moyamoya disease. Dural inversion is a technique in which flaps of dura mater centered around the middle meningeal artery are inverted, encouraging the formation of a rich collateral blood supply. This procedure has been used in combination with encephaloduroarteriosynangiosis for more than 20 years at the authors’ institution for the treatment of pediatric moyamoya disease. The objective of this study was to describe the clinical and radiographic outcomes for a cohort of consecutive pediatric moyamoya patients undergoing dural inversion.

METHODS

Clinical and radiographic data on patients who had undergone dural inversion in the period from 1997 to 2016 were reviewed. Univariate and multivariate logistic regression and Kaplan-Meier analyses were performed to assess the risk of postoperative stroke, functional outcome, and the angiographic degree of revascularization.

RESULTS

Dural inversion was performed on 169 hemispheres in 102 patients. Median follow-up was 4.3 years. Six patients (3.6% of hemispheres) suffered postoperative ischemic or hemorrhagic stroke. Overall mortality was 1.0%. Good postoperative neurological status (modified Rankin Scale [mRS] score ≤ 2) was observed in 90 patients (88%); preoperative and postoperative mRS scores showed significant improvement (p < 0.001). Eighty-six percent of hemispheres had Matsushima grade A or B revascularization. Younger age was associated with postoperative stroke and poor functional outcome. Patients with secondary moyamoya syndrome had a significantly worse radiographic outcome. The cumulative 5-year Kaplan-Meier risk for stroke was 6.4%.

CONCLUSIONS

Dural inversion is a useful technique of cerebral revascularization in pediatric moyamoya disease. A 20-year experience demonstrates the safety and efficacy of this technique with a relatively low rate of postoperative stroke, good functional outcomes, and favorable angiographic results.