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Aaron Hockley, Michael K. Tso, Mohammed A. Almekhlafi, Abhay K. Lodha, Robin Clegg, Jeremy Luntley, Muneer Eesa, and John H. Wong

OBJECTIVE

Vein of Galen aneurysmal malformations (VGAMs) in infancy have a poor natural history if left untreated. Their high-flow nature can preclude safe and accurate therapeutic vessel occlusion and the risk of inadvertent pulmonary embolism is predominant. The authors describe the technique of rapid cardiac ventricular pacing for inducing transient hypotension to facilitate the controlled embolization of VGAMs.

METHODS

Initial transjugular venous access was obtained for placing temporary pacing leads for rapid cardiac ventricular pacing immediately prior to embolization. Definitive transarterial embolization procedures for the VGAMs were then performed in the same setting in which liquid embolic agents or coils were used.

RESULTS

Beginning in 2010, a total of five procedures were performed in three infants. Transvenous rapid cardiac ventricular pacing was successfully achieved to induce systemic transient flow arrest in all but two attempts, and facilitated partial embolization with n-butyl cyanoacrylate (n-BCA) and coils in all procedures. Ventricular fibrillation occurred twice in one patient and was successfully reversed with defibrillation on both occasions. One patient failed to improve and died from refractory heart failure. Two patients stabilized following staged embolization.

CONCLUSIONS

Rapid transvenous cardiac ventricular pacing can be considered to induce transient hypotension and facilitate controlled embolization in challenging high-flow VGAMs.

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Nicholas A. Pickersgill, Gary B. Skolnick, Sybill D. Naidoo, Matthew D. Smyth, and Kamlesh B. Patel

OBJECTIVE

Metrics used to quantify preoperative severity and postoperative outcomes for patients with sagittal synostosis include cephalic index (CI), the well-known standard, and the recently described adjusted cephalic index (aCI), which accounts for altered euryon location. This study tracks the time course of these measures following endoscopic repair with orthotic helmet therapy. The authors hypothesize that CI and aCI show significant regression following endoscope-assisted repair.

METHODS

CT scans or 3D photographs of patients with nonsyndromic sagittal synostosis treated before 6 months of age by endoscope-assisted strip craniectomy and postoperative helmet therapy (n = 41) were reviewed retrospectively at three time points (preoperatively, 0–2 months after helmeting, and > 24 months postoperatively). The CI and aCI were measured at each time point.

RESULTS

Mean CI and aCI increased from 71.8 to 78.2 and 62.7 to 72.4, respectively, during helmet treatment (p < 0.001). At final follow-up, mean CI and aCI had regressed significantly from 78.2 to 76.5 and 72.4 to 69.7, respectively (p < 0.001). The CI regressed in 33 of 41 cases (80%) and aCI in 39 of 41 cases (95%). The authors observed a mean loss of 31% of improvement in aCI achieved through treatment. A strong, positive correlation existed between CI and aCI (R = 0.88).

CONCLUSIONS

Regression following endoscope-assisted strip craniectomy with postoperative helmet therapy commonly occurs in patients with sagittal synostosis. Future studies are required to determine whether duration of helmet therapy or modifications in helmet design affect regression.

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Darryl Lau, Cecilia L. Dalle Ore, Kenneth W. Martin, James F. Policy, and Peter P. Sun

Pedicle aplasia is an uncommon congenital anomaly most frequently involving the absence of a single pedicle at a single vertebral level. Bilateral pedicle aplasia at multiple levels is exceedingly rare and has only been described once previously in the literature. While single-level pedicle aplasia is often asymptomatic and discovered incidentally, pedicle aplasia of multiple levels may produce severe spinal deformities and neurological deficits. Due to the rarity of this condition, optimal management remains uncertain. In this case report, the authors describe the surgical management of a healthy 9-year-old boy who presented with frequent falls, difficulty running, and severe thoracic kyphotic deformity and was found to have bilateral pedicle aplasia from T3 to T9. A review of the literature regarding pedicle aplasia is also presented.

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Xiaolei Hao, Ruie Feng, Yalan Bi, Yuhan Liu, Chunde Li, Tao Lu, and Yongji Tian

Erdheim-Chester disease (ECD) is a rare non–Langerhans cell form of histiocytosis that can affect the central nervous system. ECD predominantly affects adults, and only a few pediatric cases have been reported. The co-occurrence of ECD and Langerhans cell histiocytosis (LCH) is exceedingly rare. An 11-year-old boy, who was diagnosed with LCH 7 years previously, presented with multiple giant intracranial lesions. At the time of his initial diagnosis, only one intracranial lesion was observed, and it began to enlarge. Currently, up to 7 intracranial lesions can be observed in this patient. However, the diagnosis of ECD was not confirmed until this most recent open resection. The BRAF V600E mutation was detected in both LCH and ECD lesions. Dabrafenib therapy exhibited dramatic efficacy in this pediatric patient. This case represents the first successful application of dabrafenib in a pediatric patient with intracranial ECD lesions as well as mixed ECD and LCH. In this article, the authors describe the intricate diagnosis and treatment processes in this patient. Recent studies regarding treatment with BRAF inhibitors for neurological involvement in mixed ECD and LCH are also reviewed.

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David F. Jimenez, Michael J. McGinity, and Constance M. Barone

OBJECTIVE

The objective of this study was to present the authors’ 19-year experience treating metopic craniosynostosis by using an endoscopy-assisted technique and postoperative cranial orthotic therapy. The authors also aimed to provide a comprehensive, comparative statistical analysis of minimally invasive surgery (MIS) versus open surgery in reports previously published in the literature (through 2014) regarding only patients with metopic synostosis.

METHODS

A total of 141 patients with single-suture metopic nonsyndromic craniosynostosis sutures were treated between 1998 and 2017 by endoscopically resecting the synostosed bone followed by postoperative custom cranial orthosis use. All data used in the case series were collected prospectively and stored in a secure database. A comprehensive literature review was performed that included all previous case series reporting common surgical performance measures. A statistical comparison of traditional open methods versus MIS techniques was performed with regard to age, length of hospital stay (LOS), surgical time, estimated blood loss (EBL), and transfusion rate.

RESULTS

The mean age at the time of surgery in the current series was 4.1 months. The mean EBL was 33 ml (range 5–250 ml). One patient underwent an intraoperative blood transfusion and 5 underwent postoperative blood transfusion for a total transfusion rate of 4.3%. The mean operating time was 56 minutes. Ninety-eight percent of patients were discharged on the 1st postoperative day. The median size of the removed synostosed bone was 0.6 cm × 10 cm. The primary goal of achieving correction of the forehead deformity was obtained in 94% of the patients. One hundred eight patients presented with hypotelorism (76.6%). Those with a minimum 1-year follow-up achieved 99% correction (n = 97). Six patients younger than 1 year had not achieved correction at the time of follow-up (6%). There were no intra- or postoperative deaths. One patient had a temporary contact dermatitis to the helmet materials and 2 patients developed pseudomeningoceles, which were successfully treated with a lumbar drain and/or spinal tap. No patient required nor underwent a second surgical procedure. Regarding the previously published literature through 2014, the reported EBL in patients who underwent MIS versus traditional open methods was 54.7 ml versus 224 ml, respectively. The reported average age for patients undergoing MIS versus traditional open methods was 3.8 months versus 11.5 months. The average LOS for patients undergoing MIS versus traditional open methods was 1.7 days versus 3.7 days. The average reported surgical time for those undergoing MIS versus traditional open methods was 66.7 minutes versus 223.7 minutes. The transfusion rate for patients undergoing MIS versus traditional open methods was 22% versus 77%. All of the above differences demonstrated statistical significance.

CONCLUSIONS

The authors’ team has safely and effectively performed 141 metopic craniosynostosis corrections over the past 19 years, with excellent outcomes. Literature review comparing metrics such as LOS, EBL, operating time, and transfusion rate demonstrates a statistically significant improvement in all commonly reported measurements. MIS techniques are safe and effective and should be offered to parents and patients as an option at craniofacial centers treating this condition.

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Kristopher G. Hooten, Klaus Werner, Mohamad A. Mikati, and Carrie R. Muh

Cortical tubers associated with tuberous sclerosis complex (TSC) are potential epileptic foci that are often amenable to resective or ablative surgeries, and controlling seizures at a younger age may lead to improved functional outcomes. MRI-guided laser interstitial thermal therapy (MRgLITT) has become a popular minimally invasive alternative to traditional craniotomy. Benefits of MRgLITT include the ability to monitor the ablation in real time, a smaller incision, shorter hospital stay, reduced blood loss, and reduced postoperative pain. To place the laser probe for LITT, however, stereotaxy is required—which classically involves head fixation with cranial pins. This creates a relative minimum age limit of 2 years old because it demands a mature skull and fused cranial sutures. A novel technique is presented for the application of MRgLITT in a 6-month-old infant for the treatment of epilepsy associated with TSC. To the authors’ knowledge this is the youngest patient treated with laser ablation. The authors used a frameless navigation technique with a miniframe tripod system and intraoperative reference points. This technique expands the application of MRgLITT to younger patients, which may lead to safer surgical interventions and improved outcomes for these children.

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Christopher M. Bonfield, Jade Basem, D. Douglas Cochrane, Ash Singhal, and Paul Steinbok

OBJECTIVE

At British Columbia Children’s Hospital (BCCH), pediatric patients with nonsyndromic craniosynostosis are admitted directly to a standard surgical ward after craniosynostosis surgery. This study’s purpose was to investigate the safety of direct ward admission and to examine the rate at which patients were transferred to the intensive care unit (ICU), the cause for the transfer, and any patient characteristics that indicate higher risk for ICU care.

METHODS

The authors retrospectively reviewed medical records of pediatric patients who underwent single-suture or nonsyndromic craniosynostosis repair from 2011 to 2016 at BCCH. Destination of admission from the operating room (i.e., ward or ICU) and transfer to the ICU from the ward were evaluated. Patient characteristics and operative factors were recorded and analyzed.

RESULTS

One hundred fourteen patients underwent surgery for single-suture or nonsyndromic craniosynostosis. Eighty surgeries were open procedures (cranial vault reconstruction, frontoorbital advancement, extended-strip craniectomy) and 34 were minimally invasive endoscope-assisted craniectomy (EAC). Sutures affected were sagittal in 66 cases (32 open, 34 EAC), coronal in 20 (15 unilateral, 5 bilateral), metopic in 23, and multisuture in 5. Only 5 patients who underwent open procedures (6%) were initially admitted to the ICU from the operating room; the reasons for direct admission were as follows: the suggestion of preoperative elevated intracranial pressure, pain control, older-age patients with large reconstruction sites, or a significant medical comorbidity. Overall, of the 107 patients admitted directly to the ward (75 who underwent an open surgery, 32 who underwent an EAC), none required ICU transfer.

CONCLUSIONS

Overall, the findings of this study suggest that patients with nonsyndromic craniosynostosis can be managed safely on the ward and do not require postoperative ICU admission. This could potentially increase cost savings and ICU resource utilization.

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Pierre-Olivier Champagne, Florence Cayouette, Anne Lortie, Jean-Claude Decarie, and Alexander G. Weil

The occipital and marginal sinuses, when present, must be sacrificed in order to open the dura in most posterior fossa surgeries in the pediatric population, including posterior fossa decompression for Type I Chiari malformation (CM-I) with duraplasty. Apart from the immediate risk of hemorrhage, the voluntary occlusion of this structure is almost universally well tolerated. The authors report a case of intracranial hypertension following the sacrifice of occipital and marginal sinuses following posterior fossa decompression with duraplasty for CM-I. The specific draining pattern variant of the occipital and marginal sinuses leading to this complication as well as avoidance and management strategies of this condition are discussed.

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Kavelin Rumalla, Kyle A. Smith, Vijay Letchuman, Mrudula Gandham, Rachana Kombathula, and Paul M. Arnold

OBJECTIVE

Posttraumatic seizures (PTSs) are the most common complication following a traumatic brain injury (TBI) and may lead to posttraumatic epilepsy. PTS is well described in the adult literature but has not been studied extensively in children. Here, the authors utilized the largest nationwide registry of pediatric hospitalizations to report the national incidence, risk factors, and outcomes associated with PTS in pediatric TBI.

METHODS

The authors queried the Kids’ Inpatient Database (KID) using ICD-9-CM codes to identify all patients (age < 21 years) who had a primary diagnosis of TBI (850.xx–854.xx) and a secondary diagnosis of PTS (780.33, 780.39). Parameters of interest included patient demographics, preexisting comorbidities, hospital characteristics, nature of injury (open/closed), injury type (concussion, laceration/contusion, subarachnoid hemorrhage, subdural hematoma, or epidural hematoma), loss of consciousness (LOC), surgical management (Clinical Classification Software code 1 or 2), discharge disposition, in-hospital complications, and in-hospital mortality. The authors utilized the IBM SPSS statistical package (version 24) for univariate comparisons, as well as the identification of independent risk factors for PTS in multivariable analysis (alpha set at < 0.05).

RESULTS

The rate of PTS was 6.9% among 124,444 unique patients hospitalized for TBI. The utilization rate of continuous electroencephalography (cEEG) was 0.3% and increased between 2003 (0.1%) and 2012 (0.7%). The most common etiologies of TBI were motor vehicle accident (n = 50,615), accidental fall (n = 30,847), and blunt trauma (n = 13,831). However, the groups with the highest rate of PTS were shaken infant syndrome (41.4%), accidental falls (8.1%), and cycling accidents (7.4%). In multivariable analysis, risk factors for PTS included age 0–5 years (compared with 6–10, 11–15, and 16–20 years), African American race (OR 1.4), ≥ 3 preexisting comorbidities (OR 4.0), shaken infant syndrome (OR 4.4), subdural hematoma (OR 1.6), closed-type injury (OR 2.3), brief LOC (OR 1.4), moderate LOC (OR 1.5), and prolonged LOC with baseline return (OR 1.8). Surgically managed patients were more likely to experience PTS (OR 1.5) unless they were treated within 24 hours of admission (OR 0.8). PTS was associated with an increased likelihood of in-hospital complications (OR 1.7) and adverse (nonroutine) discharge disposition (OR 1.2), but not in-hospital mortality (OR 0.5). The overall utilization rate of cEEG was 1.3% in PTS patients compared with 0.2% in patients without PTS. Continuous EEG monitoring was associated with higher rates of diagnosed PTS (35.4% vs 6.8%; OR 4.9, p < 0.001).

CONCLUSIONS

PTS is common in children with TBI and is associated with adverse outcomes. Independent risk factors for PTS include younger age (< 5 years), African American race, increased preexisting comorbidity, prolonged LOC, and injury pattern involving cortical exposure to blood products. However, patients who undergo urgent surgical evacuation are less likely to develop PTS.

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Neena I. Marupudi, Carolyn Harris, Tanya Pavri, Brenna Mell, Rasanjeet Singh, Steven D. Ham, and Sandeep Sood

OBJECTIVE

Lumboperitoneal (LP) shunts have a role not only in pseudotumor cerebri, but also in patients with slit-like ventricles who are treated with CSF shunting on a chronic basis. Hesitation to utilize LP shunts is based on previous conventional beliefs including the tendency for overdrainage, difficulties accessing the shunt to tap or revise, and risk of progressive cerebellar tonsillar herniation. The authors hypothesized that the use of horizontal-vertical (HV) valves may reduce the risk of these complications, particularly overdrainage and development of Chiari malformation.

METHODS

All pediatric cases involving patients treated with an LP shunt at the Children’s Hospital of Michigan were reviewed in this retrospective case series. A total of 143 patients with hydrocephalus were treated with LP shunts from 1997–2015 (follow-up range 8 months–8 years, median 4.2 years). Patients with pseudotumor cerebri underwent placement of an LP shunt as a primary procedure. In patients with slit ventricles from chronically treated hydrocephalus or repeated shunt malfunctions from proximal catheter obstruction, a lumbar drain was inserted to assess candidacy for conversion to an LP shunt. In patients who tolerated the lumbar drain and demonstrated communication of the ventricles with the spinal cisterns, treatment was converted to an LP shunt. All patients included in the series had undergone initial shunt placement between birth and age 16 years.

RESULTS

In 30% of patients (n = 43), LP shunts were placed as the initial shunt treatment; in 70% (n = 100), treatment was converted to LP shunts from ventriculoperitoneal (VP) shunts. The patients’ age at insertion of or conversion to an LP shunt ranged from 1 to 43 years (median 8.5 years). Of the patients with clear pre-LP and post-LP shunt follow-up imaging, none were found to develop an acquired Chiari malformation. In patients with pre-existing tonsillar ectopia, no progression was noted on follow-up MRIs of the brain in these patients after LP shunt insertion. In our LP shunt case series, no patient presented with acute deterioration from shunt malfunction.

CONCLUSIONS

Conversion to an LP shunt may minimize acute deterioration from shunt malfunction and decrease morbidity of repeated procedures in patients with chronically shunt-treated hydrocephalus and small ventricles. In comparison to previously published case series of LP shunt treatment, the use of LP shunts in conjunction with HV valves may decrease the overall risk of cerebellar tonsillar herniation. The use of an LP shunt may be an alternative in the management of slit ventricles when VP shunting repeatedly fails.