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Claudia L. Craven, Martyn Cooke, Clare Rangeley, Samuel J. M. M. Alberti, and Mary Murphy

OBJECTIVE

One of the greatest challenges of pediatric neurosurgery training is balancing the training needs of the trainee against patient safety and parental expectation. The traditional “see one, do one, teach one” approach to training is no longer acceptable in pediatric neurosurgery. The authors have developed the baby Modeled Anatomical Replica for Training Young Neurosurgeons (babyMARTYN). The development of this new training model is described, its feasibility as a training tool is tested, and a new approach of integrating simulation into day-to-day training is suggested.

METHODS

In part 1 (development), a prototype skull was developed using novel model-making methods. In part 2 (validation), 18 trainee neurosurgeons (at various stages in training) performed the following 4 different procedures: 1) evacuation of a posterior fossa hematoma; 2) pterional craniotomy; 3) tapping of the fontanelle to obtain a CSF specimen; and 4) external ventricular drain insertion. Completion of the procedural stages (scored using a curriculum-based checklist) was used to test the feasibility of babyMARTYN as a training tool. Likert scale–based questionnaires were used to assess the model for face and content validity. Training benefit was assessed using pre- and posttraining ratings on the Physician Performance Diagnostic Inventory Scale (PPDIS). To determine the significance of improvement in median PPDIS score, the Wilcoxon matched-pairs signed-rank test was performed.

RESULTS

In part 1 (development), the model was successfully developed with good fidelity. In part 2 (validation), the validation data demonstrated feasibility, face, and content validity. The PPDIS score significantly increased for all groups after babyMARTYN training, thereby indicating a potential future role for babyMARTYN in the training of pediatric neurosurgeons.

CONCLUSIONS

This recent collaborative neurosurgical development by the Royal College of Surgeons of England is designed to supplement current neurosurgical training. High-fidelity, portable, operation-specific models enable preoperative planning and have the potential to be used in an operating room environment prior to novel operations. A “see one, simulate one, do one” approach for pediatric neurosurgical training using babyMARTYN is suggested.

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Claudia L. Craven, Roshini Ramkumar, Linda D’Antona, Simon D. Thompson, Lewis Thorne, Laurence D. Watkins, and Ahmed K. Toma

OBJECTIVE

Chronic ventriculomegaly in the absence of raised intracranial pressure (ICP) is a known entity in adult hydrocephalus practice. The natural history and indication for treatment is, however, poorly defined. A highly heterogeneous group, some adults with ventriculomegaly are asymptomatic, while others have life-threatening deteriorations. The authors hypothesized that the various presentations can be subtyped and represent different stages of decompensation. A cluster analysis was performed on a cohort of patients with chronic ventriculomegaly with the aim of elucidating typical clinical characteristics and outcomes in chronic ventriculomegaly in adults.

METHODS

Data were collected from 79 patients with chronic ventriculomegaly referred to a single center, including demographics, presenting symptoms, and 24-hour ICP monitoring (ICPM). A statistical cluster analysis was performed to determine the presence of subgroups.

RESULTS

Four main subgroups and one highly dissimilar group were identified. Patients with ventriculomegaly commonly have a perinatal event followed by one of four main presentations: 1) incidental ventriculomegaly with or without headache; 2) highly symptomatic presentation (including reduced consciousness) and raised ICP; 3) early presenting with symptoms of headache and nausea (with abnormal pulsatility); and 4) late presenting with features common to normal pressure hydrocephalus. Each symptomatic group has characteristic radiological features, ICPM, and responses to treatment.

CONCLUSIONS

Cluster analysis has identified subgroups of adult patients with ventriculomegaly. Such groups may represent various degrees of decompensation. Surgical interventions may not be equally effective across the subgroups, presenting an avenue for further research. The identified subtypes provide further insight into the natural history of this lesser studied form of hydrocephalus.

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Claudia L. Craven, Paul Gissen, Rebecca Bower, Laura Lee, Kristian Aquilina, and Dominic N. P. Thompson

OBJECTIVE

Late infantile neuronal ceroid lipofuscinosis type 2 (CLN2) is a rare autosomal recessive disease caused by tripeptidyl peptidase 1 enzyme deficiency. At the authors’ center, the medication cerliponase alfa is administered every 2 weeks via the intracerebroventricular (ICV) route. This requires the placement of a ventricular access device (VAD) or reservoir and frequent percutaneous punctures of this device over the child’s lifetime. In this study, the authors audited the longevity and survival of these VADs and examined the causes of device failure.

METHODS

A single-center survival analysis of VAD insertions and revisions (January 2014 through June 2020) was conducted. All children received cerliponase alfa infusions through a VAD. Patient characteristics and complications were determined from a prospectively maintained surgical database and patient records. For the VAD survival analysis, the defined endpoint was when the device was removed or changed. Reservoir survival was assessed using Kaplan-Meier curves and the log-rank (Cox-Mantel) test.

RESULTS

A total of 17 patients had VADs inserted for drug delivery; median (range) age at first surgery was 4 years 4 months (1 year 8 months to 15 years). Twenty-six VAD operations (17 primary insertions and 9 revisions) were required among these 17 patients. Twelve VAD operations had an associated complication, including CSF infection (n = 6) with Propionibacterium and Staphylococcus species being the most prevalent organisms, significant surgical site swelling preventing infusion (n = 3), leakage/wound breakdown (n = 2), and catheter obstruction (n = 1). There were no complications or deaths associated with VAD insertion. The median (interquartile range) number of punctures was 59.5 (7.5–82.0) for unrevised VADs (n = 17) versus 2 (6–87.5) for revised VADs (n = 9) (p = 0.70). The median survival was 301 days for revisional reservoirs (n = 9) versus 2317 days for primary inserted reservoirs (n = 17) (p = 0.019).

CONCLUSIONS

In the context of the current interest in intrathecal drug delivery for rare metabolic disorders, the need for VADs is likely to increase. Auditing the medium- to long-term outcomes associated with these devices will hopefully result in their wider application and may have potential implications on the development of new VAD technologies. These results could also be used to counsel parents prior to commencement of therapy and VAD implantation.

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Hasan Asif, Claudia L. Craven, Almas H. Siddiqui, Syed N. Shah, Samir A. Matloob, Lewis Thorne, Fergus Robertson, Laurence D. Watkins, and Ahmed K. Toma

OBJECTIVE

Idiopathic intracranial hypertension (IIH) is commonly associated with venous sinus stenosis. In recent years, transvenous dural venous sinus stent (DVSS) insertion has emerged as a potential therapy for resistant cases. However, there remains considerable uncertainty over the safety and efficacy of this procedure, in particular the incidence of intraprocedural and delayed complications and in the longevity of sinus patency, pressure gradient obliteration, and therapeutic clinical outcome. The aim of this study was to determine clinical, radiological, and manometric outcomes at 3–4 months after DVSS in this treated IIH cohort.

METHODS

Clinical, radiographic, and manometric data before and 3–4 months after DVSS were reviewed in this single-center case series. All venographic and manometric procedures were performed under local anesthesia with the patient supine.

RESULTS

Forty-one patients underwent DVSS venography/manometry within 120 days. Sinus pressure reduction of between 11 and 15 mm Hg was achieved 3–4 months after DVSS compared with pre-stent baseline, regardless of whether the procedure was primary or secondary (after shunt surgery). Radiographic obliteration of anatomical stenosis correlating with reduction in pressure gradients was observed. The complication rate after DVSS was 4.9% and stent survival was 87.8% at 120 days. At least 20% of patients developed restenosis following DVSS and only 63.3% demonstrated an improvement or resolution of papilledema.

CONCLUSIONS

Reduced venous sinus pressures were observed at 120 days after the procedure. DVSS showed lower complication rates than shunts, but the clinical outcome data were less convincing. To definitively compare the outcomes between DVSS and shunts in IIH, a randomized prospective study is needed.