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Hani J. Marcus, Fahid T. Rasul, Ziad Hussein, Stephanie E. Baldeweg, Helen A. Spoudeas, Richard Hayward, Noor ul Owase Jeelani, Dominic Thompson, Joan P. Grieve, Neil L. Dorward and Kristian Aquilina

OBJECTIVE

The management of children with craniopharyngioma has evolved over time, with a trend toward less invasive neurosurgical approaches as surgeons have sought to balance oncological control and treatment-related morbidity. To this end, the aim of this study was to evaluate the safety and effectiveness of the current management of children with craniopharyngioma compared to the previous management methods used at the authors’ treatment center.

METHODS

A prospectively maintained database was searched over a 14-year period between January 1, 2005, and December 31, 2018, to identify all children 17 years of age or younger with a new diagnosis of craniopharyngioma. A retrospective case note review was performed for each child to extract data on the presentation, investigation, treatment, and outcome of their illness. Morbidity was assessed in the same fashion as in previous cohorts, according to the following categories: visual loss, pituitary dysfunction, hypothalamic dysfunction, neurological deficits, and cognitive impairment.

RESULTS

In total, 59 children were identified with craniopharyngioma during the study period. A total of 92 operations were performed, including cyst drainage (35/92; 38.0%), craniotomy and resection (30/92; 32.6%), and transsphenoidal resection (16/92; 17.4%). Approximately two-thirds of all operations were performed using image guidance (66/92; 71.7%) and one-third were performed using endoscopy (27/92; 29.3%). The majority of children had adjuvant therapy comprising proton beam therapy (18/59; 30.5%) or conventional radiotherapy (16/59; 27.1%). The median follow-up duration was 44 months (range 1–142 months), and approximately one-half of the children had no evidence of residual disease on MRI studies (28/59; 47.5%). Of the remaining 31 children, there was a reduction in the volume of residual disease in 8 patients (8/59; 13.6%), stable residual disease in 18 (18/59; 30.5%), and tumor growth in 5 patients (5/59; 8.5%). There was significantly reduced morbidity (p < 0.05) in all categories in the current cohort compared with our last cohort (1996–2004).

CONCLUSIONS

The authors’ institutional experience of pediatric craniopharyngioma confirms a trend toward less invasive neurosurgical procedures, most of which are now performed with the benefit of image guidance or endoscopy. Moreover, the authors have identified an expanding role for more targeted radiotherapy for children with residual disease. These advances have allowed for tumor control comparable to that achieved in previous cohorts, but with significantly reduced morbidity and mortality.

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Matthew A. Kirkman, Richard Hayward, Kim Phipps and Kristian Aquilina

OBJECTIVE

It is relatively unusual for pediatric CNS tumors to be disseminated at presentation, and the literature on the clinical features, management, and outcomes of this specific group is scarce. Surgical management in this population is often challenging, particularly in the presence of hydrocephalus. The authors present their recent experience of treating pediatric CNS tumors that were disseminated at presentation, and they compare these lesions with focal tumors.

METHODS

The authors performed a retrospective review of prospectively collected data on children presenting to a tertiary center between 2003 and 2016 inclusive.

RESULTS

Of 361 children with CNS tumors, the authors identified 53 patients with disease dissemination at presentation (male/female ratio 34:19, median age 3.8 years, age range 7 days to 15.6 years) and 308 without dissemination at presentation (male/female ratio 161:147, median age 5.8 years, age range 1 day to 16.9 years). Five tumor groups were studied: medulloblastoma (disseminated n = 29, focal n = 74), other primitive neuroectodermal tumor (n = 8, n = 17), atypical teratoid rhabdoid tumor (n = 8, n = 22), pilocytic astrocytoma (n = 6, n = 138), and ependymoma (n = 2, n = 57). The median follow-up duration in survivors was not significantly different between those with disease dissemination at presentation (64.0 months, range 5.2–152.0 months) and those without it (74.5 months, range 4.7–170.1 months) (p > 0.05). When combining data from all 5 tumor groups, dissemination status at presentation was significantly associated with a higher risk of requiring CSF diversion, a higher surgical complication rate, and a reduced likelihood of achieving gross-total resection of the targeted lesion (all variables p < 0.05). Differences between the 5 tumor groups were evident. No factors that predicted the need for permanent CSF diversion following temporary external ventricular drainage were identified on multivariate analysis, and there was no clear superiority of either ventriculoperitoneal shunt surgery or endoscopic third ventriculostomy as a permanent CSF diversion procedure.

CONCLUSIONS

Tumor type and dissemination status at initial presentation significantly affect outcomes across a range of measures. The management of hydrocephalus in patients with CNS tumors is challenging, and further prospective studies are required to identify the optimal CSF diversion strategy in this population.

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Alessandro Borghi, Silvia Schievano, Naiara Rodriguez Florez, Roisin McNicholas, Will Rodgers, Allan Ponniah, Greg James, Richard Hayward, David Dunaway and N. u. Owase Jeelani

OBJECTIVE

Scaphocephaly secondary to sagittal craniosynostosis has been treated in recent years with spring-assisted cranioplasty, an innovative approach that leverages the use of metallic spring distractors to reshape the patient skull. In this study, a population of patients who had undergone spring cranioplasty for the correction of scaphocephaly at the Great Ormond Street Hospital for Children was retrospectively analyzed to systematically assess spring biomechanical performance and kinematics in relation to spring model, patient age, and outcomes over time.

METHODS

Data from 60 patients (49 males, mean age at surgery 5.2 ± 0.9 months) who had received 2 springs for the treatment of isolated sagittal craniosynostosis were analyzed. The opening distance of the springs at the time of insertion and removal was retrieved from the surgical notes and, during the implantation period, from planar radiographs obtained at 1 day postoperatively and at the 3-week follow-up. The force exerted by the spring to the patient skull at each time point was derived after mechanical testing of each spring model—3 devices with the same geometry but different wire thicknesses. Changes in the cephalic index between preoperatively and the 3-week follow-up were recorded.

RESULTS

Stiffer springs were implanted in older patients (p < 0.05) to achieve the same opening on-table as in younger patients, but this entailed significantly different—higher—forces exerted on the skull when combinations of stiffer springs were used (p < 0.001). After initial force differences between spring models, however, the devices all plateaued. Indeed, regardless of patient age or spring model, after 10 days from insertion, all the devices were open.

CONCLUSIONS

Results in this study provide biomechanical insights into spring-assisted cranioplasty and could help to improve spring design and follow-up strategy in the future.

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Irene Mathijssen, Robbin de Goederen, Sarah L. Versnel, Koen F. M. Joosten, Marie-Lise C. van Veelen and Robert C. Tasker

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Richard Hayward, Jonathan Britto, David Dunaway and Owase Jeelani

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Ghassan S. Abu-Sittah, Owase Jeelani, David Dunaway and Richard Hayward

OBJECT

Patients with Crouzon syndrome (CS) are at risk for developing raised intracranial pressure (ICP), which has the potential to impair both vision and neurocognitive development. For this reason, some experts recommend early prophylactic cranial vault expansion on the basis that if ICP is not currently raised, it is likely to become so. The aim of this study was to examine the justification for such a policy. This was done by analyzing the incidence, causes, and subsequent risk of recurrence in a series of patients with CS, in whom raised ICP was treated only after it had been diagnosed.

METHODS

This study was a retrospective review of the medical records and imaging data of patients with a clinical diagnosis of CS.

RESULTS

There were 49 patients in the study, of whom 30 (61.2%) developed at least 1 episode of raised ICP. First episodes occurred at an average age of 1.42 years and were attributable to craniocerebral disproportion/venous hypertension (19 patients), hydrocephalus (8 patients), and airway obstruction (3 patients). They were managed, respectively, by vault expansion, ventriculoperitoneal shunt insertion, and airway improvement. Fourteen of the 30 patients developed a second episode of raised ICP an average of 1.42 years after treatment for their initial episode, and 3 patients developed a third episode an average of 3.15 years after that. Causes of subsequent episodes of raised ICP often differed from previous episodes and required different management. Patients who were < 1 year old when the first episode was diagnosed were at increased risk of recurrence.

CONCLUSIONS

Although the incidence of raised ICP in CS is high, it did not occur in nearly 40% of children during the course of this study. The several possible causes of CS require different management and may vary from episode to episode. The authors recommend an expectant policy toward these children with careful clinical, ophthalmological, respiratory, and radiological monitoring for raised ICP, reserving intervention for when it has been detected and the appropriate treatment can be initiated.

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Richard Hayward, Jonathan A. Britto, David Dunaway, Robert Evans, Noor ul Owase Jeelani and Dominic Thompson

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Aabir Chakraborty, Darach Crimmins, Richard Hayward and Dominic Thompson

Object

The prevalence of shunt-dependent hydrocephalus in patients with myelomeningocele has been reported to be in the region of 85%, and shunt-related complications are a significant cause of morbidity and mortality in these patients. Since 1997 the authors have adopted a stringent policy with respect to shunt placement in patients with myelomeningocele, reserving this treatment for those with symptomatic hydrocephalus, severe ventricular dilation at the time of presentation, and/or unequivocal progressive ventriculomegaly after primary closure. They report their experience.

Methods

The authors reviewed all cases of myelomeningocele involving patients who presented to their institution over a 10-year period. They excluded cases in which the primary closure was carried out at another institution or in which there was not at least 12 months of clinical and imaging follow-up. Data regarding shunt insertion shunt-related complications, and clinical outcome were obtained from a review of the clinical records and analyzed.

Results

Fifty-four cases satisfied the inclusion criteria for this study. Shunt insertion was performed in 28 of these cases (51.9%).

Conclusions

Applying more stringent guidelines for shunt placement, permitting moderate ventricular dilation, and accepting some mild increase in ventricular size after myelomeningocele closure has resulted in a reduced rate of shunt placement compared with previous series. The rate is comparable to that reported following in utero closure of myelomeningocele.

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Sasha Burn, Roxana Gunny, Kim Phipps, Mark Gaze and Richard Hayward

Object

Cavernous hemangiomas (cavernomas) are benign vascular malformations that may cause seizures and/or hemorrhage when they develop in the brain. The incidence of cavernoma development after brain radiotherapy is unknown. The aim of this study was to assess the prevalence of cavernoma formation in patients who had previously received radiotherapy for brain tumors during childhood.

Methods

All patients were identified who were younger than 16 years of age and had undergone radiotherapy for brain tumors within a 16-year period (January 1, 1988–December 31, 2003). The patient data that were ascertained included age at diagnosis, sex, histopathology results, initial preoperative magnetic resonance (MR) imaging results, date of radiotherapy, and date of detection of cavernoma. Children who were followed up for less than 1 month after radiotherapy or who died during treatment were excluded, as were children with brainstem tumors. All patients had undergone diagnostic MR imaging before receiving radiotherapy, and no vascular malformations were revealed.

Of the 379 patients identified, 297 satisfied the inclusion criteria. Ten patients (3.4%) developed cavernomas after radiation therapy. The ages of these patients ranged from 2 to 11 years at the time of radiotherapy (median 7 years), and the latency interval between radiotherapy and cavernoma development was 3 to 102 months (median 37 months).

Conclusions

The prevalence of cavernomas in the present study was more than six times greater than the prevalence rate cited in the literature for this population. The authors conclude that there is an increased risk of developing an intracranial cavernoma after radiotherapy for brain tumors. The possibility of this complication arising should be mentioned when informed consent is sought before treatment using radiotherapy.

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Ali Nader-Sepahi, Adrian T. H. Casey, Richard Hayward, H. Alan Crockard and Dominic Thompson

Object

The aim of this study was to audit the treatment of symptomatic atlantoaxial instability in Down syndrome and to assess factors associated with fusion failure in this group of patients.

Methods

The authors reviewed the cases of 12 children with Down syndrome presenting with symptomatic atlantoaxial instability who underwent surgery for internal fixation and fusion. A review of clinical histories, radiological investigations, and details of operative interventions was also performed.

The mode of presentation was acute spinal cord injury (five cases), progressive myelopathy (four cases), and neck pain or stiffness (three cases). The atlantodental interval ranged from 5 to 13 mm. The posterior atlantodental interval at the C-1 level was 9.5 mm (range 6–11 mm). In 10 patients an os odontoideum was present. Translocation of the odontoid process occurred in one patient, and occipitoatlantal instability was also identified in two cases. Twenty-four operations were performed in the 12 patients. A transoral odontoidectomy was required in four children. Successful fusion was demonstrated in seven patients at the first operation. Three of the five patients with acute cord injury made significant functional recovery and were left with no or mild disability.

Conclusions

Additional bone abnormalities at the CVJ are common in the Down syndrome population. Young age at the time of fusion and multiple osseous anomalies pose a higher risk factor in fusion failure. The authors recommend an aggressive surgical approach for management of symptomatic cases of CVJ instability.