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Sagittal synostosis: does choice of intervention and its timing affect the long-term aesthetic and neurodevelopmental outcome? A single-institution study of 167 children

Adnan-Mustafiz Chowdhury, Ryan Patel, Adikarige Haritha Dulanka Silva, David J. Dunaway, Noor ul Owase Jeelani, Juling Ong, Richard Hayward, and Greg James

OBJECTIVE

Sagittal craniosynostosis (SC) is the most commonly encountered form of craniosynostosis. Despite its relative frequency, there remains significant heterogeneity in both operative management and follow-up between centers and a relative paucity of long-term outcome data in the literature. At the authors’ institution, families of children presenting with SC are offered the following options: 1) conservative management with ophthalmic surveillance, 2) minimally invasive surgery at < 6 months of age (spring-assisted cranioplasty [SAC]) or 3) calvarial vault remodeling at any age (CVR). The authors reviewed outcomes for all children presenting with SC during a 5-year period, regardless of the treatment received.

METHODS

Consecutive children born between January 1, 2008, and December 31, 2012, presenting with SC were identified, and detailed chart reviews were undertaken. Demographic, surgical, perioperative, head shape, scar, and neurodevelopmental (behavioral, education, speech, and language) data were analyzed. The cohort was divided by type of surgery (none, SAC, or CVR) and by age at surgery (early, defined as ≤ 6 months; or late, defined as > 6 months) for comparison purposes.

RESULTS

A total of 167 children were identified, 129 boys and 38 girls, with a median age at presentation of 5.0 (range 0.4–135) months. Three families opted for conservative management. Of the 164 children who underwent surgery, 83 underwent SAC, 76 underwent CVR, and 5 underwent a "hybrid" procedure (CVR with springs). At a median age of 7.0 (range 0.5–12.3) years, there was no significant difference in concerns regarding head shape, scar, or neurodevelopmental outcomes between the early and late intervention groups over all procedures performed, or between the early or late SAC and CVR cohorts. There were more head shape concerns in the SAC group than in the CVR group overall (25.7% vs 11.8%, respectively; p = 0.026), although most of these concerns were minor and did not require revision.

CONCLUSIONS

In this cohort, regardless of operative intervention and timing of intervention, infants achieved similar neurodevelopmental outcomes. Minimally invasive surgery (SAC) appears to result in less complete correction of head shape than CVR, but this may be balanced by advantages in reduced operative time, hospitalization, and blood loss. SAC was equal to CVR in neuropsychological outcomes.

In Journal of Neurosurgery: Pediatrics Volume 31: Issue 2 (Feb 2023)

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Endoscopic strip craniectomy with molding helmet therapy versus spring-assisted cranioplasty for nonsyndromic single-suture sagittal craniosynostosis: a systematic review

Alexandra Valetopoulou, Maria Constantinides, Simon Eccles, Juling Ong, Richard Hayward, David Dunaway, Noor ul Owase Jeelani, Greg James, and Adikarige Haritha Dulanka Silva

OBJECTIVE

Endoscopic strip craniectomy with postoperative molding helmet therapy (ESC-H) and spring-assisted cranioplasty (SAC) are commonly used minimally invasive techniques for correction of nonsyndromic sagittal craniosynostosis, but it is unclear which, if either, is superior. Therefore, the authors undertook a systematic review to compare ESC-H with SAC for the surgical management of nonsyndromic single-suture sagittal craniosynostosis.

METHODS

Studies were identified through a systematic and comprehensive search of four databases (Embase, MEDLINE, and two databases in the Cochrane Library). Databases were searched from inception until February 19, 2021. Pediatric patients undergoing either ESC-H or SAC for the management of nonsyndromic single-suture sagittal craniosynostosis were included. Systematic reviews and meta-analyses, single-patient case reports, mixed cohorts of nonsyndromic and syndromic patients, mixed cohorts of different craniosynostosis types, and studies in which no outcomes of interest were reported were excluded. Outcomes of interest included reoperations, blood transfusion, complications, postoperative intensive care unit (ICU) admission, operative time, estimated blood loss, length of hospital stay, and cephalic index. Pooled summary cohort characteristics were calculated for each outcome of interest. Methodological quality was assessed using the Newcastle-Ottawa Scale. The study was reported in accordance with the 2020 PRISMA statement.

RESULTS

Twenty-two studies were eligible for inclusion in the review, including 1094 patients, of whom 605 (55.3%) underwent ESC-H and 489 (44.7%) underwent SAC for nonsyndromic sagittal craniosynostosis. There was no difference between the pooled estimates of the ESC-H and SAC groups for operative time, length of stay, estimated blood loss, and cephalic index. There was no difference between the groups for reoperation rate and complication rate. However, ESC-H was associated with a higher blood transfusion rate and higher postoperative ICU admission.

CONCLUSIONS

The available literature does not demonstrate superiority of either ESC-H or SAC, and outcomes are broadly similar for the treatment of nonsyndromic sagittal craniosynostosis. However, the evidence is limited by single-center retrospective studies with low methodological quality. There is a need for international multicenter randomized controlled trials comparing both techniques to gain definitive and generalizable data.

In Journal of Neurosurgery: Pediatrics Volume 30: Issue 4 (Oct 2022)

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Editorial. Disparities in access to healthcare and the neurosurgeon

Richard Hayward

In Neurosurgical Focus Volume 50 (2021): Issue 4 (Apr 2021): Controversies in the Management of Single-Suture Craniosynostosis

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Introduction. Controversies in the management of single-suture craniosynostosis

Concezio Di Rocco, John R. W. Kestle, Richard Hayward, and Jesse A. Taylor

In Neurosurgical Focus Volume 50 (2021): Issue 4 (Apr 2021): Controversies in the Management of Single-Suture Craniosynostosis

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Craniopharyngioma in children: trends from a third consecutive single-center cohort study

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.

In Journal of Neurosurgery: Pediatrics Volume 25: Issue 3 (Mar 2020)

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Surgical decision-making in the management of childhood tumors of the CNS disseminated at presentation

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.

In Journal of Neurosurgery: Pediatrics Volume 21: Issue 6 (Jun 2018)

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Assessment of spring cranioplasty biomechanics in sagittal craniosynostosis patients

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.

In Journal of Neurosurgery: Pediatrics Volume 20 (2017): Issue 5 (Nov 2017)

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Letter to the Editor. Raised intracranial pressure and cognitive delay in craniosynostosis

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

In Journal of Neurosurgery: Pediatrics Volume 20 (2017): Issue 5 (Nov 2017)

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Connecting raised intracranial pressure and cognitive delay in craniosynostosis: many assumptions, little evidence

Richard Hayward, Jonathan Britto, David Dunaway, and Owase Jeelani

In Journal of Neurosurgery: Pediatrics Volume 18 (2016): Issue 2 (Aug 2016)

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Raised intracranial pressure in Crouzon syndrome: incidence, causes, and management

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.

In Journal of Neurosurgery: Pediatrics Volume 17 (2016): Issue 4 (Apr 2016)