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Jehuda Soleman, Danil A. Kozyrev, Shlomi Constantini, and Jonathan Roth

OBJECTIVE

The aim of this cohort study was to describe and analyze the surgical treatment and outcome of posterior fossa arachnoid cysts (PFACs) in infants.

METHODS

Patients presenting with a PFAC at infancy or prenatally, between the years 2000 and 2019, and who were surgically treated before the age of 2 years, were included in this study. Patient data were retrospectively collected including baseline characteristics and surgical variables. Factors related to revision surgery were analyzed through uni- and multivariate analysis.

RESULTS

Thirty-five patients, of whom 54.3% were male, were included. The cyst was diagnosed prenatally in 23 patients (65.7%). Surgery was typically recommended after a mean cyst follow-up of 3.4 ± 3.9 months, with a mean age at surgery of 6.1 ± 5.1 months. In 54.3% of patients (n = 19), surgery was performed before the age of 6 months. The PFAC was treated purely neuroendoscopically in 57.1% of patients (n = 20), while 28.6% of patients underwent open cyst procedures (n = 10), 5.7% (n = 2) were treated with a shunt, and 8.6% (n = 3) underwent a combined procedure. Additional surgery was required in 31.4% of patients (n = 11; mean 2.36 ± 2.11 surgeries per patient). At the last follow-up (61.40 ± 55.33 months), no mortality or permanent morbidity was seen; radiological improvement was apparent in 83.9% of the patients. Those patients treated before the age of 6 months (p = 0.09) and who presented before surgery with a stable cyst size that was maintained throughout preoperative monitoring (p = 0.08) showed a trend toward higher revision rates after surgical treatment.

CONCLUSIONS

PFACs in infancy may require surgical treatment before the age of 6 months. Navigated endoscopy was a valid surgical option. Overall mortality or permanent morbidity was rare. Additional surgery was required in up to 30% of the patients; younger age and a preoperatively stable cyst might be risk factors for revision surgery.

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Leeat Granek, Shahar Shapira, Shlomi Constantini, and Jonathan Roth

OBJECTIVE

The objective of this study was to explore approaches to intraoperative decision-making in pediatric neurosurgeons when they encounter unexpected events, uncertainties, or complications while operating on children.

METHODS

Twenty-six pediatric neurosurgeons from 12 countries around the world were interviewed using a semistructured interview guide. The grounded theory method of data collection and analysis was used. Analysis involved line-by-line coding and was inductive, with codes and categories emerging from participants’ narratives.

RESULTS

When asked to discuss the strategies they used to make intraoperative decisions, neurosurgeons reported three distinct approaches that formed a philosophy of practice. This included the theme of professional practice—with the subthemes of preparing for uncertainty, doing no harm, being creative and adaptive, being systematic, and working on teams. The second theme pertained to patient and caregiver practices—with the subthemes of shared decision-making and seeing the whole patient. The third theme involved surgeon practice—with the subthemes of cultivating self-awareness and learning from experience.

CONCLUSIONS

Pediatric neurosurgeons have a structured, diverse, and well-thought-out analytical philosophy and practice regarding intraoperative decision-making that encompasses a range of approaches including the following: doing no harm, cultivating self-awareness, and seeing the whole patient; and concrete practices such as preparing in advance for uncertainty, working on teams, and learning from experience. These philosophies and practices can be structured and codified in order to teach residents how to develop intraoperative judgment techniques.

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Jonathan Roth, Or Bercovich, Ashton Roach, Francesco T. Mangano, Arvind C. Mohan, Guillermo Aldave, Howard L. Weiner, Ulrich-Wilhelm Thomale, Andreas Schaumann, Shimrit Uliel-Sibony, and Shlomi Constantini

OBJECTIVE

Resection of brain tumors may lead to new-onset seizures but may also reduce seizure rates in patients presenting with seizures. Seizures are seen at presentation in about 24% of patients with brain tumors. For lesional epilepsy in general, early resection is associated with improved seizure control. However, the literature is limited regarding the occurrence of new-onset postoperative seizures, or rates of seizure control in those presenting with seizures, following resections of extratemporal low-grade gliomas (LGGs) in children.

METHODS

Data were collected retrospectively from 4 large tertiary centers for children (< 18 years of age) who underwent resection of a supratentorial extratemporal (STET) LGG. The patients were divided into 4 groups based on preoperative seizure history: no seizures, up to 2 seizures, more than 2 seizures, and uncontrolled or refractory epilepsy. The authors analyzed the postoperative occurrence of seizures and the need for antiepileptic drugs (AEDs) over time for the various subgroups.

RESULTS

The study included 98 children. Thirty patients had no preoperative seizures, 18 had up to 2, 16 had more than 2, and 34 had refractory or uncontrolled epilepsy. The risk for future seizures was higher if the patient had seizures within 1 month of surgery. The risk for new-onset seizures among patients with no seizures prior to surgery was low. The rate of seizures decreased over time for children with uncontrolled or refractory seizures. The need for AEDs was higher in the more active preoperative seizure groups; however, it decreased with time.

CONCLUSIONS

The resection of STET LGGs in children is associated with a low rate of postoperative new-onset epilepsy. For children with preoperative seizures, even with uncontrolled epilepsy, most have a significant improvement in the seizure activity, and many may be weaned off their AEDs.

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Jonathan Roth, Neal Fischer, David D. Limbrick Jr., Travis CreveCoeur, Liat Ben-Sira, and Shlomi Constantini

OBJECTIVE

Solitary posterior fossa low-grade glial tumors (SPFLGT) in children are rarely associated with leptomeningeal dissemination (LMD). To date, there are no clear guidelines regarding the role of screening and surveillance spinal MRI (sMRI) in children with SPFLGT, at diagnosis or during follow-up periods. The current study reviews a cohort of children with SPFLGT, focusing on sMRI findings.

METHODS

In this binational retrospective study, the authors analyzed 229 patients with SPFLGT treated and followed over 13 years. One hundred twelve children had at least 1 total sMRI screening or surveillance examination. One hundred seventeen had no sMRI, but did not present with clinical spinal signs or symptoms. Collected data included demographics, disease characteristics, radiology, pathology, and clinical follow-up data.

RESULTS

For the 112 children with at least 1 sMRI, the mean duration from diagnosis to first sMRI was 11.73 ± 28.66 months (range 0–165 months). All sMRI scans were conducted as screening examinations, with no spinal-related symptoms. One patient was found to have a sacral intradural lesion concurrent to the brain tumor diagnosis. Over the course of 180 radiological and 533 clinical follow-up years for the 112 patients with sMRI, and 582 clinical follow-up years for the 117 patients with no sMRI, there were no additional cases with spinal tumor spread.

CONCLUSIONS

The yield of screening sMRI in the absence of cranial metastasis, or spinal symptoms, is extremely low. Because preoperative sMRI is recommended for medulloblastomas and ependymomas, it may be logical to acquire. During the follow-up period the authors recommend limiting sMRI in patients without symptoms suggesting a spinal lesion, in patients without known cranial metastases, or recurrence or residual SPFLGT.

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Li-tal Pratt, Shelly I. Shiran, Ronit Precel, Liat Ben-Sira, Gustavo Malinger, Emanuela Cagnano, Danil A. Kozyrev, Shlomi Constantini, and Jonathan Roth

Mature teratomas (MTs) of the posterior fossa are extremely rare. The authors present a case of a prenatal diagnosis of an MT splitting the brainstem. Representative images as well as the clinical and surgical course are presented. Literature regarding “split brainstem” and MT of the posterior fossa is discussed.

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Mitchell Schertz, Shlomi Constantini, Rina Eshel, Adi Hannah Sela, Jonathan Roth, and Aviva Fattal-Valevski

OBJECTIVE

Management of children with large temporal arachnoid cysts (TACs) remains controversial, with limited data available on their neurodevelopmental outcome. The aim of this study was to examine neurodevelopmental outcomes in children with large TACs.

METHODS

In this medical center–based cohort study, 25 patients (19 males) who were diagnosed in childhood with large TACs (9 patients [36%] with a Galassi type II and 16 patients [64%] with a Galassi type III TAC) were examined. The mean ± SD age at assessment was 11.1 ± 5.6 years (range 2.7–22 years). Twelve patients (48%) had right-sided, 12 (48%) had left-sided, and 1 (4%) had bilateral cysts. Nine patients (36%) underwent surgery for the cyst. The siblings of 21 patients (84%) served as control participants. Neurodevelopmental function was assessed using the Adaptive Behavior Assessment System (ABAS), Vanderbilt Behavioral Rating Scale (VBRS), and Developmental Coordination Disorder Questionnaire (DCDQ), and quality of life was measured using the treatment-oriented screening questionnaire (TOSQ). The results of all instruments except for TOSQ were compared with those of the sibling control participants.

RESULTS

The mean ± SD ABAS score of the patients was 93.3 ± 20.09 compared with 98.3 ± 18.04 of the sibling control participants (p = 0.251). Regarding the incidence of poor outcome (ABAS score < 80), there was a trend for more patients with TAC to have poor outcome than the sibling controls (p = 0.058). Patients who underwent surgery scored significantly worse with regard to the VBRS total score compared with those who did not (p = 0.020), but not on ABAS, DCD, or TOSQ. The mean score of the cognitive and psychological items on TOSQ was lower than that for the physical items (p < 0.001).

CONCLUSIONS

Children with a large TAC performed similarly to their sibling control participants in neurodevelopmental function. However, a subgroup of those with cysts did have an increased risk for poor outcomes in general function. Neurodevelopmental assessment should be part of the management of all patients with TAC.

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Kirsten van Baarsen, Jonathan Roth, Natalia Serova, Roger J. Packer, Ben Shofty, Ulrich-W. Thomale, Giuseppe Cinalli, Helen Toledano, Shalom Michowiz, and Shlomi Constantini

OBJECTIVE

Hemorrhage (also known as apoplexy) in optic pathway gliomas (OPGs) is rare. Because of the variable presentations and low incidence of OPG hemorrhages, little is known about their clinical course and the best treatment options. The aim of this work was to review risk factors, clinical course, and treatment strategies of optic glioma hemorrhages in the largest possible number of cases.

METHODS

A total of 34 patients were analyzed. Nine new cases were collected, and 25 were identified in the literature. Data regarding demographics, radiological and histological features, treatment, and outcome were retrospectively reviewed.

RESULTS

The majority of patients were younger than 20 years. Only 3 patients were known to have neurofibromatosis. The histopathological diagnosis was pilocytic astrocytoma in the majority of cases. Five patients had intraorbital hemorrhages, whereas 29 patients had intracranial hemorrhage; the majority of intracranial bleeds were treated surgically. Six patients, all with intracranial hemorrhage, died due to recurrent bleeding, hydrocephalus, or surgical complications. No clear risk factors could be identified.

CONCLUSIONS

Intracerebral OPG hemorrhages have a fatal outcome in 20% of cases. Age, hormonal status, neurofibromatosis involvement, and histopathological diagnosis have been suggested as risk factors for hemorrhage, but this cannot be reliably established from the present series. The goals of surgery should be patient survival and prevention of further neurological and ophthalmological deterioration.

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Ori Barzilai, Jonathan Roth, Akiva Korn, and Shlomi Constantini

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Gyang Markus Bot, Shlomi Constantini, and Jonathan Roth

Cavum septum pellucidum (CSP) cysts are relatively rare. The most common presenting symptom is headache, which is thought to be secondary to elevated intracranial pressure. Many CSP cysts are treated surgically; conservative treatment is seldom recommended. The authors describe 3 cases of pediatric CSP cysts that were managed without surgery.

The patients ranged in age from 5 months to 8 years old. Two presented with headaches, which were associated with mild ventricular enlargement in 1 case. Over the course of 5–15 months, 2 cysts became markedly reduced in size, and in one of these 2 cases a substantial reduction in ventricle size was also observed. At last follow-up, all 3 children were asymptomatic.

The authors note that CSP cysts are often associated with headaches. In the absence of hydrocephalus, they recommend conservative management with clinical and radiological follow-up.

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Jonathan Roth and Shlomi Constantini

OBJECT

Tumors leading to occlusion of the sylvian aqueduct include those of pineal, thalamic, and tectal origins. These tumors cause obstructive hydrocephalus and thus necessitate a CSF diversion procedure such as an endoscopic third ventriculostomy (ETV), often coupled with an endoscopic biopsy (EBX). Lesions located posterior to the massa intermedia pose a technical challenge, as the use of a rigid endoscope for performing both an ETV and EBX is limited. The authors describe their experience using a combined rigid and flexible endoscopic procedure through a single bur hole for both procedures in patients with posterior third ventricular tumors.

METHODS

Since January 2012, patients with posterior third ventricular tumors causing hydrocephalus underwent dual ETV and EBX procedures using the combined rigid-flexible endoscopic technique. Following institutional review board approval, data from clinical, radiological, surgical, and pathological records were retrospectively collected.

RESULTS

Six patients 3.5–53 years of age were included. Lesion locations included pineal (n = 3), fourth ventricle (n = 1), aqueduct (n = 1), and tectum (n = 1). The ETV and EBX were successful in all cases. Pathologies included pilocytic astrocytoma, pineoblastoma, ependymoma Grade II, germinoma, low-grade glioneural tumor, and atypical choroid plexus papilloma. One patient experienced an immediate postoperative intraventricular hemorrhage necessitating evacuation of the clots and resection of the tumor, eventually leading to the patient's death.

CONCLUSIONS

The authors recommend using a combined rigid-flexible endoscope for endoscopic third ventriculostomy and biopsy to approach posterior third ventricular tumors (behind the massa intermedia). This technique overcomes the limitations of using a rigid endoscope by reaching 2 distant regions.