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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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Ben Shofty, Liat Ben-Sira, Anat Kesler, George Jallo, Mari L. Groves, Rajiv R. Iyer, Alvaro Lassaletta, Uri Tabori, Eric Bouffet, Ulrich-Wilhelm Thomale, Pablo Hernáiz Driever, Shlomi Constantini and for the Isolated Optic Nerve Abnormalities (IONA) Collaboration

OBJECTIVE

Isolated optic nerve gliomas (IONGs) constitute a rare subgroup of optic pathway gliomas (OPGs). Due to the rarity of this condition and the difficulty in differentiating IONGs from other types of OPGs in most clinical series, little is known about these tumors. Currently, due to lack of evidence, they are managed the same as any other OPG.

METHODS

The authors conducted a multicenter retrospective cohort study aimed at determining the natural history of IONGs. Included were patients with clear-cut glioma of the optic nerve without posterior (chiasmatic/hypothalamic) involvement. At least 1 year of follow-up, 2 MRI studies, and 2 neuro-ophthalmological examinations were required for inclusion.

RESULTS

Thirty-six patients with 39 tumors were included in this study. Age at diagnosis ranged between 6 months and 16 years (average 6 years). The mean follow-up time was 5.6 years. Twenty-five patients had neurofibromatosis Type 1. During the follow-up period, 59% of the tumors progressed, 23% remained stable, and 18% (all with neurofibromatosis Type 1) displayed some degree of spontaneous regression. Fifty-one percent of the patients presented with visual decline, of whom 90% experienced further deterioration. Nine patients were treated with chemotherapy, 5 of whom improved visually. Ten patients underwent operation, and no local or distal recurrence was noted.

CONCLUSIONS

Isolated optic nerve gliomas are highly dynamic tumors. Radiological progression and visual deterioration occur in greater percentages than in the general population of patients with OPGs. Response to chemotherapy may be better in this group, and its use should be considered early in the course of the disease.

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Ori Barzilai, Zvi Lidar, Shlomi Constantini, Khalil Salame, Yifat Bitan-Talmor and Akiva Korn

Intramedullary spinal cord tumors (IMSCTs) represent a rare entity, accounting for 4%–10% of all central nervous system tumors. Microsurgical resection of IMSCTs is currently considered the primary treatment modality. Intraoperative neurophysiological monitoring (IONM) has been shown to aid in maximizing tumor resection and minimizing neurological morbidity, consequently improving patient outcome. The gold standard for IONM to date is multimodality monitoring, consisting of both somatosensory evoked potentials, as well as muscle-based transcranial electric motor evoked potentials (tcMEPs). Monitoring of tcMEPs is optimal when combining transcranial electrically stimulated muscle tcMEPs with D-wave monitoring. Despite continuous monitoring of these modalities, when classic monitoring techniques are used, there can be an inherent delay in time between actual structural or vascular-based injury to the corticospinal tracts (CSTs) and its revelation. Often, tcMEP stimulation is precluded by the surgeon’s preference that the patient not twitch, especially at the most crucial times during resection. In addition, D-wave monitoring may require a few seconds of averaging until updating, and can be somewhat indiscriminate to laterality. Therefore, a method that will provide immediate information regarding the vulnerability of the CSTs is still needed.

The authors performed a retrospective series review of resection of IMSCTs using the tip of an ultrasonic aspirator for continuous proximity mapping of the motor fibers within the spinal cord, along with classic muscle-based tcMEP and D-wave monitoring.

The authors present their preliminary experience with 6 patients who underwent resection of an IMSCT using the tip of an ultrasonic aspirator for continuous proximity mapping of the motor fibers within the spinal cord, together with classic muscle-based tcMEP and D-wave monitoring. This fusion of technologies can potentially assist in optimizing resection while preserving neurological function in these challenging surgeries.

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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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Liat Ben-Sira, Noam Goder, Haim Bassan, Shlomi Lifshits, Yaniv Assaf and Shlomi Constantini

OBJECT

The object of this study was to use diffusion tensor imaging (DTI) to evaluate and characterize white matter changes in hydrocephalus.

METHODS

The authors performed a retrospective analysis of DTI in a cohort of patients with hydrocephalus (n = 35), 19 of whom had both pre- and postsurgical imaging studies. These patient’s DTI values were compared with values extracted from age-dependent trend lines computed from a healthy subject group (n = 70, age span 14 months-14 years). Several DTI parameters in different regions of interest (ROIs) were evaluated to find the most sensitive parameters for clinical decision making in hydrocephalus.

RESULTS

Compared with healthy controls, patients with active hydrocephalus had a statistically significant change in all DTI parameters. The most sensitive and specific DTI parameter for predicting hydrocephalus was axial diffusivity (λ1) measured at the level of the corona radiata. Diffusion tensor imaging parameters correlated with several conventional radiological parameters in the assessment of hydrocephalus but were not superior to them. There was no convincing correlation between clinical disease severity and DTI parameters. When examining the pre- and postsurgical effect, it was found that DTI may be a sensitive tool for estimating tissue improvement.

CONCLUSIONS

This large-cohort study with a multidisciplinary approach combining clinical, neurological, radiological, and multiple DTI parameters revealed the most sensitive DTI parameters for identifying hydrocephalus and suggested that they may serve as an important tool for the disorder’s quantitative radiological assessment.

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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.

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Shlomi Constantini, Aaron Mohanty, Samuel Zymberg, Sergio Cavalheiro, Conor Mallucci, Dieter Hellwig, Yusuf Ersahin, Hiroshi Mori, Carmelo Mascari, José Aloysio Costa Val, Wolfgang Wagner, Abhaya V. Kulkarni, Spyros Sgouros and Shizuo Oi

Object

Analysis of the safety and morbidity of neuroendoscopic biopsies (NEBs), as well as the reliability in obtaining an accurate diagnosis, has until now been based on studies with relatively small sample sizes. Through the cooperative efforts of several international medical centers, authors of the present study collected data on a large number of patients to obtain better insight into this issue. When possible, they compared pathology obtained through an NEB with the “gold-standard” pathology obtained in open surgery.

Methods

Thirteen randomly chosen medical centers in 9 countries collected data for patients who had undergone NEB, which were then analyzed for universal complications, bleeding, navigation technique, pathology, mismatch between biopsy results and final diagnosis, and a number of other potentially influential factors.

Results

Data for 293 patients were analyzed. Sixty percent of the patients were male, and patient ages ranged from 0.1 to 78.7 years (median age 20.4 years). The most common tumor locations were pineal (33.1%), thalamic (16.7%), tectal (13%), and hypothalamic (4.4%). Fifty percent of the tumors were larger than 20 mm, 36% were between 10 and 20 mm, and 14% were smaller than 10 mm. Intraoperative bleeding was seen in 275 patients (94%). The amount of blood was noted as mild in 75%, moderate in 13%, and severe in 6%. Infection occurred in 8 patients (3%). Death occurred in 1 patient (0.3%), which was caused by severe intraoperative bleeding. Biopsies were informative in 265 patients (90.4%). Seventy-eight patients (26.6%) had open surgery following the NEB. For these patients, the pathology results from the NEB were compared with those from the open surgery that followed. In 14 cases (17.9%) there was disagreement on the pathology. Of these cases, a meaningful mismatch, in which the erroneous NEB pathology could have led to an inappropriate management decision, occurred in 9 cases (11.5%). Most of these meaningful mismatches were lesions diagnosed as low-grade or pilocytic astrocytoma on the NEB and later proved to be high-grade astrocytoma (4 cases) and 1 case each of meningioma, cavernoma, primitive neuroectodermal tumor, neurocysticercosis, and pineocytoma.

Conclusions

In experienced hands, NEBs can be performed with low morbidity and mortality, providing meaningful pathological data for the majority of patients with a wide range of tumor types, locations, and presentations. These biopsies also offer other advantages, such as the ability to perform concomitant endoscopic third ventriculostomy and septum pellucidotomy. However, due caution must be maintained, since pathology obtained from an NEB, as with stereotactic biopsies, may be subject to sampling errors, especially when the results seem to indicate a low-grade glial tumor.

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Jonathan Roth, Marian M. Bercu and Shlomi Constantini

Hypothalamic hamartomas (HHs) are typically located within the vicinity of the third ventricle. They can be attached to the walls of the third ventricle, within the interpeduncular cistern (third ventricle floor), and/or attached to the mammillary bodies and hypothalamus. Depending on their location, resection is performed either through the third ventricle, approaching from above, or via a frontotemporal craniotomy (pterional or frontoorbital), approaching from below. “Above” approaches typically include the transcallosal–anterior interforniceal approach, and recently, purely endoscopic approaches performed transforaminally.

The authors present a combined open and endoscopic approach for resection of HHs located within the third ventricle. They used this approach in 2 young girls with relatively small lateral and third ventricles. Following an interhemispheric, transcallosal approach and exposure of the right foramen of Monro, an endoscope was inserted through the foramen, which enabled safe resection of the HH.

The main advantage of the combined approach is when the lateral and third ventricles are relatively small, making a purely endoscopic approach more challenging and possibly riskier.