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1.
Emerg Infect Dis ; 30(3): 555-559, 2024 Mar.
Article in English | MEDLINE | ID: mdl-38407150

ABSTRACT

A neurocysticercosis-like lesion in an 11-year-old boy in the Netherlands was determined to be caused by the zoonotic Taenia martis tapeworm. Subsequent testing revealed that 15% of wild martens tested in that region were infected with T. martis tapeworms with 100% genetic similarity; thus, the infection source was most likely local.


Subject(s)
Neurocysticercosis , Taenia , Male , Child , Animals , Humans , Neurocysticercosis/diagnostic imaging , Taenia/genetics , Netherlands
2.
J Cell Sci ; 135(11)2022 06 01.
Article in English | MEDLINE | ID: mdl-35535520

ABSTRACT

Sonic hedgehog (SHH) medulloblastoma originates from the cerebellar granule neuron progenitor (CGNP) lineage, which depends on Hedgehog signaling for its perinatal expansion. Whereas SHH tumors exhibit overall deregulation of this pathway, they also show patient age-specific aberrations. To investigate whether the developmental stage of the CGNP can account for these age-specific lesions, we analyzed developing murine CGNP transcriptomes and observed highly dynamic gene expression as a function of age. Cross-species comparison with human SHH medulloblastoma showed partial maintenance of these expression patterns, and highlighted low primary cilium expression as hallmark of infant medulloblastoma and early embryonic CGNPs. This coincided with reduced responsiveness to upstream SHH pathway component Smoothened, whereas sensitivity to downstream components SUFU and GLI family proteins was retained. Together, these findings can explain the preference for SUFU mutations in infant medulloblastoma and suggest that drugs targeting the downstream SHH pathway will be most appropriate for infant patients.


Subject(s)
Cerebellar Neoplasms , Medulloblastoma , Neural Stem Cells , Animals , Cell Proliferation/physiology , Cerebellar Neoplasms/drug therapy , Cerebellar Neoplasms/genetics , Hedgehog Proteins/genetics , Hedgehog Proteins/metabolism , Humans , Medulloblastoma/drug therapy , Medulloblastoma/genetics , Mice , Neural Stem Cells/metabolism
3.
NMR Biomed ; 37(6): e5124, 2024 Jun.
Article in English | MEDLINE | ID: mdl-38403798

ABSTRACT

Advanced intraoperative MR images (ioMRI) acquired during the resection of pediatric brain tumors could offer additional physiological information to preserve healthy tissue. With this work, we aimed to develop a protocol for ioMRI with increased sensitivity for arterial spin labeling (ASL) and diffusion MRI (dMRI), optimized for patient positioning regularly used in the pediatric neurosurgery setting. For ethical reasons, ASL images were acquired in healthy adult subjects that were imaged in the prone and supine position. After this, the ASL cerebral blood flow (CBF) was quantified and compared between both positions. To evaluate the impact of the RF coils setups on image quality, we compared different setups (two vs. four RF coils) by looking at T1-weighted (T1w) signal-to-noise ratio (SNR) and contrast-to-noise ratio (CNR), as well as undertaking a qualitative evaluation of T1w, T2w, ASL, and dMR images. Mean ASL CBF did not differ between the surgical prone and supine positions in any of the investigated regions of interest or the whole brain. T1w SNR (gray matter: p = 0.016, 34% increase; white matter: p = 0.016, 32% increase) and CNR were higher (p = 0.016) in the four versus two RF coils setups (18.0 ± 1.8 vs. 13.9 ± 1.8). Qualitative evaluation of T1w, T2w, ASL, and dMR images resulted in acceptable to good image quality and did not differ statistically significantly between setups. Only the nonweighted diffusion image maps and corticospinal tract reconstructions yielded higher image quality and reduced susceptibility artifacts with four RF coils. Advanced ioMRI metrics were more precise with four RF coils as the standard deviation decreased. Taken together, we have investigated the practical use of advanced ioMRI during pediatric neurosurgery. We conclude that ASL CBF quantification in the surgical prone position is valid and that ASL and dMRI acquisition with two RF coils can be performed adequately for clinical use. With four versus two RF coils, the SNR of the images increases, and the sensitivity to artifacts reduces.


Subject(s)
Magnetic Resonance Imaging , Neurosurgical Procedures , Signal-To-Noise Ratio , Humans , Male , Female , Magnetic Resonance Imaging/methods , Child , Adult , Cerebrovascular Circulation/physiology , Spin Labels , Diffusion Magnetic Resonance Imaging , Brain/diagnostic imaging , Brain/surgery
4.
Childs Nerv Syst ; 40(1): 99-108, 2024 Jan.
Article in English | MEDLINE | ID: mdl-37436473

ABSTRACT

INTRODUCTION: The Cirq robotic alignment system (Brainlab, Munich, Germany) is a manually adjustable electronic arm with a robotic alignment module on its distal end, enabling the neurosurgeon to automatically and accurately align surgical instruments to a preoperatively planned trajectory. In this study, we share our first experiences and results using Cirq for intracranial tumor biopsy in children. METHODS: From May 2021 until October 2022, all consecutive patients that underwent a brain tumor biopsy using Cirq were included and compared to a historical cohort of patients biopsied with the non-robotic system Varioguide (Brainlab, Munich, Germany). Patient-related data, tumor-related data, and surgery-related data were collected. Registration accuracy was calculated for different patient-to-image registration methods. Pre- and postoperative images were fused, and entry error, target error, and angulation error were calculated. RESULTS: Thirty-seven patients, aged 1-19 years, were included (14 with Cirq and 23 with Varioguide). An integrated histopathological and molecular diagnosis was acquired in all cases. Patient-to-image registration was significantly more accurate when based on bone screw fiducials combined with intraoperative CT, as compared to surface matching or skin fiducials. The target error (Euclidian distance) was 5.3 mm for Cirq as compared to 8.3 mm for Varioguide, but this was not statistically significant. Entry error and angulation error were also not significantly different between both groups. CONCLUSION: Intracranial biopsy with the Cirq robotic system is feasible and safe, and its accuracy does not differ from the Varioguide system.


Subject(s)
Brain Neoplasms , Robotic Surgical Procedures , Child , Humans , Robotic Surgical Procedures/methods , Brain Neoplasms/diagnostic imaging , Brain Neoplasms/surgery , Biopsy/methods , Bone Screws , Germany
5.
Childs Nerv Syst ; 2024 Jun 11.
Article in English | MEDLINE | ID: mdl-38862795

ABSTRACT

PURPOSE: The aim of this study was to evaluate the diagnostic value and accuracy of navigated intraoperative ultrasound (iUS) in pediatric oncological neurosurgery as compared to intraoperative magnetic resonance imaging (iMRI). METHODS: A total of 24 pediatric patients undergoing tumor debulking surgery with iUS, iMRI, and neuronavigation were included in this study. Prospective acquisition of iUS images was done at two time points during the surgical procedure: (1) before resection for tumor visualization and (2) after resection for residual tumor assessment. Dice similarity coefficients (DSC), Hausdorff distances 95th percentiles (HD95) and volume differences, sensitivity, and specificity were calculated for iUS segmentations as compared to iMRI. RESULTS: A high correlation (R = 0.99) was found for volume estimation as measured on iUS and iMRI before resection. A good spatial accuracy was demonstrated with a median DSC of 0.72 (IQR 0.14) and a median HD95 percentile of 4.98 mm (IQR 2.22 mm). The assessment after resection demonstrated a sensitivity of 100% and a specificity of 84.6% for residual tumor detection with navigated iUS. A moderate accuracy was observed with a median DSC of 0.58 (IQR 0.27) and a median HD95 of 5.84 mm (IQR 4.04 mm) for residual tumor volumes. CONCLUSION: We found that iUS measurements of tumor volume before resection correlate well with those obtained from preoperative MRI. The accuracy of residual tumor detection was reliable as compared to iMRI, indicating the suitability of iUS for directing the surgeon's attention to areas suspect for residual tumor. Therefore, iUS is considered as a valuable addition to the neurosurgical armamentarium. TRIAL REGISTRATION NUMBER AND DATE: PMCLAB2023.476, February 12th 2024.

6.
Neuroendocrinology ; 113(10): 987-1007, 2023.
Article in English | MEDLINE | ID: mdl-37231961

ABSTRACT

INTRODUCTION: Survival of childhood-onset craniopharyngioma (cCP) is excellent; however, many survivors suffer from hypothalamic-pituitary dysfunction. Growth hormone replacement therapy (GHRT) is of high importance for linear growth and metabolic outcome. Optimal timing for initiation of GHRT in cCP is on debate because of concerns regarding tumor progression or recurrence. METHODS: A systematic review and cohort studys were performed for the effect and timing of GHRT on overall mortality, tumor progression/recurrence, and secondary tumors in cCP. Within the cohort, cCP receiving GHRT ≤1 year after diagnosis were compared to those receiving GHRT >1 year after diagnosis. RESULTS: Evidence of 18 included studies, reporting on 6,603 cCP with GHRT, suggests that GHRT does not increase the risk for overall mortality, progression, or recurrent disease. One study evaluated timing of GHRT and progression/recurrence-free survival and found no increased risk with earlier initiation. One study reported a higher than expected prevalence of secondary intracranial tumors compared to a healthy population, possibly confounded by radiotherapy. In our cohort, 75 of 87 cCP (86.2%) received GHRT for median of 4.9 years [0.0-17.1]. No effect of timing of GHRT was found on mortality, progression/recurrence-free survival, or secondary tumors. CONCLUSION: Although the quality of the evidence is low, the available evidence suggests no effect of GHRT or its timing on mortality, tumor progression/recurrence, or secondary neoplasms in cCP. These results support early initiation of GHRT in cCP aiming to optimize linear growth and metabolic outcome. Prospective studies are needed to increase the level of evidence upon the optimal timing to start GHRT in cCP patients.


Subject(s)
Craniopharyngioma , Human Growth Hormone , Pituitary Neoplasms , Humans , Cohort Studies , Craniopharyngioma/drug therapy , Human Growth Hormone/adverse effects , Neoplasm Recurrence, Local , Hormone Replacement Therapy/adverse effects , Pituitary Neoplasms/drug therapy , Pituitary Neoplasms/pathology , Growth Hormone
7.
Childs Nerv Syst ; 39(4): 1013-1019, 2023 04.
Article in English | MEDLINE | ID: mdl-36790491

ABSTRACT

PURPOSE: This study aimed to establish the incidence of CSF leakage in children and associated complications after intradural spinal surgery in three tertiary neurosurgical referral centers and to describe the treatment strategies applied. METHODS: Patients of 18 years or younger who underwent intradural spinal surgery between 2015 and 2021 in three tertiary neurosurgical referral centers were included. Patients who died or were lost to follow-up within six weeks after surgery were excluded. The primary outcome measure was CSF leakage within six weeks after surgery, defined as leakage of CSF through the skin. Secondary outcome measures included the presence of pseudomeningocele (PMC), meningitis, and surgical site infection (SSI). RESULTS: We included a total of 75 procedures, representing 66 individual patients. The median age in this cohort was 5 (IQR = 0-13 years. CSF leakage occurred in 2.7% (2/75) of procedures. It occurred on days 3 and 21 after the index procedure, respectively. One patient was treated with a pressure bandage and an external lumbar drain on day 4 after diagnosis of the leak, and the other was treated with wound revision surgery on day 1 after the leak occurred. In total, 1 patient developed a PMC without a CSF leak which was treated with wound revision surgery. SSI occurred in 10.7%, which included both cases of CSF leak. CONCLUSIONS: CSF leakage after intradural spinal surgery in the pediatric population is relatively rare (2.7%). Nevertheless, the clinical consequences with respect to secondary complications such as infection and the necessity for invasive treatment are serious.


Subject(s)
Cerebrospinal Fluid Leak , Cerebrospinal Fluid Rhinorrhea , Humans , Child , Infant, Newborn , Infant , Child, Preschool , Adolescent , Cerebrospinal Fluid Leak/etiology , Cerebrospinal Fluid Leak/epidemiology , Neurosurgical Procedures/adverse effects , Neurosurgical Procedures/methods , Surgical Wound Infection/epidemiology , Surgical Wound Infection/etiology , Reoperation , Postoperative Complications/epidemiology , Postoperative Complications/etiology , Retrospective Studies
8.
Childs Nerv Syst ; 38(8): 1479-1485, 2022 08.
Article in English | MEDLINE | ID: mdl-35759029

ABSTRACT

PURPOSE: Cerebellar mutism syndrome (CMS) is a severe neurological complication of posterior fossa tumour surgery in children, and postoperative speech impairment (POSI) is the main component. Left-handedness was previously suggested as a strong risk factor for POSI. The aim of this study was to investigate the relationship between handedness and the risk of POSI. METHODS: We prospectively included children (aged < 18 years) undergoing surgery for posterior fossa tumours in 26 European centres. Handedness was assessed pre-operatively and postoperative speech status was categorised as either POSI (mutism or reduced speech) or habitual speech, based on the postoperative clinical assessment. Logistic regression was used in the risk factor analysis of POSI as a dichotomous outcome. RESULTS: Of the 500 children included, 37 (7%) were excluded from the present analysis due to enrolment at a reoperation; another 213 (43%) due to missing data about surgery (n = 37) and/or handedness (n = 146) and/or postoperative speech status (n = 53). Out of the remaining 250 (50%) patients, 20 (8%) were left-handed and 230 (92%) were right-handed. POSI was observed equally frequently regardless of handedness (5/20 [25%] in left-handed, 61/230 [27%] in right-handed, OR: 1.08 [95% CI: 0.40-3.44], p = 0.882), also when adjusted for tumour histology, location and age. CONCLUSION: We found no difference in the risk of POSI associated with handedness. Our data do not support the hypothesis that handedness should be of clinical relevance in the risk assessment of CMS.


Subject(s)
Cerebellar Diseases , Cerebellar Neoplasms , Infratentorial Neoplasms , Mutism , Cerebellar Diseases/complications , Cerebellar Neoplasms/surgery , Child , Functional Laterality , Humans , Infratentorial Neoplasms/complications , Infratentorial Neoplasms/surgery , Mutism/complications , Postoperative Complications/epidemiology , Postoperative Complications/etiology , Prospective Studies , Risk Factors , Speech
9.
J Neuroophthalmol ; 42(1): e99-e108, 2022 03 01.
Article in English | MEDLINE | ID: mdl-34812765

ABSTRACT

BACKGROUND: Children with a brain tumor are prone to develop visual impairment, which to date is often underestimated and unrecognized. Our aim was to assess the prevalence of ophthalmological evaluation and abnormal ophthalmological findings, and investigate whether demographic and tumor-related characteristics are associated with abnormal ophthalmological findings in children presenting with a primary brain tumor. METHODS: Medical records of all 90 children diagnosed with a primary brain tumor between June 2018 and May 2019 and treated at the Princess Máxima Center for Pediatric Oncology, a tertiary referral center in the Netherlands, were retrospectively reviewed. Univariate regression analysis was used to investigate associations between demographic, tumor-related and clinical characteristics, and abnormal ophthalmological findings. RESULTS: Sixty children (34 male [56.7%]; median [range] age, 9.3 [0-16.9] years) underwent ophthalmological evaluation within 6 weeks before or after diagnosis, 11 children (5 male [45.5%]; median [range] age, 5.7 [0.1-17.2] years) were seen more than 6 weeks before or after diagnosis, and 19 children (7 male [36.8%]; median [range] age, 7.2 [1.9-16.6] years) did not receive ophthalmological evaluation within at least 6 months from diagnosis. A total of 19 children (21.1%) presented with visual symptoms as first sign leading to the diagnosis of a brain tumor. Children who presented with visual symptoms (odds ratio [OR], 22.52; 95% confidence interval [CI], 4.90-103.60) and/or hydrocephalus (OR, 3.60; 95% CI, 1.38-9.36) at diagnosis were more often seen for ophthalmological evaluation. The most common abnormal ophthalmological findings were eye movement disorders (66.0%), papilledema (44.1%), and visual field defects (58.1%). Eye movement disorders occurred more frequently in patients with an infratentorial tumor (OR, 4.71; 95% CI, 1.03-21.65). The risk of papilledema was associated with older age (OR, 1.19; 95% CI, 1.05-1.34), hydrocephalus (OR, 9.63; 95% CI, 2.68-34.61), and infratentorial (OR, 9.11; 95% CI, 1.77-46.78) and supratentorial (OR, 13.13; 95% CI, 1.92-89.52) tumors. CONCLUSIONS: In this study, most children with a primary brain tumor underwent ophthalmological evaluation around diagnosis, 21% of the children were not evaluated. The high prevalence of abnormal ophthalmological findings stresses the importance of early standardized ophthalmological evaluation to detect visual impairment and provide timely treatment to potentially prevent permanent visual loss.


Subject(s)
Brain Neoplasms , Hydrocephalus , Ocular Motility Disorders , Papilledema , Vision, Low , Brain Neoplasms/complications , Brain Neoplasms/diagnosis , Brain Neoplasms/epidemiology , Child , Child, Preschool , Female , Humans , Hydrocephalus/complications , Hydrocephalus/diagnosis , Hydrocephalus/epidemiology , Male , Papilledema/diagnosis , Papilledema/epidemiology , Papilledema/etiology , Retrospective Studies , Vision Disorders/diagnosis , Vision Disorders/epidemiology , Vision Disorders/etiology
10.
Int J Mol Sci ; 23(17)2022 Aug 30.
Article in English | MEDLINE | ID: mdl-36077248

ABSTRACT

Medulloblastoma is a pediatric brain malignancy that consists of four transcriptional subgroups. Structural and numerical aneuploidy are common in all subgroups, although they are particularly profound in Group 3 and Group 4 medulloblastoma and in a subtype of SHH medulloblastoma termed SHHα. This suggests that chromosomal instability (CIN), the process leading to aneuploidy, is an important player in medulloblastoma pathophysiology. However, it is not known if there is ongoing CIN in medulloblastoma or if CIN affects the developing cerebellum and promotes tumor formation. To investigate this, we performed karyotyping of single medulloblastoma cells and demonstrated the presence of distinct tumor cell clones harboring unique copy number alterations, which is suggestive of ongoing CIN. We also found enrichment for processes related to DNA replication, repair, and mitosis in both SHH medulloblastoma and in the highly proliferative compartment of the presumed tumor cell lineage-of-origin, the latter also being sensitive to genotoxic stress. However, when challenging these tumor cells-of-origin with genetic lesions inducing CIN using transgenic mouse modeling, we found no evidence for large chromosomal aberrations in the cerebellum or for medulloblastoma formation. We therefore conclude that without a background of specific genetic mutations, CIN is not tolerated in the developing cerebellum in vivo and, thus, by itself is not sufficient to initiate medulloblastoma.


Subject(s)
Cerebellar Neoplasms , Medulloblastoma , Aneuploidy , Animals , Cerebellar Neoplasms/genetics , Cerebellar Neoplasms/pathology , Cerebellum/metabolism , Chromosomal Instability , Hedgehog Proteins/metabolism , Humans , Medulloblastoma/genetics , Medulloblastoma/pathology , Mice , Mice, Transgenic
11.
Childs Nerv Syst ; 37(5): 1439-1447, 2021 05.
Article in English | MEDLINE | ID: mdl-33538867

ABSTRACT

BACKGROUND: Cerebrospinal fluid (CSF) leakage is a common complication after neurosurgical intervention. It is associated with substantial morbidity and increased healthcare costs. The current systematic review and meta-analysis aim to quantify the incidence of cerebrospinal fluid leakage in the pediatric population and identify its risk factors. METHODS: The authors followed the PRISMA guidelines. The Embase, PubMed, and Cochrane database were searched for studies reporting CSF leakage after intradural cranial surgery in patients up to 18 years old. Meta-analysis of incidences was performed using a generalized linear mixed model. RESULTS: Twenty-six articles were included in this systematic review. Data were retrieved of 2929 patients who underwent a total of 3034 intradural cranial surgeries. Surprisingly, only four of the included articles reported their definition of CSF leakage. The overall CSF leakage rate was 4.4% (95% CI 2.6 to 7.3%). The odds of CSF leakage were significantly greater for craniectomy as opposed to craniotomy (OR 4.7, 95% CI 1.7 to 13.4) and infratentorial as opposed to supratentorial surgery (OR 5.9, 95% CI 1.7 to 20.6). The odds of CSF leakage were significantly lower for duraplasty use versus no duraplasty (OR 0.41 95% CI 0.2 to 0.9). CONCLUSION: The overall CSF leakage rate after intradural cranial surgery in the pediatric population is 4.4%. Risk factors are craniectomy and infratentorial surgery. Duraplasty use is negatively associated with CSF leak. We suggest defining a CSF leak as "leakage of CSF through the skin," as an unambiguous definition is fundamental for future research.


Subject(s)
Plastic Surgery Procedures , Postoperative Complications , Cerebrospinal Fluid Leak/epidemiology , Cerebrospinal Fluid Leak/etiology , Child , Craniotomy/adverse effects , Humans , Neurosurgical Procedures/adverse effects , Postoperative Complications/epidemiology , Postoperative Complications/etiology , Retrospective Studies
12.
Neurosurg Focus ; 51(2): E14, 2021 08.
Article in English | MEDLINE | ID: mdl-34333477

ABSTRACT

OBJECTIVE: For currently available augmented reality workflows, 3D models need to be created with manual or semiautomatic segmentation, which is a time-consuming process. The authors created an automatic segmentation algorithm that generates 3D models of skin, brain, ventricles, and contrast-enhancing tumor from a single T1-weighted MR sequence and embedded this model into an automatic workflow for 3D evaluation of anatomical structures with augmented reality in a cloud environment. In this study, the authors validate the accuracy and efficiency of this automatic segmentation algorithm for brain tumors and compared it with a manually segmented ground truth set. METHODS: Fifty contrast-enhanced T1-weighted sequences of patients with contrast-enhancing lesions measuring at least 5 cm3 were included. All slices of the ground truth set were manually segmented. The same scans were subsequently run in the cloud environment for automatic segmentation. Segmentation times were recorded. The accuracy of the algorithm was compared with that of manual segmentation and evaluated in terms of Sørensen-Dice similarity coefficient (DSC), average symmetric surface distance (ASSD), and 95th percentile of Hausdorff distance (HD95). RESULTS: The mean ± SD computation time of the automatic segmentation algorithm was 753 ± 128 seconds. The mean ± SD DSC was 0.868 ± 0.07, ASSD was 1.31 ± 0.63 mm, and HD95 was 4.80 ± 3.18 mm. Meningioma (mean 0.89 and median 0.92) showed greater DSC than metastasis (mean 0.84 and median 0.85). Automatic segmentation had greater accuracy for measuring DSC (mean 0.86 and median 0.87) and HD95 (mean 3.62 mm and median 3.11 mm) of supratentorial metastasis than those of infratentorial metastasis (mean 0.82 and median 0.81 for DSC; mean 5.26 mm and median 4.72 mm for HD95). CONCLUSIONS: The automatic cloud-based segmentation algorithm is reliable, accurate, and fast enough to aid neurosurgeons in everyday clinical practice by providing 3D augmented reality visualization of contrast-enhancing intracranial lesions measuring at least 5 cm3. The next steps involve incorporation of other sequences and improving accuracy with 3D fine-tuning in order to expand the scope of augmented reality workflow.


Subject(s)
Augmented Reality , Brain Neoplasms , Algorithms , Brain Neoplasms/diagnostic imaging , Brain Neoplasms/surgery , Humans , Image Processing, Computer-Assisted
13.
J Neurooncol ; 147(2): 417-425, 2020 Apr.
Article in English | MEDLINE | ID: mdl-32088813

ABSTRACT

INTRODUCTION: Childhood, adolescent and young adult (CAYA) cancer survivors treated with cranial radiotherapy are at risk for developing subsequent meningiomas. There is insufficient evidence concerning the benefits and harms of screening for subsequent meningiomas, and uncertainty about the most appropriate clinical management of asymptomatic meningiomas. Data describing current clinical decision-making is essential to formulate surveillance recommendations. METHODS: We created an online survey to identify the current international clinical practice regarding screening for and management of subsequent asymptomatic meningiomas among CAYA cancer survivors. Fifty-nine physicians from North America and Europe with expertise relevant to meningiomas were invited to participate. RESULTS: Thirty-four physicians (58%) completed the survey. The reported number of CAYA cancer survivors that physicians are willing to screen to detect one meningioma varied widely from 0 to 750 (median 50). Physicians expressed concerns regarding harms from MRI screening, including risks of unnecessary interventions (n = 25, 73%) and overdiagnosis (n = 19, 56%). Growth pattern (n = 33, 97%), location (n = 31, 91%) and size (n = 29, 85%) were endorsed as the most important factors influencing the decision to treat asymptomatic meningiomas. A challenging location (n = 14, 52%), indolent tumor growth pattern (n = 13, 48%), and absence of symptoms (n = 12, 44%) were endorsed as the main reasons to monitor without intervention. CONCLUSIONS: There is international variation in opinions and clinical practice regarding screening for subsequent asymptomatic meningiomas among at risk CAYA cancer survivors. Decision-making regarding interventions of asymptomatic meningiomas are largely driven by clinical characteristics. These valuable insights into current clinical practice will inform surveillance guidelines for CAYA cancer survivors.


Subject(s)
Cancer Survivors/statistics & numerical data , Delivery of Health Care/statistics & numerical data , Early Detection of Cancer/statistics & numerical data , Meningeal Neoplasms/diagnosis , Meningeal Neoplasms/therapy , Meningioma/diagnosis , Meningioma/therapy , Adolescent , Adult , Child , Disease Management , Female , Follow-Up Studies , Humans , Male , Meningeal Neoplasms/pathology , Meningioma/pathology , Prognosis , Survival Rate , Young Adult
14.
Clin Otolaryngol ; 44(4): 565-571, 2019 07.
Article in English | MEDLINE | ID: mdl-30901156

ABSTRACT

OBJECTIVES: The patients' perspective on health has become increasingly important when assessing treatment outcomes. Recently, the Endoscopic Endonasal Sinus and Skull Base Surgery Questionnaire (EES-Q) was developed to determine the impact of endoscopic endonasal surgery on health-related quality of life (HRQoL). The aim of this study was to evaluate the test-retest reliability, construct validity and responsiveness of the EES-Q. DESIGN: Prospective cohort study. SETTING: University Medical Center Groningen, tertiary referral hospital, the Netherlands. PARTICIPANTS: One hundred patients who underwent endoscopic endonasal surgery because of sinus or anterior skull base pathology. MAIN OUTCOME MEASURES: Test-retest reliability, construct validity and responsiveness. RESULTS: The EES-Q domains exhibited good test-retest reliability (ICC > 0.90). Construct validity was corroborated by significant positive and negative correlations between the EES-Q and the Sino-Nasal Outcome Test-22 (SNOT-22) and postoperative health status (P < 0.01) respectively. The correlation between the social EES-Q domain and the ability to move and perform usual activities (EuroQol-5D-3L [EQ-5D-3L]) was significant positive (P < 0.01). In patients with paranasal sinus pathology, the EES-Q was responsive to clinical change (Cohen's d = 0.6). CONCLUSION: The EES-Q is a reliable and acceptable responsive disease-specific HRQoL instrument. The expected construct validity of the EES-Q is supported by the results in this study. Inconveniences in social functioning had the greatest negative impact on postoperative health status rating. This reflects the importance of a multidimensional HRQoL assessment after EES. The results indicate that the EES-Q is a promising disease-specific tool for the HRQoL assessment after endoscopic endonasal sinus or anterior skull base surgery.


Subject(s)
Endoscopy , Paranasal Sinus Diseases/surgery , Psychometrics , Skull Base/surgery , Surveys and Questionnaires , Female , Humans , Male , Middle Aged , Netherlands , Prospective Studies , Reproducibility of Results , Sino-Nasal Outcome Test
15.
Childs Nerv Syst ; 32(9): 1721-5, 2016 Sep.
Article in English | MEDLINE | ID: mdl-27289362

ABSTRACT

PURPOSE: The acquired Chiari I malformation with abnormal cranial vault thickening is a rare late complication of supratentorial shunting. It poses a difficult clinical problem, and there is debate about the optimal surgical strategy. Some authors advocate supratentorial skull enlarging procedures while others prefer a normal Chiari decompression consisting of a suboccipital craniectomy, with or without C1 laminectomy and dural patch grafting. METHODS: We illustrate three cases of symptomatic acquired Chiari I malformation due to inward cranial vault thickening. RESULTS: We describe a new surgical approach that appears to be effective in these patients. This approach includes the standard Chiari decompression combined with posterior fossa augmentation by thinning the occipital planum. CONCLUSION: Internal volume re-expansion of the posterior fossa by thinning the occipital planum appears to be an effective novel surgical strategy in conjunction with the standard surgical therapy of Chiari decompression.


Subject(s)
Arnold-Chiari Malformation/diagnostic imaging , Arnold-Chiari Malformation/surgery , Cerebrospinal Fluid Shunts/adverse effects , Ventriculostomy/methods , Adolescent , Arnold-Chiari Malformation/etiology , Cerebrospinal Fluid Shunts/trends , Child, Preschool , Female , Humans , Male , Treatment Outcome
16.
Childs Nerv Syst ; 31(8): 1247-59, 2015 Aug.
Article in English | MEDLINE | ID: mdl-25930722

ABSTRACT

PURPOSE: This study aims to develop and establish the content validity of multiple expert rating instruments to assess performance in endoscopic third ventriculostomy (ETV), collectively called the Neuro-Endoscopic Ventriculostomy Assessment Tool (NEVAT). METHODS: The important aspects of ETV were identified through a review of current literature, ETV videos, and discussion with neurosurgeons, fellows, and residents. Three assessment measures were subsequently developed: a procedure-specific checklist (CL), a CL of surgical errors, and a global rating scale (GRS). Neurosurgeons from various countries, all identified as experts in ETV, were then invited to participate in a modified Delphi survey to establish the content validity of these instruments. In each Delphi round, experts rated their agreement including each procedural step, error, and GRS item in the respective instruments on a 5-point Likert scale. RESULTS: Seventeen experts agreed to participate in the study and completed all Delphi rounds. After item generation, a total of 27 procedural CL items, 26 error CL items, and 9 GRS items were posed to Delphi panelists for rating. An additional 17 procedural CL items, 12 error CL items, and 1 GRS item were added by panelists. After three rounds, strong consensus (>80% agreement) was achieved on 35 procedural CL items, 29 error CL items, and 10 GRS items. Moderate consensus (50-80% agreement) was achieved on an additional 7 procedural CL items and 1 error CL item. The final procedural and error checklist contained 42 and 30 items, respectively (divided into setup, exposure, navigation, ventriculostomy, and closure). The final GRS contained 10 items. CONCLUSIONS: We have established the content validity of three ETV assessment measures by iterative consensus of an international expert panel. Each measure provides unique assessment information and thus can be used individually or in combination, depending on the characteristics of the learner and the purpose of the assessment. These instruments must now be evaluated in both the simulated and operative settings, to determine their construct validity and reliability. Ultimately, the measures contained in the NEVAT may prove suitable for formative assessment during ETV training and potentially as summative assessment measures during certification.


Subject(s)
Clinical Competence , Reproducibility of Results , Ventriculostomy/methods , Ventriculostomy/standards , Humans , Online Systems , Surveys and Questionnaires , Third Ventricle/surgery
17.
Childs Nerv Syst ; 30(6): 1129-33, 2014 Jun.
Article in English | MEDLINE | ID: mdl-24169867

ABSTRACT

We present a 2.5-year-old child suffering from acute hydrocephalus. First, the child was diagnosed with aseptic viral meningitis. The PCR of the cerebrospinal fluid (CSF) was positive for enterovirus. Subsequently, MRI revealed that the hydrocephalus was caused by a cyst in the third ventricle. During ventriculoscopy, the cyst had all aspects of an arachnoid cyst. An endoscopic fenestration and partial removal of the cyst was performed, combined with a ventriculocisternostomy. The coincidental finding of viral meningitis and a third ventricle arachnoid cyst in a patient with acute hydrocephalus has, to our knowledge, not been described in literature before. If there is a relation between the enteroviral meningitis, the arachnoid cyst (possibly causing a pre-existing subclinical hydrocephalus) and the rapidly evolving neurological deterioration, remains speculative. Proposed mechanisms, by which the viral meningitis could accelerate the disease process, are slight brain swelling or increased CSF production. This rare combination of diagnoses could also be coincidental.


Subject(s)
Arachnoid Cysts/complications , Arachnoid Cysts/pathology , Enterovirus Infections/complications , Hydrocephalus/etiology , Meningitis, Viral/complications , Third Ventricle/pathology , Arachnoid Cysts/surgery , Child, Preschool , Endoscopy , Enterovirus Infections/surgery , Glasgow Coma Scale , Humans , Hydrocephalus/surgery , Hydrocephalus/virology , Magnetic Resonance Imaging , Male , Meningitis, Viral/surgery , Neurologic Examination , Third Ventricle/surgery , Ventriculostomy
18.
Neurooncol Adv ; 6(1): vdae026, 2024.
Article in English | MEDLINE | ID: mdl-38476931

ABSTRACT

Background: Pediatric brain tumor patients are at risk of developing neurocognitive impairments and associated white matter alterations. In other populations, post-traumatic stress symptoms (PTSS) impact cognition and white matter. This study aims to investigate the effect of PTSS on neurocognitive functioning and limbic white matter in pediatric brain tumor patients. Methods: Sixty-six patients (6-16 years) completed neuropsychological assessment and brain MRI (1-year post-diagnosis) and parents completed PTSS proxy questionnaires (CRIES-13; 1-3 months and 1-year post-diagnosis). Mean Z-scores and percentage impaired (>1SD) for attention, processing speed, executive functioning, and memory were compared to normscores (t-tests, chi-square tests). Multi-shell diffusion MRI data were analyzed for white matter tractography (fractional anisotropy/axial diffusivity). Effects of PTSS on neurocognition and white matter were explored with linear regression models (FDR correction for multiple testing), including age at diagnosis, treatment intensity, and tumor location as covariates. Neurocognition and limbic white matter associations were explored with correlations. Results: Attention (M = -0.49, 33% impaired; P < .05) and processing speed (M = -0.57, 34% impaired; P < .05) were significantly lower than healthy peers. PTSS was associated with poorer processing speed (ß = -0.64, P < .01). Treatment intensity, age at diagnosis, and tumor location, but not PTSS, were associated with limbic white matter metrics. Neurocognition and white matter metrics were not associated. Conclusions: Higher PTSS was associated with poorer processing speed, highlighting the need for monitoring, and timely referrals to optimize psychological well-being and neurocognitive functioning. Future research should focus on longitudinal follow-up and explore the impact of PTSS interventions on neurocognitive performance.

19.
STAR Protoc ; 5(1): 102905, 2024 Mar 15.
Article in English | MEDLINE | ID: mdl-38386548

ABSTRACT

Cancer registry data on pediatric gliomas come with inherent limitations as inclusion criteria and registration practices of these tumors differ between registries due to specific guidelines that are lacking. These limitations can lead to biased estimates in incidence and survival outcomes. Here, we present a protocol to investigate data quality and comparability for retrospective population-based pediatric glioma studies. We describe steps for obtaining institutional permissions, dealing with data quality issues, regrouping tumors, and reporting tumors in a clinically relevant manner. For complete details on the use and execution of this protocol, please refer to Hoogendijk et al.1.


Subject(s)
Data Accuracy , Glioma , Humans , Child , Retrospective Studies , Glioma/epidemiology , Glioma/therapy , Glioma/pathology , Registries
20.
J Neurosurg Pediatr ; 31(4): 313-320, 2023 04 01.
Article in English | MEDLINE | ID: mdl-36738466

ABSTRACT

OBJECTIVE: The risk of cerebrospinal fluid (CSF) leakage after cranial surgery and its associated complications in children are unclear because of variable definitions and the lack of multicenter studies. In this study, the authors aimed to establish the incidence of CSF leakage after intradural cranial surgery in the pediatric population. In addition, they evaluated potential risk factors and complications related to CSF leakage in the pediatric population. METHODS: The authors performed an international multicenter retrospective cohort study in three tertiary neurosurgical referral centers. Included were all patients aged 18 years or younger who had undergone cranial surgery to reach the subdural space during the period between 2015 and 2021. Patients who died or were lost to follow-up within 6 weeks after surgery were excluded. The primary outcome measure was the incidence of CSF leakage, defined as leakage through the skin, within 6 weeks after surgery. Univariable and multivariable logistic regression analyses were performed to identify risk factors for and complications related to CSF leakage. RESULTS: In total, 759 procedures were identified, performed in 687 individual patients. The incidence of CSF leakage was 7.5% (95% CI 5.7%-9.6%). In the multivariate model, independent risk factors for CSF leakage were hydrocephalus (OR 4.5, 95% CI 2.2-8.9) and craniectomy (OR 7.6, 95% CI 3.0-19.5). Patients with CSF leakage had higher odds of pseudomeningocele (5.7, 95% CI 3.0-10.8), meningitis (21.1, 95% CI 9.5-46.8), and surgical site infection (7.4, 95% CI 2.6-20.8) than patients without leakage. CONCLUSIONS: CSF leakage risk in children after cranial surgery, which is comparable to the risk reported in adults, is an event of major concern and has a serious clinical impact.


Subject(s)
Cerebrospinal Fluid Leak , Neurosurgical Procedures , Adult , Humans , Child , Incidence , Retrospective Studies , Neurosurgical Procedures/adverse effects , Neurosurgical Procedures/methods , Cerebrospinal Fluid Leak/epidemiology , Cerebrospinal Fluid Leak/etiology , Risk Factors , Postoperative Complications/epidemiology , Postoperative Complications/etiology
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