Publications by authors named "Antoinette Schouten-van Meeteren"

The frequency and duration of imaging surveillance in children and adolescents with pediatric low-grade gliomas (pLGGs) aims for the early detection of recurrence or progression. Although surveillance of pLGGs is performed routinely, it is not yet standardized. The aim of the current review is to provide a comprehensive synthesis of published studies regarding the optimal frequency, intervals, and duration of surveillance.

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Article Synopsis
  • Low-grade glioma is the most common type of brain tumor in children, often leading to chronic issues due to residual tumors and treatment-related brain injury.
  • Research highlights various affected areas like motor skills, speech, organ function, and psychosocial well-being, indicating the complexity of long-term effects.
  • Understanding these affected domains is crucial for tailoring medical follow-ups, support strategies, and long-term care for survivors of childhood low-grade glioma.
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  • Progressive isolated optic nerve glioma (ONG) in children is a rare condition with no standard treatment consensus, and a study was conducted to compare the effectiveness of different treatment methods.
  • The study involved 21 ONGs where systemic anticancer therapy (SAT) led to visual improvements in a significant number of cases, while surgery often resulted in worsening vision, and radiotherapy had mixed effects, including a potential increase in tumor volume shortly after treatment.
  • The findings suggest that SAT is the preferred treatment for preserving vision, while surgery may help alleviate other symptoms in blind patients, but radiotherapy poses risks and needs careful evaluation before use.
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Background: Children and adolescents treated for a brain tumor suffer from more fatigue than survivors of other types of childhood cancer. As tumor location might be predictive of fatigue, our aim was to investigate the longitudinal development of fatigue in children with brain tumors and risk factors for fatigue separately for different tumor locations.

Methods: Fatigue was assessed 1235 times for 425 participants.

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Background: Children with life-threatening and life-limiting conditions can experience high levels of suffering due to multiple distressing symptoms that result in poor quality of life and increase risk of long-term distress in their family members. High quality symptom treatment is needed for all these children and their families, even more so at the end-of-life. In this paper, we provide evidence-based recommendations for symptom treatment in paediatric palliative patients to optimize care.

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In October 2022, the FDA Oncology Center of Excellence hosted an educational symposium entitled, "Considering Functional Outcomes as Efficacy Endpoints in Pediatric Low-Grade Glioma (pLGG) Clinical Trials." The symposium brought together patient advocates, regulators from the FDA and the European Medicines Agency (EMA), and an international group of academic thought leaders in the field of pediatric neuro-oncology to discuss the potential role of functional outcomes, including visual acuity, motor function, and neurocognitive performance, as endpoints in clinical trials enrolling patients with pLGG. The panel discussed challenges and opportunities regarding the selection, implementation, and evaluation of clinical outcome assessments in these functional domains and outlined key considerations for their inclusion in future clinical trial design and role in new drug development.

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Purpose: Progressive pediatric optic pathway gliomas (OPGs) are treated by diverse systemic antitumor modalities. Refined insights on the course of intra-tumoral components are limited.

Methods: We performed an exploratory study on the longitudinal volumetric course of different (intra-)tumor components by manual segmentation of MRI at the start and after 3, 6 and 12 months of bevacizumab (BVZ) treatment.

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Within the last few decades, we have witnessed tremendous advancements in the study of pediatric low-grade gliomas (pLGG), leading to a much-improved understanding of their molecular underpinnings. Consequently, we have achieved successful milestones in developing and implementing targeted therapeutic agents for treating these tumors. However, the community continues to face many unknowns when it comes to the most effective clinical implementation of these novel targeted inhibitors or combinations thereof.

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We present the case of a 15-year-old girl, with a fifth cystic progression of an adamantinomatous craniopharyngioma after multiple surgeries and previous local radiotherapy. She had severe visual impairment, panhypopituitarism including diabetes insipidus, and several components of hypothalamic damage, including morbid obesity and severe fatigue. To prevent further late effects hampering her quality of survival, she was treated biweekly with intravenous tocilizumab, an anti-interleukin-6 agent, which stabilized the cyst for a prolonged time.

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Pediatric low-grade gliomas (pLGG) show heterogeneous responses to MAPK inhibitors (MAPKi) in clinical trials. Thus, more complex stratification biomarkers are needed to identify patients likely to benefit from MAPKi therapy. Here, we identify MAPK-related genes enriched in MAPKi-sensitive cell lines using the GDSC dataset and apply them to calculate class-specific MAPKi sensitivity scores (MSSs) via single-sample gene set enrichment analysis.

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Background: Childhood craniopharyngioma (cCP) has excellent survival, but quality of life may be severely hampered by hypothalamic dysfunction. We aimed to evaluate treatment and hypothalamic outcomes of a Dutch cCP cohort, and evaluate the effect of centralization of care.

Methods: A retrospective cohort study was performed, including cCP patients diagnosed between 2004 and 2021.

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The aim of treating childhood cancer remains to cure all. As survival rates improve, long-term health outcomes increasingly define quality of care. The International Childhood Cancer Outcome Project developed a set of core outcomes for most types of childhood cancers involving relevant international stakeholders (survivors; pediatric oncologists; other medical, nursing or paramedical care providers; and psychosocial or neurocognitive care providers) to allow outcome-based evaluation of childhood cancer care.

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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.

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Background: The international, multicenter registry LOGGIC Core BioClinical Data Bank aims to enhance the understanding of tumor biology in pediatric low-grade glioma (pLGG) and provide clinical and molecular data to support treatment decisions and interventional trial participation. Hence, the question arises whether implementation of RNA sequencing (RNA-Seq) using fresh frozen (FrFr) tumor tissue in addition to gene panel and DNA methylation analysis improves diagnostic accuracy and provides additional clinical benefit.

Methods: Analysis of patients aged 0 to 21 years, enrolled in Germany between April 2019 and February 2021, and for whom FrFr tissue was available.

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Article Synopsis
  • The study aimed to determine how effectively circumpapillary retinal nerve fibre layer (RNFL) thickness and macular ganglion cell layer-inner plexiform layer (GCL-IPL) thickness can identify abnormal visual function in children newly diagnosed with brain tumors.
  • Conducted on a group of 115 children aged 0-18 across four hospitals in the Netherlands, the research assessed sensitivity, specificity, positive predictive value (PPV), and negative predictive value (NPV) using reliable visual tests and optical coherence tomography.
  • Results showed RNFL thickness identified 74.5% of abnormal visual functions, while GCL-IPL thickness detected 41.7%. The findings suggest that normal measurements for both layers indicate a high likelihood
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Backgrounds: Bevacizumab (BVZ) is used as a subsequent line of treatment for pediatric optic pathway glioma (OPG) in the case of progression. Data on the treatment effect concerning tumor progression and visual function are scarce and nationwide studies are lacking.

Methods: We performed a retrospective, nationwide, multicentre cohort study including all pediatric patients with OPG treated with BVZ in the Netherlands (2009-2021).

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Importance: Visual impairment is an irreversible adverse effect in individuals who experienced a childhood brain tumor. Ophthalmological evaluation at diagnosis enables early detection of vision loss, decision-making about treatment, and when applicable, the timely use of visual interventions. However, awareness of visual impairment in clinical practice is suboptimal, and adherence to ophthalmological evaluation needs to be improved.

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Background: Children with suprasellar low grade glioma (LGG) frequently develop problems to maintain their body weight within the normal range, due to hypothalamic dysfunction. Hypothalamic damage may result in the diencephalic syndrome (DS), characterized by underweight or failure to thrive, but also in hypothalamic obesity (HO). Children with LGG presenting with DS at young age often develop HO later in life.

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Introduction: Hypothalamic obesity (HO) in children has severe health consequences. Lifestyle interventions are mostly insufficient and currently no drug treatment is approved for children with HO. Amphetamines are known for their stimulant side-effect on resting energy expenditure (REE) and suppressing of appetite.

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Article Synopsis
  • Endocrine disorders in survivors of childhood, adolescent, and young adult (CAYA) cancers can lead to serious physical and mental health issues.
  • The International Late Effects of Childhood Cancer Guideline Harmonization Group (IGHG) is working on creating guidelines for healthcare providers to enhance screening and referrals for these conditions.
  • New recommendations for monitoring hypothalamic-pituitary dysfunction have been developed, emphasizing the importance of regular check-ups and timely referrals for survivors at risk of endocrine issues.
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Purpose: To systematically review the evidence on the diagnostic accuracy and prognostic value of retinal optical coherence tomography (OCT) to detect visual acuity (VA) or visual field (VF) loss in children with a brain tumour.

Methods: PubMed, Embase and Cochrane Library databases were searched from inception to February 2021. We included studies evaluating retinal OCT and standard visual function parameters (VA and or VF) in children with a brain tumour.

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Unlabelled: Liquid biopsies can be used to investigate tumor-derived DNA, circulating in the cell-free DNA (cfDNA) pool in blood. We aimed to develop a droplet digital polymerase chain reaction (ddPCR) assay detecting hypermethylation of tumor suppressor gene as a simple standard test to detect various pediatric tumor types in small volume blood samples and to evaluate this test for monitoring treatment response of patients with high-risk neuroblastoma.

Methods: We developed a ddPCR assay to sensitively detect tumor-derived hypermethylated DNA in liquid biopsies.

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