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  • Author: M M van den Heuvel-Eibrink x
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V G Pluimakers Princess Máxima Centre for Paediatric Oncology, Utrecht, The Netherlands

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M van Waas Department of Paediatric Oncology/Haematology, Erasmus MC–Sophia Children’s Hospital, Rotterdam, The Netherlands

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C W N Looman Department of Public Health, Erasmus MC, Rotterdam, The Netherlands

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M P de Maat Department of Haematology, Erasmus MC, Rotterdam, The Netherlands

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R de Jonge Department of Clinical Chemistry, Erasmus MC, Rotterdam, The Netherlands

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P Delhanty Section Endocrinology, Department of Medicine, Erasmus MC, Rotterdam, The Netherlands

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M Huisman Section Endocrinology, Department of Medicine, Erasmus MC, Rotterdam, The Netherlands

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F U S Mattace-Raso Section Geriatric Medicine, Department of Medicine, Erasmus MC, Rotterdam, The Netherlands

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M M van den Heuvel-Eibrink Princess Máxima Centre for Paediatric Oncology, Utrecht, The Netherlands

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S J C M M Neggers Princess Máxima Centre for Paediatric Oncology, Utrecht, The Netherlands
Section Endocrinology, Department of Medicine, Erasmus MC, Rotterdam, The Netherlands

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Purpose:

Augmented survival of childhood nephroblastoma and neuroblastoma has increased long-term side effects such as metabolic syndrome (MetS). Risk stratification is difficult after abdominal radiation because waist circumference underestimates adiposity. We aimed to develop a strategy for determining MetS in irradiated survivors using an integrated biomarker profile and vascular ultrasonography.

Methods:

The NCEP-ATPIII MetS-components, 14 additional serum biomarkers and 9 vascular measurements were assessed in a single-centre cohort of childhood nephroblastoma (n = 67) and neuroblastoma (n = 36) survivors and controls (n = 61). Multivariable regression models were used to study treatment effects. Principal component analysis (PCA) was used to study all biomarkers in a combined analysis, to identify patterns and correlations.

Results:

After 27.5 years of follow-up, MetS occurred more often in survivors (14%) than controls (3%). Abdominal radiotherapy and nephrectomy, to a lesser extent, were associated with MetS and separate components and with several biomarker abnormalities. PCA of biomarkers revealed a pattern on PC1 from favourable lipid markers (HDL-cholesterol, adiponectin) towards unfavourable markers (triglycerides, LDL-cholesterol, apoB, uric acid). Abdominal radiotherapy was associated with the unfavourable biomarker profile (β = 1.45, P = 0.001). Vascular measurements were not of added diagnostic value.

Conclusions:

Long-term childhood nephro- and neuroblastoma survivors frequently develop MetS. Additional assessment of biomarkers identified in PCA – adiponectin, LDL, apoB, and uric acid – may be used especially in abdominally irradiated survivors, to classify MetS as alternative for waist circumference. Vascular ultrasonography was not of added value.

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I M.a.a. van Roessel I van Roessel, Department of Pediatric Neuro-oncology, Prinses Maxima Centrum voor Kinderoncologie, Utrecht, Netherlands

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Je Gorter J Gorter, Department of Pediatric Neuro-oncology, Prinses Maxima Centrum voor Kinderoncologie, Utrecht, Netherlands

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Boudewijn Bakker B Bakker, Department of Pediatric Neuro-oncology, Prinses Maxima Centrum voor Kinderoncologie, Utrecht, Netherlands

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Mm van den Heuvel-Eibrink M van den Heuvel-Eibrink, Prinses Maxima Centrum voor Kinderoncologie, Utrecht, Netherlands

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M H Lequin M Lequin, Department of Radiology, Prinses Maxima Centrum voor Kinderoncologie, Utrecht, Netherlands

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J van der Lugt J van der Lugt, Department of Pediatric Neuro-oncology, Prinses Maxima Centrum voor Kinderoncologie, Utrecht, Netherlands

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L Meijer L Meijer, Department of Pediatric Neuro-oncology, Prinses Maxima Centrum voor Kinderoncologie, Utrecht, Netherlands

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A.y.n. Schouten-van Meeteren A Schouten-van Meeteren, Department of Pediatric Neuro-oncology, Prinses Maxima Centrum voor Kinderoncologie, Utrecht, Netherlands

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H M van Santen H van Santen, Department of Pediatric Neuro-oncology, Prinses Maxima Centrum voor Kinderoncologie, Utrecht, Netherlands

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Objective: Children with a supratentorial midline low grade glioma (LGG) may be at risk for impaired bone health due to hypothalamic-pituitary dysfunction, obesity, exposure to multiple treatment modalities, and/or decreased mobility. The presence of impaired bone health and/or its severity in this population has been understudied. We aimed to identify the prevalence and risk factors for bone problems in children with supratentorial midline LGG.

Design and Methods: A retrospective study was performed in children with supratentorial midline (suprasellar or thalamic) LGG between 1-1-2003 and 1-1-2022, visiting the Princess Máxima Center for Pediatric Oncology. Impaired bone health was defined as presence of vertebral fractures and/or very low bone mineral density (BMD).

Results: In total, 161 children were included, with a median age at tumor diagnosis of 4.7 years (range 0.1 – 17.9) and a median follow-up of 6.1 years (range 0.1 – 19.9). Five patients (3.1 %) had vertebral fractures. In 99 patients BMD was assessed either by Dual Energy X ray Absorptiometry (n=12) or Bone Health Index (n=95); 34 patients (34.3%) had a low BMD (≤ -2.0). Impaired visual capacity was associated with bone problems in multivariable analysis (OR 6.63, 95% CI 1.83 – 24.00, p = 0.004).

Conclusions: In this retrospective evaluation, decreased BMD was prevalent in 34.3% of children with supratentorial midline LGG. For the risk to develop bone problems visual capacity seems highly relevant. Surveillance of bone health must be an aspect for awareness in the care and follow-up of children with a supratentorial midline LGG.

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