van Roessel I M A A, Gorter J E, Bakker B, van den Heuvel-Eibrink M M, Lequin M H, van der Lugt J, Meijer L, Schouten-van Meeteren A Y N, van Santen H M
Department of Pediatric Neuro-oncology, Princess Máxima Center, Heidelberglaan, CS Utrecht, The Netherlands.
Department of Pediatric Endocrinology, Wilhelmina Children's Hospital, University Medical Center, Lundlaan, EA Utrecht, The Netherlands.
Endocr Connect. 2024 Sep 25;13(10). doi: 10.1530/EC-24-0224. Print 2024 Oct 1.
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.
A retrospective study was performed in children with supratentorial midline (suprasellar or thalamic) LGG between 1 January 2003 and 1 January 2022, visiting the Princess Máxima Center for Pediatric Oncology. Impaired bone health was defined as the presence of vertebral fractures and/or very low bone mineral density (BMD).
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).
In this retrospective evaluation, decreased BMD was prevalent in 34.3% of children with supratentorial midline LGG. For the risk of developing bone problems, visual capacity seems highly relevant. Surveillance of bone health must be an aspect of awareness in the care and follow-up of children with a supratentorial midline LGG.
Patients with supratentorial midline LGG may encounter various risk factors for impaired bone health. Bone problems in survivors of childhood supratentorial midline LGG are, however, understudied. This is the first paper to address the prevalence of bone problems in this specific patient population, revealing visual problems as an important risk factor. Diencephalic syndrome historyand/or weight problems associated with hypothalamic dysfunction were related to bone problems in univariate analyses. The results of this study can be used in the development of guidelines to adequately screen and treat these patients to subsequently minimizing bone problems as one of the endocrine complications.
患有幕上中线低级别胶质瘤（LGG）的儿童可能因下丘脑 - 垂体功能障碍、肥胖、接受多种治疗方式和/或活动能力下降而面临骨骼健康受损的风险。该人群中骨骼健康受损的存在及其严重程度尚未得到充分研究。我们旨在确定幕上中线LGG儿童骨骼问题的患病率和危险因素。
对2003年1月1日至2022年1月1日期间就诊于马克西玛公主儿科肿瘤中心的幕上中线（鞍上或丘脑）LGG儿童进行了一项回顾性研究。骨骼健康受损定义为存在椎体骨折和/或极低骨密度（BMD）。
共纳入161名儿童，肿瘤诊断时的中位年龄为4.7岁（范围：0.1 - 17.9岁），中位随访时间为6.1年（范围：0.1 - 19.9年）。5名患者（3.1%）有椎体骨折。在99名患者中，通过双能X线吸收法（n = 12）或骨健康指数（n = 95）评估了BMD；34名患者（34.3%）BMD较低（≤ -2.0）。在多变量分析中，视力受损与骨骼问题相关（比值比：6.63，95%置信区间：1.83 - 24.00，P = 0.004）。
在这项回顾性评估中，幕上中线LGG儿童中34.3%存在BMD降低的情况。对于发生骨骼问题的风险，视力似乎高度相关。骨骼健康监测必须成为幕上中线LGG儿童护理和随访中的一个关注方面。
幕上中线LGG患者可能面临多种骨骼健康受损的危险因素。然而，儿童幕上中线LGG幸存者的骨骼问题尚未得到充分研究。这是第一篇探讨这一特定患者群体中骨骼问题患病率的论文，揭示视力问题是一个重要的危险因素。在单变量分析中，间脑综合征病史和/或与下丘脑功能障碍相关的体重问题与骨骼问题有关。本研究结果可用于制定指南，以充分筛查和治疗这些患者，从而将骨骼问题作为内分泌并发症之一降至最低。
