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Increased insulin-like growth factor-1 concentrations in paediatric suprasellar low-grade glioma: an international
Ichelle M A A van Roessel1,2,3, Boudewijn Bakker2, Antoinette Y N Schouten-van Meeteren1
1Department of Pediatric Neuro-oncology, Princess Máxima Center for Pediatric Onology, Heidelberglaan 25, Utrecht 3584 CS, The Netherlands.
Insights
Elevated insulin-like growth factor (IGF)-1 is common in children with suprasellar low-grade glioma (LGG), particularly younger patients and those with NF1. This elevation often resolves naturally without impacting final height or tumor progression.
Area of Science:
- Pediatric Endocrinology
- Pediatric Oncology
- Neuro-oncology
Background:
- Suprasellar low-grade gliomas (LGG) can affect growth and hormonal balance in children.
- Insulin-like growth factor 1 (IGF-1) plays a crucial role in growth and metabolism.
- Understanding IGF-1 dynamics in pediatric LGG is important for managing growth and treatment outcomes.
Purpose of the Study:
- To determine the prevalence of elevated IGF-1 in children with suprasellar LGG.
- To investigate the longitudinal course of IGF-1 levels.
- To explore associations between elevated IGF-1 and clinical factors like age, neurofibromatosis type 1 (NF1), hypothalamic syndrome, and tumor behavior.
Main Methods:
- Retrospective analysis of pediatric patients (<18 years) with suprasellar LGG diagnosed between 2003-2023.
- Inclusion criteria: minimum 1-year follow-up and no growth hormone use.
- Elevated IGF-1 defined as IGF-1 standard deviation score (SDS) >+2.0.
Main Results:
- 31.1% of 235 children exhibited elevated IGF-1 at any point; 15.2% at diagnosis.
- Elevated IGF-1 was linked to younger age at diagnosis (P=.004), NF1 (P=.028), and diencephalic syndrome (P=.047).
- IGF-1 normalized spontaneously in 75.3% of affected children; no significant impact on final height or tumor progression was observed.
Conclusions:
- Increased IGF-1 is frequently seen in pediatric suprasellar LGG, especially in younger children, those with NF1, or underweight.
- The spontaneous resolution of elevated IGF-1 and its lack of effect on final height and tumor progression are reassuring findings.
Objective:
The objective of this study was to assess the prevalence of elevated insulin-like growth factor (IGF)-1 in children with suprasellar low-grade glioma (LGG) and explore the course of IGF-1 over time and its association with anthropometrics, hypothalamic syndrome, tumour characteristics, and tumour behaviour.
Design:
This retrospective study included children from the Netherlands and the United Kingdom diagnosed with a suprasellar LGG under the age of 18 between 2003 and 2023, with a minimum 1-year follow-up. Elevated IGF-1 was defined as IGF-1 standard deviation score (SDS) >+2.0 for age and biological sex, without growth hormone use.
Results:
We included 235 patients with a median age at brain tumour diagnosis of 3.8 years (IQR 1.7-7.1). Elevated IGF-1 was observed in 73 patients (31.1%) at any time point. At tumour diagnosis, 15.2% of the 138 children tested showed elevated IGF-1. Elevated IGF-1 was associated with younger age at tumour diagnosis (P = .004), neurofibromatosis type 1 (NF1; P = .028), and diencephalic syndrome (P = .047). In 55 of the 73 patients with elevated IGF-1 (75.3%), IGF-1 normalized spontaneously over time. Final height SDS corrected for target height SDS was not associated with having had an elevated IGF-1 (P = .113). No difference was found in the number of tumour progressions.
Conclusions:
Increased IGF-1 concentrations are commonly observed in children with suprasellar LGG, especially in younger children, those with an NF1 mutation, or during underweight. Elevation of IGF-1 can resolve over time, and the absence of an effect on final height or on tumour progressions seems reassuring.
