Increased insulin-like growth factor-1 concentrations in paediatric suprasellar low-grade glioma: an international multicentre study.

in European journal of endocrinology by Ichelle M A A van Roessel, Boudewijn Bakker, Antoinette Y N Schouten-van Meeteren, Wim J E Tissing, Hoong-Wei Gan, Hanneke M van Santen

TLDR

  • The study found that elevated IGF-1 was common in children with suprasellar LGG, especially in younger children and those with NF1 or diencephalic syndrome. IGF-1 normalization and no significant effects on final height or tumour progressions provide reassuring results.

Abstract

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. 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. 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. 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.

Overview

  • The study aimed to assess the prevalence of elevated insulin-like growth factor (IGF)-1 in children with suprasellar low-grade glioma (LGG) and explore its course over time and associations with various factors.
  • The study included 235 patients with a median age at brain tumour diagnosis of 3.8 years, and used IGF-1 standard deviation score (SDS) >+2.0 to define elevated IGF-1.
  • The primary objectives were to examine the prevalence of elevated IGF-1, its association with various factors, and its effect on final height and tumour progressions.

Comparative Analysis & Findings

  • Elevated IGF-1 was observed in 31.1% of the patients at any time point, and 15.2% showed elevated IGF-1 at tumour diagnosis.
  • Elevated IGF-1 was associated with younger age at tumour diagnosis, neurofibromatosis type 1 (NF1), and diencephalic syndrome.
  • IGF-1 normalized spontaneously over time in 75.3% of the patients with elevated IGF-1, and final height SDS was not associated with having had an elevated IGF-1.

Implications and Future Directions

  • The study provides insights into the prevalence and associations of elevated IGF-1 in children with suprasellar LGG, which can inform clinical practice and future research.
  • Future studies could investigate the potential mechanisms underlying the association between elevated IGF-1 and tumour characteristics, and explore the impact of IGF-1 elevation on quality of life and overall outcome.
  • The study highlights the importance of long-term follow-up and monitoring of IGF-1 levels in children with suprasellar LGG to identify potential changes over time.
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