Prevalence of osteopathologies in a single center cohort of survivors of childhood primary brain tumor.

in Frontiers in pediatrics by Michael M Schündeln, Sebastian Fritzemeier, Sarah C Goretzki, Pia K Hauffa, Martin Munteanu, Cordula Kiewert, Berthold P Hauffa, Gudrun Fleischhack, Stephan Tippelt, Corinna Grasemann

TLDR

  • The study looked at children who had a type of brain tumor called a childhood primary brain tumor (CPBT). These children often have hormonal problems later in life, which can affect their bones. The study found that many of these children had impaired bone health and osteopathologies. The study also found that vitamin D deficiency is common in these children and should be treated. The study suggests that these children may have a special ability to heal their bones, but more research is needed to understand this.

Abstract

Childhood primary brain tumors (CPBT) are the second largest group of childhood malignancies and associated with a high risk for endocrine late effects. To assess endocrine late effects and their relevance for the development of osteopathologies in survivors. This single center cross sectional study investigated data from 102 CPBT survivors with a mean age of 13.0 years and a mean age at diagnosis of 8.7 years. Clinical, biochemical, radiographic, and anamnestic data regarding endocrine and bone health were obtained at study visits. In addition, data regarding tumor stage and therapy was obtained by chart review. An expert opinion was applied to define presence of osteopathologies. Impaired bone health, defined by at least one pathological screening parameter, was present in 65% of patients. 27.5% were found to have overt osteopathologies per expert opinion. 37.8% displayed a severe vitamin D deficiency (25-OH vitamin D < 10 ng/ml) and 11% a secondary hyperparathyroidism. Patients with osteopathologies had lower 25-OH vitamin D levels compared to patients without osteopathologies. Multiple endocrine late effects were present: diabetes insipidus in 10.8%, aberrant pubertal development in 13.7%, central hypocortisolism in 14.9%, thyroid dysfunction in 23.8% and growth hormone deficiency in 21.8%. A total of 31.3% of survivors displayed any endocrinopathy. Tumors located near hypothalamic structures and patients who received irradiation had a higher likelihood of endocrine morbidity. This study indicates that endocrine deficiencies are common in pediatric survivors of CPBTs. Osteopathologies are present in this cohort. A prominent effect of hormonal deficiencies on bone health was not detected, possibly because patients were sufficiently treate for their endocrine conditions or indicating resilience of the childhood bone remodeling process. Vitamin D deficiency is frequent and should be treated as recommended.

Overview

  • The study investigates endocrine late effects and their relevance for the development of osteopathologies in childhood primary brain tumor (CPBT) survivors. The study is a single center cross sectional study that includes 102 CPBT survivors with a mean age of 13.0 years and a mean age at diagnosis of 8.7 years. Clinical, biochemical, radiographic, and anamnestic data regarding endocrine and bone health were obtained at study visits. In addition, data regarding tumor stage and therapy was obtained by chart review. An expert opinion was applied to define presence of osteopathologies. The primary objective of the study is to assess the prevalence of endocrine deficiencies and osteopathologies in CPBT survivors and their relationship with tumor location and therapy. The study aims to provide insights into the long-term effects of CPBT on endocrine and bone health and inform clinical practice and future research.

Comparative Analysis & Findings

  • The study found that impaired bone health, defined by at least one pathological screening parameter, was present in 65% of patients. 27.5% were found to have overt osteopathologies per expert opinion. 37.8% displayed a severe vitamin D deficiency (25-OH vitamin D < 10 ng/ml) and 11% a secondary hyperparathyroidism. Patients with osteopathologies had lower 25-OH vitamin D levels compared to patients without osteopathologies. Multiple endocrine late effects were present: diabetes insipidus in 10.8%, aberrant pubertal development in 13.7%, central hypocortisolism in 14.9%, thyroid dysfunction in 23.8% and growth hormone deficiency in 21.8%. A total of 31.3% of survivors displayed any endocrinopathy. Tumors located near hypothalamic structures and patients who received irradiation had a higher likelihood of endocrine morbidity. The study indicates that endocrine deficiencies are common in pediatric survivors of CPBTs. Osteopathologies are present in this cohort. A prominent effect of hormonal deficiencies on bone health was not detected, possibly because patients were sufficiently treated for their endocrine conditions or indicating resilience of the childhood bone remodeling process. Vitamin D deficiency is frequent and should be treated as recommended.

Implications and Future Directions

  • The study's findings highlight the importance of assessing endocrine late effects and their relevance for the development of osteopathologies in CPBT survivors. The study provides insights into the prevalence of endocrine deficiencies and osteopathologies in this cohort and their relationship with tumor location and therapy. The study suggests that endocrine deficiencies are common in pediatric survivors of CPBTs and that osteopathologies are present in this cohort. The study also highlights the importance of addressing vitamin D deficiency in CPBT survivors. Future research should focus on developing interventions to prevent and treat endocrine deficiencies and osteopathologies in CPBT survivors. The study also suggests that further research is needed to understand the resilience of the childhood bone remodeling process and its relationship with hormonal deficiencies.
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