ESPE2023 Rapid Free Communications Pituitary, neuroendocrinology and puberty 2 (6 abstracts)
Treatment and outcome of the Dutch Childhood Craniopharyngioma Cohort study; first results after centralization of care
Jiska van Schaik 1,2 , Netteke Schouten-van Meeteren 2 , Evelien Vos-Kerkhof 2 , Geert Janssens 2 , Giorgio Porro 3 , Marta Fiocco 4,2 , Boudewijn Bakker 1,2 , Wim Tissing 2,5 , Eelco Hoving 2 & Hanneke van Santen 1,2
1Wilhelmina Children’s Hospital, University Medical Center Utrecht, Utrecht, Netherlands. 2Princess Máxima Center for pediatric oncology, Utrecht, Netherlands. 3University Medical Center Utrecht, Utrecht, Netherlands. 4Leiden University, Leiden, Netherlands. 5University of Groningen, University Medical Center Groningen, Groningen, Netherlands
Introduction: Childhood craniopharyngioma (cCP) has excellent survival, but quality of life may be severely hampered by hypothalamic dysfunction. We aimed to evaluate treatment and hypothalamic outcomes of a Dutch cCP cohort, and evaluate the effect of centralization of care.
Methods: A retrospective cohort study was performed, including cCP patients diagnosed between 2004-2021. Treatment characteristics and hypothalamic outcomes were evaluated and compared before and since centralization of care in May 2018.
Results: We included 87 cCP patients. Cyst drainage/fenestration was performed in 29.9%, limited resection in 27.6%, near total resection in 16.1%, and gross total resection (GTR) in 25.4%. Radiotherapy was given in 46.0%. After a median follow-up of 6.5 years, hypothalamic obesity (HO) was present in 24.7% and panhypopituitarism with diabetes insipidus in 71.3%. Higher BMI SDS at diagnosis and Muller grade II at last MRI of follow-up were associated to overweight/obesity. No association was found between extensiveness of resection and overweight/obesity at last moment of follow-up. When comparing before and after centralization of care, rates of GTR remained similar, but BMI outcomes changed; mean ΔBMI SDS 1 year after diagnosis from 1.12 (SD 1.15) to 0.81 (SD 1.24), and HO after 1 year decreased from 33.3% to 12.0% (P= 0.067), and after 2 years from 28.6% to 6.7% (P= NS).
Conclusion: In our nationwide cohort, GTR was performed in a relatively low percentage of patients and extensiveness of resection was no longer associated to HO at follow-up. A trend towards improvement of BMI is observed since centralization of care, which needs further exploration.
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