Treatment and outcome of the Dutch Childhood Craniopharyngioma Cohort study: First results after centralization of care

Schaik, John Van; Meeteren, Antoinette Y. N. Schouten–van; Vos-Kerkhof, Evelien de; Janssens, Geert O.; Porro, Giorgio L.; Fiocco, Marta; Bakker, Boudewijn; Tissing, Wim J. E.; Hoving, Eelco W.; Santen, Hanneke M van

doi:10.1093/neuonc/noad112

Cited by 5 publications

(1 citation statement)

References 31 publications

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“…Hormonal deficits are treated by restoring hormone, but hypothalamic obesity and behavioral changes are difficult to manage, especially in the presence of visual disturbances. In a recent study on 87 patients with CP, 90.8% had one or more pituitary deficiencies at follow-up ( 10 ). In the same study BMI outcomes seemed to be better in the last years, after centralization of care, but no differences in visual disturbances and hormonal deficiencies were noted over time.…”

Section: Introductionmentioning

confidence: 99%

Childhood craniopharyngioma: a retrospective study of children followed in Hôpital Universitaire de Bruxelles

Magerman,

Boros,

Preziosi

et al. 2024

Front. Endocrinol.

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IntroductionCraniopharyngiomas (CPs) are benign brain tumors accounting for 5 - 11% of intracranial tumors in children. These tumors often recur and can cause severe morbidity. Postoperative radiotherapy efficiently controls and prevents progression and recurrence. Despite advancements in neurosurgery, endocrinological, visual, and neuropsychological complications are common and significantly lower the quality of life of patients.MethodsWe performed a retrospective study, including all patients younger than sixteen diagnosed with CP between July 1989 and August 2022 and followed up in Hôpital Universitaire de Bruxelles.ResultsNineteen children with CP were included, with median age of 7 years at first symptoms and 7.5 at diagnosis. Common symptoms at diagnosis were increased intracranial pressure (63%), visual impairment (47%), growth failure (26%), polyuria/polydipsia (16%), and weight gain (10.5%). As clinical signs at diagnosis, growth failure was observed in 11/18 patients, starting with a median lag of 1 year and 4 months before diagnosis. On ophthalmological examination, 27% of patients had papillary edema and 79% had visual impairment. When visual disturbances were found, the average preoperative volume was higher (p=0.039). Only 6/19 patients had gross total surgical resection. After the first neurosurgery, 83% experienced tumor recurrence or progression at a median time of 22 months. Eleven patients (73%) underwent postsurgical radiotherapy. At diagnosis, growth hormone deficiency (GHD) was the most frequent endocrine deficit (8/17) and one year post surgery, AVP deficiency was the most frequent deficit (14/17). Obesity was present in 13% of patients at diagnosis, and in 40% six months after surgery. There was no significant change in body mass index over time (p=0.273) after the first six months post-surgery.ConclusionCP is a challenging brain tumor that requires multimodal therapy and lifelong multidisciplinary follow-up including hormonal substitution therapy. Early recognition of symptoms is crucial for prompt surgical management. The management of long-term sequelae and morbidity are crucial parts of the clinical path of the patients. The results of this study highlight the fundamental importance of carrying out a complete assessment (ophthalmological, endocrinological, neurocognitive) at the time of diagnosis and during follow-up so that patients can benefit from the best possible care.

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Section: Introductionmentioning

confidence: 99%

Childhood craniopharyngioma: a retrospective study of children followed in Hôpital Universitaire de Bruxelles

Magerman,

Boros,

Preziosi

et al. 2024

Front. Endocrinol.

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The development of fatigue after treatment for pediatric brain tumors does not differ between tumor locations

Irestorm,

Schouten‐van Meeteren,

van Gorp

et al. 2024

Pediatric Blood & Cancer

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BackgroundChildren and adolescents treated for a brain tumor suffer from more fatigue than survivors of other types of childhood cancer. As tumor location might be predictive of fatigue, our aim was to investigate the longitudinal development of fatigue in children with brain tumors and risk factors for fatigue separately for different tumor locations.MethodsFatigue was assessed 1235 times for 425 participants. Self‐report versions of PedsQL Multidimensional Fatigue Scale were used to repeatedly assess fatigue from the end of treatment up to 8 years later. Mixed models were used to analyze fatigue over time and determinants separately for infratentorial (N = 205), supratentorial hemispheric (N = 91), and supratentorial midline tumors (N = 129).ResultsCognitive fatigue worsened with time, while sleep‐rest and general fatigue first decreased and then increased. There was no difference in fatigue between the tumor locations, but the risk factors differed when stratified by location. Radiotherapy was associated with more fatigue for infratentorial tumors, and centralization of care was associated with less fatigue for the supratentorial midline tumors. For supratentorial hemispheric tumors, female sex was associated with more fatigue. Higher parental education was associated with less fatigue regardless of tumor location.ConclusionsThe development of fatigue seems to be more related to sociodemographic and treatment variables than to tumor location. Healthcare providers need to be aware that fatigue may develop in the years following end of treatment, and that patients with a low/middle educational family background might be more vulnerable and in need of targeted support.

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Impact of stereotactic radiotherapy for craniopharyngioma: a large, academic hospital cohort

Mauro,

Da Róz,

Gico

et al. 2024

Childs Nerv Syst

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Treatment and outcome of the Dutch Childhood Craniopharyngioma Cohort study: First results after centralization of care

Cited by 5 publications

References 31 publications

Childhood craniopharyngioma: a retrospective study of children followed in Hôpital Universitaire de Bruxelles

Childhood craniopharyngioma: a retrospective study of children followed in Hôpital Universitaire de Bruxelles

The development of fatigue after treatment for pediatric brain tumors does not differ between tumor locations

Impact of stereotactic radiotherapy for craniopharyngioma: a large, academic hospital cohort

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