QOS-05AN ITALIAN NETWORK FOR THE NEUROCOGNITIVE AND PSYCHOLOGICAL FOLLOW-UP OF CHILDHOOD BRAIN TUMOR SURVIVORS

PURPOSE Childhood brain tumor survivors (CBTS) are at risk for developing obesity, which negatively influences cardiometabolic health. The prevalence of obesity in CBTS may have been overestimated in previous cohorts because of inclusion of children with craniopharyngioma. On the contrary, the degree of weight gain may have been underestimated because of exclusion of CBTS who experienced weight gain, but were neither overweight nor obese. Weight gain may be an indicator of underlying hypothalamic-pituitary (HP) dysfunction. We aimed to study prevalence of and risk factors for significant weight gain, overweight, or obesity, and its association with HP dysfunction in a national cohort of noncraniopharyngioma and nonpituitary CBTS. METHODS Prevalence of and risk factors for significant weight gain (body mass index [BMI] change ≥ +2.0 standard deviation score [SDS]), overweight, or obesity at follow-up, and its association with HP dysfunction were studied in a nationwide cohort of CBTS, diagnosed in a 10-year period (2002-2012), excluding all craniopharyngioma and pituitary tumors. RESULTS Of 661 CBTS, with a median age at follow-up of 7.3 years, 33.1% had significant weight gain, overweight, or obesity. Of the CBTS between 4 and 20 years of age, 28.7% were overweight or obese, compared with 13.2% of the general population between 4 and 20 years of age. BMI SDS at diagnosis, diagnosis of low-grade glioma, diabetes insipidus, and central precocious puberty were associated with weight gain, overweight, or obesity. The prevalence of HP dysfunction was higher in overweight and obese CTBS compared with normal-weight CBTS. CONCLUSION Overweight, obesity, and significant weight gain are prevalent in CBTS. An increase in BMI during follow-up may be a reflection of HP dysfunction, necessitating more intense endocrine surveillance.

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Prevalence and Risk Factors of Early Endocrine Disorders in Childhood Brain Tumor Survivors: A Nationwide, Multicenter Study

Journal of Clinical Oncology ◽

10.1200/jco.2016.67.5025 ◽

2016 ◽

Vol 34 (36) ◽

pp. 4362-4370 ◽

Cited By ~ 29

Author(s):

Sarah C. Clement ◽

Antoinette Y.N. Schouten-van Meeteren ◽

Annemieke M. Boot ◽

Hedy L. Claahsen-van der Grinten ◽

Bernd Granzen ◽

...

Keyword(s):

Risk Factors ◽

Brain Tumor ◽

Thyroid Stimulating Hormone ◽

Endocrine Function ◽

Hormone Deficiency ◽

Study Cohort ◽

Endocrine Disorders ◽

Childhood Brain Tumor ◽

Endocrine Disorder

Purpose To evaluate the prevalence of, and risk factors for, early endocrine disorders in childhood brain tumor survivors (CBTS). Patients and Methods This nationwide study cohort consisted of 718 CBTS who were diagnosed between 2002 and 2012, and who survived ≥ 2 years after diagnosis. Patients with craniopharyngeoma or a pituitary gland tumor were excluded. Results of all endocrine investigations, which were performed at diagnosis and during follow-up, were collected from patient charts. Multivariable logistic regression was used to study associations between demographic and tumor- and treatment-related variables and the prevalence of early endocrine disorders. Results After a median follow-up of 6.6 years, 178 CBTS (24.8%) were diagnosed with an endocrine disorder. A total of 159 CBTS (22.1%) presented with at least one endocrine disorder within the first 5 years after diagnosis. The most common endocrine disorders were growth hormone deficiency (12.5%), precocious puberty (12.2%), thyroid-stimulating hormone deficiency (9.2%), and thyroidal hypothyroidism (5.8%). The risk of hypothalamic-pituitary dysfunction (n = 138) was associated with radiotherapy (odds ratio [OR], 15.74; 95% CI, 8.72 to 28.42), younger age at diagnosis (OR, 1.09; 95% CI, 1.04 to 1.14), advanced follow-up time (OR, 1.10; 95% CI, 1.02 to 1.18), hydrocephalus at diagnosis (OR, 1.77; 95% CI, 1.09 to 2.88), and suprasellar (OR, 34.18; 95% CI, 14.74 to 79.29) and infratentorial (OR, 2.65; 95% CI, 1.48 to 4.74) tumor site. Conclusion The prevalence of early endocrine disorders among CBTS is high. The observation that 22.1% of CBTS developed at least one endocrine disorder within the first 5 years after diagnosis stresses the importance of early and regular assessment of endocrine function in CBTS who are at risk for endocrine damage.

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Prevalence of endocrine disorders in childhood brain tumor survivors in South Korea: A nationwide population-based, longitudinal study.

Journal of Clinical Oncology ◽

10.1200/jco.2019.37.15_suppl.e21517 ◽

2019 ◽

Vol 37 (15_suppl) ◽

pp. e21517-e21517

Author(s):

Jaesung Heo ◽

O Kyu Noh ◽

Jun Eun Park ◽

Minhyung Cho ◽

Seonghye Choi

Keyword(s):

Brain Tumor ◽

Longitudinal Study ◽

South Korea ◽

Odds Ratio ◽

Treatment Compliance ◽

Endocrine Function ◽

Hormone Deficiency ◽

Endocrine Disorders ◽

Childhood Brain Tumor

e21517 Background: Brain tumors are the most common solid cancers in child patients with a current expected 5-year overall survival rate of 73%. Also, patients with brain cancer tend to have a high rate of neuroendocrine disorders. These endocrine problems may have a significant negative effect on quality of life and treatment compliance. Methods: The aim of this longitudinal study was to analyze the prevalence of endocrine disorders in childhood brain tumor survivors using claims data in South Korea. We confirmed endocrine disorders in a nationwide cohort of 1,058 patients who were diagnosed with brain tumor between January 1, 2009 and March 29, 2016 and who survived > 2 years after diagnosis. Multivariable logistic regression was used to evaluate association between demographic and treatment related variables and the prevalence of endocrine disorders. Results: After a median follow-up of 60.0 months, a total of 393 (37.1%) patients were diagnosed with at least one endocrine disorder. The median follow-up time from primary brain tumor diagnosis to diagnosis of first endocrine disorders was 26.3 months (range, 0.06 to 96.7). Of those patients, 333 childhood brain tumor survivors were diagnosed with endocrine disorders during their first 5 years after diagnosis. The overall frequency of endocrine disorders peaked during 2 months after the cancer diagnosis. The most common endocrine disorders were pituitary dysfunction (21.9%), thyroidal disease (6.1%), precocious puberty (4.6%), and growth hormone deficiency (4.3%). Female patients were at a higher risk for endocrine disorders (odds ratio: 1.45, p = 0.005). The patients with radiotherapy were more likely to have endocrine disorders compared without radiotherapy (odds ratio: 1.79, p < 0.001). Conclusions: Endocrine disorders in childhood brain tumor survivors was high and showed different patterns of prevalence depending on the nature of disease and time sequence. In childhood brain tumor survivors who are risk of endocrine disorders, regular assessment of endocrine function and timely intervention were needed. [Table: see text]

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Prevalence and Risk Factors of Hypothalamic-Pituitary Dysfunction in Infant and Toddler Childhood Brain Tumor Survivors

Acta Endocrinologica ◽

10.1530/eje-21-0137 ◽

2021 ◽

Author(s):

C A Lebbink ◽

T.p Ringers ◽

A.y.n. Schouten-van Meeteren ◽

L van Iersel ◽

S.c Clement ◽

...

Keyword(s):

Risk Factors ◽

Brain Tumor ◽

Age Groups ◽

Tumor Location ◽

Childhood Brain Tumor ◽

Older Children ◽

Treatment Protocols ◽

Pituitary Dysfunction ◽

Frequent Use

Objective Childhood brain tumor survivors (CBTS) are at risk to develop hypothalamic-pituitary (HP) dysfunction (HPD). The risk for HPD may vary between different age groups due to maturation of the brain and differences in oncologic treatment protocols. Specific studies on HPD in infant brain tumor survivors (infant-BTS, 0-1 years at diagnosis) or toddler brain tumor survivors (toddler-BTS, ≥1-3 years) have not been performed. Patients and Methods A retrospective nationwide cohort study in CBTS was performed. Prevalence and risk factors for HPD were compared between infant-, toddler- and older-BTS. Subgroup analysis was performed for all non-irradiated CBTS (n=460). Results In total 718 CBTS were included, with a median follow-up time of 7.9 years. Overall, despite less frequent use of radiotherapy (RT) in infants, no differences in prevalence of HPD were found between the three groups. RT (OR 16.44; 95%CI 8.93 to 30.27), suprasellar tumor location (OR 44.76; 95%CI 19.00 to 105.49) and younger age (OR 1.11; 95%CI 1.05 to 1.18) were associated with HP dysfunction. Infant-BTS and toddler-BTS showed more weight gain (p<0.0001) and smaller height SDS (p=0.001) during follow-up. In non-irradiated CBTS, infant-BTS and toddler-BTS were significantly more frequently diagnosed with TSH-, ACTH- and ADH deficiency, compared to older-BTS. Conclusion Infant and toddler brain tumor survivors seem to be more vulnerable to develop HP dysfunction than older children. These results emphasize the importance of special infant- and toddler brain tumor treatment protocols and the need for endocrine surveillance in children treated for a brain tumor at young age.

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Prevalence and Risk Factors of Hypothalamic-Pituitary Dysfunction in Infant and Toddler Brain Tumor Survivors

Journal of the Endocrine Society ◽

10.1210/jendso/bvab048.1463 ◽

2021 ◽

Vol 5 (Supplement_1) ◽

pp. A719-A719

Author(s):

Chantal A Lebbink ◽

Tiara P Ringers ◽

Antoinette Y N Schouten-van Meeteren ◽

Laura van Iersel ◽

Sarah C Clement ◽

...

Keyword(s):

Risk Factors ◽

Brain Tumor ◽

Age Groups ◽

Tumor Location ◽

Childhood Brain Tumor ◽

Older Children ◽

Treatment Protocols ◽

Pituitary Dysfunction ◽

Oncologic Treatment

Abstract Background: Childhood brain tumor survivors (CBTS) are at risk for hypothalamic-pituitary (HP) dysfunction, mainly caused by radiation exposure or tumor involvement of the HP-region. The risk for HP dysfunction (HPD) may vary between different age groups due to maturation of the brain and differences in oncologic treatment protocols. The aim of this study was to determine the prevalence and risk factors of HPD in infant (IBTS) and toddler brain tumor survivors (TBTS) compared to older childhood brain tumor survivors (OCBTS). Patients and Methods: A retrospective analysis in a nationwide cohort of CBTS was performed. Prevalence and risk factors for HPD were compared between IBTS (aged 0-1 years at diagnosis), TBTS (aged 1-3 years at diagnosis) and OCBTS (aged >3-18 years at diagnosis). Results: In 718 included CBTS, with a median follow-up time of 7.9 years, overall no differences in percentage of HPD were found between the three age groups. Treatment with radiotherapy (RT) (OR 15.41; 95%CI 8.33 to 28.48), suprasellar tumor location (OR 46.62; 95%CI 19.64 to 110.66) and younger age (OR 1.09; 95%CI 1.02 to 1.15) were associated with HP dysfunction. Because IBTS were significantly less often treated with RT, subanalyses were performed for all CBTS not treated with radiation (n=459). In non-irradiated CBTS, IBTS and TBTS were significantly more frequently diagnosed with TSH-, ACTH- and ADH deficiency, compared to ECBTS. IBTS and TBTS showed significantly more weight gain (p<0.0001) and smaller height SDS (p=0.001) during follow-up. Conclusion: Infant and toddler brain tumor survivors seem to be more vulnerable to develop HP dysfunction than when compared to older children. These results emphasize the importance of special infant and toddlers brain tumor treatment protocols and endocrine surveillance in children treated for a brain tumor at young age.

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SWK-02. WEAVING COMFORT AND SUPPORT FOR CHILDREN WITH BRAIN TUMORS AND THEIR FAMILIES IN AN OUTPATIENT CLINIC

Neuro-Oncology ◽

10.1093/neuonc/noaa222.819 ◽

2020 ◽

Vol 22 (Supplement_3) ◽

pp. iii464-iii465

Author(s):

Ria Hawks ◽

Jane Bloom ◽

Maurine Packard ◽

Sonia Lugo ◽

Nadine Ulysses ◽

...

Keyword(s):

Brain Tumor ◽

Brain Tumors ◽

Outpatient Clinic ◽

Low Vision ◽

Medical Center ◽

Primary Brain Tumor ◽

Childhood Brain Tumor ◽

Tumor Patients ◽

Neurologic Disability

Abstract Parents of children diagnosed with brain tumors report high levels of stress at diagnosis and feelings of “being lost” on transition to outpatient follow-up care (Jackson AC, et al, 2007). Ssori is a Japanese form of free-style weaving that encourages people facing life-limiting challenges to discover inner strengths. We report our experience with Saori weaving with brain tumor patients and their families in a pediatric oncology outpatient clinic at a major university medical center. During 2019, we offered weaving sessions twice a week. We had a total of 151 encounters with hematology/oncology patients (age 5–18 years), siblings, or parents. Among these patients there were 20 with primary brain tumor diagnoses. Weaving was offered in the art therapy area of the clinic. After creating a fabric, the weavers had the opportunity to have their work sewn into functional objects, such as pillows, bags, purses, or healing pouches filled with beans that can be heated or cooled for comfort. Brain tumor patients readily engaged in weaving, despite various degrees of neurologic disability including hemiparesis or low vision. In the words of an 8 y/o weaver. “This is so cool. Daddy, can we always come when the weavers are here, so I can weave?” And from a mother: “This is great. She’s focused and busy!” Case studies, including a presentation of Legacy work, will be reported. In conclusion, Saori weaving can be an impactful intervention for childhood brain tumor patients and their families in an outpatient clinic setting.

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Reintegration Into School After Treatment for a Brain Tumor: The Child’s Perspective

Global Pediatric Health ◽

10.1177/2333794x19860659 ◽

2019 ◽

Vol 6 ◽

pp. 2333794X1986065

Author(s):

Stephanie Vanclooster ◽

Johan Bilsen ◽

Lieve Peremans ◽

Jutte Van Der Werff Ten Bosch ◽

Geneviève Laureys ◽

...

Keyword(s):

Brain Tumor ◽

Peer Relations ◽

Psychosocial Functioning ◽

School Attendance ◽

Social Contact ◽

School Attitudes ◽

Childhood Brain Tumor ◽

School Life ◽

Returning To School

This multiple case study investigated perspectives of childhood brain tumor survivors on reintegration into school over a 2-year period. Semistructured interviews were conducted with 5 children at 3 times to obtain an extensive view of their overall school experience. Thematic analysis of data resulted in 4 themes: “school life and participation,” “peer relations and friendships,” “performance and difficulties,” and “support and follow-up.” Childhood brain tumor survivors consider school attendance as part of a normal disease-free life. Social contact and friendships represent their main motivating factors for returning to school. Attitudes and feelings regarding performance, difficulties, and support vary among survivors and change over time. In conclusion, continuity in learning and social contact established before the return facilitate the reintegration process. A comprehensive assessment of their academic and psychosocial functioning should be organized on reentry. Systematic follow-up by parents, school staff, and health professionals throughout the child’s school career is required.

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Declining free thyroxine levels over time in irradiated childhood brain tumor survivors

Endocrine Connections ◽

10.1530/ec-18-0311 ◽

2018 ◽

Vol 7 (12) ◽

pp. 1322-1332 ◽

Cited By ~ 1

Author(s):

Laura van Iersel ◽

Sarah C Clement ◽

Antoinette Y N Schouten-van Meeteren ◽

Annemieke M Boot ◽

Hedi L Claahsen-van der Grinten ◽

...

Keyword(s):

Brain Tumor ◽

Reference Range ◽

Free Thyroxine ◽

Reference Ranges ◽

Childhood Brain Tumor ◽

Cranial Radiotherapy ◽

Central Hypothyroidism ◽

Future Studies ◽

Over Time

Objective The incidence of cranial radiotherapy (cRT)–induced central hypothyroidism (TSHD) in childhood brain tumor survivors (CBTS) is reported to be low. However, TSHD may be more frequent than currently suspected, as its diagnosis is challenging due to broad reference ranges for free thyroxine (FT4) concentrations. TSHD is more likely to be present when FT4 levels progressively decline over time. Therefore, we determined the incidence and latency time of TSHD and changes of FT4 levels over time in irradiated CBTS. Design Nationwide, 10-year retrospective study of irradiated CBTS. Methods TSHD was defined as ‘diagnosed’ when FT4 concentrations were below the reference range with low, normal or mildly elevated thyrotropin levels, and as ‘presumed’ when FT4 declined ≥ 20% within the reference range. Longitudinal FT4 concentrations over time were determined in growth hormone deficient (GHD) CBTS with and without diagnosed TSHD from cRT to last follow-up (paired t-test). Results Of 207 included CBTS, the 5-year cumulative incidence of diagnosed TSHD was 20.3%, which occurred in 50% (25/50) of CBTS with GHD by 3.4 years (range, 0.9–9.7) after cRT. Presumed TSHD was present in 20 additional CBTS. The median FT4 decline in GH-deficient CBTS was 41.3% (P < 0.01) to diagnosis of TSHD and 12.4% (P = 0.02) in GH-deficient CBTS without diagnosed TSHD. Conclusions FT4 concentrations in CBTS significantly decline over time after cRT, also in those not diagnosed with TSHD, suggesting that TSHD occurs more frequently and earlier than currently reported. The clinical relevance of cRT-induced FT4 decline over time should be investigated in future studies.

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