Risk of benign meningioma after childhood cancer in the DCOG-LATER cohort: contributions of radiation dose, exposed cranial volume, and age

Kok, Judith L.; Teepen, Jop C.; Leeuwen, Flora E. van; Tissing, Wim J. E.; Neggers, Sebastian J C M M; Pal, Helena J. van der; Loonen, Jacqueline; Bresters, Dorine; Versluys, Birgitta; Heuvel‐Eibrink, Marry M. van den; Broeder, Eline van Dulmen‐den; Loo, Margriet van der Heiden‐van der; Aleman, Berthe M.P.; Daniëls, Laurien A.; Haasbeek, Cornelis J.A.; Hoeben, Bianca A.W.; Janssens, Geert O.; Maduro, John H.; Oldenburger, Foppe; Rij, C. van; Tersteeg, Robbert J.H.A.; Hauptmann, Michael; Kremer, Leontien C. M.; Ronckers, Cécile M.

doi:10.1093/neuonc/noy124

Cited by 43 publications

(37 citation statements)

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“…Radiation induced meningioma (RIM) is defined as a meningioma occurring secondary to radiation treatment that satisfies specific criteria relating to: location of radiation, differing histology from previous malignancy, and at least a 5-year interval period after radiation treatment [1][2][3][4]. Meningiomas are the most common type of brain tumor to occur following cranio-spinal radiotherapy [5], with a one in eight risk of developing a RIM by the age of 40 [6][7][8]. Improvements in childhood and adult cancer survival rates have led to an increased incidence of RIM.…”

Section: Introductionmentioning

confidence: 99%

The growth rate and clinical outcomes of radiation induced meningioma undergoing treatment or active monitoring

et al. 2021

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Introduction Radiation induced meningioma (RIM) incidence is increasing in line with improved childhood cancer survival. No optimal management strategy consensus exists. This study aimed to delineate meningioma growth rates from tumor discovery and correlate with clinical outcomes. Methods Retrospective study of patients with a RIM, managed at a specialist tertiary neuroscience center (2007–2019). Tumor volume was measured from diagnosis and at subsequent interval scans. Meningioma growth rate was determined using a linear mixed-effects model. Clinical outcomes were correlated with growth rates accounting for imaging and clinical prognostic factors. Results Fifty-four patients (110 meningiomas) were included. Median duration of follow-up was 74 months (interquartile range [IQR], 41–102 months). Mean radiation dose was 41 Gy (standard deviation [SD] = 14.9) with a latency period of 34.4 years (SD = 13.7). Median absolute growth rate was 0.62 cm3/year and the median relative growth rate was 72%/year. Forty meningiomas (between 27 patients) underwent surgical intervention after a median follow-up duration of 4 months (IQR 2–35). Operated RIMs were clinically aggressive, likely to be WHO grade 2 at first resection (43.6%) and to progress after surgery (41%). Median time to progression was 28 months (IQR 13–60.5). A larger meningioma at discovery was associated with growth (HR 1.2 [95% CI 1.0–1.5], P = 0.039) but not progression after surgery (HR 2.2 [95% CI 0.7–6.6], P = 0.181). Twenty-seven (50%) patients had multiple meningiomas by the end of the study. Conclusion RIMs exhibit high absolute and relative growth rates after discovery. Surgery is recommended for symptomatic or rapidly growing meningiomas only. Recurrence risk after surgery is high.

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Section: Introductionmentioning

confidence: 99%

The growth rate and clinical outcomes of radiation induced meningioma undergoing treatment or active monitoring

et al. 2021

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“…First, there was a 40% response rate, though nonparticipants hardly differed from participants. Compared to other studies from the Dutch LATER cohort, survivors of CNS tumors seem to be underrepresented in our study [58]. This may complicate the generalization of our study's findings to all Dutch YACCS.…”

Section: Study Limitationsmentioning

confidence: 59%

A vulnerable age group: the impact of cancer on the psychosocial well-being of young adult childhood cancer survivors

Erp

Maurice‐Stam

Kremer

et al. 2021

Support Care Cancer

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Purpose This study aimed to increase our understanding of the psychosocial well-being of young adult childhood cancer survivors (YACCS) as well as the positive and negative impacts of cancer. Methods YACCS (aged 18–30, diagnosed ≤ 18, time since diagnosis ≥ 5 years) cross-sectionally filled out the “Pediatric Quality of Life Inventory Young Adults” (PedsQL-YA), “Hospital Anxiety and Depression Scale” (HADS), and “Checklist Individual Strengths” (CIS-20R) to measure fatigue and survivor-specific “Impact of Cancer - Childhood Survivors” (IOC-CS), which measures the long-term impact of childhood cancer in several domains. Descriptive statistics (IOC-CS), logistic regression (HADS, CIS-20R), and ANOVA (PedsQL-YA, HADS, CIS-20R) were performed. Associations between positive and negative impacts of childhood cancer and psychosocial outcomes were examined with linear regression analyses. Results YACCS (N = 151, 61.6% female, mean age 24.1 ± 3.6, mean time since diagnosis 13.6 ± 3.8) reported lower HRQOL (− .4 ≤ d ≤ − .5, p ≤ .001) and more anxiety (d = .4, p ≤ .001), depression (d = .4, p ≤ .01), and fatigue (.3 ≤ d ≤ .5, p ≤ .001) than young adults from the general Dutch population. They were at an increased risk of experiencing (sub)clinical anxiety (OR = 1.8, p = .017). YACCS reported more impact on scales representing a positive rather than negative impact of CC. Various domains of impact of childhood cancer were related to psychosocial outcomes, especially “Life Challenges” (HRQOL β = − .18, anxiety β = .36, depression β = .29) and “Body & Health” (HRQOL β = .27, anxiety β = − .25, depression β = − .26, fatigue β = − .47). Conclusion YACCS are vulnerable to psychosocial difficulties, but they also experience positive long-term impacts of childhood cancer. Positive and negative impacts of childhood cancer were associated with psychosocial outcomes in YACCS. Screening of psychosocial outcomes and offering targeted interventions are necessary to optimize psychosocial long-term follow-up care for YACCS.

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“…It is unclear whether the lower cumulative incidence reported here reflects differences in susceptibility to and/or surveillance for benign tumors among retinoblastoma versus other childhood cancer survivors or methodologic differences in the studies. Notably, in the Dutch cohort, meningioma represented the most common benign tumor after childhood cancer, mainly after high-dose, full-volume cranial radiotherapy [ 17 , 18 ]; however, similar treatments are uncommon for retinoblastoma. Additionally, whereas that study based ascertainment on linkage with a database containing results from all pathology examinations in the Netherlands, our study largely relied on directly surveying survivors, with subsequent confirmation of self-reported benign tumors through pathology report review (i.e., from biopsies).…”

Section: Discussionmentioning

confidence: 99%

“…Meningiomas may have been underascertained in this study, given that a substantial proportion of benign meningioma cases are based only on diagnostic imaging and lack histological confirmation (50% in a recent study based on cancer registry data in the US) [ 28 ]. Although radiotherapy for childhood cancer is a well-established risk factor for meningioma [ 18 , 29 , 30 ] and all five hereditary survivors with a meningioma in this study received external beam radiotherapy for their retinoblastoma, the small sample size precluded detailed assessment of meningioma risk factors in this study.…”

Section: Discussionmentioning

confidence: 99%

Benign Tumors in Long-Term Survivors of Retinoblastoma

Wijsard

Schonfeld

Leeuwen

et al. 2021

Cancers

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Hereditary retinoblastoma survivors have substantially increased risk of subsequent malignant neoplasms (SMNs). The risk of benign neoplasms, a substantial cause of morbidity, is unclear. We calculated the cumulative incidence of developing benign tumors at 60 years following retinoblastoma diagnosis among 1128 hereditary (i.e., bilateral retinoblastoma or unilateral with family history, mutation testing was not available) and 924 nonhereditary retinoblastoma survivors diagnosed during 1914–2006 at two US medical centers with follow-up through 2016. Using Cox proportional hazards regression, we compared benign tumor risk by hereditary status and evaluated the association between benign tumors and SMNs. There were 100 benign tumors among 73 hereditary survivors (cumulative incidence = 17.6%; 95% confidence interval [CI] = 12.9–22.8%) and 22 benign tumors among 16 nonhereditary survivors (cumulative incidence = 3.9%; 95%CI = 2.2–6.4%), corresponding to 4.9-fold (95%CI = 2.8–8.4) increased risk for hereditary survivors. The cumulative incidence after hereditary retinoblastoma was highest for lipoma among males (14.0%; 95%CI = 7.7–22.1%) and leiomyoma among females (8.9%; 95%CI = 5.2–13.8%). Among hereditary survivors, having a prior SMN was associated with 3.5-fold (95%CI = 2.0–6.1) increased risk of developing a benign tumor; the reciprocal risk for developing an SMN after a benign tumor was 1.8 (95%CI = 1.1–2.9). These large-scale, long-term data demonstrate an increased risk for benign tumors after hereditary versus nonhereditary retinoblastoma. If confirmed, the association between benign tumors and SMNs among hereditary patients may have implications for long-term surveillance.

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Risk of benign meningioma after childhood cancer in the DCOG-LATER cohort: contributions of radiation dose, exposed cranial volume, and age

Abstract: After cranial radiotherapy one in eight survivors developed late meningioma by age 40 years, associated with radiation dose- and exposure age, relevant for future treatment protocols and awareness among survivors and physicians.

Cited by 43 publications

References 19 publications

The growth rate and clinical outcomes of radiation induced meningioma undergoing treatment or active monitoring

The growth rate and clinical outcomes of radiation induced meningioma undergoing treatment or active monitoring

A vulnerable age group: the impact of cancer on the psychosocial well-being of young adult childhood cancer survivors

Benign Tumors in Long-Term Survivors of Retinoblastoma

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