Abstract
Purpose
Improved therapies for pediatric central nervous system (CNS) tumors have increased survival rates; however, many survivors experience significant long-term functional limitations. Survivors of pediatric CNS tumors can experience deficits in social attainment. The aim of this review was to systematically amalgamate findings pertaining to social attainment (i.e., educational attainment, marriage, employment outcomes) in survivors of pediatric CNS tumors.
Methods
PubMed (web-based), PsycINFO (EBSCO), EMBASE (Ovid), and Web of Science (Thomson Reuters) were used to identify articles published between January 2011 and September 2018. Eligible studies reported outcomes for survivors of pediatric CNS tumors diagnosed before age 21 years and > 5 years from diagnosis and/or > 2 years off therapy. All data were independently abstracted by two reviewers. Random-effects meta-analyses were performed using Review Manager 5.0.
Results
The search yielded 7021 unique publications. Forty-six were included in the current review. Meta-analyses revealed survivors of CNS tumors were significantly more likely to have completed compulsory education only (OR = 1.87, 95% CI = 1.66, 2.12, p < 0.00001), less likely to be married (OR = 4.70, 95% CI = 3.89, 5.68, p < 0.00001), and more likely to be unemployed (OR = 2.84, 95% CI = 2.62, 3.08, p < 0.00001) compared to non-cancer controls. Cranial radiation therapy, neurocognitive deficits, and younger age at diagnosis were associated with poorer outcomes. Hearing loss and bilateral blindness were also related to poorer outcomes. Sex did not impact social attainment outcomes.
Conclusions
Survivors of pediatric CNS tumors are at elevated risk for poor attainment of key adult social outcomes.
Implications for Cancer Survivors
There is a critical need to develop interventions to support survivors in becoming independent and productive adults.
Similar content being viewed by others
References
Ward E, et al. Childhood and adolescent cancer statistics, 2014. CA Cancer J Clin. 64:83–103.
Schulte F, Barrera M. Social competence in childhood brain tumor survivors: a comprehensive review. Support Care Cancer. 2010;18:1499–513.
Zebrack B, et al. Psychological outcomes in long-term survivors of childhood brain cancer: a report from the childhood cancer survivor study. J Clin Oncol. 2004;22:999–1006.
Gurney JG, Krull KR, Kadan-Lottick N, Nicholson HS, Nathan PC, Zebrack B, et al. Social outcomes in the childhood cancer survivor study cohort. J Clin Oncol. 2009;27(14):2390–5.
Mulhern RK, Merchant TE, Gajjar A, Reddick WE, Kun LE. Late neurocognitive sequelae in survivors of brain tumours in childhood. Lancet Oncol. 2004;5:399–408.
Group, C.s.O. Long-term follow-up guidelines for survivors of childhood, adolescent and young adult cancers. Version 5.0. Monoovia: Children’s Oncology Group; 2018.
Schulte F, Brinkman TM, Li C, Fay-McClymont T, Srivastava DK, Ness KK, et al. Social adjustment in adolescent survivors of pediatric CNS tumors: a report from the Childhood Cancer Survivor Study. Cancer. 2018;124(17):3596–608.
Moher D, et al. Preferred Reporting Items for Systematic Review and Meta-Analysis Protocols (PRISMA-P) 2015 statement. Syst Rev. 2015;4(1).
Higgins JPT, Green S. Cochrane handbook for systematic reviews of interventions version 5.1.0 [updated March 2011]. Cochrane Collaboration; 2011. 4.
Landis JR, Koch GG. The measurement of observer agreement for categorical data. Biometrics. 1977;33:159–74.
Review Manager (RevMan). 2014, Copenhagen: the Nordic Cochrane Centre, The Cochrane Collaboration.
Khelifa-Gallois N et al. Long-term functional outcome of patients with cerebellar pilocytic astrocytoma surgically treated in childhood.
Ahomaki R, et al. Non-graduation after comprehensive school, and early retirement but not unemployment are prominent in childhood cancer survivors—a Finnish registry-based study. J Cancer Surviv. 2017;11(2):284–94.
Barakat LP, et al. Health-related quality of life of adolescent and young adult survivors of childhood brain tumors. Psychooncology. 2015;24(7):804–11.
Bashore L, Breyer E. Educational and career goal attainments in young adult childhood cancer survivors. J Spec Pediatr Nurs. 2017;22(2).
Beek L, et al. Surviving a brain tumor in childhood: impact on family functioning in adolescence. Psychooncology. 2015;24(1):89–94.
Brinkman TM, et al. Attainment of Functional and Social Independence in Adult Survivors of Pediatric CNS Tumors: A Report From the St Jude Lifetime Cohort Study. J Clin Oncol. 2018;36(27):2762–9.
Brinkman TM, Bass JK, Li Z, Ness KK, Gajjar A, Pappo AS, et al. Treatment-induced hearing loss and adult social outcomes in survivors of childhood CNS and non-CNS solid tumors: results from the St. Jude Lifetime Cohort Study. Cancer. 2015;121(22):4053–61.
Brinkman TM, et al. Long-Term Neurocognitive Functioning and Social Attainment in Adult Survivors of Pediatric CNS Tumors: Results From the St Jude Lifetime Cohort Study. J Clin Oncol. 2016;34(12):1358–67.
Chen CM, Chen YC, Wong TT. Comparison of resilience in adolescent survivors of brain tumors and healthy adolescents. Cancer Nurs. 2014;37(5):373–81.
Cheung C-k, Mok B-h Psychosocial adaptation of childhood cancer survivors and their siblings.
de Blank PM, et al. Impact of vision loss among survivors of childhood central nervous system astroglial tumors. Cancer. 2016;122(5):730–9.
Dumas A, Berger C, Auquier P, Michel G, Fresneau B, Sètchéou Allodji R, et al. Educational and occupational outcomes of childhood cancer survivors 30 years after diagnosis: a French cohort study. Br J Cancer. 2016;114(9):1060–8.
Ehrstedt C, et al. Glioneuronal tumors in childhood - Before and after surgery. A long-term follow-up study. Epilepsy Behav. 2017;72:82–8.
Fjalldal S, et al. Hypothalamic involvement predicts cognitive performance and psychosocial health in long-term survivors of childhood craniopharyngioma. J Clin Endocrinol Metab. 2013;98:3253–62.
Font-Gonzalez A, Feijen EL, Sieswerda E, van Dulmen-den Broeder E, Grootenhuis M, Maurice-Stam H, et al. Social outcomes in adult survivors of childhood cancer compared to the general population: linkage of a cohort with population registers. Psychooncology. 2016;25(8):933–41.
Fox M, King T Pituitary disorders as a predictor of apathy and executive dysfunction in adult survivors of childhood brain tumors.
Frobisher C, Lancashire ER, Jenkinson H, Winter DL, Kelly J, Reulen RC, et al. Employment status and occupational level of adult survivors of childhood cancer in Great Britain: the British childhood cancer survivor study. Int J Cancer. 2017;140(12):2678–92.
Gautier A, et al. Markers of recurrence and long-term morbidity in craniopharyngioma: A systematic analysis of 171 patients. J Clin Endocrinol Metab. 2012;97:1258–67.
Ghaderi S, Engeland A, Gunnes MW, Moster D, Ruud E, Syse A, et al. Educational attainment among long-term survivors of cancer in childhood and adolescence: a Norwegian population-based cohort study. J Cancer Surviv. 2016;10(1):87–95.
Gunn ME, et al. Late new morbidity in survivors of adolescent and youngadulthood brain tumors in Finland: a registry-based study. Neuro-Oncology. 2015;17(10):1412–8.
Hoag J, et al. Feasibility of conducting long-term follow-up of children and infants treated for CNS tumors on the same cooperative group clinical trial protocol. J Clin Psychol Med Settings. 2014;21(2):136–43.
Hocking M et al. Family Functioning Mediates the Association Between Neurocognitive Functioning and Health-Related Quality of Life in Young Adult Survivors of Childhood Brain Tumors.
Holland AA, Hughes CW, Stavinoha PL. School Competence and Fluent Academic Performance: Informing Assessment of Educational Outcomes in Survivors of Pediatric Medulloblastoma. Appl Neuropsychol Child. 2015;4(4):249–56.
Hornquist L, Rickardsson J, Lannering B, Gustafsson G, Boman KK. Altered self-perception in adult survivors treated for a CNS tumor in childhood or adolescence: population-based outcomes compared with the general population. Neuro-Oncology. 2015;17(5):733–40.
Howard AF, et al. Parents' perspectives of life challenges experienced by longterm paediatric brain tumour survivors: work and finances, daily and social functioning, and legal difficulties. J Cancer Surviv. 2014;8(3):372–83.
Jervaeus A, et al. Survivors of childhood cancer report high levels of independence five years after diagnosis. J Pediatr Oncol Nurs. 2014;31(5):245–51.
Jinguji S, et al. Factors affecting functional outcomes in long-term survivors of intracranial germinomas: a 20-year experience in a single institution. J Neurosurg Pediatr. 2013;11(4):454–63.
King AA, et al. Long-term neurologic health and psychosocial function of adult survivors of childhood medulloblastoma/PNET: a report from the Childhood Cancer Survivor Study. Neuro-Oncology. 2017;19(5):689–98.
Kirchhoff AC, Krull KR, Ness KK, Park ER, Oeffinger KC, Hudson MM, et al. Occupational outcomes of adult childhood cancer survivors: a report from the childhood cancer survivor study. Cancer. 2011;117(13):3033–44.
Koch SV, Kejs AMT, Engholm G, Møller H, Johansen C, Schmiegelow K. Marriage and divorce among childhood cancer survivors. J Pediatr Hematol Oncol. 2011;33(7):500–5.
Korinthenberg R, et al. Assessing quality of life in long-term survivors after 125I brachytherapy for low-grade glioma in childhood. Neuropediatrics. 2011;42:110–5.
Kuehni CE, Strippoli MPF, Rueegg CS, Rebholz CE, Bergstraesser E, Grotzer M, et al. Educational achievement in Swiss childhood cancer survivors compared with the general population. Cancer. 2012;118(5):1439–49.
Longaud-Vales A et al. Assessment of executive functioning in children and young adults treated for frontal lobe tumours using ecologically valid tests.
Lundar T, et al. Pediatric spinal ependymomas: an unpredictable and puzzling disease. Long-term follow-up of a single consecutive institutional series of ten patients. Childs Nerv Syst. 2014;30(12):2083–8.
Massimino M, et al. Long-term results of combined preradiation chemotherapy and age-tailored radiotherapy doses for childhood medulloblastoma. J Neuro-Oncol. 2012;108(1):163–71.
Memmesheimer RM, et al. Psychological well-being and independent living of young adults with childhood-onset craniopharyngioma. Dev Med Child Neurol. 2017;59(8):829–36.
Olson R, et al. Prospective evaluation of legal difficulties and quality of life in adult survivors of childhood cancer. Pediatr Blood Cancer. 2011;56(3):439–43.
Pfitzer C, Zynda A, Hohmann C, Keil T, Borgmann-Staudt A. Educational level of childhood brain tumor survivors: results from a German survey. Klin Padiatr. 2013;225(3):138–44.
Pivetta E, Maule MM, Pisani P, Zugna D, Haupt R, Jankovic M, et al. Marriage and parenthood among childhood cancer survivors: a report from the Italian AIEOP Off-Therapy Registry. Haematologica. 2011;96(5):744–51.
Pletschko T, et al. Applying the International Classification of Functioning- Children and Youth Version to Pediatric Neuro-oncology. J Child Neurol. 2017;32(1):23–8.
de Ruiter MA, et al. Psychosocial profile of pediatric brain tumor survivors with neurocognitive complaints. Qual Life Res. 2016;25(2):435–46.
Wengenroth L, Rueegg CS, Michel G, Essig S, Ammann RA, Bergstraesser E, et al. Life partnerships in childhood cancer survivors, their siblings, and the general population. Pediatr Blood Cancer. 2014;61(3):538–45.
Wengenroth L, Sommer G, Schindler M, Spycher BD, von der Weid NX, Stutz-Grunder E, et al. Income in adult survivors of childhood cancer. PLoS One. 2016;11(5):e0155546.
Yano S, et al. Quality of Life and Clinical Features of Long-Term Survivors Surgically Treated for Pediatric Craniopharyngioma. World Neurosurg. 2016;85:153–62.
Yuen KC, et al. Clinical characteristics and effects of GH replacement therapy in adults with childhood-onset craniopharyngioma compared with those in adults with other causes of childhood-onset hypothalamic-pituitary dysfunction. Eur J Endocrinol. 2013;169(4):511–9.
Schulte F, Bartels U, Barrera M. A pilot study evaluating the efficacy of a group social skills program for survivors of childhood CNS tumors using a comparison group and teacher reports. Psycho-Oncology. 2014;23:597–600.
Devine KA, Bukowski WM, Sahler OJZ, Ohman-Strickland P, Smith TH, Lown EA, et al. Social competence in childhood brain tumor survivors: feasibility and preliminary outcomes of a peer-mediated intervention. J Dev Behav Pediatr. 2016;37(6):475–82.
Hocking MC, McCurdy M. Social competence in pediatric brain tumor survivors: application of a model from social neuroscience and developmental psychology. Pediatr Blood Cancer. 2015;62:375–84.
Olson R, et al. Prospective evaluation of legal difficulties and quality of life in adult survivors of childhood cancer Survey Construction and QoL Measure Selection; 2011. p. 439–443.
Brinkman TM, Krasin MJ, Liu W, Armstrong GT, Ojha RP, Sadighi ZS, et al. Long-term neurocognitive functioning and social attainment in adult survivors of pediatric CNS tumors: results from the St Jude Lifetime Cohort Study. J Clin Oncol. 2016;34:1358–67.
Gautier A, Godbout A, Grosheny C, Tejedor I, Coudert M, Courtillot C, et al. Markers of recurrence and long-term morbidity in craniopharyngioma: a systematic analysis of 171 patients. J Clin Endocrinol Metab. 2012;97:1258–67.
de Ruiter MA, et al. Neurocognitive consequences of a paediatric brain tumour and its treatment: a meta-analysis. Dev Med Child Neurol. 2013;55(5):408–17.
Barrera M, et al. A randomized control intervention trial to improve social skills and quality of life in pediatric brain tumor survivors. Psychooncology, 2017.
Barrera M, Schulte F. A group social skills intervention program for survivors of childhood brain tumors. J Pediatr Psychol. 2009;38(10):1108–18.
Olson R, Hung G, Bobinski MA, Goddard K. Prospective evaluation of legal difficulties and quality of life in adult survivors of childhood cancer. Pediatric Blood & Cancer. (2011);56(3):439–443
Funding
This work was supported in part by the Children’s Oncology Group grant U10CA180886 from the National Cancer Institute and the St. Baldrick’s Foundation.
Author information
Authors and Affiliations
Corresponding author
Ethics declarations
Conflict of interest
The authors declare that they have no conflict of interest.
Ethical approval
This article does not contain any studies with human participants performed by any of the authors.
Additional information
Publisher’s note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Electronic supplementary material
ESM 1
(DOCX 148 kb)
Rights and permissions
About this article
Cite this article
Schulte, F., Kunin-Batson, A.S., Olson-Bullis, B.A. et al. Social attainment in survivors of pediatric central nervous system tumors: a systematic review and meta-analysis from the Children’s Oncology Group. J Cancer Surviv 13, 921–931 (2019). https://doi.org/10.1007/s11764-019-00808-3
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s11764-019-00808-3