Long-term neurologic health and psychosocial function of adult survivors of childhood medulloblastoma/PNET

A report from the Childhood Cancer Survivor Study

Allison A. King, Kristy Seidel, Chongzhi Di, Wendy M. Leisenring, Stephanie Mabry Perkins, Kevin R. Krull, Charles A. Sklar, Daniel M. Green, Gregory Armstrong, Lonnie K. Zeltzer, Elizabeth Wells, Marilyn Stovall, Nicole J. Ullrich, Kevin C. Oeffinger, Leslie L. Robison, Roger J. Packer

Research output: Contribution to journalArticle

15 Citations (Scopus)

Abstract

Background. Medulloblastoma is the most common malignant childhood brain tumor, although long-term risks for chronic neurologic health and psychosocial functioning in aging adult survivors are incompletely characterized. Methods. The Childhood Cancer Survivor Study (CCSS) includes 380 fve-year survivors of medulloblastoma/primitive neuroectodermal tumor (PNET; median age at follow-up: 30 y, interquartile range 24-36) and sibling comparison (n = 4031). Cumulative incidence of neurologic health conditions was reported. Cox regression models provided hazard ratios (HRs) and 95% CIs. Cross-sectional outcomes were assessed using generalized linear models. Results. Compared with siblings, survivors were at increased risk of late-onset hearing loss (HR: 36.0, 95% CI: 23.6-54.9), stroke (HR: 33.9, 95% CI: 17.8-64.7), seizure (HR: 12.8, 95% CI: 9.0-18.1), poor balance (HR: 10.4, 95% CI: 6.7-15.9), tinnitus (HR: 4.8, 95% CI: 3.5-6.8), and cataracts (HR: 31.8, 95% CI: 16.7-60.5). Temporal/frontal lobe radiotherapy of 50 Gy or more increased risk for hearing loss (HR: 1.9, 95% CI: 1.1-1.3), seizure (HR: 2.1, 95% CI: 1.1-3.9), stroke (HR: 3.5, 95% CI: 1.3-9.1), and tinnitus (HR: 2.0, 95% CI: 1.0-3.9). Survivors were less likely than siblings to earn a college degree (relative risk [RR]: 0.49, 95% CI: 0.39-0.60), marry (RR: 0.35, 95% CI: 0.29-0.42), and live independently (RR: 0.58, 95% CI: 0.52-0.66). Conclusions. Adult survivors of childhood medulloblastoma/PNET demonstrate pronounced risk for hearing impairment, stroke, lower educational attainment, and social independence. Interventions to support survivors should be a high priority.

Original languageEnglish (US)
Pages (from-to)689-698
Number of pages10
JournalNeuro-Oncology
Volume19
Issue number5
DOIs
StatePublished - May 1 2017
Externally publishedYes

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Primitive Neuroectodermal Tumors
Medulloblastoma
Nervous System
Survivors
Health
Hearing Loss
Neoplasms
Siblings
Tinnitus
Stroke
Seizures
Frontal Lobe
Temporal Lobe
Proportional Hazards Models
Brain Neoplasms
Cataract
Linear Models
Radiotherapy
Incidence

All Science Journal Classification (ASJC) codes

  • Oncology
  • Clinical Neurology
  • Cancer Research

Cite this

Long-term neurologic health and psychosocial function of adult survivors of childhood medulloblastoma/PNET : A report from the Childhood Cancer Survivor Study. / King, Allison A.; Seidel, Kristy; Di, Chongzhi; Leisenring, Wendy M.; Perkins, Stephanie Mabry; Krull, Kevin R.; Sklar, Charles A.; Green, Daniel M.; Armstrong, Gregory; Zeltzer, Lonnie K.; Wells, Elizabeth; Stovall, Marilyn; Ullrich, Nicole J.; Oeffinger, Kevin C.; Robison, Leslie L.; Packer, Roger J.

In: Neuro-Oncology, Vol. 19, No. 5, 01.05.2017, p. 689-698.

Research output: Contribution to journalArticle

King, AA, Seidel, K, Di, C, Leisenring, WM, Perkins, SM, Krull, KR, Sklar, CA, Green, DM, Armstrong, G, Zeltzer, LK, Wells, E, Stovall, M, Ullrich, NJ, Oeffinger, KC, Robison, LL & Packer, RJ 2017, 'Long-term neurologic health and psychosocial function of adult survivors of childhood medulloblastoma/PNET: A report from the Childhood Cancer Survivor Study', Neuro-Oncology, vol. 19, no. 5, pp. 689-698. https://doi.org/10.1093/neuonc/now242
King, Allison A. ; Seidel, Kristy ; Di, Chongzhi ; Leisenring, Wendy M. ; Perkins, Stephanie Mabry ; Krull, Kevin R. ; Sklar, Charles A. ; Green, Daniel M. ; Armstrong, Gregory ; Zeltzer, Lonnie K. ; Wells, Elizabeth ; Stovall, Marilyn ; Ullrich, Nicole J. ; Oeffinger, Kevin C. ; Robison, Leslie L. ; Packer, Roger J. / Long-term neurologic health and psychosocial function of adult survivors of childhood medulloblastoma/PNET : A report from the Childhood Cancer Survivor Study. In: Neuro-Oncology. 2017 ; Vol. 19, No. 5. pp. 689-698.
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title = "Long-term neurologic health and psychosocial function of adult survivors of childhood medulloblastoma/PNET: A report from the Childhood Cancer Survivor Study",
abstract = "Background. Medulloblastoma is the most common malignant childhood brain tumor, although long-term risks for chronic neurologic health and psychosocial functioning in aging adult survivors are incompletely characterized. Methods. The Childhood Cancer Survivor Study (CCSS) includes 380 fve-year survivors of medulloblastoma/primitive neuroectodermal tumor (PNET; median age at follow-up: 30 y, interquartile range 24-36) and sibling comparison (n = 4031). Cumulative incidence of neurologic health conditions was reported. Cox regression models provided hazard ratios (HRs) and 95{\%} CIs. Cross-sectional outcomes were assessed using generalized linear models. Results. Compared with siblings, survivors were at increased risk of late-onset hearing loss (HR: 36.0, 95{\%} CI: 23.6-54.9), stroke (HR: 33.9, 95{\%} CI: 17.8-64.7), seizure (HR: 12.8, 95{\%} CI: 9.0-18.1), poor balance (HR: 10.4, 95{\%} CI: 6.7-15.9), tinnitus (HR: 4.8, 95{\%} CI: 3.5-6.8), and cataracts (HR: 31.8, 95{\%} CI: 16.7-60.5). Temporal/frontal lobe radiotherapy of 50 Gy or more increased risk for hearing loss (HR: 1.9, 95{\%} CI: 1.1-1.3), seizure (HR: 2.1, 95{\%} CI: 1.1-3.9), stroke (HR: 3.5, 95{\%} CI: 1.3-9.1), and tinnitus (HR: 2.0, 95{\%} CI: 1.0-3.9). Survivors were less likely than siblings to earn a college degree (relative risk [RR]: 0.49, 95{\%} CI: 0.39-0.60), marry (RR: 0.35, 95{\%} CI: 0.29-0.42), and live independently (RR: 0.58, 95{\%} CI: 0.52-0.66). Conclusions. Adult survivors of childhood medulloblastoma/PNET demonstrate pronounced risk for hearing impairment, stroke, lower educational attainment, and social independence. Interventions to support survivors should be a high priority.",
author = "King, {Allison A.} and Kristy Seidel and Chongzhi Di and Leisenring, {Wendy M.} and Perkins, {Stephanie Mabry} and Krull, {Kevin R.} and Sklar, {Charles A.} and Green, {Daniel M.} and Gregory Armstrong and Zeltzer, {Lonnie K.} and Elizabeth Wells and Marilyn Stovall and Ullrich, {Nicole J.} and Oeffinger, {Kevin C.} and Robison, {Leslie L.} and Packer, {Roger J.}",
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T1 - Long-term neurologic health and psychosocial function of adult survivors of childhood medulloblastoma/PNET

T2 - A report from the Childhood Cancer Survivor Study

AU - King, Allison A.

AU - Seidel, Kristy

AU - Di, Chongzhi

AU - Leisenring, Wendy M.

AU - Perkins, Stephanie Mabry

AU - Krull, Kevin R.

AU - Sklar, Charles A.

AU - Green, Daniel M.

AU - Armstrong, Gregory

AU - Zeltzer, Lonnie K.

AU - Wells, Elizabeth

AU - Stovall, Marilyn

AU - Ullrich, Nicole J.

AU - Oeffinger, Kevin C.

AU - Robison, Leslie L.

AU - Packer, Roger J.

PY - 2017/5/1

Y1 - 2017/5/1

N2 - Background. Medulloblastoma is the most common malignant childhood brain tumor, although long-term risks for chronic neurologic health and psychosocial functioning in aging adult survivors are incompletely characterized. Methods. The Childhood Cancer Survivor Study (CCSS) includes 380 fve-year survivors of medulloblastoma/primitive neuroectodermal tumor (PNET; median age at follow-up: 30 y, interquartile range 24-36) and sibling comparison (n = 4031). Cumulative incidence of neurologic health conditions was reported. Cox regression models provided hazard ratios (HRs) and 95% CIs. Cross-sectional outcomes were assessed using generalized linear models. Results. Compared with siblings, survivors were at increased risk of late-onset hearing loss (HR: 36.0, 95% CI: 23.6-54.9), stroke (HR: 33.9, 95% CI: 17.8-64.7), seizure (HR: 12.8, 95% CI: 9.0-18.1), poor balance (HR: 10.4, 95% CI: 6.7-15.9), tinnitus (HR: 4.8, 95% CI: 3.5-6.8), and cataracts (HR: 31.8, 95% CI: 16.7-60.5). Temporal/frontal lobe radiotherapy of 50 Gy or more increased risk for hearing loss (HR: 1.9, 95% CI: 1.1-1.3), seizure (HR: 2.1, 95% CI: 1.1-3.9), stroke (HR: 3.5, 95% CI: 1.3-9.1), and tinnitus (HR: 2.0, 95% CI: 1.0-3.9). Survivors were less likely than siblings to earn a college degree (relative risk [RR]: 0.49, 95% CI: 0.39-0.60), marry (RR: 0.35, 95% CI: 0.29-0.42), and live independently (RR: 0.58, 95% CI: 0.52-0.66). Conclusions. Adult survivors of childhood medulloblastoma/PNET demonstrate pronounced risk for hearing impairment, stroke, lower educational attainment, and social independence. Interventions to support survivors should be a high priority.

AB - Background. Medulloblastoma is the most common malignant childhood brain tumor, although long-term risks for chronic neurologic health and psychosocial functioning in aging adult survivors are incompletely characterized. Methods. The Childhood Cancer Survivor Study (CCSS) includes 380 fve-year survivors of medulloblastoma/primitive neuroectodermal tumor (PNET; median age at follow-up: 30 y, interquartile range 24-36) and sibling comparison (n = 4031). Cumulative incidence of neurologic health conditions was reported. Cox regression models provided hazard ratios (HRs) and 95% CIs. Cross-sectional outcomes were assessed using generalized linear models. Results. Compared with siblings, survivors were at increased risk of late-onset hearing loss (HR: 36.0, 95% CI: 23.6-54.9), stroke (HR: 33.9, 95% CI: 17.8-64.7), seizure (HR: 12.8, 95% CI: 9.0-18.1), poor balance (HR: 10.4, 95% CI: 6.7-15.9), tinnitus (HR: 4.8, 95% CI: 3.5-6.8), and cataracts (HR: 31.8, 95% CI: 16.7-60.5). Temporal/frontal lobe radiotherapy of 50 Gy or more increased risk for hearing loss (HR: 1.9, 95% CI: 1.1-1.3), seizure (HR: 2.1, 95% CI: 1.1-3.9), stroke (HR: 3.5, 95% CI: 1.3-9.1), and tinnitus (HR: 2.0, 95% CI: 1.0-3.9). Survivors were less likely than siblings to earn a college degree (relative risk [RR]: 0.49, 95% CI: 0.39-0.60), marry (RR: 0.35, 95% CI: 0.29-0.42), and live independently (RR: 0.58, 95% CI: 0.52-0.66). Conclusions. Adult survivors of childhood medulloblastoma/PNET demonstrate pronounced risk for hearing impairment, stroke, lower educational attainment, and social independence. Interventions to support survivors should be a high priority.

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