Longitudinal follow-up of adult survivors of Ewing sarcoma

A report from the Childhood Cancer Survivor Study

Neyssa M. Marina, Qi Liu, Sarah S. Donaldson, Charles A. Sklar, Gregory Armstrong, Kevin C. Oeffinger, Wendy M. Leisenring, Jill P. Ginsberg, Tara O. Henderson, Joseph P. Neglia, Marilyn A. Stovall, Yutaka Yasui, R. Lor Randall, David S. Geller, Leslie L. Robison, Kirsten K. Ness

Research output: Contribution to journalArticle

8 Citations (Scopus)

Abstract

BACKGROUND: Ewing sarcoma survivors (ESSs) are at increased risk for treatment-related complications. The incidence of treatment-related morbidity and late mortality with aging is unknown. METHODS: This study reports survival probabilities, estimated with the Kaplan-Meier method, and the cumulative incidence of cause-specific mortality and chronic conditions among ESSs in the Childhood Cancer Survivor Study who were treated between 1970 and 1986. Piecewise exponential models were used to estimate relative rates (RRs) and 95% confidence intervals (CIs) for these outcomes. Chronic conditions were graded with the Common Terminology Criteria for Adverse Events (version 4.03). RESULTS: Among 404 5-year ESSs (median age at last follow-up, 34.8 years; range, 9.1-54.8 years), the 35-year survival rate was 70% (95% CI, 66%-74%). Late recurrence (cumulative incidence at 35 years, 15.1%) was the most common cause of death, and it was followed by treatment-related causes (11.2%). There were 53 patients with subsequent neoplasms (SNs; cumulative incidence at 35 years, 24.0%), and 38 were malignant (14.3% at 35 years). The standardized incidence ratios were 377.1 (95% CI, 172.1-715.9) for osteosarcoma, 28.9 (95% CI, 3.2-104.2) for acute myeloid leukemia, 14.9 (95% CI, 7.9-25.5) for breast cancer, and 13.1 (95% CI, 4.8-28.5) for thyroid cancer. Rates of chronic conditions were highest for musculoskeletal (RR, 18.1; 95% CI, 12.8-25.7) and cardiac complications (RR, 1.8; 95% CI, 1.4-2.3). Thirty-five years after the diagnosis, the cumulative incidences of any chronic conditions and 2 or more chronic conditions were 84.6% (95% CI, 80.4%-88.8%) and 73.8% (95% CI, 67.8%-79.9%), respectively. CONCLUSIONS: With extended follow-up, ESSs' risk for late mortality and SNs does not plateau. Treatment-related chronic conditions develop years after therapy, and this supports the need for lifelong follow-up. Cancer 2017;123:2551–60.

Original languageEnglish (US)
Pages (from-to)2551-2560
Number of pages10
JournalCancer
Volume123
Issue number13
DOIs
StatePublished - Jul 1 2017

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Ewing's Sarcoma
Survivors
Confidence Intervals
Neoplasms
Incidence
Mortality
Therapeutics
Osteosarcoma
Thyroid Neoplasms
Terminology
Acute Myeloid Leukemia
Cause of Death
Survival Rate
Breast Neoplasms
Morbidity
Recurrence

All Science Journal Classification (ASJC) codes

  • Oncology
  • Cancer Research

Cite this

Marina, N. M., Liu, Q., Donaldson, S. S., Sklar, C. A., Armstrong, G., Oeffinger, K. C., ... Ness, K. K. (2017). Longitudinal follow-up of adult survivors of Ewing sarcoma: A report from the Childhood Cancer Survivor Study. Cancer, 123(13), 2551-2560. https://doi.org/10.1002/cncr.30627

Longitudinal follow-up of adult survivors of Ewing sarcoma : A report from the Childhood Cancer Survivor Study. / Marina, Neyssa M.; Liu, Qi; Donaldson, Sarah S.; Sklar, Charles A.; Armstrong, Gregory; Oeffinger, Kevin C.; Leisenring, Wendy M.; Ginsberg, Jill P.; Henderson, Tara O.; Neglia, Joseph P.; Stovall, Marilyn A.; Yasui, Yutaka; Randall, R. Lor; Geller, David S.; Robison, Leslie L.; Ness, Kirsten K.

In: Cancer, Vol. 123, No. 13, 01.07.2017, p. 2551-2560.

Research output: Contribution to journalArticle

Marina, NM, Liu, Q, Donaldson, SS, Sklar, CA, Armstrong, G, Oeffinger, KC, Leisenring, WM, Ginsberg, JP, Henderson, TO, Neglia, JP, Stovall, MA, Yasui, Y, Randall, RL, Geller, DS, Robison, LL & Ness, KK 2017, 'Longitudinal follow-up of adult survivors of Ewing sarcoma: A report from the Childhood Cancer Survivor Study', Cancer, vol. 123, no. 13, pp. 2551-2560. https://doi.org/10.1002/cncr.30627
Marina, Neyssa M. ; Liu, Qi ; Donaldson, Sarah S. ; Sklar, Charles A. ; Armstrong, Gregory ; Oeffinger, Kevin C. ; Leisenring, Wendy M. ; Ginsberg, Jill P. ; Henderson, Tara O. ; Neglia, Joseph P. ; Stovall, Marilyn A. ; Yasui, Yutaka ; Randall, R. Lor ; Geller, David S. ; Robison, Leslie L. ; Ness, Kirsten K. / Longitudinal follow-up of adult survivors of Ewing sarcoma : A report from the Childhood Cancer Survivor Study. In: Cancer. 2017 ; Vol. 123, No. 13. pp. 2551-2560.
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title = "Longitudinal follow-up of adult survivors of Ewing sarcoma: A report from the Childhood Cancer Survivor Study",
abstract = "BACKGROUND: Ewing sarcoma survivors (ESSs) are at increased risk for treatment-related complications. The incidence of treatment-related morbidity and late mortality with aging is unknown. METHODS: This study reports survival probabilities, estimated with the Kaplan-Meier method, and the cumulative incidence of cause-specific mortality and chronic conditions among ESSs in the Childhood Cancer Survivor Study who were treated between 1970 and 1986. Piecewise exponential models were used to estimate relative rates (RRs) and 95{\%} confidence intervals (CIs) for these outcomes. Chronic conditions were graded with the Common Terminology Criteria for Adverse Events (version 4.03). RESULTS: Among 404 5-year ESSs (median age at last follow-up, 34.8 years; range, 9.1-54.8 years), the 35-year survival rate was 70{\%} (95{\%} CI, 66{\%}-74{\%}). Late recurrence (cumulative incidence at 35 years, 15.1{\%}) was the most common cause of death, and it was followed by treatment-related causes (11.2{\%}). There were 53 patients with subsequent neoplasms (SNs; cumulative incidence at 35 years, 24.0{\%}), and 38 were malignant (14.3{\%} at 35 years). The standardized incidence ratios were 377.1 (95{\%} CI, 172.1-715.9) for osteosarcoma, 28.9 (95{\%} CI, 3.2-104.2) for acute myeloid leukemia, 14.9 (95{\%} CI, 7.9-25.5) for breast cancer, and 13.1 (95{\%} CI, 4.8-28.5) for thyroid cancer. Rates of chronic conditions were highest for musculoskeletal (RR, 18.1; 95{\%} CI, 12.8-25.7) and cardiac complications (RR, 1.8; 95{\%} CI, 1.4-2.3). Thirty-five years after the diagnosis, the cumulative incidences of any chronic conditions and 2 or more chronic conditions were 84.6{\%} (95{\%} CI, 80.4{\%}-88.8{\%}) and 73.8{\%} (95{\%} CI, 67.8{\%}-79.9{\%}), respectively. CONCLUSIONS: With extended follow-up, ESSs' risk for late mortality and SNs does not plateau. Treatment-related chronic conditions develop years after therapy, and this supports the need for lifelong follow-up. Cancer 2017;123:2551–60.",
author = "Marina, {Neyssa M.} and Qi Liu and Donaldson, {Sarah S.} and Sklar, {Charles A.} and Gregory Armstrong and Oeffinger, {Kevin C.} and Leisenring, {Wendy M.} and Ginsberg, {Jill P.} and Henderson, {Tara O.} and Neglia, {Joseph P.} and Stovall, {Marilyn A.} and Yutaka Yasui and Randall, {R. Lor} and Geller, {David S.} and Robison, {Leslie L.} and Ness, {Kirsten K.}",
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TY - JOUR

T1 - Longitudinal follow-up of adult survivors of Ewing sarcoma

T2 - A report from the Childhood Cancer Survivor Study

AU - Marina, Neyssa M.

AU - Liu, Qi

AU - Donaldson, Sarah S.

AU - Sklar, Charles A.

AU - Armstrong, Gregory

AU - Oeffinger, Kevin C.

AU - Leisenring, Wendy M.

AU - Ginsberg, Jill P.

AU - Henderson, Tara O.

AU - Neglia, Joseph P.

AU - Stovall, Marilyn A.

AU - Yasui, Yutaka

AU - Randall, R. Lor

AU - Geller, David S.

AU - Robison, Leslie L.

AU - Ness, Kirsten K.

PY - 2017/7/1

Y1 - 2017/7/1

N2 - BACKGROUND: Ewing sarcoma survivors (ESSs) are at increased risk for treatment-related complications. The incidence of treatment-related morbidity and late mortality with aging is unknown. METHODS: This study reports survival probabilities, estimated with the Kaplan-Meier method, and the cumulative incidence of cause-specific mortality and chronic conditions among ESSs in the Childhood Cancer Survivor Study who were treated between 1970 and 1986. Piecewise exponential models were used to estimate relative rates (RRs) and 95% confidence intervals (CIs) for these outcomes. Chronic conditions were graded with the Common Terminology Criteria for Adverse Events (version 4.03). RESULTS: Among 404 5-year ESSs (median age at last follow-up, 34.8 years; range, 9.1-54.8 years), the 35-year survival rate was 70% (95% CI, 66%-74%). Late recurrence (cumulative incidence at 35 years, 15.1%) was the most common cause of death, and it was followed by treatment-related causes (11.2%). There were 53 patients with subsequent neoplasms (SNs; cumulative incidence at 35 years, 24.0%), and 38 were malignant (14.3% at 35 years). The standardized incidence ratios were 377.1 (95% CI, 172.1-715.9) for osteosarcoma, 28.9 (95% CI, 3.2-104.2) for acute myeloid leukemia, 14.9 (95% CI, 7.9-25.5) for breast cancer, and 13.1 (95% CI, 4.8-28.5) for thyroid cancer. Rates of chronic conditions were highest for musculoskeletal (RR, 18.1; 95% CI, 12.8-25.7) and cardiac complications (RR, 1.8; 95% CI, 1.4-2.3). Thirty-five years after the diagnosis, the cumulative incidences of any chronic conditions and 2 or more chronic conditions were 84.6% (95% CI, 80.4%-88.8%) and 73.8% (95% CI, 67.8%-79.9%), respectively. CONCLUSIONS: With extended follow-up, ESSs' risk for late mortality and SNs does not plateau. Treatment-related chronic conditions develop years after therapy, and this supports the need for lifelong follow-up. Cancer 2017;123:2551–60.

AB - BACKGROUND: Ewing sarcoma survivors (ESSs) are at increased risk for treatment-related complications. The incidence of treatment-related morbidity and late mortality with aging is unknown. METHODS: This study reports survival probabilities, estimated with the Kaplan-Meier method, and the cumulative incidence of cause-specific mortality and chronic conditions among ESSs in the Childhood Cancer Survivor Study who were treated between 1970 and 1986. Piecewise exponential models were used to estimate relative rates (RRs) and 95% confidence intervals (CIs) for these outcomes. Chronic conditions were graded with the Common Terminology Criteria for Adverse Events (version 4.03). RESULTS: Among 404 5-year ESSs (median age at last follow-up, 34.8 years; range, 9.1-54.8 years), the 35-year survival rate was 70% (95% CI, 66%-74%). Late recurrence (cumulative incidence at 35 years, 15.1%) was the most common cause of death, and it was followed by treatment-related causes (11.2%). There were 53 patients with subsequent neoplasms (SNs; cumulative incidence at 35 years, 24.0%), and 38 were malignant (14.3% at 35 years). The standardized incidence ratios were 377.1 (95% CI, 172.1-715.9) for osteosarcoma, 28.9 (95% CI, 3.2-104.2) for acute myeloid leukemia, 14.9 (95% CI, 7.9-25.5) for breast cancer, and 13.1 (95% CI, 4.8-28.5) for thyroid cancer. Rates of chronic conditions were highest for musculoskeletal (RR, 18.1; 95% CI, 12.8-25.7) and cardiac complications (RR, 1.8; 95% CI, 1.4-2.3). Thirty-five years after the diagnosis, the cumulative incidences of any chronic conditions and 2 or more chronic conditions were 84.6% (95% CI, 80.4%-88.8%) and 73.8% (95% CI, 67.8%-79.9%), respectively. CONCLUSIONS: With extended follow-up, ESSs' risk for late mortality and SNs does not plateau. Treatment-related chronic conditions develop years after therapy, and this supports the need for lifelong follow-up. Cancer 2017;123:2551–60.

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