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Incidence of and Risk Factors for Histologically Confirmed Solid Benign Tumors Among Long-term Survivors of Childhood Cancer

DCOG LATER Study Grp, Kok, J. L., Teepen, J. C., van der Pal, H. J., van Leeuwen, F. E., Tissing, W. J. E., Neggers, S. J. C. M. M., Loonen, J. J., Louwerens, M., Versluys, B., van den Heuvel-Eibrink, M., van Dulmen-den Broeder, E., Jaspers, M. M. W., van Santen, H. M., van der Heiden-van der Loo, M., Janssens, G. O., Maduro, J. H., Bruggink, A. H., Jongmans, M. C., Kremer, L. C. M. & Ronckers, C. M., May-2019, In : JAMA oncology. 5, 5, p. 671-680 10 p.

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APA

DCOG LATER Study Grp, Kok, J. L., Teepen, J. C., van der Pal, H. J., van Leeuwen, F. E., Tissing, W. J. E., ... Ronckers, C. M. (2019). Incidence of and Risk Factors for Histologically Confirmed Solid Benign Tumors Among Long-term Survivors of Childhood Cancer. JAMA oncology, 5(5), 671-680. https://doi.org/10.1001/jamaoncol.2018.6862

Author

DCOG LATER Study Grp ; Kok, Judith L. ; Teepen, Jop C. ; van der Pal, Helena J. ; van Leeuwen, Flora E. ; Tissing, Wim J. E. ; Neggers, Sebastian J. C. M. M. ; Loonen, Jacqueline J. ; Louwerens, Marloes ; Versluys, Birgitta ; van den Heuvel-Eibrink, MarryM. ; van Dulmen-den Broeder, Eline ; Jaspers, Monique M. W. ; van Santen, Hanneke M. ; van der Heiden-van der Loo, Margriet ; Janssens, Geert O. ; Maduro, John H. ; Bruggink, Annette H. ; Jongmans, Marjolijn C. ; Kremer, Leontien C. M. ; Ronckers, Cecile M. / Incidence of and Risk Factors for Histologically Confirmed Solid Benign Tumors Among Long-term Survivors of Childhood Cancer. In: JAMA oncology. 2019 ; Vol. 5, No. 5. pp. 671-680.

Harvard

DCOG LATER Study Grp, Kok, JL, Teepen, JC, van der Pal, HJ, van Leeuwen, FE, Tissing, WJE, Neggers, SJCMM, Loonen, JJ, Louwerens, M, Versluys, B, van den Heuvel-Eibrink, M, van Dulmen-den Broeder, E, Jaspers, MMW, van Santen, HM, van der Heiden-van der Loo, M, Janssens, GO, Maduro, JH, Bruggink, AH, Jongmans, MC, Kremer, LCM & Ronckers, CM 2019, 'Incidence of and Risk Factors for Histologically Confirmed Solid Benign Tumors Among Long-term Survivors of Childhood Cancer', JAMA oncology, vol. 5, no. 5, pp. 671-680. https://doi.org/10.1001/jamaoncol.2018.6862

Standard

Incidence of and Risk Factors for Histologically Confirmed Solid Benign Tumors Among Long-term Survivors of Childhood Cancer. / DCOG LATER Study Grp; Kok, Judith L.; Teepen, Jop C.; van der Pal, Helena J.; van Leeuwen, Flora E.; Tissing, Wim J. E.; Neggers, Sebastian J. C. M. M.; Loonen, Jacqueline J.; Louwerens, Marloes; Versluys, Birgitta; van den Heuvel-Eibrink, MarryM.; van Dulmen-den Broeder, Eline; Jaspers, Monique M. W.; van Santen, Hanneke M.; van der Heiden-van der Loo, Margriet; Janssens, Geert O.; Maduro, John H.; Bruggink, Annette H.; Jongmans, Marjolijn C.; Kremer, Leontien C. M.; Ronckers, Cecile M.

In: JAMA oncology, Vol. 5, No. 5, 05.2019, p. 671-680.

Research output: Contribution to journalArticleAcademicpeer-review

Vancouver

DCOG LATER Study Grp, Kok JL, Teepen JC, van der Pal HJ, van Leeuwen FE, Tissing WJE et al. Incidence of and Risk Factors for Histologically Confirmed Solid Benign Tumors Among Long-term Survivors of Childhood Cancer. JAMA oncology. 2019 May;5(5):671-680. https://doi.org/10.1001/jamaoncol.2018.6862


BibTeX

@article{cdaa2ac25f82486dbead6aaf5d36ab32,
title = "Incidence of and Risk Factors for Histologically Confirmed Solid Benign Tumors Among Long-term Survivors of Childhood Cancer",
abstract = "IMPORTANC E Survivors of childhood cancer (CCSs) face risk of developing subsequent tumors. Solid benign tumors may be cancer precursors; benign tumors and cancers may share etiologic factors. However, comprehensive data on the risk for solid benign tumors are lacking. OBJECT; VE To quantify the incidence of and treatment-related risk factors for histologically confirmed solid nonskin benign tumors among CCSs.DESIGN, SETTING, AND PARTICIPANTS This record linkage study involves the Dutch Childhood Oncology Group-Long-Term Effects After Childhood Cancer (DCOG-LATER) cohort of 6165 individuals diagnosed with childhood cancer at younger than 18 years from January], 1963, through December 31, 2001, in 7 Dutch pediatric centers and who survived at least 5 years after the diagnosis. Study groups eligible for record linkage from 1990 onward included 5843 CCSs (94.8{\%}) and 883 siblings. Benign tumors were identified from the population-based Dutch histopathology and cytopathology registry (PALGA). Follow-up was completed on May 1, 2015. Data were analyzed from January 1, 1990, through May 1, 2015.MAIN OUTCOMES AND MEASURES Cumulative incidence of any subsequent benign tumor for cohort strata and multivariable Cox proportional hazards regression models (hazard ratios [HRs]) were used to evaluate potential risk factors for 8 major benign tumor subtypes. RESULTS Of the 5843 eligible CC5s (55.9{\%} male), 542 (9.3{\%}) developed a histologically confirmed subsequent benign tumor after a median follow-up of 22.7 years (range, 5.0-52.2 years). Among women, abdominopelvic radiotherapy inferred dose-dependent increased risks for uterine leiomyoma (n = 43) for doses of less than 20 Gy (HR, 1.9; 95{\%} CI, 0.5-7.0), 20 to less than 30 Gy (HR, 3.4; 95{\%} CI, 1.1-10.4), and at least 30 Gy (HR, 5.4; 95{\%} CI, 2.4-12.4) compared with no abdominopelvic radiotherapy (P =.002 for trend). High-dose radiotherapy to the trunk was not associated with breast fibroadenoma (n = 45). Of 23 osseous and/or chondromatous neoplasms, 16 occurred among leukemia survivors, including llafter total body irradiation (HR, 37.4; 95{\%} CI, 14.8-94.7). Nerve sheath tumors (n = 55) were associated with radiotherapy (HR at 31years of age, 2.9; 95{\%} CI, 1.5-5.5) and a crude indicator of neurofibromatosis type lor 2 status (HR, 5.6; 95{\%} CI, 2.3-13.7). Subsequent risk for benign tumors was higher than the risks for subsequent nonskin solid malignant neoplasms and for benign tumors among siblings.CONCLUSIONS AND RELEVANCE This record linkage study uses a unique resource for valid and complete outcome assessment and shows that CCSs have an approximately 2-fold risk of developing subsequent benign tumors compared with siblings. Site-specific new findings, including for uterine leiomyoma, osteochondroma, and nervous system tumors, are important to enable early diagnosis; this information will be the first step for future surveillance guidelines that include some benign tumors in CCSs and will provide leads for in-depth etiologic studies.",
keywords = "5-YEAR SURVIVORS, DCOG-LATER, NEOPLASMS",
author = "{DCOG LATER Study Grp} and Kok, {Judith L.} and Teepen, {Jop C.} and {van der Pal}, {Helena J.} and {van Leeuwen}, {Flora E.} and Tissing, {Wim J. E.} and Neggers, {Sebastian J. C. M. M.} and Loonen, {Jacqueline J.} and Marloes Louwerens and Birgitta Versluys and {van den Heuvel-Eibrink}, MarryM. and {van Dulmen-den Broeder}, Eline and Jaspers, {Monique M. W.} and {van Santen}, {Hanneke M.} and {van der Heiden-van der Loo}, Margriet and Janssens, {Geert O.} and Maduro, {John H.} and Bruggink, {Annette H.} and Jongmans, {Marjolijn C.} and Kremer, {Leontien C. M.} and Ronckers, {Cecile M.} and Aleman, {B. M. P.} and {van den Berg}, {M. H.} and D. Bresters and Caron, {H. N.} and Clement, {S. C.} and Daniels, {L. A.} and Dolsma, {W. V.} and Grootenhuis, {M. A.} and Haasbeek, {C. J. A.} and Hoeben, {B. A. W.} and {den Hartogh}, {J. G.} and N. Hollema and F. Oldenburger and A. Postma and {van Rij}, {C. M.} and Tersteeg, {R. J. H. A.}",
year = "2019",
month = "5",
doi = "10.1001/jamaoncol.2018.6862",
language = "English",
volume = "5",
pages = "671--680",
journal = "JAMA oncology",
issn = "2374-2437",
publisher = "AMER MEDICAL ASSOC",
number = "5",

}

RIS

TY - JOUR

T1 - Incidence of and Risk Factors for Histologically Confirmed Solid Benign Tumors Among Long-term Survivors of Childhood Cancer

AU - DCOG LATER Study Grp

AU - Kok, Judith L.

AU - Teepen, Jop C.

AU - van der Pal, Helena J.

AU - van Leeuwen, Flora E.

AU - Tissing, Wim J. E.

AU - Neggers, Sebastian J. C. M. M.

AU - Loonen, Jacqueline J.

AU - Louwerens, Marloes

AU - Versluys, Birgitta

AU - van den Heuvel-Eibrink, MarryM.

AU - van Dulmen-den Broeder, Eline

AU - Jaspers, Monique M. W.

AU - van Santen, Hanneke M.

AU - van der Heiden-van der Loo, Margriet

AU - Janssens, Geert O.

AU - Maduro, John H.

AU - Bruggink, Annette H.

AU - Jongmans, Marjolijn C.

AU - Kremer, Leontien C. M.

AU - Ronckers, Cecile M.

AU - Aleman, B. M. P.

AU - van den Berg, M. H.

AU - Bresters, D.

AU - Caron, H. N.

AU - Clement, S. C.

AU - Daniels, L. A.

AU - Dolsma, W. V.

AU - Grootenhuis, M. A.

AU - Haasbeek, C. J. A.

AU - Hoeben, B. A. W.

AU - den Hartogh, J. G.

AU - Hollema, N.

AU - Oldenburger, F.

AU - Postma, A.

AU - van Rij, C. M.

AU - Tersteeg, R. J. H. A.

PY - 2019/5

Y1 - 2019/5

N2 - IMPORTANC E Survivors of childhood cancer (CCSs) face risk of developing subsequent tumors. Solid benign tumors may be cancer precursors; benign tumors and cancers may share etiologic factors. However, comprehensive data on the risk for solid benign tumors are lacking. OBJECT; VE To quantify the incidence of and treatment-related risk factors for histologically confirmed solid nonskin benign tumors among CCSs.DESIGN, SETTING, AND PARTICIPANTS This record linkage study involves the Dutch Childhood Oncology Group-Long-Term Effects After Childhood Cancer (DCOG-LATER) cohort of 6165 individuals diagnosed with childhood cancer at younger than 18 years from January], 1963, through December 31, 2001, in 7 Dutch pediatric centers and who survived at least 5 years after the diagnosis. Study groups eligible for record linkage from 1990 onward included 5843 CCSs (94.8%) and 883 siblings. Benign tumors were identified from the population-based Dutch histopathology and cytopathology registry (PALGA). Follow-up was completed on May 1, 2015. Data were analyzed from January 1, 1990, through May 1, 2015.MAIN OUTCOMES AND MEASURES Cumulative incidence of any subsequent benign tumor for cohort strata and multivariable Cox proportional hazards regression models (hazard ratios [HRs]) were used to evaluate potential risk factors for 8 major benign tumor subtypes. RESULTS Of the 5843 eligible CC5s (55.9% male), 542 (9.3%) developed a histologically confirmed subsequent benign tumor after a median follow-up of 22.7 years (range, 5.0-52.2 years). Among women, abdominopelvic radiotherapy inferred dose-dependent increased risks for uterine leiomyoma (n = 43) for doses of less than 20 Gy (HR, 1.9; 95% CI, 0.5-7.0), 20 to less than 30 Gy (HR, 3.4; 95% CI, 1.1-10.4), and at least 30 Gy (HR, 5.4; 95% CI, 2.4-12.4) compared with no abdominopelvic radiotherapy (P =.002 for trend). High-dose radiotherapy to the trunk was not associated with breast fibroadenoma (n = 45). Of 23 osseous and/or chondromatous neoplasms, 16 occurred among leukemia survivors, including llafter total body irradiation (HR, 37.4; 95% CI, 14.8-94.7). Nerve sheath tumors (n = 55) were associated with radiotherapy (HR at 31years of age, 2.9; 95% CI, 1.5-5.5) and a crude indicator of neurofibromatosis type lor 2 status (HR, 5.6; 95% CI, 2.3-13.7). Subsequent risk for benign tumors was higher than the risks for subsequent nonskin solid malignant neoplasms and for benign tumors among siblings.CONCLUSIONS AND RELEVANCE This record linkage study uses a unique resource for valid and complete outcome assessment and shows that CCSs have an approximately 2-fold risk of developing subsequent benign tumors compared with siblings. Site-specific new findings, including for uterine leiomyoma, osteochondroma, and nervous system tumors, are important to enable early diagnosis; this information will be the first step for future surveillance guidelines that include some benign tumors in CCSs and will provide leads for in-depth etiologic studies.

AB - IMPORTANC E Survivors of childhood cancer (CCSs) face risk of developing subsequent tumors. Solid benign tumors may be cancer precursors; benign tumors and cancers may share etiologic factors. However, comprehensive data on the risk for solid benign tumors are lacking. OBJECT; VE To quantify the incidence of and treatment-related risk factors for histologically confirmed solid nonskin benign tumors among CCSs.DESIGN, SETTING, AND PARTICIPANTS This record linkage study involves the Dutch Childhood Oncology Group-Long-Term Effects After Childhood Cancer (DCOG-LATER) cohort of 6165 individuals diagnosed with childhood cancer at younger than 18 years from January], 1963, through December 31, 2001, in 7 Dutch pediatric centers and who survived at least 5 years after the diagnosis. Study groups eligible for record linkage from 1990 onward included 5843 CCSs (94.8%) and 883 siblings. Benign tumors were identified from the population-based Dutch histopathology and cytopathology registry (PALGA). Follow-up was completed on May 1, 2015. Data were analyzed from January 1, 1990, through May 1, 2015.MAIN OUTCOMES AND MEASURES Cumulative incidence of any subsequent benign tumor for cohort strata and multivariable Cox proportional hazards regression models (hazard ratios [HRs]) were used to evaluate potential risk factors for 8 major benign tumor subtypes. RESULTS Of the 5843 eligible CC5s (55.9% male), 542 (9.3%) developed a histologically confirmed subsequent benign tumor after a median follow-up of 22.7 years (range, 5.0-52.2 years). Among women, abdominopelvic radiotherapy inferred dose-dependent increased risks for uterine leiomyoma (n = 43) for doses of less than 20 Gy (HR, 1.9; 95% CI, 0.5-7.0), 20 to less than 30 Gy (HR, 3.4; 95% CI, 1.1-10.4), and at least 30 Gy (HR, 5.4; 95% CI, 2.4-12.4) compared with no abdominopelvic radiotherapy (P =.002 for trend). High-dose radiotherapy to the trunk was not associated with breast fibroadenoma (n = 45). Of 23 osseous and/or chondromatous neoplasms, 16 occurred among leukemia survivors, including llafter total body irradiation (HR, 37.4; 95% CI, 14.8-94.7). Nerve sheath tumors (n = 55) were associated with radiotherapy (HR at 31years of age, 2.9; 95% CI, 1.5-5.5) and a crude indicator of neurofibromatosis type lor 2 status (HR, 5.6; 95% CI, 2.3-13.7). Subsequent risk for benign tumors was higher than the risks for subsequent nonskin solid malignant neoplasms and for benign tumors among siblings.CONCLUSIONS AND RELEVANCE This record linkage study uses a unique resource for valid and complete outcome assessment and shows that CCSs have an approximately 2-fold risk of developing subsequent benign tumors compared with siblings. Site-specific new findings, including for uterine leiomyoma, osteochondroma, and nervous system tumors, are important to enable early diagnosis; this information will be the first step for future surveillance guidelines that include some benign tumors in CCSs and will provide leads for in-depth etiologic studies.

KW - 5-YEAR SURVIVORS

KW - DCOG-LATER

KW - NEOPLASMS

U2 - 10.1001/jamaoncol.2018.6862

DO - 10.1001/jamaoncol.2018.6862

M3 - Article

VL - 5

SP - 671

EP - 680

JO - JAMA oncology

JF - JAMA oncology

SN - 2374-2437

IS - 5

ER -

ID: 78909564