Endocrine disorders among long-term survivors of childhood head and neck rhabdomyosarcoma

S. C. Clement, R. A. Schoot, O. Slater, J. C. Chisholm, C. Abela, A. J. M. Balm, M. W. van den Brekel, W. B. Breunis, Y. C. Chang, R. Davila Fajardo, D. Dunaway, E. Gajdosova, M. N. Gaze, S. Gupta, B. Hartley, L. C. M. Kremer, M. van Lennep, G. A. Levitt, H. C. Mandeville, B. R. Pieters & 7 others P. Saeed, L. E. Smeele, S. D. Strackee, C. M. Ronckers, H. N. Caron, H. M. van Santen, J. H. M. Merks

Research output: Contribution to journalArticleAcademicpeer-review

Abstract

Purpose Head and neck rhabdomyosarcoma (HNRMS) survivors are at increased risk of developing pituitary dysfunction as an adverse event of radiotherapy. Our aim was to investigate the frequency and risk factors for pituitary dysfunction in these survivors. Secondly, we aimed to compare the prevalence of pituitary dysfunction between survivors treated with external beam radiation therapy (EBRT) and survivors treated with the ablative surgery, moulage technique after loading brachytherapy, and surgical reconstruction (AMORE) procedure. Methods Eighty HNRMS survivors treated in London (EBRT based) and Amsterdam (AMORE based: AMORE if feasible, otherwise EBRT) in the period 1990-2010 and alive ≥2 years post-treatment were evaluated. Survivors were evaluated in multidisciplinary late-effects clinics, with measurement of linear growth, determination of thyroid function, and growth hormone parameters. Additional data, such as baseline characteristics, anthropometrics, pubertal stage, and the results of additional laboratory investigations, were retrieved from patient charts. Results Pituitary dysfunction was diagnosed in 24 in 80 (30%) survivors, after a median follow-up time of 11 years. Median time to develop pituitary dysfunction after HNRMS diagnosis was 3.0 years. Risk factors were EBRT-based therapy (odds ratio [OR] 2.06; 95% confidence interval [CI] 1.79-2.46), parameningeal tumour site (OR 1.83; 95% CI 1.60-2.17) and embryonal RMS histology (OR 1.49; 95% CI 1.19-1.90). Conclusions Radiotherapy used for the treatment of HNRMS confers a significant risk of the development of pituitary dysfunction. AMORE-based treatment in children with HNRMS resulted in less pituitary dysfunction than treatment with conventional EBRT. Our findings underscore the importance of routine early endocrine follow-up in this specific population.
Original languageEnglish
Pages (from-to)1-10
JournalEuropean Journal of Cancer
Volume54
DOIs
Publication statusPublished - 2016
Externally publishedYes

Cite this

Clement, S. C., Schoot, R. A., Slater, O., Chisholm, J. C., Abela, C., Balm, A. J. M., ... Merks, J. H. M. (2016). Endocrine disorders among long-term survivors of childhood head and neck rhabdomyosarcoma. European Journal of Cancer, 54, 1-10. https://doi.org/10.1016/j.ejca.2015.10.064
Clement, S. C. ; Schoot, R. A. ; Slater, O. ; Chisholm, J. C. ; Abela, C. ; Balm, A. J. M. ; van den Brekel, M. W. ; Breunis, W. B. ; Chang, Y. C. ; Davila Fajardo, R. ; Dunaway, D. ; Gajdosova, E. ; Gaze, M. N. ; Gupta, S. ; Hartley, B. ; Kremer, L. C. M. ; van Lennep, M. ; Levitt, G. A. ; Mandeville, H. C. ; Pieters, B. R. ; Saeed, P. ; Smeele, L. E. ; Strackee, S. D. ; Ronckers, C. M. ; Caron, H. N. ; van Santen, H. M. ; Merks, J. H. M. / Endocrine disorders among long-term survivors of childhood head and neck rhabdomyosarcoma. In: European Journal of Cancer. 2016 ; Vol. 54. pp. 1-10.
@article{fb8f3ed5c7bd49ec864f0b73a6cb3b37,
title = "Endocrine disorders among long-term survivors of childhood head and neck rhabdomyosarcoma",
abstract = "Purpose Head and neck rhabdomyosarcoma (HNRMS) survivors are at increased risk of developing pituitary dysfunction as an adverse event of radiotherapy. Our aim was to investigate the frequency and risk factors for pituitary dysfunction in these survivors. Secondly, we aimed to compare the prevalence of pituitary dysfunction between survivors treated with external beam radiation therapy (EBRT) and survivors treated with the ablative surgery, moulage technique after loading brachytherapy, and surgical reconstruction (AMORE) procedure. Methods Eighty HNRMS survivors treated in London (EBRT based) and Amsterdam (AMORE based: AMORE if feasible, otherwise EBRT) in the period 1990-2010 and alive ≥2 years post-treatment were evaluated. Survivors were evaluated in multidisciplinary late-effects clinics, with measurement of linear growth, determination of thyroid function, and growth hormone parameters. Additional data, such as baseline characteristics, anthropometrics, pubertal stage, and the results of additional laboratory investigations, were retrieved from patient charts. Results Pituitary dysfunction was diagnosed in 24 in 80 (30{\%}) survivors, after a median follow-up time of 11 years. Median time to develop pituitary dysfunction after HNRMS diagnosis was 3.0 years. Risk factors were EBRT-based therapy (odds ratio [OR] 2.06; 95{\%} confidence interval [CI] 1.79-2.46), parameningeal tumour site (OR 1.83; 95{\%} CI 1.60-2.17) and embryonal RMS histology (OR 1.49; 95{\%} CI 1.19-1.90). Conclusions Radiotherapy used for the treatment of HNRMS confers a significant risk of the development of pituitary dysfunction. AMORE-based treatment in children with HNRMS resulted in less pituitary dysfunction than treatment with conventional EBRT. Our findings underscore the importance of routine early endocrine follow-up in this specific population.",
author = "Clement, {S. C.} and Schoot, {R. A.} and O. Slater and Chisholm, {J. C.} and C. Abela and Balm, {A. J. M.} and {van den Brekel}, {M. W.} and Breunis, {W. B.} and Chang, {Y. C.} and {Davila Fajardo}, R. and D. Dunaway and E. Gajdosova and Gaze, {M. N.} and S. Gupta and B. Hartley and Kremer, {L. C. M.} and {van Lennep}, M. and Levitt, {G. A.} and Mandeville, {H. C.} and Pieters, {B. R.} and P. Saeed and Smeele, {L. E.} and Strackee, {S. D.} and Ronckers, {C. M.} and Caron, {H. N.} and {van Santen}, {H. M.} and Merks, {J. H. M.}",
year = "2016",
doi = "10.1016/j.ejca.2015.10.064",
language = "English",
volume = "54",
pages = "1--10",
journal = "European Journal of Cancer",
issn = "0959-8049",
publisher = "Pergamon",

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Clement, SC, Schoot, RA, Slater, O, Chisholm, JC, Abela, C, Balm, AJM, van den Brekel, MW, Breunis, WB, Chang, YC, Davila Fajardo, R, Dunaway, D, Gajdosova, E, Gaze, MN, Gupta, S, Hartley, B, Kremer, LCM, van Lennep, M, Levitt, GA, Mandeville, HC, Pieters, BR, Saeed, P, Smeele, LE, Strackee, SD, Ronckers, CM, Caron, HN, van Santen, HM & Merks, JHM 2016, 'Endocrine disorders among long-term survivors of childhood head and neck rhabdomyosarcoma' European Journal of Cancer, vol. 54, pp. 1-10. https://doi.org/10.1016/j.ejca.2015.10.064

Endocrine disorders among long-term survivors of childhood head and neck rhabdomyosarcoma. / Clement, S. C.; Schoot, R. A.; Slater, O.; Chisholm, J. C.; Abela, C.; Balm, A. J. M.; van den Brekel, M. W.; Breunis, W. B.; Chang, Y. C.; Davila Fajardo, R.; Dunaway, D.; Gajdosova, E.; Gaze, M. N.; Gupta, S.; Hartley, B.; Kremer, L. C. M.; van Lennep, M.; Levitt, G. A.; Mandeville, H. C.; Pieters, B. R.; Saeed, P.; Smeele, L. E.; Strackee, S. D.; Ronckers, C. M.; Caron, H. N.; van Santen, H. M.; Merks, J. H. M.

In: European Journal of Cancer, Vol. 54, 2016, p. 1-10.

Research output: Contribution to journalArticleAcademicpeer-review

TY - JOUR

T1 - Endocrine disorders among long-term survivors of childhood head and neck rhabdomyosarcoma

AU - Clement, S. C.

AU - Schoot, R. A.

AU - Slater, O.

AU - Chisholm, J. C.

AU - Abela, C.

AU - Balm, A. J. M.

AU - van den Brekel, M. W.

AU - Breunis, W. B.

AU - Chang, Y. C.

AU - Davila Fajardo, R.

AU - Dunaway, D.

AU - Gajdosova, E.

AU - Gaze, M. N.

AU - Gupta, S.

AU - Hartley, B.

AU - Kremer, L. C. M.

AU - van Lennep, M.

AU - Levitt, G. A.

AU - Mandeville, H. C.

AU - Pieters, B. R.

AU - Saeed, P.

AU - Smeele, L. E.

AU - Strackee, S. D.

AU - Ronckers, C. M.

AU - Caron, H. N.

AU - van Santen, H. M.

AU - Merks, J. H. M.

PY - 2016

Y1 - 2016

N2 - Purpose Head and neck rhabdomyosarcoma (HNRMS) survivors are at increased risk of developing pituitary dysfunction as an adverse event of radiotherapy. Our aim was to investigate the frequency and risk factors for pituitary dysfunction in these survivors. Secondly, we aimed to compare the prevalence of pituitary dysfunction between survivors treated with external beam radiation therapy (EBRT) and survivors treated with the ablative surgery, moulage technique after loading brachytherapy, and surgical reconstruction (AMORE) procedure. Methods Eighty HNRMS survivors treated in London (EBRT based) and Amsterdam (AMORE based: AMORE if feasible, otherwise EBRT) in the period 1990-2010 and alive ≥2 years post-treatment were evaluated. Survivors were evaluated in multidisciplinary late-effects clinics, with measurement of linear growth, determination of thyroid function, and growth hormone parameters. Additional data, such as baseline characteristics, anthropometrics, pubertal stage, and the results of additional laboratory investigations, were retrieved from patient charts. Results Pituitary dysfunction was diagnosed in 24 in 80 (30%) survivors, after a median follow-up time of 11 years. Median time to develop pituitary dysfunction after HNRMS diagnosis was 3.0 years. Risk factors were EBRT-based therapy (odds ratio [OR] 2.06; 95% confidence interval [CI] 1.79-2.46), parameningeal tumour site (OR 1.83; 95% CI 1.60-2.17) and embryonal RMS histology (OR 1.49; 95% CI 1.19-1.90). Conclusions Radiotherapy used for the treatment of HNRMS confers a significant risk of the development of pituitary dysfunction. AMORE-based treatment in children with HNRMS resulted in less pituitary dysfunction than treatment with conventional EBRT. Our findings underscore the importance of routine early endocrine follow-up in this specific population.

AB - Purpose Head and neck rhabdomyosarcoma (HNRMS) survivors are at increased risk of developing pituitary dysfunction as an adverse event of radiotherapy. Our aim was to investigate the frequency and risk factors for pituitary dysfunction in these survivors. Secondly, we aimed to compare the prevalence of pituitary dysfunction between survivors treated with external beam radiation therapy (EBRT) and survivors treated with the ablative surgery, moulage technique after loading brachytherapy, and surgical reconstruction (AMORE) procedure. Methods Eighty HNRMS survivors treated in London (EBRT based) and Amsterdam (AMORE based: AMORE if feasible, otherwise EBRT) in the period 1990-2010 and alive ≥2 years post-treatment were evaluated. Survivors were evaluated in multidisciplinary late-effects clinics, with measurement of linear growth, determination of thyroid function, and growth hormone parameters. Additional data, such as baseline characteristics, anthropometrics, pubertal stage, and the results of additional laboratory investigations, were retrieved from patient charts. Results Pituitary dysfunction was diagnosed in 24 in 80 (30%) survivors, after a median follow-up time of 11 years. Median time to develop pituitary dysfunction after HNRMS diagnosis was 3.0 years. Risk factors were EBRT-based therapy (odds ratio [OR] 2.06; 95% confidence interval [CI] 1.79-2.46), parameningeal tumour site (OR 1.83; 95% CI 1.60-2.17) and embryonal RMS histology (OR 1.49; 95% CI 1.19-1.90). Conclusions Radiotherapy used for the treatment of HNRMS confers a significant risk of the development of pituitary dysfunction. AMORE-based treatment in children with HNRMS resulted in less pituitary dysfunction than treatment with conventional EBRT. Our findings underscore the importance of routine early endocrine follow-up in this specific population.

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UR - https://www.ncbi.nlm.nih.gov/pubmed/26707591

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VL - 54

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JO - European Journal of Cancer

JF - European Journal of Cancer

SN - 0959-8049

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