Risk of meningioma in European patients treated with growth hormone in childhood: results from the SAGhE cohort

Anthony J Swerdlow; Rosie Cooke; Dominique Beckers; Gary Butler; Jean-Claude Carel; Stefano Cianfarani; Peter Clayton; Joël Coste; Annalisa Deodati; Emmanuel Ecosse; Anita C S Hokken-Koelega; Aysha J Khan; Wieland Kiess; Claudia E Kuehni; Christa E Flück; Roland Pfaffle; Lars Sävendahl; Grit Sommer; Muriel Thomas; Anders Tidblad; Sally Tollerfield; Gladys R J Zandwijken

doi:10.1210/jc.2018-01133

Risk of meningioma in European patients treated with growth hormone in childhood: results from the SAGhE cohort

Anthony J Swerdlow, Rosie Cooke, Dominique Beckers, Gary Butler, Jean-Claude Carel, Stefano Cianfarani, Peter Clayton, Joël Coste, Annalisa Deodati, Emmanuel Ecosse, Anita C S Hokken-Koelega, Aysha J Khan, Wieland Kiess, Claudia E Kuehni, Christa E Flück, Roland Pfaffle, Lars Sävendahl, Grit Sommer, Muriel Thomas, Anders TidbladSally Tollerfield, Gladys R J Zandwijken

Ospedale Pediatrico Bambino Gesu

Research output: Contribution to journal › Article › peer-review

Abstract

Context: There has been concern that growth hormone (GH) treatment of children might increase meningioma risk. Results of published studies have been inconsistent and limited.

Objective: To examine meningioma risks in relation to GH treatment.

Design: Cohort study with follow-up via cancer registries and other registers.

Setting: Population-based.

Patients: A cohort of 10,403 patients treated in childhood with recombinant GH (r-hGH) in 5 European countries since this treatment was first used in 1984. Expected rates from national cancer registration statistics.

Main Outcome Measures: Risk of meningioma incidence.

Results: During follow-up 38 meningiomas occurred. Meningioma risk was greatly raised in the cohort overall (SIR=75.4; 95% confidence interval (CI) 54.9-103.6), as a consequence of high risk in subjects who had received radiotherapy for underlying malignancy (SIR= 658.4; 95% CI 460.4-941.7). Risk was not significantly raised in patients who did not receive radiotherapy. Risk in radiotherapy-treated patients was not significantly related to mean daily dose of GH, duration of GH treatment or cumulative dose of GH.

Conclusions: Our data add to evidence of very high risk of meningioma in patients treated in childhood with GH after cranial radiotherapy, but suggest that GH may not affect radiotherapy-related risk, and that there is no material raised risk of meningioma in GH-treated patients who did not receive radiotherapy.

Original language	English
Journal	The Journal of clinical endocrinology and metabolism
DOIs	https://doi.org/10.1210/jc.2018-01133
Publication status	E-pub ahead of print - Aug 17 2018

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Swerdlow, A. J., Cooke, R., Beckers, D., Butler, G., Carel, J-C., Cianfarani, S., Clayton, P., Coste, J., Deodati, A., Ecosse, E., Hokken-Koelega, A. C. S., Khan, A. J., Kiess, W., Kuehni, C. E., Flück, C. E., Pfaffle, R., Sävendahl, L., Sommer, G., Thomas, M., ... Zandwijken, G. R. J. (2018). Risk of meningioma in European patients treated with growth hormone in childhood: results from the SAGhE cohort. The Journal of clinical endocrinology and metabolism. https://doi.org/10.1210/jc.2018-01133

Risk of meningioma in European patients treated with growth hormone in childhood : results from the SAGhE cohort. / Swerdlow, Anthony J; Cooke, Rosie; Beckers, Dominique; Butler, Gary; Carel, Jean-Claude; Cianfarani, Stefano; Clayton, Peter; Coste, Joël; Deodati, Annalisa; Ecosse, Emmanuel; Hokken-Koelega, Anita C S; Khan, Aysha J; Kiess, Wieland; Kuehni, Claudia E; Flück, Christa E; Pfaffle, Roland; Sävendahl, Lars; Sommer, Grit; Thomas, Muriel; Tidblad, Anders; Tollerfield, Sally; Zandwijken, Gladys R J.

In: The Journal of clinical endocrinology and metabolism, 17.08.2018.

Research output: Contribution to journal › Article › peer-review

Swerdlow, AJ, Cooke, R, Beckers, D, Butler, G, Carel, J-C, Cianfarani, S, Clayton, P, Coste, J, Deodati, A, Ecosse, E, Hokken-Koelega, ACS, Khan, AJ, Kiess, W, Kuehni, CE, Flück, CE, Pfaffle, R, Sävendahl, L, Sommer, G, Thomas, M, Tidblad, A, Tollerfield, S & Zandwijken, GRJ 2018, 'Risk of meningioma in European patients treated with growth hormone in childhood: results from the SAGhE cohort', The Journal of clinical endocrinology and metabolism. https://doi.org/10.1210/jc.2018-01133

Swerdlow, Anthony J ; Cooke, Rosie ; Beckers, Dominique ; Butler, Gary ; Carel, Jean-Claude ; Cianfarani, Stefano ; Clayton, Peter ; Coste, Joël ; Deodati, Annalisa ; Ecosse, Emmanuel ; Hokken-Koelega, Anita C S ; Khan, Aysha J ; Kiess, Wieland ; Kuehni, Claudia E ; Flück, Christa E ; Pfaffle, Roland ; Sävendahl, Lars ; Sommer, Grit ; Thomas, Muriel ; Tidblad, Anders ; Tollerfield, Sally ; Zandwijken, Gladys R J. / Risk of meningioma in European patients treated with growth hormone in childhood : results from the SAGhE cohort. In: The Journal of clinical endocrinology and metabolism. 2018.

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title = "Risk of meningioma in European patients treated with growth hormone in childhood: results from the SAGhE cohort",

abstract = "Context: There has been concern that growth hormone (GH) treatment of children might increase meningioma risk. Results of published studies have been inconsistent and limited.Objective: To examine meningioma risks in relation to GH treatment.Design: Cohort study with follow-up via cancer registries and other registers.Setting: Population-based.Patients: A cohort of 10,403 patients treated in childhood with recombinant GH (r-hGH) in 5 European countries since this treatment was first used in 1984. Expected rates from national cancer registration statistics.Main Outcome Measures: Risk of meningioma incidence.Results: During follow-up 38 meningiomas occurred. Meningioma risk was greatly raised in the cohort overall (SIR=75.4; 95% confidence interval (CI) 54.9-103.6), as a consequence of high risk in subjects who had received radiotherapy for underlying malignancy (SIR= 658.4; 95% CI 460.4-941.7). Risk was not significantly raised in patients who did not receive radiotherapy. Risk in radiotherapy-treated patients was not significantly related to mean daily dose of GH, duration of GH treatment or cumulative dose of GH.Conclusions: Our data add to evidence of very high risk of meningioma in patients treated in childhood with GH after cranial radiotherapy, but suggest that GH may not affect radiotherapy-related risk, and that there is no material raised risk of meningioma in GH-treated patients who did not receive radiotherapy.",

author = "Swerdlow, {Anthony J} and Rosie Cooke and Dominique Beckers and Gary Butler and Jean-Claude Carel and Stefano Cianfarani and Peter Clayton and Jo{\"e}l Coste and Annalisa Deodati and Emmanuel Ecosse and Hokken-Koelega, {Anita C S} and Khan, {Aysha J} and Wieland Kiess and Kuehni, {Claudia E} and Fl{\"u}ck, {Christa E} and Roland Pfaffle and Lars S{\"a}vendahl and Grit Sommer and Muriel Thomas and Anders Tidblad and Sally Tollerfield and Zandwijken, {Gladys R J}",

year = "2018",

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doi = "10.1210/jc.2018-01133",

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T1 - Risk of meningioma in European patients treated with growth hormone in childhood

T2 - results from the SAGhE cohort

AU - Swerdlow, Anthony J

AU - Cooke, Rosie

AU - Beckers, Dominique

AU - Butler, Gary

AU - Carel, Jean-Claude

AU - Cianfarani, Stefano

AU - Clayton, Peter

AU - Coste, Joël

AU - Deodati, Annalisa

AU - Ecosse, Emmanuel

AU - Hokken-Koelega, Anita C S

AU - Khan, Aysha J

AU - Kiess, Wieland

AU - Kuehni, Claudia E

AU - Flück, Christa E

AU - Pfaffle, Roland

AU - Sävendahl, Lars

AU - Sommer, Grit

AU - Thomas, Muriel

AU - Tidblad, Anders

AU - Tollerfield, Sally

AU - Zandwijken, Gladys R J

PY - 2018/8/17

Y1 - 2018/8/17

N2 - Context: There has been concern that growth hormone (GH) treatment of children might increase meningioma risk. Results of published studies have been inconsistent and limited.Objective: To examine meningioma risks in relation to GH treatment.Design: Cohort study with follow-up via cancer registries and other registers.Setting: Population-based.Patients: A cohort of 10,403 patients treated in childhood with recombinant GH (r-hGH) in 5 European countries since this treatment was first used in 1984. Expected rates from national cancer registration statistics.Main Outcome Measures: Risk of meningioma incidence.Results: During follow-up 38 meningiomas occurred. Meningioma risk was greatly raised in the cohort overall (SIR=75.4; 95% confidence interval (CI) 54.9-103.6), as a consequence of high risk in subjects who had received radiotherapy for underlying malignancy (SIR= 658.4; 95% CI 460.4-941.7). Risk was not significantly raised in patients who did not receive radiotherapy. Risk in radiotherapy-treated patients was not significantly related to mean daily dose of GH, duration of GH treatment or cumulative dose of GH.Conclusions: Our data add to evidence of very high risk of meningioma in patients treated in childhood with GH after cranial radiotherapy, but suggest that GH may not affect radiotherapy-related risk, and that there is no material raised risk of meningioma in GH-treated patients who did not receive radiotherapy.

AB - Context: There has been concern that growth hormone (GH) treatment of children might increase meningioma risk. Results of published studies have been inconsistent and limited.Objective: To examine meningioma risks in relation to GH treatment.Design: Cohort study with follow-up via cancer registries and other registers.Setting: Population-based.Patients: A cohort of 10,403 patients treated in childhood with recombinant GH (r-hGH) in 5 European countries since this treatment was first used in 1984. Expected rates from national cancer registration statistics.Main Outcome Measures: Risk of meningioma incidence.Results: During follow-up 38 meningiomas occurred. Meningioma risk was greatly raised in the cohort overall (SIR=75.4; 95% confidence interval (CI) 54.9-103.6), as a consequence of high risk in subjects who had received radiotherapy for underlying malignancy (SIR= 658.4; 95% CI 460.4-941.7). Risk was not significantly raised in patients who did not receive radiotherapy. Risk in radiotherapy-treated patients was not significantly related to mean daily dose of GH, duration of GH treatment or cumulative dose of GH.Conclusions: Our data add to evidence of very high risk of meningioma in patients treated in childhood with GH after cranial radiotherapy, but suggest that GH may not affect radiotherapy-related risk, and that there is no material raised risk of meningioma in GH-treated patients who did not receive radiotherapy.

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DO - 10.1210/jc.2018-01133

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JO - Journal of Clinical Endocrinology and Metabolism

JF - Journal of Clinical Endocrinology and Metabolism

SN - 0021-972X

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