Gonadectomy in conditions affecting sex development: A registry-based cohort study

Angela K. Lucas-Herald; Jillian Bryce; Andreas Kyriakou; Marie Lindhardt Ljubicic; Wiebke Arlt; Laura Audi; Antonio Balsamo; Federico Baronio; Silvano Bertelloni; Markus Bettendorf; Antonia Brooke; Hedi L.Claahsen Van Der Grinten; Justin H. Davies; Gloria Hermann; Liat De Vries; Ieuan A. Hughes; Rieko Tadokoro-Cuccaro; Feyza Darendeliler; Sukran Poyrazoglu; Mona Ellaithi; Olcay Evliyaoglu; Simone Fica; Lavinia Nedelea; Aneta Gawlik; Evgenia Globa; Nataliya Zelinska; Tulay Guran; Ayla Güven; Sabine E. Hannema; Olaf Hiort; Paul Martin Holterhus; Violeta Iotova; Vilhelm Mladenov; Vandana Jain; Rajni Sharma; Farida Jennane; Colin Johnston; Gil Guerra; Daniel Konrad; Odile Gaisl; Nils Krone; Ruth Krone; Katherine Lachlan; Dejun Li; Corina Lichiardopol; Lidka Lisa; Renata Markosyan; Inas Mazen; Klaus Mohnike; Marek Niedziela; Anna Nordenstrom; Rodolfo Rey; Mars Skaeil; Lloyd J.W. Tack; Jeremy Tomlinson; Naomi Weintrob; Martine Cools; S. Faisal Ahmed

doi:10.1530/EJE-20-1058

Gonadectomy in conditions affecting sex development: A registry-based cohort study

Angela K. Lucas-Herald, Jillian Bryce, Andreas Kyriakou, Marie Lindhardt Ljubicic, Wiebke Arlt, Laura Audi, Antonio Balsamo, Federico Baronio, Silvano Bertelloni, Markus Bettendorf, Antonia Brooke, Hedi L.Claahsen Van Der Grinten, Justin H. Davies, Gloria Hermann, Liat De Vries, Ieuan A. Hughes, Rieko Tadokoro-Cuccaro, Feyza Darendeliler, Sukran Poyrazoglu, Mona EllaithiOlcay Evliyaoglu, Simone Fica, Lavinia Nedelea, Aneta Gawlik, Evgenia Globa, Nataliya Zelinska, Tulay Guran, Ayla Güven, Sabine E. Hannema, Olaf Hiort, Paul Martin Holterhus, Violeta Iotova, Vilhelm Mladenov, Vandana Jain, Rajni Sharma, Farida Jennane, Colin Johnston, Gil Guerra, Daniel Konrad, Odile Gaisl, Nils Krone, Ruth Krone, Katherine Lachlan, Dejun Li, Corina Lichiardopol, Lidka Lisa, Renata Markosyan, Inas Mazen, Klaus Mohnike, Marek Niedziela, Anna Nordenstrom, Rodolfo Rey, Mars Skaeil, Lloyd J.W. Tack, Jeremy Tomlinson, Naomi Weintrob, Martine Cools, S. Faisal Ahmed^*

^*Corresponding author for this work

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

Abstract

Objectives: To determine trends in clinical practice for individuals with D SD requiring gonadectomy. Design: Retrospective cohort study. Methods: Information regarding age at gonadectomy according to diagnosis ; reported sex; time of presentation to specialist centre; and location of centre from cases reported t o the International DSD Registry and who were over 16 years old in January 2019. Results: Data regarding gonadectomy were available in 668 (88%) individu als from 44 centres. Of these, 248 (37%) (median age (range) 24 (17, 75) years) were male and 420 (63%) (median age (range) 26 (16, 86) years) were female. Gonadectomy was reported from 36 centres in 351/668 cas es (53%). Females were more likely to undergo gonadectomy (n = 311, P < 0.0001). The indication for gonadectomy was reported in 268 (7 6%). The most common indication was mitigation of tumour risk in 172 (64%). Variatio ns in the practice of gonadectomy were observed; of the 351 cases from 36 centres, 17 (5%) at 9 centres had undergo ne gonadectomy before their first presentation to the specialist centre. Median age at gonadectomy of cases from high -income countries and low-/middle-income countries (LMIC) was 13.0 years (0.1, 68) years and 16.5 years (1, 28), r espectively (P < 0.0001) with the likelihood of long-term retention of gonads being higher in LMIC countries. Conclusions: The likelihood of gonadectomy depends on the underlying diagnos is, sex of rearing and the geographical setting. Clinical benchmarks, which can be studied across all f orms of DSD will allow a better understanding of the variation in the practice of gonadectomy.

Original language	English
Pages (from-to)	791-801
Number of pages	11
Journal	European Journal of Endocrinology
Volume	184
Issue number	6
DOIs	https://doi.org/10.1530/EJE-20-1058
State	Published - Jun 2021

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10.1530/EJE-20-1058

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Lucas-Herald, A. K., Bryce, J., Kyriakou, A., Ljubicic, M. L., Arlt, W., Audi, L., Balsamo, A., Baronio, F., Bertelloni, S., Bettendorf, M., Brooke, A., Van Der Grinten, H. L. C., Davies, J. H., Hermann, G., De Vries, L., Hughes, I. A., Tadokoro-Cuccaro, R., Darendeliler, F., Poyrazoglu, S., ... Ahmed, S. F. (2021). Gonadectomy in conditions affecting sex development: A registry-based cohort study. European Journal of Endocrinology, 184(6), 791-801. https://doi.org/10.1530/EJE-20-1058

@article{c8bf06f577694ec7a6c74fa594b1974f,

title = "Gonadectomy in conditions affecting sex development: A registry-based cohort study",

abstract = "Objectives: To determine trends in clinical practice for individuals with D SD requiring gonadectomy. Design: Retrospective cohort study. Methods: Information regarding age at gonadectomy according to diagnosis ; reported sex; time of presentation to specialist centre; and location of centre from cases reported t o the International DSD Registry and who were over 16 years old in January 2019. Results: Data regarding gonadectomy were available in 668 (88%) individu als from 44 centres. Of these, 248 (37%) (median age (range) 24 (17, 75) years) were male and 420 (63%) (median age (range) 26 (16, 86) years) were female. Gonadectomy was reported from 36 centres in 351/668 cas es (53%). Females were more likely to undergo gonadectomy (n = 311, P < 0.0001). The indication for gonadectomy was reported in 268 (7 6%). The most common indication was mitigation of tumour risk in 172 (64%). Variatio ns in the practice of gonadectomy were observed; of the 351 cases from 36 centres, 17 (5%) at 9 centres had undergo ne gonadectomy before their first presentation to the specialist centre. Median age at gonadectomy of cases from high -income countries and low-/middle-income countries (LMIC) was 13.0 years (0.1, 68) years and 16.5 years (1, 28), r espectively (P < 0.0001) with the likelihood of long-term retention of gonads being higher in LMIC countries. Conclusions: The likelihood of gonadectomy depends on the underlying diagnos is, sex of rearing and the geographical setting. Clinical benchmarks, which can be studied across all f orms of DSD will allow a better understanding of the variation in the practice of gonadectomy.",

author = "Lucas-Herald, {Angela K.} and Jillian Bryce and Andreas Kyriakou and Ljubicic, {Marie Lindhardt} and Wiebke Arlt and Laura Audi and Antonio Balsamo and Federico Baronio and Silvano Bertelloni and Markus Bettendorf and Antonia Brooke and {Van Der Grinten}, {Hedi L.Claahsen} and Davies, {Justin H.} and Gloria Hermann and {De Vries}, Liat and Hughes, {Ieuan A.} and Rieko Tadokoro-Cuccaro and Feyza Darendeliler and Sukran Poyrazoglu and Mona Ellaithi and Olcay Evliyaoglu and Simone Fica and Lavinia Nedelea and Aneta Gawlik and Evgenia Globa and Nataliya Zelinska and Tulay Guran and Ayla G{\"u}ven and Hannema, {Sabine E.} and Olaf Hiort and Holterhus, {Paul Martin} and Violeta Iotova and Vilhelm Mladenov and Vandana Jain and Rajni Sharma and Farida Jennane and Colin Johnston and Gil Guerra and Daniel Konrad and Odile Gaisl and Nils Krone and Ruth Krone and Katherine Lachlan and Dejun Li and Corina Lichiardopol and Lidka Lisa and Renata Markosyan and Inas Mazen and Klaus Mohnike and Marek Niedziela and Anna Nordenstrom and Rodolfo Rey and Mars Skaeil and Tack, {Lloyd J.W.} and Jeremy Tomlinson and Naomi Weintrob and Martine Cools and Ahmed, {S. Faisal}",

note = "Publisher Copyright: {\textcopyright} 2021 The authors.",

year = "2021",

month = jun,

doi = "10.1530/EJE-20-1058",

language = "אנגלית",

volume = "184",

pages = "791--801",

journal = "European Journal of Endocrinology",

issn = "0804-4643",

publisher = "BioScientifica Ltd.",

number = "6",

}

Lucas-Herald, AK, Bryce, J, Kyriakou, A, Ljubicic, ML, Arlt, W, Audi, L, Balsamo, A, Baronio, F, Bertelloni, S, Bettendorf, M, Brooke, A, Van Der Grinten, HLC, Davies, JH, Hermann, G, De Vries, L, Hughes, IA, Tadokoro-Cuccaro, R, Darendeliler, F, Poyrazoglu, S, Ellaithi, M, Evliyaoglu, O, Fica, S, Nedelea, L, Gawlik, A, Globa, E, Zelinska, N, Guran, T, Güven, A, Hannema, SE, Hiort, O, Holterhus, PM, Iotova, V, Mladenov, V, Jain, V, Sharma, R, Jennane, F, Johnston, C, Guerra, G, Konrad, D, Gaisl, O, Krone, N, Krone, R, Lachlan, K, Li, D, Lichiardopol, C, Lisa, L, Markosyan, R, Mazen, I, Mohnike, K, Niedziela, M, Nordenstrom, A, Rey, R, Skaeil, M, Tack, LJW, Tomlinson, J, Weintrob, N, Cools, M & Ahmed, SF 2021, 'Gonadectomy in conditions affecting sex development: A registry-based cohort study', European Journal of Endocrinology, vol. 184, no. 6, pp. 791-801. https://doi.org/10.1530/EJE-20-1058

TY - JOUR

T1 - Gonadectomy in conditions affecting sex development

T2 - A registry-based cohort study

AU - Lucas-Herald, Angela K.

AU - Bryce, Jillian

AU - Kyriakou, Andreas

AU - Ljubicic, Marie Lindhardt

AU - Arlt, Wiebke

AU - Audi, Laura

AU - Balsamo, Antonio

AU - Baronio, Federico

AU - Bertelloni, Silvano

AU - Bettendorf, Markus

AU - Brooke, Antonia

AU - Van Der Grinten, Hedi L.Claahsen

AU - Davies, Justin H.

AU - Hermann, Gloria

AU - De Vries, Liat

AU - Hughes, Ieuan A.

AU - Tadokoro-Cuccaro, Rieko

AU - Darendeliler, Feyza

AU - Poyrazoglu, Sukran

AU - Ellaithi, Mona

AU - Evliyaoglu, Olcay

AU - Fica, Simone

AU - Nedelea, Lavinia

AU - Gawlik, Aneta

AU - Globa, Evgenia

AU - Zelinska, Nataliya

AU - Guran, Tulay

AU - Güven, Ayla

AU - Hannema, Sabine E.

AU - Hiort, Olaf

AU - Holterhus, Paul Martin

AU - Iotova, Violeta

AU - Mladenov, Vilhelm

AU - Jain, Vandana

AU - Sharma, Rajni

AU - Jennane, Farida

AU - Johnston, Colin

AU - Guerra, Gil

AU - Konrad, Daniel

AU - Gaisl, Odile

AU - Krone, Nils

AU - Krone, Ruth

AU - Lachlan, Katherine

AU - Li, Dejun

AU - Lichiardopol, Corina

AU - Lisa, Lidka

AU - Markosyan, Renata

AU - Mazen, Inas

AU - Mohnike, Klaus

AU - Niedziela, Marek

AU - Nordenstrom, Anna

AU - Rey, Rodolfo

AU - Skaeil, Mars

AU - Tack, Lloyd J.W.

AU - Tomlinson, Jeremy

AU - Weintrob, Naomi

AU - Cools, Martine

AU - Ahmed, S. Faisal

PY - 2021/6

Y1 - 2021/6

N2 - Objectives: To determine trends in clinical practice for individuals with D SD requiring gonadectomy. Design: Retrospective cohort study. Methods: Information regarding age at gonadectomy according to diagnosis ; reported sex; time of presentation to specialist centre; and location of centre from cases reported t o the International DSD Registry and who were over 16 years old in January 2019. Results: Data regarding gonadectomy were available in 668 (88%) individu als from 44 centres. Of these, 248 (37%) (median age (range) 24 (17, 75) years) were male and 420 (63%) (median age (range) 26 (16, 86) years) were female. Gonadectomy was reported from 36 centres in 351/668 cas es (53%). Females were more likely to undergo gonadectomy (n = 311, P < 0.0001). The indication for gonadectomy was reported in 268 (7 6%). The most common indication was mitigation of tumour risk in 172 (64%). Variatio ns in the practice of gonadectomy were observed; of the 351 cases from 36 centres, 17 (5%) at 9 centres had undergo ne gonadectomy before their first presentation to the specialist centre. Median age at gonadectomy of cases from high -income countries and low-/middle-income countries (LMIC) was 13.0 years (0.1, 68) years and 16.5 years (1, 28), r espectively (P < 0.0001) with the likelihood of long-term retention of gonads being higher in LMIC countries. Conclusions: The likelihood of gonadectomy depends on the underlying diagnos is, sex of rearing and the geographical setting. Clinical benchmarks, which can be studied across all f orms of DSD will allow a better understanding of the variation in the practice of gonadectomy.

AB - Objectives: To determine trends in clinical practice for individuals with D SD requiring gonadectomy. Design: Retrospective cohort study. Methods: Information regarding age at gonadectomy according to diagnosis ; reported sex; time of presentation to specialist centre; and location of centre from cases reported t o the International DSD Registry and who were over 16 years old in January 2019. Results: Data regarding gonadectomy were available in 668 (88%) individu als from 44 centres. Of these, 248 (37%) (median age (range) 24 (17, 75) years) were male and 420 (63%) (median age (range) 26 (16, 86) years) were female. Gonadectomy was reported from 36 centres in 351/668 cas es (53%). Females were more likely to undergo gonadectomy (n = 311, P < 0.0001). The indication for gonadectomy was reported in 268 (7 6%). The most common indication was mitigation of tumour risk in 172 (64%). Variatio ns in the practice of gonadectomy were observed; of the 351 cases from 36 centres, 17 (5%) at 9 centres had undergo ne gonadectomy before their first presentation to the specialist centre. Median age at gonadectomy of cases from high -income countries and low-/middle-income countries (LMIC) was 13.0 years (0.1, 68) years and 16.5 years (1, 28), r espectively (P < 0.0001) with the likelihood of long-term retention of gonads being higher in LMIC countries. Conclusions: The likelihood of gonadectomy depends on the underlying diagnos is, sex of rearing and the geographical setting. Clinical benchmarks, which can be studied across all f orms of DSD will allow a better understanding of the variation in the practice of gonadectomy.

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U2 - 10.1530/EJE-20-1058

DO - 10.1530/EJE-20-1058

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C2 - 33780351

AN - SCOPUS:85105443791

SN - 0804-4643

VL - 184

SP - 791

EP - 801

JO - European Journal of Endocrinology

JF - European Journal of Endocrinology

IS - 6

ER -

Gonadectomy in conditions affecting sex development: A registry-based cohort study

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