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

被引：7

作者：

Lucas-Herald, Angela K. ^{[1
]}

Bryce, Jillian ^{[1
]}

Kyriakou, Andreas ^{[1
]}

Ljubicic, Marie Lindhardt ^{[2
]}

Arlt, Wiebke ^{[3
,4
]}

Audi, Laura ^{[5
,6
]}

Balsamo, Antonio ^{[7
]}

Baronio, Federico ^{[7
]}

Bertelloni, Silvano ^{[8
]}

Bettendorf, Markus ^{[9
]}

Brooke, Antonia ^{[10
]}

van der Grinten, Hedi L. Claahsen ^{[11
]}

Davies, Justin H. ^{[12
]}

Hermann, Gloria ^{[13
]}

de Vries, Liat ^{[14
,15
]}

Hughes, Ieuan A. ^{[16
]}

Tadokoro-Cuccaro, Rieko ^{[16
]}

Darendeliler, Feyza ^{[17
]}

Poyrazoglu, Sukran ^{[17
]}

Ellaithi, Mona ^{[18
]}

Evliyaoglu, Olcay ^{[19
]}

Fica, Simone ^{[20
]}

Nedelea, Lavinia ^{[20
]}

Gawlik, Aneta ^{[21
]}

Globa, Evgenia ^{[22
]}

Zelinska, Nataliya ^{[22
]}

Guran, Tulay ^{[23
]}

Guven, Ayla ^{[24
]}

Hannema, Sabine E. ^{[25
,26
]}

Hiort, Olaf ^{[27
]}

Holterhus, Paul-Martin ^{[28
]}

Iotova, Violeta ^{[29
]}

Mladenov, Vilhelm ^{[29
]}

Jain, Vandana ^{[30
]}

Sharma, Rajni ^{[30
]}

Jennane, Farida ^{[31
]}

Johnston, Colin ^{[32
]}

Guerra Junior, Gil ^{[33
,34
]}

Konrad, Daniel ^{[35
]}

Gaisl, Odile ^{[35
]}

Krone, Nils ^{[36
]}

Krone, Ruth ^{[37
]}

Lachlan, Katherine ^{[38
]}

Li, Dejun ^{[39
]}

Lichiardopol, Corina ^{[40
]}

Lisa, Lidka ^{[41
]}

Markosyan, Renata ^{[42
]}

Mazen, Inas ^{[43
]}

Mohnike, Klaus ^{[44
]}

Niedziela, Marek ^{[45
]}

机构：

[1] Univ Glasgow, Dev Endocrinol Res Grp, Glasgow, Lanark, Scotland

[2] Univ Copenhagen, Rigshosp, Dept Growth & Reprod, Copenhagen, Denmark

[3] Univ Birmingham, Inst Metab & Syst Res IMSR, Birmingham, W Midlands, England

[4] Univ Hosp Birmingham NHS Fdn Trust, Ctr Endocrinol Diabet & Metab, Birmingham, W Midlands, England

[5] Hosp Valle De Hebron, Vall dHebron Res Inst VHIR, Dept Pediat, Barcelona, Spain

[6] Hosp Valle De Hebron, Vall dHebron Res Inst VHIR, Pediat Endocrinol Unit, Barcelona, Spain

[7] Univ Bologna Hosp Bologna, Dept Pediat, St Orsola Malpighi, Bologna, Italy

[8] Univ Hosp Pisa, Dept Pediat, Pisa, Italy

[9] Univ Hosp Heidelberg, Dept Paediat, Div Paediat Endocrinol & Diabet, Heidelberg, Germany

[10] Royal Devon & Exeter Hosp, Macleod Diabet & Endocrine Ctr, Exeter, Devon, England

[11] Radboud Univ Nijmegen Med Ctr, Dept Pediat, Nijmegen, Netherlands

[12] Univ Southampton, Fac Med, Dept Paediat Endocrinol, Southampton, Hants, England

[13] Univ Med Ctr, Dept Pediat & Adolescent Med, Ulm, Germany

[14] Tel Aviv Univ, Tel Aviv Sackler Fac Med, Paediat, Tel Aviv, Israel

[15] Schneider Childrens Med Ctr Israel, Jesse Z & Sara Lea Shafer Inst Endocrinol & Diabe, Petah Tiqwa, Israel

[16] Univ Cambridge, Dept Paediat, Cambridge, England

[17] Istanbul Univ, Istanbul Med Sch, Dept Pediat Endocrinol, Istanbul, Turkey

[18] Al Neelain Univ, Fac Med Lab Sci, Khartoum, Sudan

[19] Istanbul Univ Cerrahpasa, Cerrahpa Med Sch, Dept Pediat Endocrinol, Istanbul, Turkey

[20] Univ Med & Farmacie Carol Davila, Fac Med, Endocrinol, Bucharest, Romania

[21] Med Univ Silesia, Dept Pediat & Pediat Endocrinol, Katowice, Poland

[22] MOH Ukraine, Ukrainian Res Ctr Endocrine Surg Endocrine Organs, Kiev, Ukraine

[23] Marmara Univ, Dept Pediat Endocrinol, Istanbul, Turkey

[24] Zeynep Kamil Women & Children Hosp, Pediat Endocrinol Clin, Istanbul, Turkey

[25] Univ Med Ctr Rotterdam, Erasmus Med Ctr, Pediat Endocrinol, Rotterdam, Netherlands

[26] Leiden Univ Med Ctr, Dept Pediat, Leiden, Netherlands

[27] Univ Lubeck, Div Paediat, Lubeck, Germany

[28] Christian Albrechts Univ Kiel, Div Pediat Endocrinol & Diabet, Univ Hosp Schleswig Holstein, Dept Pediat, Campus Kiel, Kiel, Germany

[29] Med Univ Varna, UMHAT Sveta Marina, Varna, Bulgaria

[30] All India Inst Med Sci, Dept Pediat, Div Pediat Endocrinol, New Delhi, India

[31] Hop Enfants, Unite Endocrinol Diabetol & Gynecol Enfant & Adol, Casablanca, Morocco

[32] West Hertfordshire Hosp Trust, Dept Endocrinol, St Albans City Hosp, St Albans, England

[33] Univ Estadual Campinas, Fac Ciencias Med, Dept Pediat, Disciplina Endocrinol & Metabol, Sao Paulo, Brazil

[34] Dept Clin Med, Sao Paulo, Brazil

[35] Univ Childrens Hosp, Div Pediat Endocrinol & Diabetol & Childrens Res, Zurich, Switzerland

[36] Univ Sheffield, Acad Unit Child Hlth, Dept Oncol & Metab, Sheffield, S Yorkshire, England

[37] Birmingham Childrens Hosp, Endocrinol, Birmingham, W Midlands, England

[38] Univ Southampton, Fac Med, Dept Human Dev & Hlth, Southampton, Hants, England

[39] Jilin Univ First Hosp, Ctr Prenatal Diag, Jilin, Jilin, Peoples R China

[40] Univ Med & Pharm Craiova, Endocrinol, Craiova, Romania

[41] Inst Prague, Endocrinol, Prague, Czech Republic

[42] Yerevan State Med Univ, Endocrinol, Endocrinol Clin, Yerevan, Armenia

[43] Natl Res Ctr, Dept Clin Genet, Cairo, Egypt

[44] Otto von Guericke Univ, Dept Pediat, Magdeburg, Germany

[45] Poznan Univ Med Sci, Dept Pediat Endocrinol, Poznan, Poland

[46] Karolinska Inst, Dept Womens & Childrens Hlth, Stockholm, Sweden

[47] Ctr Invest Endocrinol Dr Cesar Bergada CEDIE, CONICET FEI Div Endocrinol, Buenos Aires, DF, Argentina

[48] Royal Manchester Childrens Hosp, Dept Paediat Endocrinol, Manchester, Lancs, England

[49] Univ Hosp Ghent, Pediat Endocrinol Serv, Ghent, Belgium

[50] Univ Oxford, Churchill Hosp, Oxford Ctr Diabet Endocrinol & Metab, NIHR Oxford Biomed Res Ctr, Oxford, England

来源：

EUROPEAN JOURNAL OF ENDOCRINOLOGY | 2021年 / 184卷 / 06期

关键词：

GERM-CELL CANCER; DEVELOPMENT DSD; DISORDERS; RISK;

D O I：

10.1530/EJE-20-1058

中图分类号：

R5 [内科学];

学科分类号：

1002 ; 100201 ;

摘要：

Objectives: To determine trends in clinical practice for individuals with DSD 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 to the International DSD Registry and who were over 16 years old in January 2019. Results: Data regarding gonadectomy were available in 668 (88%) individuals 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 cases (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%). Variations 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), respectively (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 diagnosis, sex of rearing and the geographical setting. Clinical benchmarks, which can be studied across all forms of DSD will allow a better understanding of the variation in the practice of gonadectomy.

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页码：791 / 801

页数：11

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