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      Gonadotropin-Dependent Precocious Puberty in an 8-Year-Old Boy with Leydig Cell Testicular Tumor

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          Abstract

          Leydig cell testicular tumors are very rare in children. They can present as gonadotropin-independent precocious puberty due to excess androgen secretion. We report the case of an 8-year-old boy with isosexual precocity whose hormonal investigation showed luteinizing hormone-independent testosterone hypersecretion. Although no palpable mass was present, scrotal ultrasound revealed a testicular tumor. Testis-sparing tumor resection was performed and the histopathology analysis showed a Leydig cell tumor. After surgery the testosterone levels remained high and further examination showed gonadotropin-dependent precocious puberty, which is believed to be likely caused by the activation of the hypothalamic-pituitary axis due to a long-term exposition to sex steroids. He is currently being treated with a long-acting gonadotropin-releasing hormone analog and the process of sexual precocity has until now been suppressed.

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          Most cited references20

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          Leydig-cell tumors caused by an activating mutation of the gene encoding the luteinizing hormone receptor.

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            Leydig cell tumors of the testis

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              Testicular tumors in children and adolescents.

              To analyze the spectrum of testicular tumors in children in an unselected population-based series, as well as the results of testis-preserving surgery. Our hospital database was analyzed for operations for testicular tumors from 1981 to 2006. The clinical data and findings during follow up (4.7 years) were recorded. Thirty-four patients were operated on because of testicular tumors. In 23 (68%) the tumor was benign: benign teratoma (16), Leydig-cell tumor (2), epidermoid cyst (2), Sertoli-cell tumor (1), cystic dysplasia (1), intratesticular focal fibrosis (1). Eleven patients (32%) had a malignant tumor: yolk-sac tumor (6), embryonal carcinoma (5). Twenty out of the 26 (77%) prepubertal boys had a benign tumor in contrast to only three of the eight (38%) adolescent males (P=0.079). Testis-preserving surgery was performed in 10 patients. In eight, the tumor was curatively excised and remaining testis preserved. Two patients with benign teratoma had a recurrence due to incomplete primary resection. In one patient who underwent orchiectomy for benign teratoma, two metachronous teratomas were detected in the contralateral testis 6 years after primary surgery. In children, most testicular tumors are benign, especially before puberty. If testis-preserving surgery is contemplated, complete excision of the tumor should be ascertained. The possibility of metachronous bilateral tumors should be considered in the follow up of testicular teratomas.
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                Author and article information

                Journal
                HRP
                Horm Res Paediatr
                10.1159/issn.1663-2818
                Hormone Research in Paediatrics
                S. Karger AG
                1663-2818
                1663-2826
                2014
                August 2014
                24 May 2014
                : 82
                : 2
                : 133-137
                Affiliations
                Departments of aPediatrics, and bPediatric Surgery, Hospital S. João, Porto, and cDivision of Pediatric Endocrinology, Department of Pediatrics, Hospital S. João, Oporto Medical School, Porto, Portugal
                Author notes
                *Rita Santos-Silva, Rua Diogo Afonso 71-41, PT-4150-254 Porto (Portugal), E-Mail ritasantossilva@gmail.com
                Article
                358084 Horm Res Paediatr 2014;82:133-137
                10.1159/000358084
                24862970
                27e85ffc-e1d6-4e82-9807-3111eff335cc
                © 2014 S. Karger AG, Basel

                Copyright: All rights reserved. No part of this publication may be translated into other languages, reproduced or utilized in any form or by any means, electronic or mechanical, including photocopying, recording, microcopying, or by any information storage and retrieval system, without permission in writing from the publisher. Drug Dosage: The authors and the publisher have exerted every effort to ensure that drug selection and dosage set forth in this text are in accord with current recommendations and practice at the time of publication. However, in view of ongoing research, changes in government regulations, and the constant flow of information relating to drug therapy and drug reactions, the reader is urged to check the package insert for each drug for any changes in indications and dosage and for added warnings and precautions. This is particularly important when the recommended agent is a new and/or infrequently employed drug. Disclaimer: The statements, opinions and data contained in this publication are solely those of the individual authors and contributors and not of the publishers and the editor(s). The appearance of advertisements or/and product references in the publication is not a warranty, endorsement, or approval of the products or services advertised or of their effectiveness, quality or safety. The publisher and the editor(s) disclaim responsibility for any injury to persons or property resulting from any ideas, methods, instructions or products referred to in the content or advertisements.

                History
                : 30 July 2013
                : 17 December 2013
                Page count
                Figures: 4, Tables: 1, Pages: 5
                Categories
                Novel Insights from Clinical Practice

                Endocrinology & Diabetes,Neurology,Nutrition & Dietetics,Sexual medicine,Internal medicine,Pharmacology & Pharmaceutical medicine
                Testicular tumor,Triptorelin ,Precocious puberty,Leydig cell tumor

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