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Τρίτη 14 Μαΐου 2019

Atypical presentation of Leydig cell tumour in three prepubertal patients: diagnosis, treatment and outcomes

Miriam García GonzálezORCID iD: https://orcid.org/0000-0002-1563-1863 / Isabel Casal-Beloy / Iván Somoza Argibay / Teresa Dargallo Carbonell
Published Online: 2019-03-15 | DOI: https://doi.org/10.1515/jpem-2018-0467

Abstract

Background

Testicular tumours are uncommon in children, accounting for only 1% of all childhood tumours. Prepubertal Leydig cell tumours actively secrete testosterone and as a result, patients typically present with isosexual precocious pseudopuberty, this being the first cause of consultation. We present three cases of Leydig cell tumours in prepubertal patients with an atypical presentation.

Methods

We studied three cases of Leydig cell tumours in prepubertal boys, who either consulted for testicular asymmetry or were incidentally found to have the tumour in the absence of systemic signs of systemic hyperandrogenism or precocious puberty. In all cases, a well-circumscribed testicular mass was found by testicular ultrasound. The diagnosis was confirmed by histology. In all three cases, testicular enucleation was performed with satisfactory follow-up.

Results

Following the surgical procedure, during the follow-up, all patients showed a normal testicular volume in comparison with the contralateral testis. No complications were seen during follow-up.

Conclusions

A testicular ultrasound in children developing asymptomatic testicular asymmetry might be recommended due to its possible hormonal action locally. An early testicular ultrasound, testicular swelling discrepancies, tumour size and androgen production are key factors in the prognosis and management of this type of tumour.
Keywords: enucleationLeydig cellpaediatricprecocious pubertytesticular tumour

References

  • 1.
    Petkovic V, Salemi S, Vassella E, Karamitopoulou-Diamantis E, Meinhardt UJ, et al. Leydig-cell tumour in children: variable clinical presentation, diagnostic features, follow-up and genetic analysis of four cases. Horm Res 2007;67:89–95.Web of SciencePubMedGoogle Scholar
  • 2.
    Guana R, Mussa A, Lala R, Tessaris D, Tessiatore PM, et al. Surgical enucleation of testicular leydigioma in a young child: case report and literature review. J Pediatr Endocrinol Metab 2011;24:839–42.Web of ScienceGoogle Scholar
  • 3.
    Ross JH, Kay R. Prepubertal testis tumors. Rev Urol 2004;6:11–8.PubMedGoogle Scholar
  • 4.
    Kayemba-KaY’S S, Fromont-Hankard G, Lettelier G, Gabriel S, Levard G. Leydig cell tumour revealed by bilateral gynecomastia in a 15 year old adolescent: a patient report. J Pediatr Endocrinol Metab 2010;23:1195–9.Web of ScienceGoogle Scholar
  • 5.
    Farkas LM, Székely JG, Pusztai C, Baki M. High frequency of metastatic leydig cell testicular tumours. Oncology 2000;59:118–21.CrossrefPubMedGoogle Scholar
  • 6.
    Henderson CG, Ahmed AA, Sesterhenn I, Belman AB, Rushton HG. Enucleation for prepubertal Leydig cell tumor. J Urol 2006;176:703–5.CrossrefPubMedGoogle Scholar
  • 7.
    Santos-Silva R, Bonito-Vítor A, Campos M, Fontoura M. Gonadotropin-dependent precocious puberty in an 8-year-old boy with leydig cell testicular tumor. Horm Res Paediatr 2014;82:133–7.CrossrefWeb of ScienceGoogle Scholar
  • 8.
    Leonhartsberger N, Ramoner R, Aigner F, Stoehr B, Pichler R, et al. Increased incidence of Leydig cell tumours of the testis in the era of improved imaging techniques. Br J Urol Int 2011;108:1603–7.CrossrefGoogle Scholar
  • 9.
    García M, Dargallo T, Palacios M, Carames J, Gómez J, et al. Tumor de células de Leydig: enucleación como tratamiento en un caso de presentación atípica. Arch Españoles Urol 2012;65:897–9.Google Scholar
  • 10.
    Mameli C, Selvaggio G, Cerini C, Bulfamante G, Madia C, et al. Atypical Leydig cell tumor in children: report of 2 cases. Pediatrics 2016;138:e20160151.CrossrefPubMedWeb of ScienceGoogle Scholar
  • 11.
    Suardi N, Strada E, Colombo R, Freschi M, Salonia A, et al. Leydig cell tumour of the testis: presentation, therapy, long-term follow-up and the role of organ-sparing surgery in a single-institution experience. Br J Urol Int 2009;103:197–200.CrossrefGoogle Scholar
  • 12.
    Wegner HE, Dieckmann KP, Herbst H, Andresen R, Miller K. Leydig cell tumor-comparison of results of radical and testis-sparing surgery in a single center. Urol Int 1997;59:170–3.CrossrefGoogle Scholar
  • 13.
    Bozzini G, Ratti D, Carmignani L. Tratamiento de tumores de células Leydig del testículo: ¿puede la cirugía conservadora de testículo reemplazar la orquiectomía radical? Resultados de una revisión sistemática. Actas Urol Esp 2017;41:146–54.CrossrefGoogle Scholar
  • 14.
    Konrad D, Schoenle EJ. Ten-year follow-up in a boy with Leydig cell tumor after selective surgery. Horm Res 1999;51:96–100.Google Scholar
  • 15.
    Guana R, Mussa A, Lala R, Tessaris D, Tessiatore PM, et al. Surgical enucleation of testicular leydigioma in a young child: case report and literature review. J Pediatr Endocrinol Metab 2011;24:839–42.Web of ScienceGoogle Scholar
  • 16.
    Emre S, Ozcan R, Elicevik M, Emir H, Soylet Y, et al. Testis sparing surgery for Leydig cell pathologies in children. J Pediatr Urol 2017;13:51.e1–e4.Google Scholar

About the article

Corresponding author: Miriam García González, MD, PhD, Department of Paediatric Surgery, Section of Paediatric Urology, Hospital Materno-Infantil Teresa Herrera, Complexo Hospitalario Universitario de A Coruña (CHUAC), As Xubias s/n, 15006 A Coruña, Spain, Phone: 0034-651367338, E-mail: Miriam.ciruped@gmail.com

Received: 2018-10-29
Accepted: 2019-01-30
Published Online: 2019-03-15
Published in Print: 2019-04-24

Author contributions: All the authors have accepted responsibility for the entire content of this submitted manuscript and approved submission.
Research funding: None declared.
Employment or leadership: None declared.
Honorarium: None declared.
Competing interests: The funding organization(s) played no role in the study design; in the collection, analysis, and interpretation of data; in the writing of the report; or in the decision to submit the report for publication.

Citation Information: Journal of Pediatric Endocrinology and Metabolism, Volume 32, Issue 4, Pages 369–374, ISSN (Online) 2191-0251, ISSN (Print) 0334-018X, DOI: https://doi.org/10.1515/jpem-2018-0467.
©2019 Walter de Gruyter GmbH, Berlin/Boston.Get Permission

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