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

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.

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Sertoli-Leydig Cell Tumour and DICER1 Mutation: A Case Report and Review of the Literature

Case Reports in Obstetrics and Gynecology ◽

10.1155/2018/7927362 ◽

2018 ◽

Vol 2018 ◽

pp. 1-4

Author(s):

B. Wormald ◽

S. Elorbany ◽

H. Hanson ◽

J. W. Williams ◽

S. Heenan ◽

...

Keyword(s):

Case Report ◽

Gene Mutation ◽

Leydig Cell ◽

Autosomal Dominant ◽

Review Of The Literature ◽

Autosomal Dominant Fashion ◽

Rare Tumours ◽

Leydig Cell Tumour ◽

Underlying Pathology ◽

Leydig Cell Tumours

Sertoli-Leydig cell tumours of the ovary (SLCT) are rare tumours predominantly caused by mutations in the DICER1 gene. We present a patient with a unilateral SLCT who had an underlying germline DICER1 gene mutation. We discuss the underlying pathology, risks, and screening opportunities available to those with a mutation in this gene as SLCT is only one of a multitude of other tumours encompassing DICER1 syndrome. The condition is inherited in an autosomal dominant fashion. As such, genetic counselling is a key component of the management of women with SLCT.

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Histamine H4 receptor as a novel therapeutic target for the treatment of Leydig-cell tumours in prepubertal boys

European Journal of Cancer ◽

10.1016/j.ejca.2017.12.003 ◽

2018 ◽

Vol 91 ◽

pp. 125-135 ◽

Cited By ~ 3

Author(s):

Adriana María Belén Abiuso ◽

María Luisa Varela ◽

Luis Haro Durand ◽

Marcos Besio Moreno ◽

Alejandra Marcos ◽

...

Keyword(s):

Leydig Cell ◽

Therapeutic Target ◽

Histamine H4 Receptor ◽

Prepubertal Boys ◽

H4 Receptor ◽

Leydig Cell Tumours ◽

Novel Therapeutic

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A transplantable rat Leydig cell tumour.

Acta Endocrinologica ◽

10.1530/acta.0.1190291 ◽

1988 ◽

Vol 119 (2) ◽

pp. 291-300 ◽

Cited By ~ 2

Author(s):

Aa. Erichsen ◽

O. P. F. Clausen ◽

P. Torjesen ◽

E. Dahl ◽

P. Wetteland ◽

...

Keyword(s):

Leydig Cell ◽

Tumour Growth ◽

Tumour Size ◽

Pituitary Hormones ◽

Cell Tumour ◽

Plasma Testosterone ◽

Testosterone Levels ◽

Leydig Cell Tumour ◽

Hypophysectomized Rats ◽

Tumour Weight

Abstract. Growth rate and morphology were studied in a transplantable rat Leydig cell tumour (H-540) grown in intact, castrated and hypophysectomized rats. The plasma levels of pituitary hormones and testosterone were measured in the same rats. The results can be summarized as follows: 1. The growth curves of tumours in intact and castrated rats were S-shaped and very similar during the observation period of three weeks. 2. In hypophysectomized rats, the onset and progression of tumour growth were delayed, compared with intact and castrated rats. 3. The thymidine labelling index as well as the size of the S and G2 phase compartments were decreased in tumours greater than 10 g compared with smaller tumours and tumours grown in hypophysectomized rats. 4. Testosterone concentrations in plasma correlate well with increasing tumour weight up to approximately 10–15 g in intact and castrated rats. 5. Plasma testosterone levels in tumour-bearing hypophysectomized rats were 7-fold higher than those of corresponding intact and castrated rats. 6. In castrated rats, suppression of LH production occurs by very small tumours (< 2 g), whereas FSH levels show a gradual decrease with increasing tumour size. PRL production was independent of castration, tumour weight, and testosterone levels. 7. In spite of no major differences in cell morphology, the morphometric analysis revealed a reduction in tumour cell size and nuclear size in hypophysectomized rats compared with intact and castrated rats. It is concluded that pituitary hormones stimulate tumour growth, but surprisingly appear to reduce the secretion of testosterone from these tumours.

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Presentation, clinical features and long-term follow-up of Leydig cell tumours of the testis: 77 cases from a single center experience

Endocrine Abstracts ◽

10.1530/endoabs.49.gp149 ◽

2017 ◽

Author(s):

Andrea M. Isidori ◽

Vincenzo Giannetta ◽

Daniele Gianfrilli ◽

Rosa Lauretta ◽

Maria Grazia Tarsitano ◽

...

Keyword(s):

Clinical Features ◽

Leydig Cell ◽

Single Center ◽

Single Center Experience ◽

Long Term Follow Up ◽

Leydig Cell Tumours

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Leydig-Cell Tumour in Children: Variable Clinical Presentation, Diagnostic Features, Follow-Up and Genetic Analysis of Four Cases

Hormone Research in Paediatrics ◽

10.1159/000096356 ◽

2006 ◽

Vol 67 (2) ◽

pp. 89-95 ◽

Cited By ~ 7

Author(s):

Vibor Petkovic ◽

Souzan Salemi ◽

Erik Vassella ◽

Evanthia Karamitopoulou-Diamantis ◽

Udo J. Meinhardt ◽

...

Keyword(s):

Genetic Analysis ◽

Leydig Cell ◽

Clinical Presentation ◽

Cell Tumour ◽

Diagnostic Features ◽

Leydig Cell Tumour ◽

Variable Clinical Presentation

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Use of novel serum markers in clinical follow-up of Sertoli-Leydig cell tumours

Clinical Chemistry and Laboratory Medicine (CCLM) ◽

10.1515/cclm.2007.120 ◽

2007 ◽

Vol 45 (5) ◽

Cited By ~ 6

Author(s):

Miriam Lenhard ◽

Caroline Kuemper ◽

Nina Ditsch ◽

Joachim Diebold ◽

Petra Stieber ◽

...

Keyword(s):

Leydig Cell ◽

Serum Markers ◽

Leydig Cell Tumours

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Hyperandrogenism caused by ovarian Leydig cell tumour: finding the needle in a haystack

BMJ Case Reports ◽

10.1136/bcr-2020-238012 ◽

2020 ◽

Vol 13 (12) ◽

pp. e238012

Author(s):

Chin Voon Tong ◽

Yong Ting Tai

Keyword(s):

Leydig Cell ◽

Specific Treatment ◽

Cell Tumour ◽

Imaging Modalities ◽

Surgical Removal ◽

Venous Sampling ◽

Ovarian Tumours ◽

Potentially Malignant ◽

Leydig Cell Tumour ◽

Leydig Cell Tumours

Leydig cell tumours (LCTs) of the ovary are rare ovarian tumours that usually present with hyperandrogenism. Conventional radiological imagings are helpful in localising these tumours. However, some tumours may be too small to be localised before curative surgical removal. It is important to identify these androgen-secreting neoplasms which originate mostly from adrenals or ovaries because they are potentially malignant and require specific treatment. When conventional imagings are unrevealing, selective ovarian and adrenal venous sampling (SOAVS) is the next option. We report a case of LCT that was localised by SOAVS after results from other imaging modalities remained inconclusive.

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Do small leydig cell tumours require follow up?

European Urology Supplements ◽

10.1016/s1569-9056(17)31945-0 ◽

2017 ◽

Vol 16 (10) ◽

pp. e2819

Author(s):

B. Yang ◽

G. Faust ◽

P. Furness

Keyword(s):

Leydig Cell ◽

Leydig Cell Tumours

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817 Loss of Libido in A Man with An Incidental Leydig Cell Tumour Of the Testis: A Rare Tumour Discovered Following an Isolated Common Complaint

British Journal of Surgery ◽

10.1093/bjs/znab259.325 ◽

2021 ◽

Vol 108 (Supplement_6) ◽

Author(s):

D Brown ◽

G Tsampoukas

Keyword(s):

Testicular Cancer ◽

Leydig Cell ◽

Biochemical Markers ◽

Hormonal Activity ◽

Rare Tumour ◽

Common Complaint ◽

Leydig Cell Tumour ◽

Loss Of Libido ◽

High Index Of Suspicion ◽

Leydig Cell Tumours

Abstract Introduction Leydig cell tumours (LCTs) are rare testicular stromal neoplasms classically presenting with a painless testicular mass or swelling in adults. Symptoms secondary to hypogonadism may occur resulting from the hormonal activity of these tumours. Loss of libido is described in LCTs in conjunction with other symptoms; however, no case has reported this as the sole presenting feature. Case report We describe the case of a 42-year-old man presenting to his General Practitioner with loss of libido and no other features suspicious of testicular cancer. Ultrasound performed due to an unrelated epididymal cyst detected an incidental mass confirmed as a benign LCT following radical orchidectomy. Biochemical markers remained normal throughout and following treatment his libido returned to normal. Conclusions This case may serve as a reminder for clinicians to maintain a high index of suspicion for testicular neoplasms in patients with features of hypogonadism in the absence of classical features for testicular cancer.

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