Metachronous Bilateral Leydig Cell Tumor in a Male Adult Aged 20

Abstract

We report a young male aged 20 who has suffered two episodes of Leydig cell tumor of the testis, the second occurring 5 years after the first in the contralateral testis. The case is outlined briefly, with references taken from the literature. This young man’s history is exceptional as this type of tumor is infrequent, and metachronous bilateral presentation extremely rare.

Case – A tale of two boys: An atypical cause of pubertal precocity

“…Parenchyma preservation surgery is pursued to maximally preserve fertility, whereby radical orchiectomy is reserved for malignant or atrophic cases. 2,15 Central precocious puberty developed after tumor resection in our second case as previously shown in the literature. 3,16 The mechanism is unknown but is theorized due to early maturation of the hypothalamic-pituitary-gonadal (HPG) axis from premature exposure of elevated testosterone via the LCT.…”

“…LCT CUAJ -Resident's Room Akilan et al Case: Atypical cause of pubertal precocity 4 © 2020 Canadian Urological Association traditionally presents as unilateral and benign with an estimated 3-10% appearing as bilateral, in adult series of patients. 4,15 There are no prior cases found in the literature describing the presentation of a multifocal and bilateral LCT as seen with our patients. It is important to understand this unique presentation as Leydig Cell Hyperplasia (LCH) would present in a multifocal manner and is difficult to differentiate from LCT.…”

Sexual Pseudo-Precocity Caused by a Somatic Activating Mutation of the LH Receptor Preceding True Sexual Precocity

“…Leydig cell tumors are usually benign, but cases of malignant Leydig cell tumors are reported [19]. Other cases are described in the literature, when tumors occur in the contralateral testis even after years [20]. The performed molecular analysis of the DNA extracted from tumor material showed a heterozygous mutation in exon 11 leading to a replacement of aspartic acid with histidine at position 578 (Asp 578 His).…”