Unilateral pubic hair growth: a paracrine finding indicative of underlying leydig cell tumor in a prepubertal boy

Abstract

Leydig cell tumors (LCTs) are rare in children and typically present with testicular masses and signs of early puberty. We report a case of a 6-year-old boy presenting solely with precocious pubic hair growth. Ultrasound revealed a small right testicular mass and elevated serum testosterone. Intra-operatively, hair growth was more prominent on the affected hemi-scrotum. Testis-sparing surgery confirmed a benign LCT with adjacent increased spermatogenesis. This is the first known report of ipsilateral pubic hair as a presenting sign of LCT. Localized androgen excess may be an early clinical indicator to guide timely diagnosis and treatment.

Keywords: Leydig cell tumor; Paracrine effects; Precocious puberty; Pubic hair; Sex cord-gonadal stromal tumor; Testis mass; Testis neoplasm; Testis-sparing surgery.

Fig. 1

A. Scrotal US demonstrating 3x5x4mm right intratesticular hypoechoic mass. Right testis volume 1.19 cc, left testis volume of 0.53 cc. B. US demonstrating mass with internal vascularity.

Fig. 2

Illustration of patient's scrotum with pubic hair growing preferentially on right hemi-scrotum.

Fig. 3

A. Microscopic evaluation of the tumor at low, medium and high magnification: The lesion is well demarcated (low magnification), with few entrapped seminiferous tubules (medium magnification, arrow) and is composed of Leydig cells without atypia, mitosis or necrosis (high magnification). B. Seminiferous tubules adjacent to the tumor demonstrating increased spermatogenesis for age, with formation of spermatozoa. (arrow).