V Khadilkar and R Jehagirdar

Pre-pubertal gynecomastia is a rare condition in adolescent boys, and may be idiopathic or may result from excessive estrogen production by adrenal or testicular tumors, in association with congenital adrenal hyperplasia or to over expression of aromatase(1). We report a patient presenting with prepubertal gynecomastia, which on histopatho-logical examination was diagnosed as a hamartoma. A 3½ year old male child was brought with the sole complaint of left sided breast enlargement since 3 years of age. On examination he had unilateral left sided breast enlargement 5 cm × 5 cm, non-tender, soft, without any palpable nodule or discharge (Fig.1). Anthropometric examination was normal for age.

Fig. 1 Child with unilateral left sided gynecomastia.

Investigations showed serum thyroid stimulating hormone of 2.9 mU/L, free thyroxine 100nmol/L, leutinising hormone 0.7IU/L, follicle stimulating hormone 0.8 IU/L, prolactin 2.6 µg/L, oestradiol 20 pmol/L, 17-hydroxy-progesterone  4  nmol/L, dehydro epiandrosterone sulfate 0.1µmol/L, andro-stenedione 0.2 nmol/L and testosterone 0.2nmol/L which were all within the normal reference range for his age. Liver and renal function tests, a-feto protein and g-hCG were also normal. Karyotyping showed normal 46XY male. CT scan of the abdomen and pelvis was normal. USG of the breast showed a well-circumscribed, solid, hypoechoic mass with posterior acoustic shadowing. CT scan of thorax showed a unilateral breast tumor, which was well demarcated, and not infiltrating the chest wall.

Over the next 6 months, his breast swelling increased to 7 cm × 5 cm and hence a subcutaneous mastectomy was performed. Histopathological examination showed well capsulated fleshy mass measuring 6×5×5 cm, which was soft in consistency, with yellow islands of fat tissue. Microscopy showed breast tissue with many cystically dilated ducts with irregular lumina, lobular cells forming acini and occasional foamy and myoepithelial cells. Islands of adipose tissue and dense fibrous tissue were seen. Histopathology thus confirmed hamartomatous fibrocystic breast mass(2).

In summary, we describe a rare case of a three-year-old child presenting as pre-pubertal gyne-comastia diagnosed as hamartoma on excision biopsy. As the underlying pathophysiology of pre pubertal gynecomastia has not yet been determined, detailed evaluation of such patients is necessary before surgical intervention is undertaken.