Background: Premature thelarche, a benign condition that affects young girls, has been associated with elevated estrogen levels. On the contrary, labial fusion, a common pediatric gynecological problem, has been associated with low estrogen status. These two conditions are not known to coexist in the same patient.

Case: We report the case of a 2-yr-old girl with premature thelarche and labial fusion occurring contemporaneously. She presented with a 7-month history of perivaginal itchiness and a palpable painless breast bud. Examination revealed unilateral breast enlargement (Tanner stage 2) and partially fused labia minora. Pelvic ultrasound findings and gonadotrophin responses to intravenous LHRH were consistent with a diagnosis of isolated premature thelarche. Furthermore, elevated serum estradiol levels were found.

Comment: The combination of isolated premature thelarche and labial adhesions in our patient suggests the existence of other factors as well as estrogen insufficiency in the etiology of the latter.

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