Endometrial estrogen and progesterone receptor activity in unusual chronology of pubertal development: a case report

Abstract An 8-year-old girl presented with vaginal bleeding without puberty signs. During investigation at 10 years old, the patient presented with advanced bone age, prepubertal hormone levels and internal genitalia. Hysteroscopy and vaginoscopy at 10 years and 10 months of age revealed endometrial proliferation despite infantile genitalia. Histopathology showed positive receptors for estrogen and progesterone. This is the first report of endometrial estrogen and progesterone receptor activity in this context. Despite normal serum estradiol, the findings suggest a local action of estrogen. Follow-up until 11 years of age showed progressive development of secondary sexual characteristics. This case report emphasizes the need to consider isolated vaginal bleeding, without the development of secondary sexual characteristics, as a result of endometrial hypersensitivity to low estrogen levels during pubertal development, which may alter the chronology of pubertal development in girls.