Presumptive ovarian hyperthecosis in an adolescent girl with sustained remission after oral contraceptive therapy: a 23-year follow-up case report.

OBJECTIVES: To report the first documented case of spontaneous resolution of presumptive ovarian hyperthecosis in an adolescent and to highlight the potential for conservative medical management in this age group.

CASE PRESENTATION: A 14-year-old girl presented with 14 months of secondary amenorrhea, voice deepening, and clitoromegaly. Laboratory evaluation revealed markedly elevated testosterone (498 ng/dL) with normal DHEAS (185 μg/dL), localizing androgen production to the ovary. After exclusion of androgen-secreting tumors and nonclassic congenital adrenal hyperplasia through imaging and ACTH stimulation testing, she was diagnosed with presumptive ovarian hyperthecosis. Treatment with combined oral contraceptives (COCs) resulted in normalization of testosterone to 22 ng/dL over 10 months. Discontinuation of COCs led to recurrence of severe hyperandrogenism (testosterone 571 ng/dL), which again normalized with reinitiation of therapy. After 4 years of COC treatment, therapy was discontinued at age 18. Remarkably, androgens remained normal off therapy at 3 months, menses resumed and became regular by 6 months, and she has remained asymptomatic with normal ovarian function for 23 years of follow-up (to age 37 years). Her most recent testosterone level at age 37 is 38 ng/dL.

CONCLUSIONS: This case represents the first documented spontaneous resolution of ovarian hyperthecosis and suggests that adolescent-onset disease may have a different natural history than postmenopausal ovarian hyperthecosis. Combined oral contraceptives provide effective first-line therapy for adolescent ovarian hyperthecosis. Long-term follow-up with periodic trials off medical therapy is warranted to assess for spontaneous resolution, supporting conservative management and fertility preservation rather than immediate surgical intervention in this age group.