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CASE REPORT
Korean J Pediatr 2011 May;54(5) :224-227.
doi:https://doi.org/10.3345/kjp.2011.54.5.224
A sclerosing stromal tumor of the ovary with masculinization in a premenarchal girl
Soo Min Park (Park SM)1, Young Nam Kim (Kim YN)1, Young Jong Woo (Woo YJ)1, Ho Sun Choi (Choi HS)2, Ji Shin Lee (Lee JS)3, Suk Hee Heo (Heo SH)4, Chan Jong Kim (Kim CJ)1
1Departments of Pediatrics, Chonnam National University Medical School, Gwangju, Korea
2Departments of Obstetrics and Gynecology, Chonnam National University Medical School, Gwangju, Korea
3Departments of Pathology, Chonnam National University Medical School, Gwangju, Korea
4Departments of Radiology, Chonnam National University Medical School, Gwangju, Korea
Corresponding Author: Chan Jong Kim ,Tel: +82-62-220-6645, Fax: +82-62-222-6103, Email: cjkim@jnu.ac.kr
Copyright © 2011 by The Korean Pediatric Society
ABSTRACT
A sclerosing stromal tumor of the ovary is an extremely rare benign tumor; it usually is found during the second and third decades of life. Patients present with pelvic pain or a palpable abdominal mass. Hormonal effects such as masculinization are uncommon. Here, an 11-year old premenarchal girl presented with deepening of the voice. In addition, clitoromegaly and hirsutism with a male suprapubic hair pattern were observed. The laboratory findings showed that the testosterone level was elevated to 3.67 ng/mL, andostenedione to above 10 ng/mL, dehydroepiandrosterone-sulfate to 346 g/dL and 17-hydroxy progesterone (17-OHP) to 11.28 ng/mL. The chromosome evaluation revealed a 46,XX female karyotype. An adrenocorticotropic hormone stimulation test was performed. The 17-OHP to cortisol ratio in 30 minutes was 0.045, which suggested a heterozygote for the 21-hydroxylase deficiency. However, the CYP21A2 gene encoding steroid 21-hydroxylase showed normal. The pelvic ultrasound showed a heterogeneous mass consisting of predominantly solid tissue in the pelvic cavity. The pelvic magnetic resonance imaging revealed an 8.9 6.26.6 cm mass of the left ovary. A left oophrectomy was performed and microscopic examination confirmed a sclerosing stromal tumor. Immunohistochemical studies showed that the tumor was positive for smooth muscle actin and vimentin, but negative for S-100 protein and cytokeratin. Following surgery, the hormone levels returned to the normal range and the hirsutism resolved.
Keywords: Ovary | Ovarian neoplasms | Sex cord-gonadal stromal tumors | Virilism
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