ESPE Abstracts (2015) 84 P-2-432

A Perioperative Change of Anti-Mullerian Hormone and E2 in a Patient with Sex Cord Tumour with Annular Tubules

Aya Shimadaa,b, Masaki Takagia, Kentaro Miyaia, Ryuji Fukuzawac & Yukihiro Hasegawaa


aDepartment of Endocrinology and Metabolism, Tokyo Metropolitan Children’s Medical Center, Tokyo, Japan; bDepartment of Endocrinology and Metabolism, Kanagawa Children’s Medical Center, Kanagawa, Japan; cDepartment of Pathology, Tokyo Children’s Medical Center, Tokyo, Japan


Background: Sex Cord Tumour with Annular tubules (SCTAT) is a rare ovarian benign tumour accounted approximately for 5% of ovarian tumour. SCTAT is an oestrogen producing ovarian tumour and can cause precocious puberty. For SCTAT, oophorectomy is recommended as the first-line therapy. Since SCTAT has a high rate of recurrence, it is clinically important to find a monitor method that can detect cancer recurrence in an early stage. In the previous reports, E2 and anti-mullerian hormone (AMH) were reported as useful tumour markers. Here, we firstly describe perioperative change of these markers in a pediatric patient.

Case presentation: 6 years-old girl. She presented with GnRH independent precocious puberty (LH, FSH undetectable, E2 131 pg/ml). Ultrasonography showed a multiple cystic tumour sized in 5 cm in diameter arising from left ovary. Left oophorectomy was performed. The tumour was pathologically diagnosed as SCTAT. We examined AMH and E2 of cystic fluid of the tumour and perioperative blood sample. Cystic fluid and preoperative blood sample showed elevated AMH and E2 (Cystic fluid: AMH 1 830 ng/ml, E2 38 499 pg/ml, Blood sample: AMH 75.7 ng/ml, E2 153 pg/ml). Other ovarian tumour markers such as CEA,CA19-9, CA125 and hCG were all negative. Serum E2 became undetectable after 24 h from operation. AMH was also normalised after 1 month from operation. After 2 years from operation, no signs of relapse has been noted so far.

Conclusion: AMH and E2 may be used as useful tumour markers in a paediatric patient with SCTAT as it had been reported with adult patient. AMH can be a more useful marker in a paediatric patient since AMH level remains relatively stable from 4 to 8 years of age regardless of patients’ pubertal stage.

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