September 1st, 2014

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Title:
Endometrial Adenocarcinoma Arising in a Turnerís Syndrome Patient with Spontaneous Menstruation: A Case Report
Authors:  Naoko Sasamoto, M.D., Yutaka Ueda, M.D., Ph.D., Kyoka Amemiya, M.D., Ph.D., Takayuki Enomoto, M.D., Ph.D., Eiichi Morii, M.D., Ph.D., and Kazushige Adachi, M.D., Ph.D
 
BACKGROUND: Women with Turnerís syndrome exhibit anovulation, and the majority do not spontaneously menstruate. We present an unusual case of endometrial adenocarcinoma developing in a Turnerís syndrome patient who was exhibiting spontaneous menstruation while not receiving regular hormone therapy.

CASE: The patientís karyotype from blood lymphocytes was a mosaic of 45,XO/ 46,XX. Menarche and sexual development were normal. Her menstrual cycle had been regular for one year, but then became noticeably irregular. At age 26 she was referred to our hospital after bleeding for almost 1 year. An endometrial adenocarcinoma was detected during performance of diagnostic endometrial curettage. A total abdominal hysterectomy with bilateral salpingo-oophorectomy and pelvic lymphadenectomy was conducted. The final histological diagnosis was endometrial adenocarcinoma, Grade 1, pT1a N0 M0. Fluorescence in situ hybridization analysis of the right and left ovaries revealed a mosaic karyotype of 45,XO/ 46,XX/47,XXX.

CONCLUSION: Previous reports regarding Turnerís syndrome detected spontaneous menstruation in only 16% of patients; however, spontaneous menstruation was observed in 8 of 10 (80%) Turnerís syndrome cases that developed endometrial carcinoma without receiving regular hormone therapy (p<0.0001). Hormone therapy may be indicated for an irregular menstrual cycle in Turnerís syndrome patients.
Keywords:  adenocarcinoma, endometrial adenocarcinoma, endometrial carcinoma, endometrial neoplasms, menstruation, Turnerís syndrome
   
   
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