TY - JOUR
T1 - Female adnexal tumor of probable wolffian origin (FATWO) with recurrence 3 years postsurgery
T2 - C-kit gene analysis and a possibility of a new molecular targeted therapy
AU - Syriac, Susanna
AU - Durie, Nicole
AU - Kesterson, Joshua
AU - Lele, Shashrikant
AU - Mhawech-Fauceglia, Paulette
PY - 2011/5
Y1 - 2011/5
N2 - This is the case report of a 38-year-old woman who presented with a mass of the right broad ligament that was diagnosed as a female adnexal tumor of probable Wollfian origin (FATWO). The patient was treated with a simple mass excision. Three years after the excision, the patient presented with uterine bleeding. A total abdominal hysterectomy was advised. Intraoperative histologic consultation showed a poorly differentiated tumor on the surface of the left ovary. After extensive immunohistochemistry analysis and after reviewing the histology slides from the primary tumor, the final diagnosis was concluded to be recurrent FATWO on the surface of the ovary. C-kit immunohistochemistry was found to be strongly positive. Polymerase chain reaction amplification of C-kit genes on exons 9, 11, 13, and 17 and of PDGFR gene on exons 12 and 18 showed no mutational changes. Owing to the limited options in treating recurrent disease and the lack of prognostic factors for recurrence or metastasis, the patient was started on 400 mg of imatinib mesylate therapy for 6 months. In addition, the patient is undergoing continuous follow-up by computed tomographic imaging every 6 months. As chemotherapy and radiation therapy for recurrent or metastatic FATWO are most often unsuccessful, a molecular targeted therapy, such as tyrosine kinase inhibitor, could be considered. However, collective data are needed from multiple centers to determine its effectiveness in these patients.
AB - This is the case report of a 38-year-old woman who presented with a mass of the right broad ligament that was diagnosed as a female adnexal tumor of probable Wollfian origin (FATWO). The patient was treated with a simple mass excision. Three years after the excision, the patient presented with uterine bleeding. A total abdominal hysterectomy was advised. Intraoperative histologic consultation showed a poorly differentiated tumor on the surface of the left ovary. After extensive immunohistochemistry analysis and after reviewing the histology slides from the primary tumor, the final diagnosis was concluded to be recurrent FATWO on the surface of the ovary. C-kit immunohistochemistry was found to be strongly positive. Polymerase chain reaction amplification of C-kit genes on exons 9, 11, 13, and 17 and of PDGFR gene on exons 12 and 18 showed no mutational changes. Owing to the limited options in treating recurrent disease and the lack of prognostic factors for recurrence or metastasis, the patient was started on 400 mg of imatinib mesylate therapy for 6 months. In addition, the patient is undergoing continuous follow-up by computed tomographic imaging every 6 months. As chemotherapy and radiation therapy for recurrent or metastatic FATWO are most often unsuccessful, a molecular targeted therapy, such as tyrosine kinase inhibitor, could be considered. However, collective data are needed from multiple centers to determine its effectiveness in these patients.
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U2 - 10.1097/PGP.0b013e3182005340
DO - 10.1097/PGP.0b013e3182005340
M3 - Article
C2 - 21464731
AN - SCOPUS:79955075607
SN - 0277-1691
VL - 30
SP - 231
EP - 235
JO - International Journal of Gynecological Pathology
JF - International Journal of Gynecological Pathology
IS - 3
ER -