Vaginal adenosarcoma arising from refractory endometriosis: A case report

Primary extra-uterine and extra-ovarian mullerian adenosarcoma: case report and literature review

“…Tumor markers were reported in 13/41 (31.7%) patients, two patients had normal value [ 20 , 31 ] and 11 patients had elevated value [ 12 , 15 , 17 , 18 , 23 , 24 , 27 , 30 , 32 – 34 ] (Table 1 ). Seven of eleven (63.6%) patients had elevated serum levels of CA 125 [ 12 , 15 , 18 , 23 , 27 , 30 , 32 ], 2/11 (18.2%) patients had elevated serum levels of both CA 125 and CA 19–9 [ 17 , 34 ], 1/11 (9.1%) patient had elevated serum levels of both CA125 and CEA [ 24 ], 1/11 (9.1%) had elevated serum level of LDH [ 33 ].…”

“…AS was treated by surgical resection in 38/41 (92.7%) patients: 5/38 (13.2%) patients underwent partial resection, and 33/38 (86.8%) underwent total resection. Of the 38 patients who received surgical treatment, 18 (47.4%) underwent resection of only the tumour [5abc, 7, 8, 9, 11, 13, 18, 19d, 20, 22, 23, 24, 30, 34, 36, 37]; four (10.5%) underwent tumour resection, hysterectomy and bilateral salpingo-oophorectomy [ 14 , 28 , 32 , 38 ]; and 16 (42.1%) underwent extensive surgery involving other organs such as the bowel [12, 15, 19a, 19b, 19c, 25, 29, 31, 35] and liver [ 17 ].…”

“…Overall, 17/25 (68%) patients with endometriosis had an AS with extra-genital localization [5c-10-11-17-19abc-22-23-24-29-30-31-33-36-37-38], and 8/25 (32%) patients had a vaginally localized tumour [15–16–21-25-26-27-32-39]. Moreover, 8/25 (32%) patients showed elevated levels of tumour markers [ 15 , 17 , 23 , 24 , 27 , 30 , 32 , 33 ]. A total of 7/25 patients received previous hormonal therapy [ 17 , 21 , 25 , 29 , 30 , 33 , 39 ]: 2/7 (28,6%) received HRT [ 17 , 30 ], 1/7 received (14.3%) ERT [ 25 ], 1/7 (14.3%) received TMX [ 33 ], 1/7 (14.3%) received oestrogenic-progestinic therapy [ 39 ], and 2/7 (28,6%) received unspecified hormonal therapy [ 21 , 29 ].…”

“…Their number of relapses ranged from one to four with a mean of 1.8. In 3/8 (37.5%) patients, the first relapse was in the pelvis [ 12 , 26 , 33 ]; in 1/8 (12.5%) patient, it was in the peritoneum [ 31 ]; in 3/8 (37.5%) patients, it was in the vagina [ 16 , 32 , 34 ]; and in 1/8 (12.5%) patient, it was in the parametrium [ 21 ]. The first relapse was surgically treated in 2/8 (25%) patients [ 33 , 34 ], it was treated with surgery and adjuvant therapy in 2/8 (25%) patients [ 16 , 26 ], and it was treated with only chemotherapy [ 12 , 31 , 32 ] or only radiotherapy and brachytherapy [ 21 ] in 4/8 (50%) patients.…”

“…In 3/8 (37.5%) patients, the first relapse was in the pelvis [ 12 , 26 , 33 ]; in 1/8 (12.5%) patient, it was in the peritoneum [ 31 ]; in 3/8 (37.5%) patients, it was in the vagina [ 16 , 32 , 34 ]; and in 1/8 (12.5%) patient, it was in the parametrium [ 21 ]. The first relapse was surgically treated in 2/8 (25%) patients [ 33 , 34 ], it was treated with surgery and adjuvant therapy in 2/8 (25%) patients [ 16 , 26 ], and it was treated with only chemotherapy [ 12 , 31 , 32 ] or only radiotherapy and brachytherapy [ 21 ] in 4/8 (50%) patients. Overall, 4/8 (50%) patients experienced a second relapse [ 12 , 32 – 34 ]: 1/4 (25%) patient’s second relapse location was in the pouch of Douglas [ 12 ], 1/4 (25%) patient’s relapse was in the pelvic peritoneum [ 33 ], and 2/4 (50%) patients’ relapses were in the vagina [ 32 , 34 ].…”