A 49-year-old woman presented to our hospital in August 2020, complaining of post-coital bleeding with no abnormal vaginal discharging. The patient began having vaginal bleeding after sexual intercourse around 1 month before admission. She had a history of didelphic uterus with cervix duplex. The time of first discovery of her genital malformation was uncertain. And she'd been pregnant four times, with one cesarean section and three induced abortions, including one abnormal pregnancy in the left uterus that resulted in a supracervical hysterectomy around 16 years ago. Previously, she had regular menstrual cycles of 30 days with menstruations of 5-6 days. She had moderate menstruation volume and no dysmenorrhea. The patient had not previously had a routine physical examination. Her latest cervical screening was three years ago, and she underwent an appendectomy eight years ago. There was no family history of tumors or genital malformations in her family.
Gynecological examination indicated a normal-appearing right cervix, the surface was smooth, but it was easy to bleed when touched, and the non-dominant side of the left cervix was concealed by the side wall of the left vagina. After exposing, the left side cervix presented a mature appearance which was smaller than a normal cervix ( Figure 1 ). Bimanual rectovaginal examination can detect the lack of uterine body, pelvic cavity emptiness, and no abnormalities in bilateral parauterine regions.
Her blood carbohydrate antigen 125 (CA125), carcinoembryonic antigen (CEA), alpha fetoprotein (AFP), and carbohydrate antigen 153 (CA153) levels were all within normal ranges (CA125 17.2 U/ml, CEA < 0.5 ng/ml, AFP 1.5ng/ml, CA153 5.5 U/ml), however the carbohydrate antigen-19-9 (CA19-9) level increased considerably, the corresponding results was 112.59 U/ml. Colonoscopy and gastroscopy revealed no visible abnormalities. The cytology result of liquid-based cell testing in the right cervix was normal, whereas the left cervix revealed atypical glandular epithelial cells. Both cervical smears were negative for human papilloma virus (HPV), including high risk HPV16, 18, 31, 33, 35, 39, 45, 51, 52, 53, 56, 58, 59, 66, 68, 73, 82, and low risk HPV6, 11, 42, 43, 81, 83. Histopathology of the two cervices from colposcopy-directed punch biopsy (CDB) and endocervical curettage (ECC) revealed: 1. (RIGHT CDB) adenocarcinoma at 3,6,12 o'clock on the right cervix, IHC: ER (focal weak+), CK7 (+), CDX2 (-), Muc-6 (-), P53 (+, positive rate of 20%), Ki67 (approximately 30%); 2. (RIGHT ECC) the submitted tissue is a small blood clot; 3. (LEFT ECC) the submitted tissue is a little mucus; 4. (LEFT CDB) chronic inflammation at the left cervix. Pelvic magnetic resonance imaging (MRI) revealed two cervical canals and a 1.9cmx1.6cm mass inside the left cervix ( Figure 2 ). The left wall of the right cervix may be involved by the tumor in the left cervix. A few small lymph nodes on both sides of the pelvic could be seen. And there was no lymphovascular space invasion or enlarged pelvic lymph node identified. In addition, there was only one right kidney, with no left kidney.
The biopsy of the patient's right cervix indicated adenocarcinoma, while MRI revealed that the tumor was situated in the left cervical canal and had invaded the neighboring right cervix. After much deliberation, the diagnosis of adenocarcinoma of the left cervix stage [IB1, International Federation of Gynecology and Obstetrics (FIGO), ] and stage T1b1N0M0 [American Joint Committee on Cancer (AJCC), ] was made. In the first step, we performed cystoscopy, the right ureteral D-J tube implantation, and pelvic sentinel lymph node biopsy under laparoscope. The hyperstaging of pathology revealed that three sentinel lymph nodes had no cancer metastases. Then, we conducted radical cervical resection with bilateral salpingo-oophorectomy, and bilateral pelvic lymphadenectomy without the use of a manipulator. The intraoperative diagnosis was consistent with the preoperative diagnosis. The double cervices were dissected after surgery.
After the resection, the morphology of the double cervices was seen to be irregular. Tumors were seen between the duplex cervices' circumferential walls. The tumor was around 2cmx2cmx2cm in the left cervix and penetrated the neighboring right cervical tissue without infiltrating the vagina or the parametrium. Microscopically, the morphology and immunohistochemistry of the duplicated cervices revealed gastric-type adenocarcinoma with high and intermediate differentiation, as well as intestinal differentiation in some regions. The tumor grew endogenously, penetrating the whole layer of the left cervical wall as well as the shallow layer of the right cervical wall (about 1/3 of the cervical wall). There was lymphatic vascular space infiltration (LVSI), but no clear nerve invasion or malignancy in the bilateral parauterine, vaginal stump, or vaginal vault. Pathological examination revealed no tumor involvement in the lymph nodes. There was no evidence of malignancy in the ovarian and fallopian tube tissues on either side. Additionally, no cancer metastases was discovered in the lymph nodes that were removed (0/18). Immunohistochemistry of the tumor cells were as follows: Muc-6 (+, focally), P16 (+, focally), P53 (20% medium intensity +), ER (+, focally), PR (-), CK7 (+++), CK20 (+, focally), Muc-2 (+, focally), Muc-5 (++), CDX-2 (+), SATB2 (-), Pax-8 (-), and the ki67 proliferative index was around 15% ( Figure 3 ). All 23 HPV subtypes tested negative, including high-risk HPV16, 18, 31, 33, 35, 39, 45, 51, 52, 53, 56, 58, 59, 66, 68, 73, 82, and low-risk HPV6, 11, 42, 43, 81, 83. Following surgery, she had concomitant chemoradiation, a total dosage of 60 Gy pelvic irradiation, and three cycles of chemotherapy comprising paclitaxel and carboplatin. Currently, 29 months after the final treatment, the patient has normal cytology and colposcopy results with negative HPV tests.