Gynecological tumor triplicity

Overview

Objective:

Information about an interesting real clinical case.

Design:

Case repor.

Setting:

Clinic of Gynecology and Obstetrics, University hospital in Ostrava.

Methods and results:

A 64-year-old female patient with no family history of gynecologic tumors was indicated to total laparoscopic radical hysterectomy due to confirmed squamous cervical carcinoma stage IB1. The operation was converted to laparotomy due to unexpected peroperative finding of ovarian malignancy. Peroperatively, left ovary borderline tumor and tiny implants on the surface of uterus and small pelvis walls were confirmed. Radical abdominal hysterectomy, bilateral salpingo-oophrectomy, omentectomy, appendectomy, pelvic lymphadenectomy and peritonectomy were performed.

Conclusion:

Definitive postoperative histology further revealed an endometroid adenocarcinoma of the uterus, serous left ovary borderline tumor with non-invasive implants and no residual tumor of cervical carcinoma. In our clinic, several tumor duplicities were reported, but this is the first case of a gynecological tumor triplicity. As far we know, this is the first case report refering to a gynecological tumor triplicity in one time.

Keywords

gynecologic, multiple, malignancy, radical surgery

INTRODUCTION

Incidence of cervical cancer in the Czech Republic is 17/100 000 while that of endometrial carcinoma is 34/100 000 [1]. The incidence of borderline tumor in the Czech Republic is not actually available, but based upon registry data from the United States, Denmark and Sweden, the estimated incidence of these tumors ranged from 1.8 to 5.5 per 100 000 women per year [4, 9, 13]. Danish study reported that the incidence of borderline ovarian tumors increased from 2.6 to 5.5 per 100 000 women-years between 1978 and 2006 [4]. Incidence of multiple tumors is very rare. In a Dutch population study, 7% of patients with any kind of malignancy also had another malignant tumor, which is 0.2% of the total population of the Netherlands. The most frequent combination was breast and genital tract cancer [7]. In case of endometrial cancer, the co-incidence of another malignancy might be as high as 20 percent [14].

The aim of this article is to inform about a very rare case which has been detected and managed in our department.

CASE REPORT

In December 2014, a 64-year-old female patient who previously underwent conization with histologically confirmed squamous cervical carcinoma (spinocellular carcinoma grade 3, with diameter 25×33×9 mm with positive resection edge) was referred to our hospital for treatment. The patient had no history of vaginal bleeding, no family history of malignancies and her medical history was without severe diseases, she underwent only three orthopedic operations – replacement of both hips and the left knee joint replacement. Her gynecologic history include two spontaneous deliveries and no history of oral contraceptions or postmenopausal hormonal replacement therapy use. Patient attended regular preventive examinations by a gynecologist and she has not been a smoker. Her body mass index was 33.

Preoperative examination with expert ultrasound examination did not show any suspicion on other tumor (endometrium height of 4.5 mm) and according to the clinical stage IB1 the patient was indicated for total laparoscopic radical hysterectomy. The laparoscopy operation in January 2015 had to be converted to laparotomy due to unexpected findings of ovarian malignancy. A serous borderline tumor of the left ovary was found together with tiny implants on the surface of the uterus and small pelvis walls. Radical abdominal hysterectomy in radicality C2 according Querleu-Morrow, bilateral salpingo-oophrectomy, omentectomy, appendectomy, pelvic lymphadenectomy and peritonectomy were performed. The postoperative course was complicated by wound infection and dehiscence with the necessity of a resuture. After reoperation the healing was without complications.

Definitive postoperative histology revealed small endometrial adenocarcinoma of the uterus stage IA grade 1, with no lymphangioinvasion, serous borderline tumor of left ovary with non-invasive implants on surface of uterus, both fallopian tubes and small pelvis peritoneum stage IIB and no residual tumor of cervical carcinoma (spinocellular carcinoma from conization grade 3, with diameter 25×33×9 mm with positive resection edge) stage IB1. Due to negative prognostic factors patient was indicated to brachyradiotherapy.

DISCUSSION

As far we know, only few similar, but not same case reports were published in literature. Takori reported about cervix/endometrium/ovary tumor triplicity, but all tumors were adenocarcinomas [15]. Isin et al. reported about tumor triplicity including well-differentiated ovarian mucinous cystadenocarcinoma, well-differentiated endometrial endometrioid adenocarcinoma, and uterine leiomyosarcoma [10] and Phupong et al. reported about adenosquamous carcinoma of the endocervix, adenocarcinoma of the endometrium, low malignant potential of the right ovary and mucinous cystadenocarcinoma of the left ovary [2, 6].

This is the first case report referring to a gynecological tumor triplicity in one time consist of spinocelullar carcinoma of cervix, endometroid adenocarcinoma of uterine corpus and borderline tumor of ovary. It raises questions whether such an incidental finding could have been avoided.

This combination of tumors is not associated with any hereditary predisposition as far we know. Ethiopathogenesis of cervical carcinoma is known [15] and differs from endometrial carcinoma and ovarian borderline tumor, whose causes are not exactly known. Endometrial cancer is associated with an imbalance between estrogens and progesteron. These carcinomas are thought to be caused by a combination of hormonal factors and mutational events [15]. Gene expression in endometrial epithelium varies with the hormonal changes during menstrual cycle. Endometrial cancer is much more common in women with Lynch syndrome (27–71% vs 3% in general population) while this syndrome has no relevance to other mentioned malignacies [15].

A meta-analysis of eight case-control studies reported a significant increase in the risk of borderline tumors in women with an unknown cause of infertility [11]. Other risk factors including the relationship between BRCA gene mutations and borderline ovarian tumors is uncertain. It appears that the prevalence of BRCA mutations is lower among women with borderline tumors than in women with invasive ovarian carcinoma [3, 8, 12].

Generally, the number of cancer survivors continues to increase by 3–5% annually and thus prognosis improves with each year survived, multiple malignancies are becoming more important in the field of cancer surveillance [5].

There is no relevant recommendation for treatment in case of tumors combination. Due to low stages of each tumor we are expecting a good patient prognosis.

CONCLUSION

In an effort of the best possible preoperative staging of oncogynecologial cases and with respect to potential finding of gynecological duplicities, all oncogynecological patients in our department undergo expert preoperative ultrasound examination including ultrasound examination of adnexal masses according to IOTA This approach clearly decreases the risk of unexpected severe peroperative findings. However in our case, the size of ovaries of both sides was bigger than atrophic postmenopausal ovaries and the surface was very irregular, which our ultrasound examination did not find out. The endometrial tumor was very small and without any symptoms. There is no history of a similar case in our department. Finding of tumor triplicity in one time is very unlikely. We didn‘t find any connection in etiopathogenesis of these tumors in literature.

MUDr. Aleš Mladěnka

Gynekologicko-porodnická klinika FN

17. listopadu 1790

708 52 Ostrava Poruba

e-mail: ales.mladenka@fno.cz