Transvaginal Direct Puncture and Ethanol Sclerotherapy for Cervicovaginal Venous Malformations: A Case Report and Literature Review.

Cervicovaginal venous malformations are extremely rare. Sclerotherapy is proven to be effective for superficial venous malformations but not for venous malformations in the lower genital tract of women. A 52-year-old female presented with intermittent vaginal bleeding. The amount of vaginal bleeding gradually increased over 3 months. Contrast-enhanced pelvis CT showed several phleboliths and dilated vessels, but pelvic angiography showed no early draining veins, nidus, or feeding artery. We performed transvaginal direct puncture and ethanol sclerotherapy rather than surgical treatment because she wanted to preserve the uterus. After four sessions of sclerotherapy, she had significantly decreased vaginal bleeding without complications. Here, we report the first case of cervicovaginal venous malformations successfully treated with transvaginal direct puncture and ethanol sclerotherapy. Copyrights

INTRODUCTION

Vascular anomalies are divided into two broad biologic categories: vascular or vasoproliferative neoplasms and vascular malformations, according to the 1996 International Society for the Study of Vascular Anomalies classification proposed by Mulliken and Glowacki's (1). Vascular malformations are also subdivided into high-flow versus low-flow lesions according to their rheology. According to the classification, venous malformations (VMs) are included in slow-flow malformations (12).

About 40% of VMs are found in the head and neck region (3). Especially, VMs in the female lower genital tract were very rarely reported (4). Management of VM have various treatment options such as observation, sclerotherapy, surgical excision or a combination of these treatments (2). Among them surgical treatment is preferred in VM of the female lower genital tract (4). We report here the first case of successful direct puncture and ethanol sclerotherapy for cervicovaginal VM of a 52-year-old female via transvaginal route.

CASE REPORT

A 52-year-old female, para 2-0-0-2, presented with history of intermittent vaginal bleeding for the last two years. She looked a little pallor, but physical examination findings and laboratory studies were unremarkable. Also, she has no previous history of abortion, trauma and abdominal surgery. She has been on conservative treatment for the last two years, but the amount of vaginal bleeding began to increase over the late three months.

Transvaginal ultrasonography and contrast enhanced pelvis CT revealed several phleboliths and dilated vessels around the cervicovaginal wall without early draining veins (Fig. 1A). Pelvic angiography showed no feeding artery, nidus and early draining veins (Fig. 1B, C). On these findings, the lesion was considered most likely to be the VM rather than the arteriovenous malformation.

Fig. 1

Transvaginal ethanol scleortherapy in a 52-year-old female with cervicovaginal venous malformations.

A. Contrast-enhanced pelvis CT shows several phleboliths and dilated vessels without early draining veins around the cervicovaginal wall.

B, C. Pretreatment pelvic angiography shows no feeding artery, nidus, or early draining veins.

D, E. First (D) and third (E) sessions of transvaginal direct puncture and ethanol sclerotherapy was done for cervicovaginal venous malformations.

F. One-year follow-up contrast-enhanced CT shows a decrease in dilated vessels around the right cervicovaginal wall.

We considered percutaneous ethanol sclerotherapy rather than surgical treatment because she wanted to preserve the uterus. A procedure was performed under general anesthesia with lithotomy position. A 21-gauge Chiba needle (Cook Inc., Bloomington, IN, USA) attached to transvaginal probe was inserted through the vagina. Under ultrasonography guidance, the dilated vessels around the cervicovaginal wall was punctured with a 21-gauge Chiba needle. To identify abnormal dilated veins, a test injection of contrast medium was performed under fluoroscopy. And ethanol sclerotherapy were performed using 80% ethanol as an embolic agent (Fig. 1D, E). She received 4 sessions of ethanol sclerotherapy treatment at three month intervals. After one year, a follow-up enhanced CT scans showed that previous dilated vessels decreased around the right cervicovaginal wall (Fig. 1F). The patient had significantly decreased vaginal bleeding without complication after 4 sessions of sclerotherapy. If the amount of vaginal bleeding increases, additional sclerotherapy may be considered, but additional treatment is not currently planned due to a significan reduction in vaginal bleeding and increased patient satisfaction.

DISCUSSION

VMs are the most common vascular malformation in the body with a prevalence of 1% of the population (5). More than 40% of cases are found in the head and neck (35). VMs are congenital lesions and become symptomatic with the child's growth. However, they may not be noticed for life without rapid enlargement of VMs (24).

Cervical or vaginal VMs are extremely rarely reported. Wang et al. (4) reported that only three patients involved the cervicovaginal lesions among 646 female patients with unclassified vascular anomalies. Karpathiou et al. (6) reported 8 cases of VM lesions in vagina or cervix of 50000 gynecological tract specimens during a 20 years period. In most cases, cervicovaginal VMs are asymptomatic and require no treatment. However, if the patient has symptoms such as abnormal vaginal bleeding or protruding mass, treatment is needed (47).

In the localized lesion of cervicovaginal VMs, it is preferred to selective embolization, local excision or a combined procedure rather than hysterectomy. However, hysterectomy is preferred in the case of diffuse lesion or older patients (4). In diffuse lesion of our case, we performed direct puncture and ethanol sclerotherapy rather than hysterectomy. We report the first case of successful treatment of the cervicovaginal VM as transvaginal direct puncture and ethanol sclerotherapy.

In conclusion, our case provide evidence that transvaginal direct puncture and ethanol sclerotherapy is relatively noninvasive, safely, and effective alternative treatment option in the cervicovaginal VM.