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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.


INTRODUCTION
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.
We considered percutaneous ethanol sclerotherapy rather than surgical treatment because she wanted to preserve the uterus. 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 (3,5). VMs are congenital lesions and become symptomatic with the child's growth. However, they may not be  vaginal VMs are asymptomatic and require no treatment. However, if the patient has symptoms such as abnormal vaginal bleeding or protruding mass, treatment is needed (4,7).
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.