A huge prolapsed cervical leiomyoma: A case report

Abstract

Introduction and importance

Uterine leiomyoma is the most common pelvic tumor in women. Its cervical location is rare and may extend into the vagina in 2.5 % of cases. Treatment of cervical fibroids includes either myomectomy or hysterectomy, depending on the patient's profile and the tumor's characteristics. These fibroids challenge the surgeon because of their proximity to vital pelvic structures and their likelihood of causing surgical complications.

Case presentation

A 47-year-old woman presented with abdominopelvic pain and a bulky necrotic mass protruding out of her vagina. CT scan showed a large heterogeneous anterior mass of the cervix measuring 30 cm prolapsed in the vagina. She underwent a total hysterectomy with complete resection of the cervical mass. The histopathological report confirmed the diagnosis of a cervical leiomyoma with no signs of malignancy.

Clinical discussion

Three types of cervical leiomyoma are known: interstitial, supra-vaginal, and polypoidal. The last one, observed in our case, is the rarest type. When prolapsed in the vagina, cervical leiomyoma can outgrow its blood supply and become necrotic. Several approaches are available for the management of cervical leiomyomas. The approach choice depends on many factors such as the tumor size and location, its extent, and the desire for fertility.

Conclusion

This report describes the case of a large gangrenous and prolapsed non-pedunculated cervical leiomyoma which remains a rare and disabling complication of this benign tumor for which hysterectomy remains the treatment of choice.

Highlights

• •Uterine leiomyoma should be considered, in front of a mass delivered through the vagina.
• •Prolapsed cervical leiomyoma is a rare entity.
• •Surgery is essential for symptomatic cervical leiomyoma.
• •Total hysterectomy remains the treatment of choice for huge non-pedunculated cervical leiomyoma.

1. Introduction

Uterine leiomyomas are the most common benign smooth muscle tumors of the genital tract in women between 35 and 40 years old [1], [2]. Asymptomatic, slow-growing fibroids are seen among perimenopausal women constituting 30 % of the total. However, they can cause vaginal bleeding, pelvic pain, urinary disorders, pregnancy loss, dyspareunia, and in some cases infertility [1]. These tumors are estrogen-dependent [3].

Cervical leiomyomas are a rare entity, their prevalence is considered to be less than 5 %, where abnormal vaginal bleeding, dyspareunia and constipation are common presentations [4]. It can pass through the cervical canal to the vagina causing necrosis which was seen in our present case [1].

Ultrasound, magnetic resonance imaging (MRI) and CT scan play an important role in the management of patients with cervical leiomyomas [5] in order to detect their number, size, and location. The management of cervical fibroid includes either myomectomy or hysterectomy depending upon patient’'s profile. These prove to be a challenge to the surgeon in view of their close proximity to the vital pelvic structures and likelihood to cause surgical complications.

This case report was prepared according to the SCARE Criteria [6].

2. Presentation of case

A 47-year-old woman gravida-3 para-3, premenopausal with no past medical history presented to our outpatient clinic with a four-month history of a mass protruding per vagina associated with sporadic vaginal bleeding and pelvic pain.

The physical examination revealed abdominal distension with pelvic tenderness.

Gynecological examination showed a 30 cm mass protruding from the vagina with areas of necrosis. The cervix was not visualized (Fig. 1).

Fig. 1.

A 30 cm necrotic leiomyoma protruding from the vagina.

The complete blood count showed hemoglobin of 8.7 g/dl, hematocrit of 28.4 %, and 12,100 white blood cells.

The abdominopelvic CT scan showed a large heterogeneous anterior mass of the cervix measuring 30 cm prolapsed in the vagina. The urinary bladder was drawn up over the uterus and there was a dilation of the right excretory urinary tract due to the mass compression (Fig. 2).

Fig. 2.

CT scan with a coronal (A) and sagittal (B) section shows a large heterogeneous anterior mass of the uterine cervix.

Exploratory laparotomy revealed a large gangrenous cervical fibroid measuring 30 cm. The uterus was normal in size sitting on top and both ovaries were normal. The surgery consisted of a total hysterectomy with complete resection of the vaginal mass (Fig. 3). The patient developed well in the postoperative period and was discharged by day four. The histopathological report confirmed the diagnosis of a cervical leiomyoma with no signs of malignancy.

Fig. 3.

Hysterectomy and bilateral salpingo-oophorectomy specimen showing a huge and necrotic cervical leiomyoma.

3. Discussion

Leiomyoma, a benign smooth muscle tumor of the uterus, is the most common pelvic tumor which occurs in women of reproductive age group given that their growth is influenced by the hormones estrogen and progesterone [3]. Cervical localization in leiomyomas is rare due to the paucity of smooth muscles in the cervix. It represents 1–2 % of the total cases [2] and it presents both a diagnostic and therapeutic challenge.

Cervical myomas are usually solitary in nature in contrast to uterine myomas [7]. They arise mainly from the supravaginal portion of the cervix. Depending upon their location, cervical fibroids may be classified as anterior, posterior, lateral or central. Anterior fibroid bulges forward & undermines the bladder, leading to urinary frequency or retention [5]. Posterior fibroid flattens the pouch of Douglas and compresses rectum against sacrum causing constipation. Central cervical fibroid expands the cervix equally in all directions, pushing the uterus upwards to give the typical “Lantern of St Paul's dome” appearance.

Three types of cervical leiomyoma are known: interstitial, supra-vaginal, and polypoidal. The last one, observed in our case, is the rarest type [2], [8].

Cervical leiomyoma can change the shape of the cervix and cause it to lengthen. It can also cause the uterus to push upward or the bladder to be drawn up when its size gets bigger which predisposes to urinary tract disorders and infections. When prolapse in the vagina, cervical leiomyoma can outgrow its blood supply and become necrotic [8], [9], [10] which is the case of our patient.

Patients with cervical leiomyoma are often asymptomatic. However, they can present some debilitating, vague and non-specific symptoms as a result of mass effect on the surrounding pelvic organs. Common complaints include uterine bleeding, dyspareunia, lower abdominal pain, urinary frequency, tenesmus, pregnancy loss, and some cases of infertility [8], [11], [12], [13]. Menstrual-related complaints are rare. This differs from FIGO type 0–5 myomas where symptoms of abnormal uterine bleeding predominates [14].

Differentials for a large pelvic mass originating from the uterus in a premenopausal female patient will include: 1. Pregnancy; 2. uterine corpus lesions: benign (leiomyomas, adenomyosis and polyps) or malignant (leiomyosarcoma, endometrial stromal sarcomas, carcinosarcoma and endometrial carcinoma); 3. uterine cervix lesions: benign (leiomyomas) or malignant (cervical carcinoma).

Although ultrasound is the preferred method for initial evaluation, MRI and CT scan are the most accurate methods for preoperative localization of fibroids and surgical planning once pregnancy has been excluded [15]. MRI helps in providing imaging planes that are not available on transabdominal or transvaginal ultrasound, a feature that permits better visualization of the more lateral and posterior area of pelvis. Fibroids appear as sharply marginated areas of low to intermediate signal intensity on T1 and T2 weighted MRI scans [16].

Several approaches are available for the management of cervical leiomyomas. The approach choice depends on many factors such as the tumor size and localization, the extensions, and the desire for fertility [4], [17].

The surgical treatment of cervical myomas can be challenging and it requires a great experience and expertise of the surgeon. In fact, the presence of a cervical leiomyoma has been identified as an independent factor affecting operation time in minimally invasive surgery [13]. The surgical risks are related to the position of the cervical leiomyoma in the pelvis since it can have close relations with pelvic structures, making difficult the identification of a correct cleavage plane for the surgeon; procedures can be further complicated by more restricted and inaccessible surgical spaces [18]. Furthermore, voluminous cervical myomas can alter the position of these structures as mentioned before, subverting the anatomy of the pelvis, resulting in a high degree of difficulty in performing the surgery [19].

Surgery performed for cervical fibroids can be vaginal or abdominal [12]. Vaginal myomectomy is a conservative procedure that can be performed in small and or pedunculated cervical fibroid polyp where there is adequate vaginal access and mobility of the mass owing to long pedicle. Central cervical fibroid is difficult to operate because uterine vessels are so elevated as to run parallel to ovarian vessels forming a vascular leash close to the uterus [5], [20].

Hysterectomy is considered the definitive treatment for uterine fibroids especially, in the case of voluminous non-pedunculated mass localized to the cervix and in the case of old women with more than one myoma, but vaginal myomectomy presents a conservative way to treat small and or pedunculated fibroids for young patients who still want to maintain their reproductive function [20]. Although hysterectomy is the definitive treatment for fibroids, it represents the gold standard in locally extensive leiomyomas like our case which was a technically challenging situation as the patient presented a huge infected gangrenous and prolapsed cervical leiomyoma with extension outward and inward the uterine cavity. We believe that this is the biggest prolapsed cervical leiomyoma reported in the literature. In addition to surgical therapy, interventional radiology techniques for the treatment of cervical leiomyomas have reported promising but still limited results. These techniques can be considered in women with a desire to preserve the uterus or who have contraindications to surgery [4], [15].

4. Conclusion

Cervical leiomyoma is a rare entity that can cause debilitating symptoms in young women. If prolapsed into the vagina, it can cause necrosis. It has various manifestations leading to diagnostic and therapeutic difficulties. Good anatomical and clinical judgment is essential for successful management.

Consent

Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.

Provenance and peer review

Not commissioned, externally peer-reviewed.

Ethical approval

This study was exempt from ethical approval in our institution.

Funding

This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

Guarantor

Ines Zemni and Marwa Aloui.

Research registration number

Not applicable.

CRediT authorship contribution statement

Ines Zemni: Conceptualization, Writing – original draft, Data analysis, Writing – review & editing, Visualization. Marwa Aloui: Conceptualization, Writing – original draft, Data analysis, Writing – review & editing. Fatma Saadallah: Writing – review & editing. Houyem Mansouri: Writing – original draft, Data analysis. Riadh Chargui: Conceptualization, Writing – review & editing, Supervision. Tarek Ben Dhieb: Conceptualization, Writing – review & editing, Supervision.

Declaration of competing interest

The authors declare they have no conflicts of interest.

Availability of data and materials

Not applicable.