Symptomatic Giant Skene’s Gland Cyst During Second Trimester Pregnancy and Surgical Excision: A Case Report

Abstract

Introduction: A Skene’s gland cyst is a rare gynecological disorder, and the cause of an adult onset is largely unknown. However, periurethral and perineal cystic lesions are common, causing often indistinguishable symptoms. Therefore, accurate diagnosis is crucial because it can significantly alter clinical management. Methods: A 27-year-old woman was gravida 2 para 1 with the onset of a unique, progressively large vulvar mass that began at approximately 6 weeks of gestation. At 22 weeks of gestation, the mass became more symptomatic and measured approximately 9 cm × 7 cm × 6 cm in size, with some areas of excoriation on the lateral surface. Results: The patient had a pelvic MRI without contrast, and it showed that the mass was most likely a giant Skene’s gland cyst. At 24 weeks, the mass was surgically removed under spinal anesthesia and followed by vulvar reconstruction. Histopathology showed a benign cyst lined by transitional and squamous epithelium cells, which was consistent with a benign Skene’s gland cyst. She recovered fully, continued with the pregnancy and delivered vaginally at 39 weeks and 3 days without complications. Conclusions: A case of excoriating giant Skene’s gland cyst in pregnancy that caused significant discomfort to the patient. To this day, there has been no reported literature of a Skene’s gland cyst this large, especially during pregnancy. Hence, Skene’s gland cysts should be part of the differential diagnosis of a large vulvar mass of this magnitude during pregnancy.

1. Introduction

Skene’s glands are paired paraurethral glands that connect to the anterior vagina at the introitus via its ducts to provide lubrication during orgasm. They form paraurethral cysts that require differentiation from the urethral diverticulum [1]. The Skene’s glands arise from the urogenital sinus and are considered the female homolog to the male prostate [2]. The function of the Skene’s gland is to produce a mucoid secretion after sexual stimulation to protect and lubricate the urethral opening [3]. The glands are located downward and laterally to the urethra. Skene’s gland cysts are occasionally reported in newborns as a congenital abnormality and are rarely reported in adults [4]. The cause of Skene’s gland cyst at birth is unclear, but in adults, it is speculated that obstruction of the ducts may be secondary to infection and inflammation [2]. Skene’s gland cysts are rare at any age, and the incidence varies from 1 in 7000 to 1 in 2000 newborns [5,6].

A Skene’s gland cyst typically presents as an asymptomatic bulging interlabial mass. However, symptoms can include dysuria, frequent urination, dyspareunia, urinary tract infections, and a moveable mass near the opening of the urethra [7]. The diagnosis of Skene’s gland cyst can be made on physical examination alone, but it can be assisted by imaging studies such as pelvic MRI to determine its location and its association with adjacent organs. Differential diagnoses are Gartner’s duct cysts, cysts of the canal of Nuck, imperforate hymen with hydrocolpos, cysts with urethral origin (urethral caruncle, urethral diverticulum), Bartholin’s gland cysts and, rarely, congenital lipoma, epithelial inclusion cysts, urethrocele, endometriosis, ectopic ureterocele and a uterovaginal prolapse [8,9,10,11,12]. Pathology can be confirmed when histology shows transitional or squamous epithelium [13]. There is no clear standard management; however, some methods include needle aspiration, marsupialization, partial excision and total excision [2,14].

In the present case report, we aim to present a case of a female with a large symptomatic Skene’s gland cyst during pregnancy that was associated with significantly progressive worsening symptoms.

2. Patient’s Information

The patient was a 27-year-old gravida 2 para 1 with late prenatal care due to a delay in obtaining medical insurance, and she presented to her primary obstetrician’s office at 21 weeks and 2 days with the onset of a unique, progressively large vulvar mass that began at approximately 6 weeks of gestation (Figure 1). The previous pregnancy was uncomplicated, and she had a spontaneous vaginal delivery at 40 weeks of a female neonate. The patient did not have any significant medical, surgical, social or family history.

Clinical Findings: The patient’s primary obstetrician and gynecologist performed a pelvic exam and ultrasound, and her physician was concerned because of the large size of the mass, which appeared to be obstructing the vaginal introitus and compressing the external urethral meatus (Figure 2 and Figure 3). The fetal anatomy ultrasound performed did not show any evidence of significant fetal anomalies. She was immediately sent to the emergency department for further evaluation, where a pelvic MRI without contrast was performed to assess the origin of the mass and its extent (Figure 4 and Figure 5).

Methods: This work has been reported in line with the SCARE criteria [15]. This case report was exempted from ethical approval by the Texas Tech Health Sciences Center Institutional Review Board (TTUHSC/I RB). However, informed consent of the patient was required, and it was obtained.

Diagnostic Assessment: The MRI found a lesion that abuts and indents the posterior wall of the urethra (Figure 2). It did not arise or connect to the urethra. It was a benign-appearing cystic lesion between the posterior wall of the urethra and the anterior wall of the vagina. The cystic lesion measured approximately 9 cm × 7 cm × 6 cm. It appeared to be a cyst with hemorrhagic or proteinaceous debris with a thin, smooth wall (Figure 2). There was no surrounding inflammation or tissue infiltration.

Timeline: The patient was started on pain medication for symptomatic relief and sent for Maternal-Fetal Medicine (MFM) consultation at 22 weeks and 2 days. At the MFM’s office, a detailed fetal anatomy ultrasound survey was performed, and it showed no significant fetal anomalies, but there were two soft markers: echogenic intracardial focus and fetal echogenic bowels. Past obstetric history involved a spontaneous vaginal delivery at 40 weeks of a female infant in an uncomplicated pregnancy. She reported no exposure to teratogenic substances or history of fetal anomalies in her last pregnancy. The patient had normal prenatal lab tests, low-risk age-appropriate fetal aneuploidy screening with cell-free DNA, negative maternal expanded carrier screening for single gene disorder, including cystic fibrosis, and negative TORCHES infection (Toxoplasmosis, Rubella, Cytomegaly, Herpes and Syphilis) and sexually transmitted infections of Gonorrhea and Chlamydia, and the urine culture was negative. The patient reported that she first noticed the mass soon after conception at approximately 6 weeks of gestation and that it progressively grew larger. It was initially painless, but she started feeling some discomfort and, later, pain at mid-trimester. However, she denied dysuria and an increase in urinary frequency or urgency, and there was no evidence of genital infection. A pelvic exam was performed, a 7–9 cm interlabial mass below the clitoris was identified, and the urethral meatus was located at position 11 o’clock of the mass (Figure 4). The bimanual exam did not show evidence of extension of the mass into the anterior vaginal wall. The patient was briefed about the necessity of excision and the risks under spinal anesthesia.

Therapeutic Intervention: There is no specific unique treatment for Skene’s gland cyst exclusive to pregnant women. However, ancillary intervention with analgesics to relieve pain and swelling and treatment of underlying infections like a urinary tract infection or sexually transmitted infections may be the first approach before surgical interventions. Asymptomatic Skene’s gland cysts outside of pregnancy can be expectantly managed by watch and wait, and it will resolve over time. However, if it is symptomatic, it may require therapeutic fine-needle aspiration, incision and drainage, unroofing the cyst, partial excision and marsupialization. However, surgical excision is a definitive treatment if it does not resolve with other interventions [16]. Surgical excision was chosen for this patient because of the very large size of the cyst, symptomatic pain, clinical presentation at mid-trimester and the potential risk of recurrence of other conservative interventions that might warrant re-operation.

In this patient, the Skene’s gland cyst was very large and symptomatic, which included pain, urinary hesitancy and an inability to attain a good urinary stream, thereby splattering urine on her legs. Hence, she was admitted for excision of the Skene’s gland cyst and vulvar reconstruction at 24 weeks and 3 days (Figure 5). She received 2 g of Cephazolin intravenously prior to the commencement of surgery. A superficial linear vertical incision was made on the left lateral edge of the mass close to the labia minora up to the cyst wall using a size 15 Scalpel. Two Allis forceps were applied to grab the edges of the incision. Using a combination of blunt and sharp dissection with Metzenbaum scissors and Bovie, the cyst wall was carefully dissected off the underlined lateral, medial, inferior and superior vaginal tissues until the base of the cyst’s attachment to the lateral wall of the urethral was reached. The base of the cyst was double-clamped with two Heaney clamps, cut in between the clamps using Metzenbaum scissors and then completely excised. The cyst was removed entirely, and the base of the cyst was transfixed with 0-Vicryl suture. Excessive vaginal tissue was excised, and the cavity was closed using 0-Vicryl in a triple-layers pattern to secure hemostasis. Hemostasis was further secured with a combination of cauterization with Bovie and ligation of bleeding blood vessels using 2-0 Vicryl suture. The vulvar mucosa was closed with 2-0 Vicryl in a running baseball-like technique.

Finally, hemostasis was achieved with the Bovie and using multiple figures-of-eight transverse mattress sutures, and the estimated blood loss was 150 mL. Cystoscopy was not performed because there was an in-dwelling Foley catheter in place, and there was no evidence of entering the urinary bladder or injury to the urethral. The patient was seen two weeks post-operation in the MFM clinic for a follow-up visit, and the vulvar was completely healed. Hence, it took approximately two weeks for the wound to heal. The histopathology of the mass was a benign cyst with the walls lined by transitional and squamous epithelium consistent with a benign Skene’s gland cyst. It was negative for dysplasia and malignancy.

The patient continued to follow up with her primary obstetrician throughout the remainder of the pregnancy, and she was again admitted on 2 January 2024 for induction of labor at 39 weeks and 2 days. Labor was uncomplicated, she received epidural anesthesia, and she eventually had spontaneous vaginal delivery on 3 January 2024 at 39 weeks and 3 days. She delivered a live male infant with a birth weight of 6 lbs 9 oz, Apgar scores were 8 and 9 in 1 and 5 min, respectively, and there was no associated genital laceration.

3. Discussion

Skene’s gland cyst is defined as a sac-like pocket of membranous tissue that contains fluid, air or other substances located in the Skene’s gland. It is diagnosed through a physical exam where a mass is palpated or visualized near the urethra, laterally and inferiorly [2]. Treatment of a Skene’s gland cyst depends on the size of the cyst, the presence of symptoms and other factors. It also depends on the time of diagnosis during pregnancy. In early pregnancy, it may be expedient to observe. The same goes for late pregnancy, where it can be expectantly managed and then undergo surgical management postpartum. Non-invasive management options and/or minimally invasive intervention may be chosen depending on the timing of diagnosis, the size of the mass and the associated symptoms. However, these treatment modalities are fraughted with either persistent mass or recurrence after the initial surgical intervention. Hence, it may be expedient to tackle this problem once and for all with definitive surgical excision. The invasive nature of surgical excision and the potential increased risk of excessive bleeding and scar formation following surgery may limit the desire of the surgeon to undergo this route. However, with meticulous surgical techniques by an experienced surgeon and post-operative ancillary interventions, these potential complications are grossly minimized.

In mid-pregnancy, like in this case, especially when symptomatic, if some general approaches like observation, warm compresses, antibiotics, incision and drainage and marsupialization do not work, surgical removal is appropriate [2,14]. Small asymptomatic cysts do not usually require acute intervention. However, in our case, observation and warm compression did not help to resolve the cyst over time. If the cyst was associated with an infection, antibiotics would have been prescribed. In some cases, more minimally invasive treatments, such as incision and drainage, are administered if it causes significant pain, persistence or recurrence after initial non-surgical treatment. Marsupialization is a surgical procedure where the cyst is opened and sutured to create a permanent opening, and this technique helps to prevent the cyst from recurring. However, it often requires a long period for complete healing, which is usually by secondary intention, and it may heal with a large scar with associated dyspareunia. If the cyst is large, painful or persistent, surgical removal is performed when other conservative measures are not effective or in extenuating circumstances like in this case report [14]. In our patient, surgical excision of the cyst was chosen for definitive management to eliminate room for recurrence that may warrant re-operation.

Skene’s gland cysts are not very common, but they have some key potential associations. The most well-understood one is infection of the Skene’s gland. Bacterial infections may cause inflammation and blockage in the ducts, resulting in the accumulation of fluid and the formation of a cyst [2]. Other associations include inflammation, hormonal changes and trauma [13]. Inflammatory conditions in the genital area may contribute to changes in the glandular tissue and ducts. Although infections of the Skene’s gland, called Skenitis, are rare, infections can arise from sexually transmitted organisms, such as Gonorrhea and Chlamydia, or common urinary tract infection organisms. Hence, antibiotics use for the treatment of sexually transmitted infections and urinary tract infections has been shown to treat Skenitis. Hormonal fluctuations in pregnancy, such as in estrogen, progesterone, human placental lactogen, prolactin and growth hormonal-like hormone, can impact the normal function of the glands and their secretions. Estrogen and growth hormones cause proliferation and swelling of Skene’s glands and epithelial cells, which may cause obstruction of the drainage of glandular secretion, thereby resulting in swelling. While pregnancy itself does not directly cause Skene’s gland cysts, the hormonal changes that occur during pregnancy can affect the pelvic area. The increased pressure in the pelvic region from the growing uterus may contribute to the development of cysts or infections [13]. The hormonal changes during pregnancy and the compression pressure from the gravid uterus may influence recurrence if other minimally invasive surgical procedures are performed. Moreover, in view of the presently discussed cyst’s large size, it could cause labor obstruction. Hence, it is expedient that a large Skene’s gland cyst of this magnitude should be removed entirely in order to minimize risks of labor obstruction or recurrence before the end of the pregnancy that may warrant re-operation.

In our case report, the patient was seen at 21 weeks and 2 days with her second pregnancy when she started manifesting symptoms of great concern to her, and the mass measured approximately 9 cm × 7 cm × 6 cm. Giant Skene’s gland cyst needs to be differentiated from other large vulvar masses in pregnancy, such as angiomyxoma and malignant melanoma. There have been case reports by Xu and Gulino et al. of aggressive angiomyxoma, which has a high propensity of recurrence after surgical excision, unlike Skene’s gland cyst, which does not usually recur if it is completely excised [17,18]. The patient exhibited no signs of mass infiltration to adjacent tissues or infection, ruling out malignancy and infection as potential causes. While Skene’s gland cyst is a benign vulvar mass and rarely causes vulvar and vaginal cancer, there have been cases of vulvar melanoma in pregnancy reported by Mendizabal et al. and Gulino et al., which is a malignant tumor that can metastasize to other parts of the body and can alter perinatal outcomes [19,20]. Due to the pain, discomfort, size of the cyst, occurrence in the mid-trimester of the pregnancy and the potential risk of recurrence if minimal invasive surgical interventions were instituted, it was determined that surgical removal was the best course of action. The entire cyst was removed, and it confirmed the diagnosis of a benign Skene’s gland cyst. The prognosis for Skene’s gland cysts is generally good, especially with prompt and appropriate medical attention. It is essential to consult with a healthcare professional for a proper diagnosis and personalized treatment plan [2].

The strength of this case report is that, to the best of our knowledge, this is the first case report of a very large Skene’s gland cyst in pregnancy. The literature on the subject suggests that Skene’s gland cysts have a link to infection and hormonal fluctuations. This Skene’s gland cyst was symptomatic and very large, and it could have caused an obstruction to the delivery of the fetus because it partially occluded the vaginal introitus. Moreover, during pregnancy, Skene’s gland cyst can be incredibly large due to hormonal sensitivity. Hence, it needed to be completely removed surgically before her delivery, followed by vulvar reconstruction. However, this is a case report, and its outcome cannot be inferred to reflect a trend in the general population with similar vulvar masses. This case report was also limited due to the inability to perform a pelvic MRI with contrast during pregnancy, but it did not interfere with our clinical suspicion of Skene’s gland cyst.

Most of the literature on Skene’s gland cysts are case reports and case series. There have been numerous case reports on newborns’ Skene’s gland cysts, but limited cases in adults and during pregnancy have been reported. Hence, further study should be conducted to explore this rare but unique vulvar mass during pregnancy, its differential diagnosis and management and the impact it has on pregnancy outcomes. The knowledge of this case report is to discuss the unique presentation of Skene’s gland cyst in pregnancy and also highlight various management modalities, including the definitive treatment, which is surgical excision.

4. Conclusions

Adult-onset Skene’s gland cysts are rare and are thought to occur due to infection. In our case report, there was no obvious sign of infection or malignancy. We speculated that the possible cause was due to hormonal fluctuations in pregnancy. The Skene’s gland cyst was excised due to its large size and the potential risks it posed to pregnancy. The patient underwent surgical resection of the cyst, and she subsequently had a successful vaginal delivery. Large vulvar masses of this magnitude in pregnancy should be meticulously and systematically evaluated to rule out other causes, and prompt intervention with complete surgical excision should be carried out like in this case. Although Skene’s gland cysts in pregnancy are rare, a multicenter study to compare various treatment modalities and the timing of intervention is recommended.