An Unusual Presentation of a Huge Nabothian Cyst of Cervix With Manifestation of Uterine Prolapse: A Case Report.

BACKGROUND: Nabothian cysts are common benign cervical lesions in women of reproductive age. They usually occur due to childbirth or minor trauma; mostly are small and asymptomatic. However, huge nabothian cysts may have variable presentation and can even mimic malignancy thus, biopsy is recommended to rule out malignancy. The unusual presentation of nabothian cyst as uterine prolapse makes it a diagnostic challenge for management of the patient similar to the present case. CASE REPORT: We report a case of a huge nabothian cyst in a perimenopausal, multiparous woman who presented with complaints of something coming out of vagina suddenly after mild heavyweight lifting and pain abdomen. Pelvic examination revealed a second degree uterine prolapse with a large lump protruding outside the vagina. Transvaginal ultrasonography demonstrated a multicystic cervical mass of size 9.5 cm × 8.0 cm arising from the posterior lip of cervix that was protruding through the vaginal canal. After excision of mass, prolapse became first degree. Histopathologic diagnosis was a cervical nabothian cyst. Patient had persisting first degree uterine prolapse along with posterior vaginal wall prolapse on subsequent postoperative follow ups. Hence, vaginal hysterectomy with repair to the vaginal wall was performed later.
CONCLUSION: The present case of nabothian cyst is reported for its unusual presentation as uterine prolapse. The risk factors for pelvic organ prolapse (POP) in the present case are advancing age, multiparity and perimenopausal status. The connective tissue and levator ani muscle became weak and the nabothian cyst unmasked the POP that was latent and asymptomatic by increasing the prolapse from first degree to second degree.

Introduction

A nabothian cyst is a mucus-filled cyst that can be visible as a nodule on the surface of the uterine cervix. It is also called mucinous retention cyst or epithelial cyst. It occurs commonly in reproductive age as a solitary or multiple, translucent to opaque nodule with a rare clinical significance. Nabothian cysts may be identified in 12% of routine pelvic magnetic resonance imaging (MRI) scans.[1] The cysts vary in size from few millimeters to several centimeters.[2] Most of the cysts are free of symptoms because of their small size (2-10 mm in diameter).[3,4] They may occasionally reach about 4 cm growing up to 8 cm that can cause a variety of symptoms and may clinically resemble as uterine tumors.[5]

If diagnosis is uncertain, and cysts are deep seated or large causing bothersome symptoms, excision is required for histopathologic evaluation (HPE) to rule out malignancy.[6,7] Only few cases of nabothain cysts, those presented with uterine prolapse are reported till date. The etiologies of pelvic organ prolapse (POP) are weakness of pelvic muscles, connective tissues, and pelvic fascia. It may even occur due to pelvic masses.[8] There are various risk factors for POP such as multiparity, increased intra-abdominal pressure (eg, bearing heavyweights, exerting physical effort), obesity, advanced age, menopause, past hysterectomies, crouching at labor, prolonged labor.[9,10] POP due to nabothian cyst similar to the present case is a rare condition.[3,11] The present case is a report of 52-year-old perimenopausal, multiparous woman who presented with symptoms of uterine prolapse due to a large nabothian cyst.

Case Report

A 52-year-old perimenopausal, multiparous woman with gravida 4, para 4 came to an Emergency Department at Chitwan Medical College Teaching Hospital (CMC-TH) with complaints of something coming out of vagina suddenly after mild heavyweight lifting and pain abdomen for 4 hours. Her last menstrual period was 18 days back and menstrual bleeding was lighter. She was having irregular and longer cycles since last 1.5 year. She had Pap smear done 4 months back. The report was negative for intraepithelial lesion or malignancy.

No urinary or bowel complaints, chronic cough, constipation, past medical history and family history of malignancy, and any previous cervical surgery. She had all vaginal deliveries. General physical and systemic examination was unremarkable. Abdomen was soft and non-tender on palpation. Pelvic examination revealed a second-degree cervical prolapse with a mass coming out of introitus. Mass was firm to soft in consistency and slightly reducible. Uterus was parous size, mobile, firm, mildly tender, and the fornices were free. Initial differential diagnosis was uterovaginal prolapse and cervical fibroid. Tumors markers such as, cancer antigen 125, cancer antigen 19-9 and carcinoembryonic antigen were within normal limits. Transvaginal ultrasonography demonstrated a multicystic cervical mass of size 9.5 cm × 8.0 cm arising from the posterior lip of cervix that was protruding through the vaginal canal. The differential diagnosis made after transvaginal ultrasonography was nabothian cyst and cervical leiomyoma with multicystic degeneration causing uterine prolapse. The uncertainty of diagnosis still existed after imaging hence; laparoscopic biopsy was performed before definitive surgery to confirm malignancy. Benign lesion (nabothian cyst) was confirmed on biopsy. Thus, excision of the mass was planned. Per operative finding was a polypoid mass arising from the posterior lip of cervix. After excision of mass, prolapse became first degree. The cystic mass was sent for HPE. The patient was informed and counseled about the possibility of requirement of further surgical intervention if the first degree uterine prolapse progresses, and affects her quality of life.

On gross examination the mass was large; measuring 9.5 cm × 8.0 cm. Focal epidermidization (Figure 1) with an ulcer was observed on external surface. Cut section showed multiloculated, cystic spaces of varying size filled with mucinous material (Figure 2). On microscopy, the outer cyst wall was lined by keratinized stratified squamous epithelium (Figure 3) and the inner cyst wall was lined by single layer of ciliated low columnar epithelium without cellular atypia (Figures 3 and 4). Histopathological diagnosis was a huge cervical nabothian cyst. At postoperative follow up after 1 week, 1 month, 3 months, and 6 months; first-degree cervical prolapse was still persisting along with posterior vaginal wall prolapse (coming up into the introitus). Hence, vaginal hysterectomy with repair to the vaginal wall was performed.

Figure 1.

Gross picture of specimen shows a cystic mass with focal epidermidization (arrow).

Figure 2.

Cut section of mass revealed multiloculated, cystic spaces of varying size filled with mucinous material.

Figure 3.

Nabothian cyst; outer cyst wall lined by keratinized stratified squamous epithelium (arrow) [H&E; ×100 magnification].

Figure 4.

Nabothian cyst; inner cyst wall lined by single layer of ciliated low columnar epithelium (arrow) [H&E; ×400 magnification].

Discussion

In chronic cervicitis and its reparative processes, or as part of the physiologic metaplasia, the squamous epithelium of the uterine cervix proliferates and covers the columnar epithelium of the endocervical glands which is thought to play role in the pathogenesis of nabothian cysts.[12] Inflammatory process can block an endocervical gland opening that leads to accumulation of mucosal secretion in small subdermal pockets.[13] The weight of the large nabothian cyst on the cervix might have caused expansion of the vaginal portion of the cervix and it could be the reason for prolapse in the present case.[3,11,14] Nigam et al[3] and Çift et al[11] had reported uterine prolapse in nulliparous women due to large nabothian cyst. However, Ulubay et al[14] had discussed a case of large nabothian cyst in a multiparous woman similar to the present case.

POP mostly occurs in postmenopausal and multiparous women. There are many risk factors for POP such as multiparity, increased intra-abdominal pressure (eg, bearing heavyweights, exerting physical effort), obesity, advanced age, menopause, past hysterectomies, crouching at labor, prolonged labor.[9,10] POP is rare in nulliparous women. The risk factors for POP in the nulliparous include inherent defect in pelvic supports for example Ehler-Danlos syndrome, congenital shortness of vagina, and deep uterovesical and uterorectal pouches.[3] It may also occur due to spina bifida occulta and split pelvis which result in inherent weakness of pelvic floor support. Family history of prolapse also suggests its congenital nature.

Herath et al[15] had reviewed the literature on nonpuerperal uterine inversion (NPUI) by collecting the data of published case reports from 1911 to September 2018. NPUI is a rare gynecological condition with diagnostic and surgical challenges. Leiomyoma is the most common etiology. The other common causes are malignancies and idiopathic. However; POP, fibrosarcoma, epidermoid carcinoma, endometrial sarcoma, carcinosarcoma, rhabdomyosarcoma, endometrial polyp, immature teratoma, and combination of fibroid and a cervical carcinoma are rare causes of NPUI.

Large nabothian cysts may compress a surrounding organ leading to pressure symptoms. When the rectum is compressed, it may cause abnormal defecation and tenesmus.[1] Harou et al[16] had reported a case of primary infertility in a 33-year-old woman due to obstruction of the cervix by multiple nabothain cysts.

Various cervical pathologies should be kept in mind for cystic cervical lesions. The differential diagnoses of a cystic lesion in the cervical stroma are adenoma malignum, deep nabothian cysts, other benign tumors of cervix and well-differentiated adenocarcinoma.[7,12] Benign tumors of cervix are endocervical polyps, leiomyomas, endometriosis, squamous papilloma, microglandular hyperplasia, and mesonephric duct remnants.[7]

According to Yıldız et al[17] transvaginal ultrasonography and MRI are the most useful imaging techniques for cervical cystic lesions. However, huge nabothian cysts must be sent for HPE to exclude other malignant glandular cervical lesions and adenoma malignum (also called minimal deviation adenocarcinoma, a rare entity), the appearance of which mimic deep and large nabothian cysts. These malignant tumors are deeply invading into the cervical stroma and a solid component surrounding or separating multiple cysts are regarded as histological clues to distinguish such malignant tumors from nabothian cysts but exact differentiation is not always easy to establish diagnosis. Hence, it is a big challenge for pathologists to make accurate diagnosis. The high suspicion of gynecologist also gives a clue for diagnosis.[18-21] Thus, preoperative pelvic examination and radiographic imaging are important for huge nabothain cysts to avoid unnecessary hysterectomy.[22] In the present case, transvaginal ultrasonography demonstrated a multicystic cervical mass that was protruding through the vaginal canal. Exclusion of malignancy and excision of the causative benign tumor are necessary before repair or surgery of prolapse. Laparoscopic biopsy was performed before definitive surgery to confirm malignancy in the present case. Nabothian cyst was confirmed on biopsy. Thus, excision of the mass was done.

D’Alessandro et al[23] had reported an interesting case of gastric-type mucinous endocervical adenocarcinoma in presence of nabothian cysts. This tumor has aggressive clinical behavior. They had discussed their possible relationship with the nabothian cysts as the transvaginal ultrasound scan of the patient showed multiple nabothian cysts, associated with an uneven appearance and a plentiful vascularization of the uterus. Many benign and malignant endocervical glandular lesions, showing gastric differentiation unrelated to human papillomavirus (HPV) have been mentioned.[24] Minimal deviation adenocarcinoma and lobular endocervical glandular hyperplasia have a gastric phenotype and immunophenotype.[25]

Nabothian cysts usually do not require any treatment unless the patient becomes symptomatic or when the characteristic of lesion is unclear and malignancy cannot be ruled out. Cryocautery, electrocautery, and excision of cyst/cystectomy can be performed whenever required.[26] Few patients may require hysterectomy to relieve the symptoms. The cases reported by Temur et al,[1] Yıldız et al,[17] and Yelikar et al[26] required hysterectomy to relieve persisting symptoms caused by nabothian cysts due to compression of rectum, chronic pelvic pain, and continuous copious per vaginal thin watery discharge, respectively.

Generally, patients with first degree prolapse are advised to do Kegel’s exercise and do not require surgery. However, women with first degree cervical prolapse should be counseled and reviewed on regular follow ups. In some cases the cervix might have only first-degree prolapse, but vaginal wall prolapse is more than cervical prolapse that cause the symptoms. Thus, such cases require vaginal hysterectomy.[27] Chhabra et al[27] mentioned 2.6% women with first degree cervico-uterine prolapse accompanying varying degrees of anterior/posterior vaginal wall prolapse who underwent vaginal hysterectomy. In the present case, the patient had persisting first-degree cervical prolapse along with posterior vaginal wall prolapse. Hence, vaginal hysterectomy with repair to the vaginal wall was performed.

A case of a spontaneous pregnancy after treatment of obstructive nabothian cyst was reported in the literature.[28] Vural et al[4] had discussed a case of a large nabothian cyst in 38 weeks pregnant woman obstructing labor passage. Simple drainage was performed to allow the spontaneous vaginal delivery and a persisting cystic mass was removed by excision after 2 months of the delivery. Wu et al[22] had performed a simple incision for local drainage to reduce size of giant nabothian cyst in a 45-year old woman. Therefore, simple drainage can manage a huge nabothian cyst in the emergency to improve the outcome.

Conclusion

Huge nabothian cysts may have variable presentation that may cause a diagnostic challenge. We reported a case of nabothian cyst for its unusual presentation as uterine prolapse. The risk factors for POP in the present case are advancing age, multiparity, and perimenopausal status. The connective tissue and levator ani muscle became weak and the nabothian cyst unmasked the POP that was latent and asymptomatic by increasing the prolapse from first degree to second degree.