Clinical Manifestations and Treatment of Postmenopausal Labial Adhesion: A Case Series and Literature Review

Introduction

Labial adhesion (LA), also known as labial fusion or agglutination, is a disorder characterized by the complete or partial fusion of the labia minora. This condition typically affects girls before puberty, but it can also rarely be seen in postmenopausal women (1). LA can occur due to congenital anomalies or acquired conditions and is often the result of a combination of estrogen deficiency and chronic inflammation (2, 3). In severe cases of postmenopausal LA, the labia minora completely adhere to each other at the midline, leaving no opening at the introitus. This condition has been associated with pseudo incontinence or vaginal voiding in women (4). LA can be managed simply with topical estrogen, steroids, or manual separation during the prepuberty period. However, there is currently no established standard treatment for postmenopausal women (1). In this article, we review the relevant literature and present the symptoms, treatment, and follow-up course of eight cases of LA in postmenopausal or hypoestrogenic women. To the best of our knowledge, this is the largest case series on LA in the literature.

Case Presentations

Case 1

A 69-year-old woman was referred to our medical center complaining of difficult voiding, postvoid dribbling, and straining. These symptoms had first appeared 6 months prior to admission and had been progressively worsening. Her past medical and surgical history was unremarkable, and she had not engaged in sexual intercourse for the past 10 years. Urine analysis and culture results came back normal. An ultrasound study revealed bilateral renal fullness and a postvoid residual volume (PVR) of 400 cc. The patient had undergone multiple conservative and medical treatment courses, but none had provided a cure. During the physical examination, it was observed that the labia minora were fused together, resulting in a complete LA. The urethral meatus was not visible, but a small opening was found in the midline. A hidden clitoris was also noted. As a result, the patient underwent clitoral release and surgical separation of the labia minora (Figures 1a and 1b).

Case 2

A 59-year-old female was referred to urology clinic complaining of frequency and postvoid dribbling. She was a known case of lichen sclerosus. She was sexually inactive for 5 years. During genital examination, we observed complete LA, a pin-point orifice in the posterior part of the fusion and skin discoloration due to lichen sclerosus. Following surgical intervention, prolonged use of topical estrogen and corticosteroids and intermittent use of a vaginal cone, were advised (Figures 1c and 1d).

Figure 1. a: adhesion of the labia minora and a pin hole opening at introitus, b: clitoral release and eversion of the introital mucosa with separate absorbable sutures, c: severe LA and skin hypopigmentation due to lichen sclerosus, d: same patient after surgical lysis and clitoral release.

Case 3                                                                                                    

A 68-year-old woman presented with difficult voiding, postvoid dribbling, and recurrent urinary tract infections (UTIs). In an ultrasound study, a postvoid residual volume of 200 cc was reported. She was sexually inactive in the past 7 years. She mentioned a history of abdominal hysterectomy due to abnormal uterine bleeding 15 years ago. On physical examination, a complete LA and a pin-point orifice were seen. The patient underwent surgical labia minora separation. Due to severe urethral stricture, cysto-urethroscopy and urethral dilatation were performed at the same session. In cystoscopic evaluation, multiple translucent submucosal cystic lesions on the posterior wall of the bladder were observed. Pathological evaluation confirmed the diagnosis of cystitis cystica (Figures 2a and 2b).

Case 4

A 57-year-old virgin woman was admitted to the emergency department with urinary retention. She mentioned a history of frequency, urgency, and postvoid dribbling for 2 years. Physical examination revealed LA. Emergent surgical intervention for the separation of adhesions and urethral catheterization was performed successfully (Figure 2c).

Figure 2. a: severe LA and a pinpoint orifice, b: cystoscopic view of the same patient showing cystitis cystica, c: complete LA

Case 5

A 62-year-old virgin female, who complained of prolonged dysuria, a burning sensation in the genital area, and spotting, was referred to our clinic. Urine analysis revealed microscopic hematuria, and urine cytology results were negative for malignancy. During the physical examination, we detected partial LA with a fragile, easily bleeding mucosa. Despite treatment with topical estrogen for 12 weeks, there was no improvement, so the patient was scheduled for surgical intervention (Figure 3a).

Case 6

A 56-year-old patient presented with postvoid dribbling. In the physical examination, partial LA was observed. Lab tests and imaging studies were all normal. She had not engaged in sexual activity for the past 3 years. Topical treatment including estrogen and steroids proved ineffective, leading to the decision to manage her condition through surgery (Figure 3b).

Case 7

A 55-year-old female complaining of stress urinary incontinence and postvoid dribbling referred to urology clinic. She reported that a significant amount of urine comes out of her vagina immediately after voiding while standing. She was sexually inactive in the past 11 years. In the physical examination, complete LA was noted. She underwent surgical separation of the LA (Figure 3c).

Case 8

A 42-year-old virgin female patient complaining of vulvar pain, dysuria, intermittency, postvoid dribbling and recurrent UTI was referred to the urology clinic. Urine analysis showed urinary infection, and in ultrasound evaluation, a PVR of 150 cc was reported. She mentioned a history of premature ovarian failure in the past 6 years. In the physical examination, complete LA with a hidden clitoris was revealed (Figure 3d).

Figure 3. a: partial LA with fragile mucosa and an open urethral orifice, b: partial LA with covered urethral orifice, c: complete LA, d: complete LA with a pinhole orifice

Surgical technique and follow-up

In two patients diagnosed with partial LA, the first optional medical treatment option was topical estrogen application with simultaneous gentle traction of the labia minora for 3 months. However, neither of them achieved a complete response, so surgical separation of the labial fusion was performed. For all patients with severe and complete LA, the initial treatment modality was surgical intervention under spinal anesthesia in a lithotomy position. The surgical technique was a combination of blunt and sharp dissection starting from the presumed urinary orifice. The adhesions were usually easily released, and cautery was used for hemostasis if necessary. To prevent early fusion and recurrence, vaginal mucosal eversion in the introitus region was achieved with separate 3-0 absorbable stitches. In cases of hidden clitoris, hoodoplasty or simple release of the clitoris was also considered. Following the surgery, a vaginal tampon and Foley catheter were inserted for one day. All patients were discharged the day after the procedure and advised to take oral antibiotics for 3-5 days. The postoperative course was uneventful. Topical estrogen was prescribed for all patients to be used every day at bedtime for 1 month, every other day for 1 month, and then twice a week for at least one year. Short-term topical corticosteroids, topical emollients, and vaginal cones were also prescribed for selected patients. None of the patients experienced recurrence of LA or any related complications during the 27- month (range: 18-36) follow-up period.

Discussion

LA typically affects prepubertal girls. There are a few case reports on this condition in postmenopausal women (Table 1).The prevalence of this disorder in the elderly is still unclear (1). In infancy, this condition usually occurs as a result of local contamination and irritation caused by diapers. It can also be associated with adrenogenital syndrome and adrenocortical hyperplasia. In the postmenopausal period, LA develops due to low estrogen levels, poor hygiene, eczema, local trauma, recurrent UTIs, and lack of sexual intercourse (2, 3). Vulvar dystrophies, including lichen sclerosus, have also been suggested as underlying condition (3). In our study, all patients were either virgins or had not engaged in sexual activity for a long time.

Table 1. Reported cases of LA in postmenopausal women

One patient had a past medical history positive for lichen sclerosus. Vulvar lichen sclerosus is a chronic inflammatory disease that can affect women of all ages and is managed with topical corticosteroids. Due to an increased risk of malignancy, close follow-up is recommended (4). LA can be asymptomatic or present with vaginal symptoms such as pruritus and vulvodynia. In some cases, it may cause voiding symptoms (5). The severity of urinary symptoms may not necessarily match the severity of LA. In early stages, adhesion is seen in the posterior part of the vaginal introitus. However, in severe cases, the vaginal and urethral orifice are entirely covered by adhesion. If urine fails to exit the vagina freely, it can lead to urinary retention, recurrent UTIs or pseudo incontinence (3). Recurrent UTIs can be both a causative factor and a result of LA (2, 3). One patient in our study suffered from recurrent UTIs and was also diagnosed with cystitis cystica. Similar concordance has been reported in two elderly women (6, 7). In children, separation of the labia can be accelerated with topical application of estrogen, and surgery is rarely needed. However, in adult women surgical treatment is usually necessary for symptomatic and severe cases (5). Nonetheless, a recurrence rate ranging from 14 to 20% has been reported following the surgical separation of the labia in prepubertal girls (8).

In our study, we did not encounter any LA relapse during the mean 27-month (range:18-36) follow- up. Based on our experience, several measures can help reduce the recurrence rate of labial fusion. These include placing separate absorbable sutures at the introitus area, minimizing the use of electrocautery, prolonged treatment with topical estrogens, maintaining regular intercourse, and/or using vaginal cones to dilate the vaginal introitus following surgery.

Conclusion

The increasing number of adults LA case reports highlights the importance of conducting a thorough genital examination in all females with hypoestrogenic state and voiding complaints, especially those who are not sexually active. A surgical procedure and proper prolonged application of topical estrogen would serve as suitable treatments for postmenopausal LA. Maintaining regular sexual intercourse or using vaginal cones in selected patients would help decrease the recurrence rate of labial fusion.

Authors’ Contributions

NMn: Conception and design, critical revision of the manuscript for important intellectual content; FSH: Supervision; ShT: Acquisition of data; MHm: Drafting of the manuscript; AKr: Drafting of the manuscript; NS: Acquisition of data

Acknowledgment

Special thanks to Shahid Beheshti University of Medical Sciences, Tehran, Iran.

Conflict of interest

The authors declare that they have no competing interests.

Funding

No funding was received for this study.

Ethics Statements

Written informed consent was obtained from all participants for the use of data and pictures in the study.

Data Availability

The datasets used during the current studyare available from the corresponding author on reasonable request.

Abbreviations

LA       Labial Adhesion

PVR     Post-void Residual Volume

UTI      Urinary Tract Infection