Nearly-complete labial adhesions diagnosed with repetitive cystitis in postmenopausal women: A case report

Abstract

BACKGROUND

Nearly-complete labial adhesions diagnosed with repetitive cystitis in postmenopausal women: A case report

CASE SUMMARY

The case of an 83-year-old woman who presented with dysuria, urination disorders, recurrent cystitis, and bacteriuria and was admitted to a private hospital after 1 mo of antibiotic treatment without improvement of her symptoms. Upon examination, labial adhesions were observed with nearly-complete labial fusion with a pinpoint opening. Bacteriuria was detected in urine analysis, and the urine culture test was positive for Escherichia coli. Therefore, a parenteral antibiotic (Fosfomycin) and topical estrogen cream were administered. However, since the adhesion did not separate after 2 wk of treatment, surgical correction was performed. First, adhesiolysis was conducted with a blunt instrument. Then, hysteroscopy and cystoscopy were performed. Hysteroscopic findings showed no abnormalities of the endometrium and endocervix, and the cystoscopic results were also normal. Finally, labiaplasty was completed to prevent adhesion recurrence. One month after the surgery, the discomfort while urinating was eliminated and the adhesion did not recur.

CONCLUSION

Labial adhesions in postmenopausal women cannot be successfully treated with estrogen creams, and surgical treatment should be considered.

Key Words: Post menopause; Genitalia, female; Cystitis; Gynecologic Surgical Procedures; Atrophy; Urinary Retention; Case report

Core Tip: Labial adhesions have a prevalence of approximately 1.8% in infants aged 13-23 mo, however, but they are rarely found in adult women, especially in women of reproductive age. Few cases have been reported in postmenopausal women. Labial adhesion is caused by various inflammatory diseases and estrogen deficiency. The beginning of adhesion occurs most frequently around the clitoris, and depending on the extent of the adhesion, it is classified as complete or partial type. Our case represents a therapeutic example of complete labial adhesion with a pinpoint-sized opening in a postmenopausal woman.

INTRODUCTION

Labial adhesions have a prevalence of approximately 1.8% in infants aged 13-23 mo[1,2], however, they are rarely found in adult women, especially in women of reproductive age. Few cases have been reported in postmenopausal women. Labial adhesion is caused by various inflammatory diseases[3,4] and estrogen deficiency[5-7]. The beginning of adhesion occurs most frequently around the clitoris and depending on the extent of the adhesion, it is classified as type[1,8]. Women who are not sexually active are difficult to diagnose because of the absence of symptoms when the labia adhere. In most cases, such as in the case of children, a gynecologist is required. In addition, unlike in children, the application of estrogen cream is less effective in women, and they can only be treated by surgery; when there is adhesion to the urethra, there is a risk of urethral injury, which requires caution. Furthermore, when only the adhesions are removed, the surgical procedure carries a risk of recurrence, necessitating appropriate plastic surgery. Our case represents a therapeutic example of complete labial adhesion with a pinpoint-sized opening in a postmenopausal woman.

CASE PRESENTATION

Chief complaints

An 83-year-old woman was referred to our hospital with a complaint of urinary symptoms. She complained of dysuria and voiding difficulty, and the urinalysis did not improve despite three weeks of antibiotic treatment at a private hospital. She complained of no other gynecological symptoms besides urinary discomfort.

History of present illness

She presented with dysuria, urination disorders, and recurrent cystitis.

History of past illness

The patient presented with urinary symptoms and bacteriuria and was admitted to a private hospital after one month of antibiotic treatment without improvement of her symptoms.

Personal and family history

Her parity was P 6-0-2-4 with normal spontaneous delivery, and her medical history included hypertension and asthma.

Physical examination

The vulva was fully fused with a unique pinpoint-sized opening (Figure 1A).

Figure 1 Near- total fused vulva examined in an outpatient setting before surgery and intraoperative hysteroscopy and cystoscopy and immediate postoperative appearance. A: Near-total fused clitoris and labia majora; B: Hysteroscopic finding: Endocervix and endometrium were clear; C: Cystoscopy finding; there was no urethral adhesion and stenosis; D: Labiaplasty was done to prevent a recurrence.

Laboratory examinations

Bacteriuria was detected in urine analysis, and the urine culture test was positive for E. coli. Therefore, a parenteral antibiotic (Fosfomycin) and topical estrogen cream were administered. However, since the adhesion did not separate after 2 wk of treatment, surgery was indicated. Urine and blood tests were performed before surgery and showed normal results (Tables 1 and 2).

Table 1 Laboratory exam.

Hb	12.7 g/dL
AST/ALT	15/12 IU/L
BUN/Creatinine	14.55/0.94 mg/dL
eGFR	56.9 mL/min

AST: Aspartate transaminase; ALT: Alanine transaminase.

Table 2 Urine analysis.

	Initial	After antibiotics treatment with fosfomycin
UA	Pyuria/Many WBC	Clear WBC 0-1
Urine culture	E. coli	Negative

UA: Ursolic acid; WBC: White blood cell.

FINAL DIAGNOSIS

Combined with the patient’s medical history, the final diagnosis was nearly-complete labial adhesions with a pinpoint opening.

TREATMENT

The surgery was performed under spinal anesthesia, and it was completed in 30 minutes. First, adhesiolysis was conducted with a blunt instrument. Then, hysteroscopy and cystoscopy were performed. Hysteroscopic findings showed no abnormalities of the endometrium and endocervix (Figure 1B), and the cystoscopic results were also normal (Figure 1C). Finally, labiaplasty was completed to prevent adhesion recurrence (Figure 1D).

OUTCOME AND FOLLOW-UP

One month after the surgery, the discomfort while urinating was eliminated, and the adhesion did not recur. The patient will be followed up later.

DISCUSSION

Labial adhesion is a rare condition in postmenopausal women, and its prevalence in South Korea is unknown. Few cases of labial adhesion in older women have been reported. In China, Laih et al[6] reported one case. In Japan, there are three cases, each reported by Griffin et al[9], Olumi et al[10], Imamura et al[5] reported one Italian case, and Tanvir et al[11] reported one in India. No racial differences appeared to influence the prevalence. This is the first report of labial adhesion in an older patient in South Korea. The main symptom in these reports was difficulty in urinating. There was no response with topical estrogen treatment, and all cases required surgery. In patients aged 60-70 years, labial adhesion is thought to result from a genitourinary syndrome due to estrogen deficiency, which could explain this case. Labial adhesions are difficult to detect in elderly women because they are typically not sexually active. The main complaint is urinary disorders, and since urinary tract infections and urinary disorders are very common in the elderly, adhesions in the vulva may not be detected. However, urinary tract infections and urethral stenosis caused by labial adhesions can also occur, so it is important to be aware of this.

The patient in this case presented with urinary tract infection symptoms that did not improve with conventional treatment. Further research is needed to better understand this condition and its relation to the genitourinary syndrome. There are many reports of congenital labial adhesions with a high success rate and a low recurrence rate when treated with the application of estrogen cream[7,13]. Estrogen cream may be necessary to prevent recurrence after surgery, which can also help in the treatment of genitourinary syndrome. In the present case, since the labial adhesion did not improve after 2 wk of topical estrogen cream and antibiotics, surgical correction was performed. First, adhesiolysis was conducted, and afterward, labiaplasty was completed to prevent adhesion recurrence. One month after the surgery, the discomfort while urinating was not present, and the adhesion did not recur. Maeda T et al[13] published a case report in 2021 on the treatment of labial adhesion with a combination of Z- and Y-V plasty. In our case, we molded by designing a similar Z-shape and performed interrupted suture with Vicryl^R 3-0 and continuous suture with Vicryl^R 6-0 (Figure 2A and B).

Figure 2 The appearance of Z plasty during vulvar plastic surgery and how to suture. A: Design to detach the adhesion tissue into a Z shape; B: Detached and sutured incised tissue.

CONCLUSION

Labial adhesions in postmenopausal women cannot be successfully treated with estrogen creams, and surgical treatment is likely to be considered. In addition, it requires plastic surgery aimed at preventing recurrence and reconstruction.