Abstract
Abstract
Introduction
A number of nonsurgical and surgical treatments have been described in the literature over the past 30 years.4–8 The most permanent and most efficacious results have been achieved by one form or another of surgical excision of the inflamed vestibule.9–11 Modifications of the total vulvectomy procedure have produced less-favorable outcomes.12–14
Prior to diagnosis and focused therapy, many of these patients have had the condition managed inappropriately. These women fall into a category of patients who have chronic vulvar pain and chronic dyspareunia, leading to chronic despair and depression. The inability to engage in sexual consortium inevitably creates marital conflict and stress within the family unit. Although psychologic/psychiatric counseling can never cure an organically based disorder, such counseling can be of value in helping patients cope with the psychosocial problems that can occur with the disorder.
Much of the confusion relative to the more-encompassing diagnosis of vulvodynia—separate from its component portion of vestibulitis—is a condition termed pudendal neuralgia or essential vulvodynia. Although both conditions have a component of neuropathic pain, the differences and treatment strategies are distinct.2,9
This report covers a span of 20 years and a large number of women treated by a single gynecologist.
Materials and Methods
Between 1991 and 2011, 559 women were diagnosed and treated for vulvar vestibulitis syndrome (Table 1). The criteria for establishing a diagnosis included history, physical examination, and limitation of pathologic findings to the confines of the vulvar vestibule. Of the total number of patients treated, 502 had to have a follow-up for a minimum of 12 months to be included in this report.
History
The pertinent historical data included descriptions of burning, stinging pain within the area of the vestibule that was aggravated by urination, tampon insertion, sexual intercourse, tight undergarments, tight jeans, or activities producing pressure within the vestibule (e.g., bicycle riding). The patients commonly reported chronic itching, feelings of rawness, and dryness. The itching, which persisted, was interpreted by the patients as well as by treating gynecologists as consistent with “yeast” infections. Unfortunately, the patients were not cured by antifungal therapy and were subsequently referred to a vulvar specialist with the incorrect diagnosis of “chronic yeast infection.”
Symptoms
Symptoms related to painful intercourse were described as a burning pain at entry as well as during and following coitus. Many patients stated that this pain was worse after intercourse, compared to during the sexual act. Uniformly, sexual relations were described as an unpleasant experiences, which the patients generally opted to avoid. A number of afflicted women gave a history of urinary frequency, urgency, and nocturia coincident with the onset of vestibular pain. The majority of patients had received one form or another of topical medications including corticosteroid creams, local anesthetic gels, antifungals, vitamin A & D ointment, Vaseline, and Desitin cream, as well as systemic antibiotics and antifungals.
Physical findings
The pelvic examination revealed normal external genitalia without tenderness to light touch with a cotton-tipped applicator. The clitoral and periclitoral tissues, perineum, and perianal tissues were likewise normal. The vestibule was exposed and showed redness, which was graded 1–10 on an analogue scale, with 1 representing pink tissue and 10 representing deep-red erythema and vascular ectasia. Similarly, as a cotton-tip applicator was drawn over the area of the Bartholin duct, discomfort was quantified, utilizing a digital scale of 1–10, for which 1 represented minimal pain and 10 represented very severe discomfort, even resulting in withdrawal on the examination table. The paraurethral duct areas were likewise gently drawn across by the applicator and scored. Next, pressure was applied over the Bartholin glands via the applicator and was similarly scored. The posterior fourchette and fossa navicularis were also examined with the cotton-tip applicator as well as the lower vagina. The examination was finished with a vaginal-speculum examination.
Cultures
Samples from each patient in the study group were cultured for aerobic bacteria, mycoplasma, ureaplasma, fungi, chlamydia, and gonorrhea. Fungal cultures were plated directly onto Sabaraud's medium.
Conservative regimen
Every patient was initially treated with a set medical protocol, which included techniques previously reported in the literature as decreasing the patient's level of pain and allowing intercourse. This regimen included prescribing tricyclic antidepressant drugs or gabapentin for treating neuropathic pain. Every woman was placed on a low-oxalate diet and was given a prescription for calcium citrate (either 400 mg t.i.d or 1200 mg as a single daily dose).
Every patient was also given a prescription to receive biofeedback from a physical therapist. The therapist visited the laboratory of H.I. Glazer's, PhD, at Cornell University in New York City to learn his technique directly. 15
All patients were advised to stop intercourse for a minimum period of 6 weeks and to avoid all topical medications.
At the 6-week second visit, patients were asked to attempt intercourse once per week while all other treatments were continued as during first 6 weeks. At the third 6-week visit, patients who had improvement and who could tolerate intercourse continued on the conservative regimen. Patients who showed no improvement were given a detailed explanation—which included a drawing and a scientific paper to take home and study—of a surgical treatment option. Patients who elected to have surgery were then scheduled and seen for a preoperative visit prior to surgery. At the aforesaid visit prior to surgery, the procedure was reviewed again, risks and complications were discussed, and the postoperative course was discussed, including a clear statement that healing would require a full 8 weeks.
Surgery
During the initial period, between the years 1991 and 1994, both simple vestibulectomy and radical Bartholin-gland excision plus vestibulectomy were performed. From 1994 to 2002, Bartholin-gland excision was the operation of choice. From 2002 to 2011, simple vestibulectomy, with paraurethral-duct excision was the operative procedure exclusively performed. Regardless of the time period, vaginal advancement was always a requirement of surgery.
Every surgical procedure was performed utilizing a Zeiss operating microscope and a carbon-dioxide laser coupled to the lens system of the microscope via a micromanipulator. The laser was focused tightly and utilized as a cutting tool only. No electrosurgical device was utilized for hemostasis, and all vessels were suture ligated with 3–0 or 4–0 Vicryl stitches.
Vestibulectomy, excision of paraurethral ducts, and vaginal advancement. 11
After suitable induction of general anesthesia, the labia were sutured back to allow exposure of the vestibule. A 1:100 diluted solution of vasopressin was injected into the vestibular tissue subdermally. The scope of the injection followed a “U-shaped” distribution, extending from the paraurethral ducts to the posterior fourchette on either side. Upon completion of the injection, the carbon-dioxide laser was coupled to the Zeiss microscope, and the beam diameter was focused tightly to 1–1.5 mm. The laser was activated at 12 Watts superpulse, and a cut was made into the vestibule just lateral to the hymenal ring and extended in a U-shaped fashion from the paraurethral duct on one side through the posterior fourchette and back up to the area of the opposite paraurethral duct. The wound was again traced with the laser into deeper tissue planes. Next, utilizing Steven's Tenotomy Scissors, the vestibule was cut to the level of Colles fascia, and the excision was carried from laterally to medially to encompass the lower vagina. When the dissection was completed the vestibule was removed in its entirety with a margin of vagina. Hemostasis was obtained by placing pleating-type sutures of 4–0 Vicryl, beginning on one side at the top of the “U” and finishing at the top of the opposite side. When hemostasis was completed (with 30–40 sutures), the vaginal margins were undermined and advanced outward. The vaginal area was closed to the vestibular remnant by interrupted sutures with 4–0 Vicryl. No tension was placed on the suture line. The goal of the surgery was to remove the vestibule, including the paraurethral ducts, and to reconstruct the tissues to obtain a good cosmetic result, and, at the same time, enlarge the vaginal opening. At the end of the procedure, the vulva and vagina were irrigated with warm saline, dried, and then Silvadene cream was placed over the wound.
Bartholin-gland removal, vestibulectomy, and vaginal advancement. 11
For the gland excision, a more-radical operation was required. The labia were sutured back for exposure and 1:100 vasopressin solution was injected into the vestibule in a manner identical to that performed with simple vestibulectomy. The initial incision with the 12-Watt superpulsed laser beam was made parallel to the lower vagina and just peripheral to the remnants of the hymenal ring. This incision was carried deeply to a depth of 1.5[[inch mark]]] from the surface and was enlarged by spreading mosquito clamps along the incision's length. The laser beam was then moved laterally ∼ 2 cm to the farther, lateral vestibule, which was cut parallel to the first incision. This dissection was carried into the underlying fat of the labium. The bulbocavernosus muscle was now isolated between the two incisions. Beginning at the lowest pole, mosquito clamps were used to grasp the tissue between the incisions and secure the tissue. Utilizing Steven's Scissors the wedge of tissue was cut and suture-ligated with 3–0 Vicryl. Several clampings and cuttings were repeated until the proper depth of excision had been reached. This procedure was repeated for the upper pole of the incision. At this point, the Bartholin gland was identified clinging to the inner aspect of the bulbocavernosus muscle. The gland, with a portion of muscle, was excised and sent to the pathology laboratory. The remaining vestibular skin was removed. The dead space was closed with figure-of-eight, 3–0 Vicryl sutures. The vagina was advanced outward and the skin was closed with an interrupted 4–0 Vicryl suture. The wounds were irrigated with warm saline and Silvadene cream was used to cover the wounds.
Postoperative routine
All patients went into instant-ocean baths 2–3 times daily. All patients applied Silvadene cream to the wounds t.i.d and h.s. If patients were allergic to sulfa, then Cleocin cream was prescribed. All patients took 500 mg of Cipro b.i.d for 14 days.
The first postoperative visit occurred 1 week from the surgical-procedure date. The second visit happened 2 weeks later, the third visit was 4 weeks after that.
At the 6-week postoperative visit, if all the sutures were gone, a small vaginal form was fitted and sent home with the patient with instructions to insert to form for 10 minutes twice daily with relaxation of the pelvic-floor muscles. This was done for 2 weeks, then, the patient returned for an office visit, during which a medium form was fitted. Two weeks later, a large form was fitted, and the patient was advised to restart intercourse 2 weeks from the date the large form was fitted. Subsequently, patients were followed at 6-month intervals.
Biopsies
From 1991 to 1993, patients underwent vestibular biopsies. This practice was stopped after 1993 unless a specific vulvar abnormality was seen that warranted a biopsy (i.e., routine biopsies of the vestibule ceased to happen). An article published last year in the British Journal of Dermatology supports this policy. 16 The latter investigation stated without equivocation that routine vulval biopsy is not recommended and offers little clinical benefit. 16
Ancillary treatments
During the early years of this study, a variety of therapeutic measures were attempted, which included: empirical antifungal therapy; vestibular corticosteroid injections; topical local anesthetics; interferon-α injections, and Botulinum toxin injections. These techniques were unsuccessful in producing lasting resolution for the patients' pain problems and were abandoned.
Evaluation of therapeutic results
It was difficult to measure “pain reduction.” Therefore, the gauge of success in the study was the elimination of all vestibular pain and the ability to have pain-free intercourse. This was the basis for the success of both nonsurgical and surgical treatment strategies.
Pathology
All specimens were submitted for pathologic evaluation.
Results
Conservative (medical) management
Every patient underwent a comprehensive medical management program, as detailed under Materials and Methods, for a minimum period of 4 months. Of the 502 patients available for follow-up, 98 continued the conservative program and had tolerably low pain during intercourse. Objectively, one third of this group continued to have pain that was elicited, albeit at low levels (≤5–6/10), with cotton-tipped applicator touch and pressure over the Bartholin duct area and gland area of the vestibule. Three hundred and five patients reported the ability to have intercourse, which was associated with significant discomfort, while 99 patients continued to have intolerable pain with intercourse, such that the experience was very very unpleasant. Objective findings with cotton tip applicator touch and pressure yielded high levels of discomfort (≥8–10/10) and there were continued observations of erythematous tissue on microscopic (colposcopic) examination. The overall success of conservative therapy was 20%. Any patient who desired to continue the conservative regimen, despite a lack of subjective or objective success was continued in that program.
Biopsies
Approximately 60 vestibular biopsy samples of the vestibule were obtained during the first 3–4 years of the study. The pathology reports were nonspecific. The findings were also repetitious (i.e., describing vascular ectasia [venule and capillary dilatation], chronic lymphocytic infiltration, and, variably, parakeratosis and acanthosis).
Cultures
Every woman in the study had many vaginal and cervical cultures at intake and follow-up. The latter were done only as indicated. The incidence of positive cultures mirrored the nonvestibulitis population of the practice. The most common organisms cultured were mycoplasma/ureaplasma, which were treated with doxycycline at 100 mg b.i.d for 14 days; ß-Streptococcus, which was treated with Augmentin at 500 mg b.i.d for 7 days; and a variety of fungal infections, which were treated with Diflucan at 100 mg b.i.d for 7 days. Candida glabrata infections were treated with Sporanox at 100 mg b.i.d for 30 days. Every treated vaginal infection was recultured following treatment for a test of cure. When indicated, vulvar-skin scrapings were performed with a scalpel, and the scrapings were inoculated directly onto Sabaraud's plates.
Urologic symptomatology
Seventy-five women complained of associated urologic symptoms, which focused on urgency and frequency of urination. Only 18 women actually underwent cystoscopic examination. The cystoscopies revealed normal urinary bladders without any evidence of interstitial cystitis. No potassium chloride tests were performed. The urinary symptoms disappeared following paraurethral-duct incision coincident with vestibulectomy in every case of a patient undergoing surgery, and in every case of a patient responding to conservative management of the vestibulitis.
Surgery
Bartholin-gland excision, vestibulectomy, and vaginal advancement.
Two hundred and thirty-four women underwent vestibulectomy, Bartholin-gland excision, and vaginal advancement. Of this group, 228 women reported pain-free intercourse. In contrast, to the conservative treatment group, objective examination at 2–3 month postoperation revealed absence of any pain elicited by cotton-tip applicator touch or pressure over the vestibular remnant. Only 6 women then underwent excision of the paraurethral ducts, because of subsequent discomfort at these sites. No patient required transfusion because of blood loss. No patient developed a postoperative infection. One patient required readmission for postoperative vulvar edema (Table 2).
All patients had paraurethral-duct excision.
Bilateral vestibulectomy, bilateral excision paraurethral ducts, and vaginal advancement.
One hundred and seventy women underwent “simple vestibulectomy,” paraurethral duct-excision, and vaginal advancement. Of this group, 161 reported pain-free intercourse at the end of the 8-week postoperative convalescence and the 6-week vaginal-form course of therapy. These findings were consistent with lack of discomfort demonstrated on physical examination utilizing a lubricated speculum, a cotton-tipped applicator, and bimanual examination, and having no pain on inserting a large vaginal form into the vagina. Blood loss for this operation was ∼100–200mL. No patient required blood transfusion. No infections were observed. No wound breakdown was seen. Other than women who had traveled from an out-of-town location, patients were sent home the same day they had the surgery. There were no readmissions. All patients returned to the office for a postoperative examination 1 week from the date of surgery. Subsequent visits occurred at 2 weeks, 4 weeks, 6 weeks, and 8 weeks (Table 3).
These patients had cyst formation.
Complications
The most troubling side effect of Bartholin-gland excision was the occurrence of pudendal neuralgia, which was observed in 15% of the patients. This was notable after 3 months postoperation and was unilateral involving the right or left side. The sensation was described as sticking and sharp. Pain was present at the lowest portion of the labium majus or at the ischial tuber at the point where the pudendal nerve emerges from Alcocks canal. The pain was also described as continuous and, on examination, sensitivity to pressure produced similar pain to that which the patient had described. This pain was initially treated by either Neurontin or Elavil. Patients, who did not respond to medications for the neuropathic pain were offered injections of 2 mg of Decadron into each pudendal nerve area at the ischial tuberosity every 2 months. The patients' pain responded to treatment in all but 8 patients within 1 year. The 8 nonresponders required continuation of Decadron for pain control. Because of the neuralgia risk, Bartholin-gland excision was abandoned other than for the most severe cases of vestibulitis in favor of vestibulectomy only. All patients were advised, however, that since the Bartholin duct was effectively removed during vestibulectomy, there was a risk of mucous-cyst development postoperatively, and this would require reoperation and gland removal.
Nine (51%) of the 170 women undergoing vestibulectomy only developed, at variable intervals, postoperative mucous-retention cysts. These cysts were most apparent during sexual stimulation when the patient reported swelling at the prior operative site. On occasion, the swelling produced discomfort. The diagnosis was not difficult to make. The presence of a soft, cystic mass, which was not tender to light touch, but was sensitive to deep pressure, made the diagnosis of retention cyst emanating from a functioning Bartholin gland with a duct that had been excised. The treatment for this condition was excision of the cyst together with the Bartholin gland on the affected side. Clearly, a surgeon who performs a vestibulectomy should be prepared to deal with this complication. Goetsch reported cyst formation in 14 of 155 women who underwent simple vulvectomy (i.e., 9%). 17 As in the current study, these symptoms were related to sexual arousal.
All patients complained of dryness postoperatively. Every patient was advised to use an artificial lubricant for intercourse. The author preferred the more liquid-type of lubrication (e.g., KY Silk or Astroglide).
Discussion
The study reported herein involved nearly 600 women diagnosed with vulvar vestibulitis syndrome (provoked vulvodynia). The basis for success of treatment in contrast to the majority of studies reported in the literature was not simply reduction of pain-related signs and symptoms but rather elimination of vestibular pain.
The true prevalence of this disorder is not known but several reports1–3 have estimated how commonly this problem occurs among the gynecologic population. Two studies from the department of epidemiology, at the University of Medicine and Dentistry of New Jersey, reported, over the course of 1 year, 1 in 20 women may experience new-onset vulvar pain and that vulvar pain lasting at least 6 months has a lifetime prevalence of 9.9%.18,19 A Harvard study estimated the lifetime cumulative incidence at 16%, which would translate to ∼ 14 million women experiencing chronic vulvar pain during their lifetimes. 20
Women—or men for that matter—can deal with acute pain provided they are given analgesic drugs. A short pain cycle terminated by effective treatment uncommonly results in psychologic sequelae. However, persisting chronic pain with no finite termination, particularly when it interferes with normal, physiologic function(s) (e.g., eating, sleeping, sexual activity, excretory function, and ambulation) will lead and to anxiety, depression, and a hopeless outlook on life. The psychologic sequelae are, of course, secondary to the chronic pain. Numerous articles have been published relating to the psychologic treatment of vestibulitis, which even suggest psychotherapy as a means of treating the primary chronic pain.21–25 Of course, psychotherapy is not an effective treatment and can never eliminate the root cause of the pain associated with vulvar vestibulitis. The conservative therapeutic regime applied to all patients in this study utilized a battery of techniques, which included medications directed at neuropathic pain, biofeedback, dietary changes, and restriction of topical medication. Nevertheless, the conservative regime failed to eliminate the vulvar pain in 70%–80% of patients, whereas surgery resulted in elimination of pain in >90% of treated patients. Curiously, a report based on a comprehensive practice of members of the International Society for the study of Vulvovaginal Diseases revealed treatment efficacy as very or somewhat effective for tricyclic antidepressants by 57% of respondents, physical therapy by 81%, and psychologic counseling by 84%, and surgery was rated not effective by 63% of the respondents. 26 Based on the aforesaid data, it is obvious that the poor surgical results reported in the survey could have stemmed from several possible factors: (1) the skill of the surgeon(s) was substandard; (2) patient selection for surgery was imperfect; and/or (3) the operation performed was incorrect.
A variety of nonsurgical treatments for vestibulitis have been reported in the literature. Many of these techniques utilize topical Xylocaine.27,28 The authors of the current report have avoided the use of chronic applications of 2%–5% Xylocaine for a number of reasons. Topical applications of “caine”-type drugs induce hypersensitivity such that a serious allergic reaction may ensue when such a drug is injected for analgesia. The application of a local anesthetic masks the symptoms of the inflammatory reaction and may actually make the condition worse. Topical Xylocaine therapy does nothing to treat the cause of the vestibulitis.
Botulinum toxin injected into the bulbocavernosus muscles was reported to reduce pain caused by vestibulitis. 29 However, a randomized, placebo-controlled double-blinded study of 64 women with vestibulodynia revealed that both Botox and placebo resulted in significant pain reduction at 6 months follow-up. The conclusion of the study was that Botox did not reduce pain, did not improve sexual functioning, or affect quality of life, compared to placebo. 30
A case study reported the salutary effects of sacral neuromodulation. 31 Applications of topical nifedipine, 32 topical nitroglycerin, 33 and cromolyn cream 34 have been reported with variable response. Finally, a recent study combining oral desipramine and topical lidocaine failed to reduce vulvar pain, compared to a placebo. 35
Numerous hypotheses about the origins of vulvar vestibulitis have been proposed, but, to date, none have been shown to be a unifying etiologic factor. Past and recent reports have eliminated the human papilloma virus (HPV) as a factor related to vestibulitis. The earlier work of Bergeron et al. 36 and, more recently, Gaunt et al., documented no evidence of an association for HPV with vulvar vestibulitis. 37 The use of interferon therapy for the treatment of vestibulitis, which was based on the incorrect belief that HPV was a causative factor, was discredited in a previous publication. 2 Nevertheless, this useless and risky therapy continues to be recommended. 38 Recent data defining structural abnormalities in the vulvae of women diagnosed with vulvar vestibulitis attempt to uncover an etiologic factor responsible for the disorder. Tympanidis et al. studied 12 women with a diagnosis of vulvar vestibulitis and compared immunohistochemistry findings with 8 normal controls. These investigators demonstrated a significant increase in area and density of protein-gene product 9.5 neural fibers in vestibulitis patients' papillary dermis, compared to controls. 39 Bohm-Starke et al. utilized laser Doppler perfusion imaging to map the superficial blood flow in the vestibular skin in 20 women with vestibulitis and 21 controls. Significant differences in erythema and perfusion were revealed at the posterior fouchette between vestibulitis patients and controls. 40
Goetsch et al., 41 compared biopsies of tender and nontender sites in primary and secondary vestibulodynia, and also in control patients for histology as well as hormone receptors. Lymphocytes and mast-cell density were greater in tender areas in vulvodynia patients compared, with controls. No differences were observed in steroid-receptor protein expression between the groups. However, androgen receptors were increased in tender sites versus nontender sites in vulvodynia patients. An earlier study with 20 controls found significantly reduced expression of α-estrogen receptors in biopsies of vulvar vestibulitis patients, compared to controls. 42
Tchoudomirova et al. studied vaginal microbiological flora in 1077 women in Sweden. Of this group, 79 patients had symptoms and signs of vestibulitis. As in the current study, no evidence of an infectious etiology was found. 43
Conflicting data have been published about the presence of inflammatory markers within vulvar tissue samples or within blood samples in women diagnosed with vulvodynia.44–48 The data reported in the study showed generally poor results associated with conservative therapy when the endpoint for success was the elimination of pain in addition to a lack of pain recurrence. The success of surgery in eradication of pain and resumption of pain-free sexual intercourse for 9 of 10 patients suggests that this method of treatment should not be withheld or delayed for inordinate periods of time. The advantages of magnification and illumination provided by the operating microscope plus the precision of focused laser beam cutting via a coupled micromanipulator offer clear-cut benefits to patients undergoing vestibulectomy surgery. No significant blood loss occurred in any of the 406 patients in the current study. Patients' wounds completely healed within 6–8 weeks postoperatively. The cosmetic results of vaginal advancement were highly satisfactory with no notable gross scar formation.
Conclusions
The large number of women diagnosed and treated for vulvar vestibulitis syndrome over a 20-year period provided substantial data detailing which therapeutic measures work and which ones do not work. Every woman in this study was given a minimum 3-4 month trial of conservative methodology before considering a surgical option. Likewise >400 pathologic specimens were available for review. The pathology always showed mild chronic inflammation of the lymphocytic variety.
Patients in pain are always relieved by an accurate diagnosis. Similarly, a detailed therapeutic program with specific benchmark expectations will reassure even the most cynical patient. Patients who have chronic pain need and deserve timely progress toward the end goal of pain elimination.
This report has shown that yeast infections are erroneously diagnosed in most women with vulvar vestibulitis syndrome. Other than a support role, psychologic counseling does little, if anything, to advance the definitive treatment of vestibulitis. This report has shown that medical therapy for vestibulitis results in objective pain elimination in a minority of patients.
This report has clearly demonstrated that simple vestibulectomy excision of paraurethral ducts and vaginal advancement results in pain elimination in >90% of treated patients.
Footnotes
Disclosure Statement
No competing financial conflicts exist.
