Martha F. Goetsch

Vulvar vestibulitis: Prevalence and historic features in a general gynecologic practice population

All gynecologic patients seen by the author during a 6-month period were questioned and examined by means of a swab test to determine the prevalence of vulvar vestibulitis and the normal variation in sensitivity of vestibular skin. Of 210 patients, 78 (37%) had some degree of positive testing. A total of 31 patients (15%) were found to fulfill the definition of vulvar vestibulitis. A questionnaire was administered to these patients as well as to seven patients in whom vestibulitis had been previously diagnosed. A total of 50% had always had pain, most since their teenage years. Their history was not suggestive of a cyclic or remittent pattern of symptoms. Those with secondary dyspareunia or resolution of pain were usually either in a post partum phase or had group B streptococcus or human papillomavirus. The two most severe cases of vestibulitis occurred after use of fluoroucil cream. A total of 32% had some female relative with dyspareunia or tampon intolerance, raising the issue of a genetic predisposition.

Simplified surgical revision of the vulvar vestibule for vulvar vestibulitis.

OBJECTIVE The prototype of surgical treatment for vulvar vestibulitis has been the Woodruff vulvoplasty. A simpler surgery could be less morbid, technically easier, and equally effective. STUDY DESIGN Twelve patients underwent vestibular revision, nine with local anesthesia. They were followed up for between 6 months and 6 years. Outcome was judged by ease of healing and relief of tenderness. This was a feasibility study. RESULTS Ten of 12 patients had complete resolution of vestibulitis. Two others had improvement. Other causes of dyspareunia remain in 2 subjects. Issues of patient histories, postoperative healing, and functional outcome are reported. CONCLUSION A simple surgery seems well suited to this problem. Additional causes of dyspareunia need to be recognized preoperatively and clarified. Development of granulation tissue in areas of wound separation can create sites of continued pain. Postoperatively, reflex vaginismus should be expected and needs therapy to complement the surgical treatment.

Histologic and receptor analysis of primary and secondary vestibulodynia and controls: a prospective study

OBJECTIVE The objective of the study was to assess the association between hormone receptor densities, pain nerves, and inflammation in vestibulodynia patients. STUDY DESIGN In a prospective study, tender and nontender biopsies from 10 primary and 10 secondary vestibulodynia patients were compared with biopsies in 4 nontender controls. Hormone receptors were evaluated using immunohistochemistry for estrogen receptor-alpha and -beta, androgen, and progesterone receptors. Inflammation, nerves, and mast cells were assessed histologically. Statistical analysis was by Fishers exact test, analysis of variance, paired Student t test, and Wilcoxon rank test. RESULTS Tender sites from primary vestibulodynia had increased nerve density compared with secondary and control biopsies (P = .01). Tender sites in secondary vestibulodynia had more lymphocytes than tender primary sites and control biopsies (P < .0001). Mast cells were increased in tender sites compared with nontender and controls. There were no differences in hormone receptor expression. CONCLUSION Markers of inflammation differed between primary and secondary vestibulodynia and controls.

A Practical Solution for Dyspareunia in Breast Cancer Survivors: A Randomized Controlled Trial

PURPOSE Dyspareunia is common in breast cancer survivors because of low estrogen. This study explored whether dyspareunia is introital pain, preventable with analgesic liquid. PATIENTS AND METHODS In a randomized, controlled, double-blind trial, estrogen-deficient breast cancer survivors with severe penetrative dyspareunia applied either saline or 4% aqueous lidocaine to the vulvar vestibule for 3 minutes before vaginal penetration. After a 1-month blinded trial of patient-assessed twice-per-week tampon insertion or intercourse, all patients received lidocaine for 2 months in an open-label trial. The primary outcome was patient-related assessment of penetration pain on a scale of zero to 10. Secondary outcomes were sexual distress (Female Sexual Distress Scale), sexual function (Sexual Function Questionnaire), and resumption of intercourse. Comparisons were made with the Mann-Whitney U and Wilcoxon signed rank test with significance set at P < .05. RESULTS In all, 46 patients, screened to exclude those with pelvic muscle and organ pain, uniformly had clinical evidence of severe vulvovaginal atrophy, dyspareunia (median pain score, 8 of 10; interquartile range [IQR], 7 to 9), increased sexual distress scores (median, 30.5; IQR, 23 to 37; abnormal, > 11), and abnormal sexual function. Users of lidocaine reported less pain during intercourse in the blinded phase (median score of 1.0 compared with saline score of 5.3; P = .007). After open-label lidocaine use, 37 (90%) of 41 reported comfortable penetration. Sexual distress decreased (median score, 14; IQR, 3 to 20; P < .001), and sexual function improved in all but one domain. Of 20 prior abstainers from intercourse who completed the study, 17 (85%) had resumed comfortable penetrative intimacy. No partners reported penile numbness. CONCLUSION Breast cancer survivors with menopausal dyspareunia can have comfortable intercourse after applying liquid lidocaine compresses to the vulvar vestibule before penetration.

Differences in Primary Compared With Secondary Vestibulodynia by Immunohistochemistry

OBJECTIVE: To assess whether primary and secondary vestibulodynia represent different pathologic pathways. METHODS: This was an analysis of archived vestibulectomy specimens from 88 premenopausal women with vestibulodynia (2002–2008). Patient records were reviewed to classify the type of vestibulodynia, duration of symptoms, and hormone status. Histologic sections were stained for hematoxylin and eosin to grade inflammation, S100 to highlight nerves, CD117 for mast cells, estrogen receptor &agr;, and progesterone receptor. Differences between primary and secondary vestibulodynia were tested by t tests, chi-square analysis, and linear and logistic regression. RESULTS: Primary vestibulodynia showed significant neural hypertrophy and hyperplasia (P=.02, adjusted odds ratio [OR] 3.01, 95% confidence interval [CI] 1.2–7.6) and increased progesterone receptor nuclear immunostaining (P=.004, adjusted OR 3.94, CI 1.6–9.9) compared with secondary vestibulodynia. Estrogen receptor &agr; expression was also greater in primary vestibulodynia when symptom diagnosis was less than 5 years (P=.004, adjusted OR 5.53 CI 1.71–17.91). CONCLUSION: Primary and secondary vestibulodynia have significantly different histologic features, suggesting that they may have separate mechanistic pathways. Clinically, this may mean the discovery of distinct conditions. LEVEL OF EVIDENCE: II

CD4-positive T-cell recruitment in primary-provoked localized vulvodynia: potential insights into disease triggers.

Objective To better understand the potential disease triggers of neurogenic inflammation in provoked localized vulvodynia (PLV), our objective was to determine whether the types of infiltrating lymphocytes were different in vestibular biopsies from women with primary PLV, secondary PLV, and unaffected controls. Methods Secondary retrospective analysis of archived vestibular biopsies from a series of adult premenopausal women with primary PLV (n = 10), secondary PLV (n = 10), and unaffected controls (n = 4) was performed. All study patients had severe entry dyspareunia for more than 1 year. Subjects were excluded if pregnant, or they had a known infection, or history of generalized vulvodynia. Biopsies were performed during the midfollicular phase. Lymphocyte subtypes were highlighted in histologic sections using antibodies against CD3, CD4, and CD8 and scored as the mean number of T-cell subtypes per high-power field. Flow cytometry was also used to test fresh biopsies from a de novo prospective series of primary PLV (n = 4) and unaffected controls (n = 2). Results Unaffected control biopsies showed more CD8-positive than CD4-positive T cells, similar to previous reports of the gynecologic tract. In contrast, biopsies from women with primary PLV showed significantly more CD4-positive T cells than those from women with secondary PLV and unaffected controls (p = .003). This observation was further supported by flow cytometry. Conclusions CD4-positive T cells are more numerous in vestibular biopsies from premenopausal women with primary PLV. This may be important because subtypes of CD4-positive T cells are specifically recruited by infectious, allergic, or autoimmune triggers. Future studies distinguishing these subtypes may lead to new insights into this common disease.

Unprovoked Vestibular Burning in Late Estrogen-Deprived Menopause: A Case Series

Objective This study aimed to document cases of severe menopausal vulvar burning localized to the vestibule. Materials and Methods Seven postmenopausal women presented to a vulvar clinic between 2007 and 2011 complaining of debilitating constant vulvar burning pain. They were treated according to the vulvar findings. Statistical tools were descriptive. Results The women’s ages ranged from 56 to 79 years (mean age = 67 years). Pain had begun 1 to 4 years before presentation (mean = 1.8 years) and was vestibular. Five had contraindications to estrogen supplements. Only 1 patient was using estrogen; the mean number of years from menopause to onset of burning was16 years (range = 4–27 years). Three patients developed pain during or after aromatase inhibitor therapy for breast cancer. Pelvic floor myalgia was present in 3 patients. Of the patients, 3 improved on systemic estrogen, 3 improved using topical vestibular estrogen therapy, and 1 was managed with reassurance alone. Vestibulodynia regressed in those using estrogen supplementation. One patient noted resolution after localized removal of vestibular mucosa. Conclusions Severe unprovoked vestibulodynia can present as unprovoked generalized pain in late menopause, and topical lidocaine can aid the diagnosis. Constant pain can arise after years of only provoked pain or in association with further lowering of estrogen from antiestrogen therapy for breast cancer. Therapy to the vestibule can provide relief. Lidocaine and local application of estrogen cream to the vestibule are effective therapies, and physical therapy can be important. With encouragement to avoid estrogen during menopause and with the increasing use of aromatase inhibitors for breast cancer, menopausal unprovoked vestibulodynia may be increasing and can be challenging to diagnose and treat.

A solution for dyspareunia in breast cancer survivors: a randomized controlled study

INTRODUCTION: Research has focused on vaginal atrophy as the cause of dyspareunia in postmenopausal women. This study explored whether penetrative pain was prevented after hypoestrogenic patients applied analgesic liquid to the vulvar vestibule. METHODS: In a randomized controlled, double-blind trial, estrogen-deficient breast cancer survivors with severe penetrative dyspareunia applied either saline or 4% aqueous lidocaine to the vulvar vestibule for 3 minutes before vaginal penetration. After a 1-month blinded trial using diary documentation of twice-weekly tampon insertion or intercourse, all patients received lidocaine in an open-label trial for 2 months. The primary outcome was penetration pain (0–10 numeric rating scale). Secondary outcomes were sexual distress (Female Sexual Distress Scale, abnormal greater than 11) and resumption of intercourse. Comparisons were made with the Wilcoxon rank sum and Wilcoxon signed rank test, with significance set at P<.05. RESULTS: Forty-six patients, screened to exclude pelvic muscle and organ pain, uniformly had severe vulvovaginal atrophy, dyspareunia (median pain 8/10, interquartile range 7–9), and elevated sexual distress scores (median 30.5, interquartile 23–37). Users of lidocaine had less intercourse pain in the blinded phase (median score 1.0 compared with saline 5.3, P=.015). After open-label lidocaine use, 37 of 41 (90%) reported comfortable penetration. Sexual distress had decreased (median 14, interquartile range 3–20, P<.001). Of 20 abstainers who completed the study, 17 (85%) had resumed penetrative intimacy. No partners complained of numbness. CONCLUSION: Breast cancer survivors with severe menopausal dyspareunia associated with atrophy can have comfortable intercourse after applying topical liquid lidocaine to the vulvar vestibule before penetration.

Incidence of Bartholin's duct occlusion after superficial localized vestibulectomy

OBJECTIVE The objective of the study was to analyze the incidence of occlusion of Bartholins ducts after the procedure of superficial localized vestibulectomy for severe vulvar vestibulitis. STUDY DESIGN One hundred fifty-five women underwent modified superficial vestibulectomy for severe primary or secondary vestibulitis between 1989 and 2007. Charts were reviewed and data were calculated regarding occlusion of Bartholins ducts. Database software was FileMaker Pro 5 (FileMaker, Inc, Santa Clara, CA). SPSS 16.0 (SPSS, Inc, Chicago, IL) calculated means. RESULTS Fourteen of 155 women (9%) had small blisters noted at a Bartholins duct site after healing. Of these, 6 (43%) noted local symptoms related to sexual arousal. Surgical unroofing was attempted in 8 (62%) and corrected symptoms in all but 1. The subject with remaining symptoms notes deep pain with arousal suggestive of Bartholins adenitis. CONCLUSION The incidence of duct ostium occlusion after superficial modified vestibulectomy was 9%, and only half had symptoms. Methods of surgical treatment of the occlusion are compared.

Low-grade fibromyxoid sarcoma of the vulva: a case report.

Objective The study aimed to describe a case of low-grade fibromyxoid sarcoma arising from the vulva and to discuss the diagnostic challenges, clinical management, and epidemiology of this rare malignancy. Case A 36-year-old woman presented to 3 separate emergency departments with complaints of a painful and slowly enlarging vulvar mass. Eventual gynecologic referral resulted in excision of a 6-cm, noncystic vulvar mass. Pathological diagnosis revealed low-grade fibromyxoid sarcoma. Later, a right radical hemivulvectomy ensured adequate margins, and 2 years later, the patient is free of recurrent and metastatic disease. Conclusions Low-grade fibromyxoid sarcoma is a rare malignancy that may present in the lower genital tract. Definitive diagnosis is essential because low-grade fibromyxoid sarcoma may metastasize many years after diagnosis, thereby requiring indefinite clinical surveillance.