Are virtual consultations suitable for patients with vulval disease? A multicentre audit of outcomes in the COVID-19 pandemic.

Background: During the COVID-19 pandemic, virtual consultation (VC) was used to replace in-person consultations. This raises specific questions when dealing with vulval conditions. Objectives: To assess the feasibility and the efficiency of VC with and without supplementary imaging, in patients with vulval conditions, and to evaluate the images provided as an aid to diagnosis. Methods: This prospective multicentre audit took place in three specialized vulval clinics in London and Paris. Anonymized data on patients' clinical characteristics, consultation characteristics (including the number and quality of any supplementary images provided) and consultation outcomes (diagnostic certainty and physician satisfaction) were collected. Characteristics and outcomes in those with or without supplementary imaging were compared amongst both new and follow-up consultations. Results: A total of 316 VCs were included. In total, 18.7% (n = 59) were new patient consultations and 81.3% (n = 257) were follow-up. Supplementary imaging (photographs and/or video recordings) were provided by 28.5% (n = 90) of the total cohort. Median photographic quality was significantly higher on a five-point Likert-type scale when photographs were taken by a third party as opposed to the patient themselves (4 vs. 3, Mann-Whitney U-test, p < 0.0001). There was no association between the provision of supplementary imaging and diagnostic certainty amongst new patient consultations. However, a higher proportion of follow-up patients who provided supplementary imaging received definitive management decisions (χ 2 test, p < 0.001) and physician satisfaction with these consultations, as measured on a five-point Likert-type scale, was significantly higher (Mann-Whitney U-test, p < 0.0001). Furthermore, median physician satisfaction scores ≥4 were observed in follow-up consultations for candidiasis, lichen simplex/eczema and vulvodynia. Conclusions: Although in-person consultation remains the gold standard of care, VC may have a role in the management of selected patients with vulval disease. It is possible to provide good-quality photographs for clinical assessment, particularly with the help of a third party and follow-up patients with an established, cancer-unrelated diagnosis may be best suited for this consultation modality.

Vulvar pseudotumoral acyclovir-resistant herpes in an HIV-negative, non-immunosuppressed patient: A therapeutic challenge.

BACKGROUND: Vulvar pseudotumoral herpes infections have been reported in HIV-positive patients. A 32-year-old HIV-negative woman presented with a 6-month history of a vulvar pseudotumor that had been unresponsive to oral acyclovir and valacyclovir, as well as topical imiquimod. OBJECTIVE: This study aimed to evaluate the therapeutic efficacy of a multidrug regimen for vulvar pseudotumor herpes infection in an HIV-negative patient. METHODS: Histology revealed multinucleated giant cells, consistent with a herpes infection. The patient's herpes simplex virus type 2 was resistant to acyclovir. Immunomodulatory agents (thalidomide and topical imiquimod) were started. RESULTS: The lesion enlarged after 6 weeks of treatment. Topical cidofovir 1% gel was added. There was gradual decrease in the pseudotumor size. After 7 months, the Pseudotumor had resolved. CONCLUSION: This is the first reported case of vulvar pseudotumoral herpes in an immunocompetent, HIV-negative patient. Oral thalidomide, in association with topical imiquimod and topical cidofovir, was effective in treating acyclovir-resistant pseudotumoral herpes of the vulva.

The Clinical Role of LASER for Vulvar and Vaginal Treatments in Gynecology and Female Urology: An ICS/ISSVD Best Practice Consensus Document.

In this best practice document, we propose recommendations for the use of LASER for gynecologic and urologic conditions such as vulvovaginal atrophy, urinary incontinence, vulvodynia, and lichen sclerosus based on a thorough literature review. Most of the available studies are limited by their design; for example, they lack a control group, patients are not randomized, follow-up is short term, series are small, LASER is not compared with standard treatments, and most studies are industry sponsored. Because of these limitations, the level of evidence for the use of LASER in the treatment of these conditions remains low and does not allow for definitive recommendations for its use in routine clinical practice. Histological evidence is commonly reported as proof of tissue regeneration after LASER treatment. However, the histological changes noted can also be consistent with reparative changes after a thermal injury rather than necessarily representing regeneration or restoration of function. The use of LASER in women with vulvodynia or lichen sclerosus should not be recommended in routine clinical practice. There is no biological plausibility or safety data on its use on this population of women. The available clinical studies do not present convincing data regarding the efficacy of LASER for the treatment of vaginal atrophy or urinary incontinence. Also, although short-term complications seem to be uncommon, data concerning long-term outcomes are lacking. Therefore, at this point, LASER is not recommended for routine treatment of the aforementioned conditions unless part of well-designed clinical trials or with special arrangements for clinical governance, consent, and audit.

The clinical role of LASER for vulvar and vaginal treatments in gynecology and female urology: An ICS/ISSVD best practice consensus document.

BACKGROUND: The clinical role of LASER for vulvar and vaginal treatments in gynecology and female urology is controversial. AIMS: In this best practice document, we propose recommendations for the use of LASER for gynecologic and urologic conditions such as vulvovaginal atrophy, urinary incontinence, vulvodynia, and lichen sclerosus based on a thorough literature review. MATERIALS & METHODS: This project was developed between January and September 2018. The development of this document followed the ICS White Paper Standard Operating Procedures. RESULTS: Most of the available studies are limited by their design; for example they lack a control group, patients are not randomized, follow up is short term, series are small, LASER is not compared with standard treatments, and studies are industry sponsored. Due to these limitations, the level of evidence for the use of LASER in the treatment of these conditions remains low and does not allow for definitive recommendations for its use in routine clinical practice. Histological evidence is commonly reported as proof of tissue regeneration following LASER treatment. However, the histological changes noted can also be consistent with reparative changes after a thermal injury rather than necessarily representing regeneration or restoration of function. The use of LASER in women with vulvodynia or lichen sclerosus should not be recommended in routine clinical practice. There is no biological plausibility or safety data on its use on this population of women. DISCUSSION: The available clinical studies do not present convincing data regarding the efficacy of LASER for the treatment of vaginal atrophy or urinary incontinence. Also, while short-term complications seem to be uncommon, data concerning long-term outcomes are lacking. CONCLUSION: At this point, LASER is not recommended for routine treatment of the aforementioned conditions unless part of well-designed clinical trials or with special arrangements for clinical governance, consent, and audit.

Epidemiological and economic burden of potentially HPV-related cancers in France.

Human papillomaviruses (HPV) infection is now known to be responsible for almost all cervical cancers, and for a substantial fraction of Head and Neck cancers (HNCs). However, comprehensive epidemiological and economic data is lacking in France, especially for rarer potentially HPV-related cancers, which include anal, vulvar and vaginal cancers. Using the national comprehensive database of French public and private hospital information (PMSI), we assessed prevalence and incidence of patients with in-hospital diagnosis for potentially HPV-related cancers in 2013, and estimated costs related to their management over a 3-year period after diagnosis in France. Concerning female genital cancers, 7,597, 1,491 and 748 women were hospitalized for cervical, vulvar and vaginal cancer in 2013, respectively, with 3,120, 522 and 323 of them being new cases. A total of 4,153 patients were hospitalized for anal cancer in 2013, including 1,661 new cases. For HNCs, 8,794 and 14,730 patients were hospitalized for oral and oropharyngeal cancer in 2013, respectively; 3,619 and 6,808 were new cases. Within the 3 years after cancer diagnosis, the average cost of hospital care per patient varied from €28 K for anal cancer to €41 K for oral cancer. Most expenditures were related to hospital care, before outpatient care and disability allowance; they were concentrated in the first year of care. The total economic burden associated with HPV-potentially related cancers was about €511 M for the French National Health Insurance over a 3 years period (2011 to 2013), ranging from €8 M for vaginal cancer to €222 M for oropharyngeal cancer. This study reported the most up-to-date epidemiological and economic data on potentially HPV-related cancers in France. These results may be used to evaluate the potential impact of new preventive strategies, namely the generalized organized screening of cervical cancer and the nine-valent HPV vaccine, indicated in the prevention of cervical, vaginal, vulvar and anal cancers.

International Society for the Study of Vulvovaginal Disease Recommendations Regarding Female Cosmetic Genital Surgery.

Female genital cosmetic surgeries (FGCSs) and procedures are increasingly being advertised as common, simple, and complication-free, capable of not only improving aesthetic appearance but also increasing self-esteem and sexual pleasure.Guidelines for physicians and clear, scientifically correct information for patients must be made available, to minimize the number of ineffective or deleterious procedures.The International Society for the Study of Vulvovaginal Disease positions/recommendations regarding FGCS are as follows:1. There is a wide variation regarding genital normalcy; providers must be able to explain this to women.2. There are no data supporting FGCS including, G-spot augmentation, hymenoplasty, vulvar and perianal bleaching/whitening, vaginal tightening procedures, and other procedures aimed at increasing sexual function.3. Women should not be offered FGCS before the age of 18 years.4. Women undergoing FGCS should be evaluated by a provider with expertise in vulvovaginal diseases, including attention to their psychological, social, and sexual context. Evaluation by an experienced mental health provider should be considered when the motivation for seeking surgery and/or expectations are not clear or realistic.5. Female genital cosmetic surgery is not exempt from complications.6. Informed consent must always be obtained.7. Surgeons performing FGCS should refrain from solicitous advertising or promoting procedures without scientific basis, including on Web sites.8. Surgeons should not perform surgery that they do not agree with and explain their rationale/position when pressured by patients.9. The genital surgeon must be adequately trained in performing FGCS including knowledge of the anatomy, physiology and pathophysiology of the vulva, vagina and adjacent organs.

Vulvar hidradenoma papilliferum (HP) is located on the sites of mammary-like anogenital glands (MLAGs): Analysis of the photographs of 52 tumors.

BACKGROUND: Hidradenoma papilliferum (HP) is a benign tumor that primarily affects the anogenital area of adult women. Previously considered apocrine tumors, anogenital HP tumors are now interpreted as adenomas of mammary-like anogenital glands based on their histologic features. OBJECTIVE: This clinical study was undertaken to determine whether vulvar HP is located on mammary-like anogenital gland sites and to describe its morphologic features. METHODS: The clinical photographs of 52 histologically confirmed vulvar HP provided by 7 vulva specialists were analyzed. RESULTS: In all, 90.4% of the HP were located on the interlabial sulcus, adjacent zone, or the perineum. These tumors were polymorphous in terms of number (1 or multiple), size (<1-4.5 cm), color (pink, red, blue), surface (ulcerated or not), and architecture (unilobular or multilobular). LIMITATIONS: Eight histologic reports could not be reviewed by the authors but the contributors confirmed that the photographs sent were only those of histologically confirmed HPs. CONCLUSIONS: Vulvar HP is mainly located on mammary-like anogenital gland sites, thereby providing further evidence to their histogenesis. Although a nonulcerated or ulcerated tumor of the interlabial sulcus should evoke a HP diagnosis, the latter must be confirmed histologically.

Vulvar dermatosis.

Vulvar dermatoses are inflammatory conditions responsible for chronic or recurrent itching and soreness. The lesions are either circumscribed to the vulva or associated with extragenital localizations which may help to assess the diagnosis. They should be differentiated from infectious or neoplastic diseases which may have clinical similarities. As opposed to the majority of all dermatoses that have a benign and regular course, lichen sclerosus or lichen planus could exceptionally foster the occurrence of an epithelial cancer precursor which may evolve to squamous cell carcinoma. Topical corticosteroids are the mainstay treatment of vulvar dermatosis. We do not know if the treatment of vulvar lichen sclerosus and vulvar lichen planus prevents squamous cell carcinoma.

Unilateral vulval swelling in cyclists: a report of 8 cases.

OBJECTIVE: To define the clinical features, imaging results, histopathologic patterns, and clinical outcomes seen in patients who develop vulval swelling as a result of intensive cycling. METHODS: The case notes of 8 female cyclists were retrospectively reviewed. RESULTS: The mean age of the patients was 45 years, and all were cycling long distances each week (range, 125-450 km; median, 210 km). All patients had a unilateral swelling of the labium majus. Five patients had magnetic resonance imaging showing asymmetry of the vulva with no enhanced signaling associated with inflammation. Histologic examination in 3 cases revealed dermal fibrosis and dermal edema associated with dilatation of lymphatic vessels. In 1 case, a perifollicular inflammatory infiltrate containing epithelioid granulomas was seen but Crohn's disease was excluded. In another case, fibromuscular hyperplasia was seen. Six patients had surgical excision of the swollen area, and 4 patients were able to resume cycling without problems up to 5 years of follow-up. The cosmetic appearance was initially considered satisfactory by 5 patients. However, 2 patients required a second surgical procedure to further improve the cosmetic appearance. In 1 case, there was a postoperative hematoma followed by an immediate recurrence of the swelling, which persisted. CONCLUSIONS: Unilateral swelling of the labium majus occurring in cyclists can be related to dermal fibroedema associated with lymphatic dilatation or to an increase in adipose tissue. Surgical results were satisfactory in 5 of the 6 patients who underwent excision.

Anti-HPV16 E2 protein T-cell responses and viral control in women with usual vulvar intraepithelial neoplasia and their healthy partners.

T-cell responses (proliferation, intracellular cytokine synthesis and IFNγ ELISPOT) against human papillomavirus 16 (HPV16) E2 peptides were tested during 18 months in a longitudinal study in eight women presenting with HPV16-related usual vulvar intraepithelial neoplasia (VIN) and their healthy male partners. In six women, anti-E2 proliferative responses and cytokine production (single IFNγ and/or dual IFNγ/IL2 and/or single IL2) by CD4+ T lymphocytes became detectable after treating and healing of the usual VIN. In the women presenting with persistent lesions despite therapy, no proliferation was observed. Anti-E2 proliferative responses were also observed with dual IFNγ/IL2 production by CD4+ T-cells in six male partners who did not exhibit any genital HPV-related diseases. Ex vivo IFNγ ELISPOT showed numerous effector T-cells producing IFNγ after stimulation by a dominant E2 peptide in all men and women. Since the E2 protein is absent from the viral particles but is required for viral DNA replication, these results suggest a recent infection with replicative HPV16 in male partners. The presence of polyfunctional anti-E2 T-cell responses in the blood of asymptomatic men unambiguously establishes HPV infection even without detectable lesions. These results, despite the small size of the studied group, provide an argument in favor of prophylactic HPV vaccination of young men in order to prevent HPV16 infection and viral transmission from men to women.

Incomplete efficacy of 5-aminolevulinic acid (5 ALA) photodynamic therapy in the treatment of widespread extramammary Paget's disease.

Photodynamic therapy (PDT) using 5-aminolevulinic acid (5-ALA) is an effective treatment for several conditions such as Bowen's disease, subsets of basal cell carcinomas and actinic keratosis. Surgical resection is the first-choice therapy for extramammary Paget's disease (EMPD), but extensive resection is highly invasive and recurrences are frequent. We report two cases of genital EMPD treated by PDT with partial efficacy. The first patient, a 78-year-old male, suffered from pubic and scrotal Paget's disease for 6 years despite numerous treatments. The second patient, a 78-year-old female, had vulvar involvement for 2 years that was resistant to multiple treatments. The disease was recurrent and chronic with important pruritus and significant impact on the quality of life. Methyl 5-aminolevulinate was applied for 3 h, and irradiation was applied with red light (630 nm) using a total light dose of 37 J/cm(2) for a period of 10 min. The patients were treated every 2 to 4 weeks for a total of at least three treatments. Both patients experienced a partial transient reduction in their symptoms. One patient had a partial transient remission (< 50% reduction of the involved surface), whereas in the other patient, PDT failed to reduce the surface area of the lesions.

Vulvar verruciform xanthoma: ten cases associated with lichen sclerosus, lichen planus, or other conditions.

BACKGROUND: Verruciform xanthoma (VX) is a rare benign tumor that usually involves the oral cavity. Since the first report of this tumor in 1971, only 9 cases have been reported on the vulva, and 3 of these were associated with another vulvar condition. We describe the clinicopathologic features of 10 patients with vulvar VX and focus on their associated conditions. OBSERVATION: The mean age of the patients was 68 years (range, 51-80 years). The VX lesions were asymptomatic, yellowish-orange verrucous plaques. The diagnosis was clinically suspected in 2 cases; other suggested diagnoses were condyloma or squamous cell carcinoma. All of the patients had an associated vulvar condition: lichen sclerosus (6 patients), lichen planus (2 patients), Paget disease, or radiodermatitis. Under microscopy, the VX lesions displayed parakeratosis, acanthosis without atypia, and elongated rete ridges. Xanthomatous cells were aggregated in the papillary dermis. CONCLUSIONS: Vulvar VX is a benign tumor with misleading clinical features. All 10 cases were associated with a vulvar condition, mainly a lichen sclerosus. Therefore, VX might represent a reaction pattern induced by different conditions, mainly characterized by damage to the dermoepidermal junction. When confronted with the diagnosis of vulvar VX, clinicians may look for an associated vulvar condition.

The vulvodynia guideline.

OBJECTIVE: To provide a review of the literature and make known expert opinion regarding the treatment of vulvodynia. MATERIALS AND METHODS: Experts reviewed the existing literature to provide new definitions for vulvar pain and to describe treatments for this condition. RESULTS: Vulvodynia has been redefined by the International Society for the Study of Vulvovaginal Disease as vulvar discomfort in the absence of gross anatomic or neurologic findings. Classification is based further on whether the pain is generalized or localized and whether it is provoked, unprovoked, or both. Treatments described include general vulvar care, topical medications, oral medications, injectables, biofeedback and physical therapy, dietary changes with supplementations, acupuncture, hypnotherapy, and surgery. No one treatment is clearly the best for an individual patient. CONCLUSIONS: Vulvodynia has many possible treatments, but very few controlled trials have been performed to verify efficacy of these treatments. Provided are guidelines based largely on expert opinion to assist the patient and practitioner in dealing with this condition.

Spontaneous regression of grade 3 vulvar intraepithelial neoplasia associated with human papillomavirus-16-specific CD4(+) and CD8(+) T-cell responses.

Cell-mediated immunity directed against human papillomavirus 16 (HPV-16) antigens was studied in six patients affected with grade 3 vulvar intraepithelial neoplasia (VIN3, also known as bowenoid papulosis). Five of the patients presented with a chronic and persistent disease that relapsed after destructive treatments. They showed no detectable anti-HPV blood T-cell responses and no T-cell intraepidermal vulvar infiltrate containing both CD4+ and CD8+ lymphocytes. The last patient had a complete clearance of viral lesions, 8 months after disease onset and 2 months after electrocoagulation of <50% of the VIN3 lesions. She showed high frequency anti-E6 and anti-E7 effector blood T cells by ex vivo ELISpot-IFNgamma assay before clinical regression. Immunohistochemical study of her vulvar biopsy revealed a marked dermal infiltrate containing a majority of CD4+ T lymphocytes and an epidermal infiltrate made up of both CD4(+) and CD8(+) T cells. This seems to be the first evidence of an association between spontaneous regression of VIN3 lesions and HPV-specific T-cell responses detectable in the blood. Hence, an increase of HPV-specific effector T lymphocyte responses by vaccine-based therapeutic strategies might be useful to clear the lesions in bowenoid papulosis disease.

Treatment of undifferentiated vulvar intraepithelial neoplasia with 5% imiquimod cream: a prospective study of 12 cases.

OBJECTIVE: To assess the efficacy of 5% imiquimod cream on undifferentiated vulvar intraepithelial neoplasia (VIN), a disease caused by high-risk human papillomavirus. DESIGN: Prospective, uncontrolled study. SETTING: University hospital vulvar clinic. Patients Twelve consecutive patients treated with 5% imiquimod cream for undifferentiated VIN between March 1, 1999, and May 31, 2001. INTERVENTION: Self-application of 5% imiquimod cream, initially 3 times a week, then adjusted according to tolerance, for up to 7 months according to clinical response. MAIN OUTCOME MEASURES: Therapeutic response, clinically assessed by successive photographs and histologically confirmed for complete responders, was scored as complete, partial (> or =50% decrease in lesion size), or failure. Tolerance was evaluated at each visit. RESULTS: A total of 3, 4, and 5 patients achieved complete response, partial response (> or =75% reduction in lesion size for all such cases), and failure, respectively. Mean duration of treatment was 3.6 months (37.3 applications), 5.0 months (50.7 applications), and 3.4 months (25.2 applications) for complete responders, partial responders, and failures, respectively. Follow-up after treatment was 5 to 18, 14 to 32, and 2 to 28 months, respectively, with 1 partial responder lost to long-term follow-up. No patient developed invasive carcinoma. All but 2 patients experienced vulvar discomfort, resulting in treatment withdrawal for 3. Two patients had flulike symptoms. CONCLUSIONS: Imiquimod cream could be a therapeutic option for undifferentiated VIN. Although poorly tolerated, this self-applied treatment could spare patients, either totally or partially, the classic painful and sometimes mutilating treatments of VIN. Controlled, randomized studies are needed to evaluate its efficacy and tolerance.

Perineoplasty for the treatment of introital stenosis related to vulvar lichen sclerosus.

OBJECTIVE: The objective of this study is to assess the usefulness of perineoplasty for introital stenosis related to vulvar lichen sclerosus. STUDY DESIGN: The records of 64 patients who underwent perineoplasty for this indication were reviewed retrospectively. The median age of patients was 49 years, and the median duration of lichen sclerosus was 60 months. Ninety percent of patients complained of dyspareunia. Patient satisfaction with the outcome was assessed by means of a questionnaire. Persistence of dyspareunia and impaired quality of sexual intercourse were considered as treatment failure. Risk factors of failure that were evaluated included duration of lichen sclerosus, age, previous topical steroid therapy, previous perineotomy, time since surgery, and histologic stage. Statistical analysis was performed by use of Fisher exact test. RESULTS: Of the 64 patients, 12 were lost to follow-up and 2 patients did not respond to the questionnaire. Perineoplasty improved dyspareunia in 45 of the 50 patients (90%) and quality of sexual intercourse in 43 of 50 patients (86%). None of the risk factors evaluated were associated with failure of perineoplasty. CONCLUSION: Perineoplasty provides good functional results for women with introital stenosis related to vulvar lichen sclerosus.