Lichen sclerosus is a chronic, progressive dermatologic

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1 A Novel Approach to the Surgical Management of Clitoral Phimosis Jamie Kroft, MD, FRCSC, Michael Shier, MD, FRCSC Department of Obstetrics and Gynecology, Sunnybrook Health Sciences Centre, University of Toronto, Toronto ON Abstract Background: The objective of this case series was to outline a novel method for surgical correction of clitoral phimosis caused by vulvar lichen sclerosus (LS) or lichen planus (LP) and to review the postoperative outcomes. Case Series: We used the CO 2 laser to treat clitoral phimosis in 20 women with LS and three women with LP. All patients underwent individualized preoperative and postoperative topical therapy with steroids or immunomodulators. Five women with LS had mild reagglutination during follow-up but were satisfied with the results, and three required reoperation, with satisfactory results in followup. Two women with LP required reoperation. Conclusion: This novel surgical technique has enabled the treatment of clitoral phimosis secondary to LS or LP, but further studies are required. Medical maintenance therapy postoperatively is a vital component of treatment. Résumé Contexte : Cette série de cas avait pour objectif de décrire une méthode novatrice de correction chirurgicale du phimosis clitoridien causé par le lichen scléreux (LS) ou le lichen plan (LP) vulvaire et d en analyser les issues postopératoires. Série de cas : Nous avons utilisé un laser à CO 2 pour traiter le phimosis clitoridien chez 20 femmes présentant un LS et trois femmes présentant un LP. Toutes les patientes ont reçu, avant et après l opération, un traitement topique personnalisé faisant appel à des stéroïdes ou à des immunomodulateurs. Cinq femmes présentant un LS ont connu une légère réagglutination au cours du suivi, mais ont été satisfaites des résultats; trois réopérations se sont avérées requises et ont donné lieu à des résultats satisfaisants au cours du suivi. Deux femmes présentant un LP ont nécessité une réopération. J Obstet Gynaecol Can 2012;34(5): Key Words: Lichen sclerosus et atrophicus, lichen planus, clitoris, phimosis, gynaecologic surgical procedures Competing Interests: None declared. Received on September 25, 2011 Accepted on November 21, 2011 Conclusion : Cette technique chirurgicale novatrice a permis le traitement du phimosis clitoridien attribuable au LS ou au LP; toutefois, la tenue d autres études s avère requise. La mise en œuvre postopératoire d un traitement d entretien médical constitue une composante cruciale du traitement. INTRODUCTION Lichen sclerosus is a chronic, progressive dermatologic condition most commonly affecting the anogenital epithelium and characterized by marked inflammation and epithelial thinning. Lichen planus is an inflammatory, autoimmune disorder that often affects the oral and vulvovaginal mucosa. 1 Both of these conditions can cause burning, itching, pain, and dyspareunia. As both conditions progress, the vulvar architecture can become distorted, and scar tissue can form between the clitoral prepuce and the glans clitoris, leading to clitoral phimosis. This frequently causes loss of clitoral sensitivity, which may cause anorgasmia and sexual dysfunction. 2 It can also result in the formation of smegmatic pseudocysts, which can become inflamed or infected. 3 The mainstay of treatment for both LS and LP is the use of superpotent topical corticosteroids, 4,5 which treat the symptoms and prevent further architectural changes. However, if clitoral phimosis has developed, this will often need to be surgically corrected. Historically, surgical correction was not performed because of the fear of further scarring. 6 The use of corticosteroids postoperatively has allowed good results surgically for treatment of LS, with future scarring prevented with maintenance therapy. Two small retrospective series of surgical correction of clitoral phimosis caused by LS have been published, each describing promising results. 1,7 Most descriptions of surgical management of LP focus on correction of vaginal involvement since, unlike LS, erosive LP involves the vagina in up to 70% of patients. 5 Although surgery MAY JOGC MAI

2 is not recommended for active LP, it may be necessary to help restore a woman s sexual function when there is severe scarring of the vulva or vagina once the disease is well controlled. 5 The CO 2 laser has multiple applications in gynaecologic surgery. Because CO 2 laser energy is almost completely absorbed by a thin layer of water and human cells have a high water content, deep penetration into tissue is minimal. This makes the CO 2 laser an ideal tool because it can dissect tissue very accurately in small increments. 8 We therefore began using this instrument to perform surgical correction of clitoral phimosis caused by LS or LP, and had satisfactory results. This is a new application of the CO 2 laser, and has allowed us to perform fine dissection of the scar tissue without damaging the underlying clitoris. We outline here the method for using this novel approach to surgical correction of clitoral phimosis caused by LS or LP. We also describe the outcomes of all patients with LS or LP and clitoral phimosis who have had surgical management using the CO 2 laser. CASE SERIES Surgical Technique Scar tissue over the clitoris is excised using the UltraPulse mode on the CO 2 laser at 6 watts and 200 millijoules per pulse, using glass rods to protect the clitoris during dissection. Once the clitoris is fully exposed, the periclitoral skin is sutured around the clitoris to keep it exposed with 4 0 polysorb sutures. The surgical practice in our centre has changed to include suturing of the periclitoral skin, as this has provided subjectively improved results. Patients are treated postoperatively with individualized maintenance steroids or immunomodulators to prevent further phimosis, as well as local estrogen, progesterone, testosterone, and/or mechanical dilators as needed (Table 1). Postoperative Results This approach has been used in our centre for over 10 years with follow-up ranging from three months to 10 years. Twenty women with LS and three with LP have been treated (Table 2). All women with LP had both oral and vulvar involvement. Two of the women with LS had previous treatment for clitoral abscesses secondary to phimosis. All patients who underwent surgical management ABBREVIATIONS LP lichen planus LS lichen sclerosus failed prior individualized vulvar medical treatment. Medical management before surgery consisted of highpotency corticosteroids or immunomodulators, and some patients also had topical progesterone, estrogen, and/or testosterone as needed. Of the 20 women with LS in this series, three had a mild amount of reagglutination, still present at their most recent follow-up. Two of these patients are still satisfied with their results despite mild reagglutination. The other did not use local estrogen despite our recommendations; this may have contributed to the further anatomical changes and her ongoing symptoms. Two women with LS experienced reagglutination during the follow-up period, but this resolved with medical treatment. Both of these patients had normal-appearing anatomy without agglutination at their most recent follow-up and were satisfied with the results. Only three women with LS have required reoperation. Outcomes and individualized postoperative treatment for those who experienced reagglutination and/or reoperation are summarized in Table 3. Two women with LP had recurrence of agglutination. Both patients had resection of inclusion cysts and suturing of the periclitoral skin. Neither patient had recurrence of agglutination at the most recent follow-up, and both patients were satisfied with their results. The outcomes and individualized postoperative treatment for those who experienced reagglutination and/or reoperation are summarized in Table 3. One woman with LS developed a wound infection following the surgical treatment and was treated with a seven-day course of clindamycin, after which she had satisfactory results without reagglutination. In follow-up, all patients reported subjectively improved psychosexual well-being and reduced vulvar symptoms. Examples of clitoral phimosis prior to surgery and outcomes for women with LS and LP after surgical management are shown in Figures 1 3. DISCUSSION This case series demonstrates the promising results of this novel surgical procedure, which uses the CO 2 laser for a new gynaecologic application with a low complication rate. Although there was recurrence in a total of eight out of 20 women with LS, by the end of follow-up only three patients had some degree of agglutination; two of these 466 MAY JOGC MAI 2012

3 A Novel Approach to the Surgical Management of Clitoral Phimosis Table 1. Medical management used postoperatively Regimen number Treatment regimen 1 Clobetasol 0.5% BID once weekly with topical progesterone 33% in aquafor base BID all other days* 2 Clobetasol 0.5% BID twice weekly with topical progesterone 33% in aquafor base BID all other days* 3 Desoximetasone 0.25% cream BID once weekly with topical progesterone 33% in aquafor base BID all other days 4 Vagifem 25 μg twice weekly in addition to one of clobetasol regimens above (marked by *) or immunomodulator 5 Topical testosterone 2% in white petrolatum base once daily in addition to steroid treatment or immunomodulator on all other days 6 Hydrocortisone 1% to apply as needed in addition to one of clobetasol regimens above (marked by *) 7 Pimecrolimus 1% cream BID after initial use of steroid treatment Number of LS patients (n = 20) 14 (70%) 2/14 changed to clobetasol once every 2 weeks with topical progesterone on all other days during follow-up 5 (25%) 3/5 changed to clobetasol once weekly with topical progesterone on all other days during follow-up 1 (5%) 0 4 (20%) 1 (33%) 9 (45%) 7/9 only used temporarily to prevent reagglutination; the remainder still using at follow-up 2 (10%) 0 1 (5%) 1 (33%) 8 Dilators 2 (10%) 1 (33%) Number of LP patients (n = 3) 2 (67%) 0 2 (67%) 2/2 only used treatment temporarily Table 2. Patient demographics Lichen sclerosus: Mean age ± SD, years 47 ± 14 (range 25 to 68) Mean follow-up length ± SD, months 31 ± 31 (range 3 to 120) Postmenopausal 45% (9/20 patients) Previous systemic postmenopausal hormone therapy 15% (3/20 patients) Proportion of patients with suturing of periclitoral skin 60% (12/20 patients) Lichen planus: Mean age ± SD, years 64 ± 2 (range 62 to 66) Mean follow-up length ± SD, months 7 ± 6 (range 3 to 14) Postmenopausal 100% (3/3 patients) Previous systemic postmenopausal hormone therapy 0 Proportion of patients with suturing of periclitoral skin 67% (2/3 patients) MAY JOGC MAI

4 Table 3. Summary of outcomes Lichen Sclerosus Reagglutination at most recent follow-up Reagglutination resolved with medical treatment Reoperation (no recurrence at most recent follow-up since reoperation) n (%) 3 (15%) 1 Total recurrence 8 (40%) Lichen Planus Reagglutination at most recent follow-up Reagglutination resolved with medical treatment Reoperation (no recurrence at most recent follow-up since reoperation) Patient no. 2 3 Suturing Yes (Y) No (N) Y N N Postoperative medical treatment until most recent follow-up (regimen number from Table 2) 1 (recommended 4, but patient declined) (10%) 4 Y (5 used for 8 months until resolution) 5 N (5 used since reagglutination, ongoing) 3 (15%) 6 Y (5 used for 7 months, then stopped) 0 N/A Total recurrence 2 (67%) 7 Y (5 used after reagglutination for 21 months until reoperation) 7 (started 2 months after reoperation, ongoing) Length of postoperative follow-up until reagglutination/ reoperation (months) Length of total postoperative follow-up (months) until re-agglutination and reoperation 8 until reagglutination, 29 until reoperation 8 N 2 3 until re-agglutination and reoperation 2 (67%) 9 Y (5 started 1 month after initial surgery, stopped after reoperation) 10 Y (5 started after reagglutination, then stopped at most recent follow-up) 12 until reagglutination and reoperation 1 until reagglutination and manual separation under local anaesthetic in office were satisfied with the results overall, and the third did not comply with postoperative medical management. Although some patients do develop reagglutination postoperatively, this can be treated with either medical or additional surgical management with minimal morbidity. Considering that LS is a chronic disease that often recurs (84% recurrence at 4 years in a study in which patients were treated with an initial longer course of high-potency corticosteroids until remission), 9 our results were encouraging. The technique described represents a good option for treating women with clitoral phimosis leading to sexual dysfunction when medical management has failed. The recurrence rates were high in women with LP in our series; however, recurrence rates are generally high in this group. 10,11 In one study, despite maintenance therapy with topical corticosteroids, only 9% of patients followed prospectively achieved complete resolution of clinical signs with the exception of scarring. 10 Since we have only recently started to treat these patients with this technique, we cannot draw any conclusions based on the small sample size. As previously described, it is important to ensure that the disease is under good control before any surgical management. 4 We will continue to follow this group of patients prospectively to determine whether the surgical 468 MAY JOGC MAI 2012

5 A Novel Approach to the Surgical Management of Clitoral Phimosis Figure 1. Case of lichen sclerosus with clitoral phimosis treated with the CO 2 laser: (a) pre-treatment (b) after laser dissection (c) 12 weeks post-treatment (a) (b) (c) Figure 2. Case of lichen sclerosus with clitoral phimosis treated with the CO 2 laser: (a) pre-treatment (b) after laser dissection (c) after suturing (d) six weeks post-treatment (a) (b) (c) (d) Figure 3. Case of lichen planus with clitoral phimosis treated with the CO 2 laser: (a) pre-treatment (b) after laser dissection (c) after suturing (d) four weeks postoperative with labia spread (a) (b) (c) (d) MAY JOGC MAI

6 technique described can adequately treat clitoral phimosis. Certainly the initial results are promising for short-term success with minimal risk of complication. Despite LS being recognized as a chronic condition, most clinicians treat the disease with only a short (6 to 12 week) course of super-potent corticosteroid ointment, and then either wean patients off corticosteroids completely or reduce to a low dose on an as-needed basis. 12 The rationale for weaning patients off super-potent corticosteroid therapy is to reduce the risk of developing atrophy, telangiectasia, and striae, which can be seen after treatment with high-potency topical corticosteroids on other areas of the body. However, the modified mucous membranes of the labia and clitoris are relatively resistant to the side effects of topical corticosteroids, and longterm follow-up of women with LS has not generally demonstrated these corticosteroid-induced changes. 9,13 Since symptoms often recur after initial treatment, and vulvar architectural changes can continue to worsen even in asymptomatic women once treatment is stopped, 14 some physicians support continuing treatment with a maintenance regimen. We support this regimen in our centre, and all patients in this review were maintained on topical high-potency corticosteroids or immunomodulators postoperatively. Immunomodulators were used only in severe cases when high potency corticosteroids failed, because immunomodulators are not approved for use in this setting. Testosterone was also used when necessary because it sometimes prevented the need for reoperation in women with reagglutination by causing mild clitoral hypertrophy; however, testosterone also is not approved for use in this setting. We attribute the high success and low recurrence rates associated with this surgical technique partly to local medical maintenance therapy postoperatively. In another small study of surgical management of clitoral phimosis secondary to LS, 5 patients were also maintained on high potency topical corticosteroids postoperatively, with promising results. Only one patient in that series had partial recurrence of phimosis, at between 12 and 36 months of follow-up, and she had stopped clobetasol therapy. Given the retrospective nature of our series, we cannot be sure that patients with recurrence were compliant with the medical treatment prescribed, and future prospective studies are warranted to determine the ideal medical treatment strategy for women with LS. Although a subjective improvement was noted with suturing the periclitoral skin with respect to surgical outcomes, and practice was changed over the 10 years of performing this surgical technique, we cannot be sure from this series that suturing decreases recurrence rates. Further prospective studies are warranted to determine whether suturing of the periclitoral skin is indicated. Because of the high cost of the CO 2 laser and its unavailability in many centres, we recognize that this is a limitation for the generalizability of this technique. With postoperative maintenance corticosteroid therapy, it is unclear whether or not similar results could be achieved using a scalpel with or without suturing. Although using the CO 2 laser is ideal because of its precision and the decreased collateral damage to tissues, prospective studies comparing use of the scalpel and the CO 2 laser for surgical correction of clitoral phimosis would be useful. Because the follow-up times varied between patients, we cannot conclude for how long the procedure is effective or if patients lost to follow-up experienced recurrence. As demonstrated in our series, many women presenting with severe LS are premenopausal. The treatment we describe can offer women of any age an option for treatment of clitoral phimosis, which can have significant vulvar side effects and, especially for premenopausal women, can have major effects on psychosexual well-being. CONCLUSION The surgical technique using the CO 2 laser that we describe has enabled the successful treatment of clitoral phimosis secondary to LS or LP. Local medical maintenance therapy after surgical correction is a vital component of treatment. Further long-term research is needed to compare the effectiveness of the novel method outlined in this case series with traditional management approaches. 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