A Case of Uterine Lymphangioleiomyomatosis Complicated by Tuberous Sclerosis Complex.

Lymphangioleiomyomatosis (LAM) is one of the presentations of perivascular epithelioid cell neoplasm that is frequently complicated by tuberous sclerosis complex (TSC). Here, we report an uncommon case of uterine LAM treated with everolimus, which is a mechanistic target of rapamycin (mTOR) inhibitor. A 42-year-old female patient (gravida 0) with a history of TSC presented with abdominal pain. Pelvic magnetic resonance imaging showed multiple masses in the uterine myometrium, suggesting tumors that may contain internal hemorrhagic components. The lesions were suspected as the root cause of her symptoms. After everolimus was administered for a previously diagnosed renal angiolipoma, her uterine tumors temporarily decreased in size. Subsequently, laparoscopic hysterectomy and bilateral salpingectomy were performed since she could not tolerate everolimus for a long period due to the medication's side effects. Furthermore, the patient was diagnosed with LAM through histopathological examination after surgical resection. Therefore, it is advisable to suspect and investigate uterine LAM when a patient with a history of TSC presents with irregular genital bleeding or abdominal pain. Moreover, mTOR inhibitors may be a treatment option, in addition to surgery, in cases of uterine LAM exacerbation.

Imiquimod for vaginal intraepithelial neoplasia 2-3: A systematic review and meta-analysis.

OBJECTIVE: The treatment strategy for vaginal intraepithelial neoplasia (VaIN) 2-3 has not been established. This study aimed to investigate the efficacy of imiquimod in VaIN 2-3. METHODS: Electronic databases (PubMed, EMBASE, ClinicalTrials.gov, and Cochrane Central Register of Controlled Trials) were searched from their inception until October 2019 and articles reporting imiquimod treatment for VaIN 2-3 were extracted. Additionally, the clinical records of women with VaIN 2-3 who had been treated with imiquimod in Shizuoka General Hospital from January 2016 to May 2020 were investigated. The data from the systematic search and the data from our hospital were analyzed, and a pooled complete response (CR) rate and response rate of imiquimod treatment for VaIN 2-3 were estimated. As a subgroup analysis, the CR rates and response rates were compared between women with and without a history of hysterectomy, and the rate ratio was calculated. RESULTS: Five articles described 28 women with VaIN 2-3 who underwent imiquimod treatment, and nine women with VaIN 2-3 were treated with imiquimod in our hospital. The discontinuation of the treatment was required in only one patient of the reported cases. The pooled CR rate and response rate of imiquimod, regardless of a history of hysterectomy, was 0.76 (95% CI, 0.59-0.87) and 0.89 (95% CI, 0.71-0.97), respectively. In the subgroup analysis, the CR rate in patients with hysterectomy was 0.98 (95% CI, 0.11-1.0) and in those without hysterectomy was 0.60 (95% CI, 0.30-0.84), and the rate ratio was 0.83 (95% CI, 0.48-1.19). The pooled response rates with and without a history of hysterectomy were not estimated, and the rate ratio was 0.83 (95% CI, 0.54-1.09). CONCLUSION: Imiquimod can be an effective treatment for vaginal intraepithelial neoplasia 2-3.

Recurrent vaginal intraepithelial neoplasia successfully treated with topical imiquimod: A case report.

Vaginal intraepithelial neoplasia (VAIN) is a rare disease associated with human papillomavirus infection. High-grade VAIN is typically treated with either excisional or ablative therapy. However, recurrent VAIN lesions are common and these treatments cause vaginal scarring. Recent studies have indicated that 5% imiquimod is an effective treatment for VAIN. The present report describes a case of a woman diagnosed with recurrent VAIN 3 who was treated with a 5% topical imiquimod cream and achieved a complete response after excision and CO2 laser vaporization. A 53-year-old, gravida 5, para 2 postmenopausal woman who was diagnosed with papillary squamous cell carcinoma by biopsy underwent conization, total abdominal hysterectomy and bilateral salpingo-oophorectomy. A histological examination revealed grade 3 cervical intraepithelial neoplasia with free surgical margins. At 3 years after the hysterectomy, the vaginal smear revealed atypical squamous cells, leading to a pathological diagnosis of VAIN 3. Partial vaginectomy was performed, and VAIN 3 was detected in the lesion with positive margins. At 4 months into follow-up, the vaginal smear revealed a high-grade squamous intraepithelial lesion (HSIL), and subsequent biopsy during colposcopy revealed a pathological diagnosis of VAIN 3. At 3 months after CO2 laser vaporization, the vaginal smear revealed HSIL with suspected recurrence and imiquimod treatment was initiated. One sachet of 5% imiquimod cream (0.25 g) was placed in the entire vagina three times per week for 14 weeks with no apparent complications. At 3 years after the treatment, there has been no recurrence. This case demonstrated that topical imiquimod with careful follow-up is an effective treatment for VAIN and is well-tolerated. Further clinical evidence of the effectiveness and safety of imiquimod in patients diagnosed with VAIN is required.