Ovarian adult granulosa cell tumor and vulval lichenoid inflammation

Mucocutaneous conditions such as dermatomyositis have been found to be associated with malignant ovarian disease 1, 2. We call attention to a possible association between ovarian adult granulosa cell tumor (AGCT) and lichenoid inflammation through the case of a 65-year-old woman whose lichenoid eruption in the vulva disappeared after treatment of AGCT. The patient was hospitalized due to lower abdominal pain attributed to diverticulitis, treated with antibiotics, and in the diagnostic process an ultrasound of the pelvic organs was performed. The ovaries showed no pathology, but the endometrium was thickened, suggesting raised estrogen levels. Histology showed simple endometrial hyperplasia with focal complex hyperplasia and suspicion of mild atypia. She had not experienced any vaginal bleeding. She was gravida 2, para 2 and menopausal since 53 years. Her BMI was 23 kg/m2. Three years earlier she had been clinically diagnosed with erosive lichen planus in the vulva. The biopsy showed lichenoid inflammation. The eruption had been treated with alternately estradiol vaginal tablets, antimycotic drugs and topical glucocorticoids, but with poor effect and she still suffered from a burning sensation and dyspareunia. Another elevated and sore lesion on the shin of her right leg was noticed, but not biopsied. One month after the initial hospitalization a laparoscopically assisted vaginal hysterectomy and bilateral salpingoophorectomy was performed. CA-125 was 61 μ/mL (reference value <35μ/mL). Serum levels of estradiol or inhibin were not analyzed. Histology showed an 8-mm-large AGCT of FIGO stage Ia in the right ovary, with a low number of mitoses. The endometrium showed focal complex hyperplasia of the endometrium with small areas of mild atypia. The serum levels of CA-125, inhibin A and -B and estradiol 14 days postoperatively were normal. During the following 12 months the symptoms from the vulva and shin gradually disappeared. A vulval biopsy 9 months after the surgery showed no lichenoid inflammation. There were no clinical signs of the two separate gynecologic conditions 20 months after the surgery. Lichenoid inflammation is a histologic change characteristic of certain immunoinflammatory and reactive lesions of skin and mucous membranes. The histologic pattern of the inflammatory infiltrate in lichen planus is the prototype of lichenoid interface inflammation. The epithelial histology features of lichen planus may not be present in mucosal lesions 3. The etiology of lichen planus is unknown but it is considered to be an autoimmune disease influenced by genetic susceptibility. Anogenital lichen planus is a chronic condition. Topical glucocorticoid and tacrolimus can relieve the symptoms, but complete remission is uncommon 4. Lichenoid mucocutaneous changes have been found in association with internal malignant disease 5. A search in the literature revealed no published articles regarding an association between lichenoid eruptions and ovarian neoplasms. Because of the mucosal response to the removal of the AGCT in this patient, we propose the possibility of an association between AGCT and vulval lichenoid inflammation through raised estrogen levels. Further cases must be published to substantiate this.