Genital ulcer in an adolescent girl

Sir,

Lipschütz ulcer is an acute disease characterized by fever, ulceration of the external genitalia, and lymphadenopathy.[1] It is an important cause of acute nonvenereal vulvar ulcer.[1] It usually occurs in young girls aged 14–20 years but may occur in children. Majority (70%) of the cases involve virgins. The common sites of ulcer are inner aspect of labia minora, labia majora, introitus, external urethral orifice, or posterior commissure.[1] Although it is a rare entity, it often misdiagnosed. We report a case of 18-year-old unmarried girl with multiple ulcerations over genitalia closely mimicking a sexually transmitted infection. Initially, she noticed single painful swelling over labia minora for 2 months, sudden in onset, which ruptured within 5 days to form an ulcer. There was a history of difficulty in walking and urination. The patient did not give any history of trauma or sexual contact. There was no prior history of similar lesions or of oral ulcers. The patient did not have any preceding infection. On examination, labia majora was swollen and erythematous. Multiple nonindurated ulcers measuring 1.5 cm × 1.5 cm to 2 cm × 3 cm with yellowish slough on the floor were present over the labia majora, minora, and introitus [Figure 1a]. The ulcers were tender. The hymen was intact. Oral mucosa was normal. On investigations, there was raised total leukocyte count (14000 mg/dl). Urine showed 7–8 pus cells, and urine culture grew Klebsiella spp. sensitive to meropenem, colistin, and polymyxin B. No pathogens were isolated in Tzanck smear, Gram stain. Pathergy test and Mantoux test were negative. Serology for human immunodeficiency virus, venereal disease research laboratory, hepatitis B surface antigen, herpes simplex virus, Epstein–Barr virus (EBV), and antinuclear antibody were negative. Histopathology showed orthokeratosis, acanthosis, wedge-shaped hypergranulosis, spongiosis, basal cell liquefaction, and lymphocytic exocytosis [Figure 2]. Dermis showed dense chronic inflammatory infiltrate and many eosinophils. Chest radiograph and ultrasound of the abdomen were normal. The patient was treated for urine infection with tablet cefixime twice daily for 7 days. She had also prescribed oral antibiotic amoxy-clavulanic acid for secondary infection. There was slight improvement initially followed by worsening of ulcers and became deeper. We had sent repeat bacterial culture from ulcer and started her again on oral antibiotics in the form of tablet doxycycline 100 mg and tablet metronidazole 400 mg twice daily for 14 days. In spite of this, there was no improvement, and the patient was started on tablet prednisolone 20 mg for 7 days followed by 10 mg for 1 month. Pain disappeared within 3 days of starting corticosteroids, and ulcers started healing in 7 days with complete healing in 2 weeks with no recurrence till 2 months of follow-up [Figure 1b]. In clinical practice, the diagnosis of genital ulcers is a challenge. The cause is mostly infectious, but it may be a variety of causes, such as autoimmune (e.g., Behçet's disease), cancer, or inflammatory processes. Etiology is unknown although some suggested an association with acute EBV or other viral or bacterial infections.[2,3,4] Management is mainly symptomatic. Topical antibiotics have been used. However, oral corticosteroids and oral antibiotics have been tried for painful acute genital ulcers. Our patient showed a dramatic response to oral prednisolone. Lipschütz ulcers should be considered as differential diagnosis in adolescent girls presenting with sudden onset of painful deep ulcers over genitalia as timely management can prevent considerable morbidity associated with this condition.

Figure 1.

(a) Multiple ulcers over genitalia. (b) Posttreatment picture showing healing of ulcers

Figure 2.

Microphotograph showing spongiosis, irregular elongation of rete ridges, basal cell liquefaction, and lymphocytic exocytosis (×40)

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