C1 (Clinical Case) Case Presentation: Acute Genital Ulceration in a 15-Year-Old Girl

Abstract

Abstract Introduction/Background Genital ulceration may occur in various conditions, and requires a systematic approach to its diagnosis. This should include consideration of both infectious and non-infectious etiologies. The most common causes of genital ulceration are herpes simplex virus (HSV) types 1 and 2 and syphilis (Treponema pallidum). Genital HSV may be asymptomatic or painful, and may present with inguinal lymphadenopathy, and fever. Primary syphilis may escape recognition as it presents with a single, painless, well-demarcated “chancre” that heals spontaneously in 3-6 weeks. Non-infectious etiologies of genital ulceration can include cutaneous drug reactions, and mechanical or chemical trauma. Granulomatous genital inflammation can be an extra-intestinal manifestation of Crohn’s disease characterized by pathognomonic “knife-cut” vulvar ulcers. This report presents a case of Lipschütz’s ulceration, or reactive non-sexually-acquired genital ulceration, as an under-recognized cause of genital ulcers in children. Case Description A previously healthy 15-year-old female presented to the emergency department with six days of daily fevers, and 72 hours of non-bloody diarrhea that had self-resolved. In addition, she complained of nausea and a headache. Notably, in the last day, she had developed labial swelling, perianal pruritus, and vaginal burning. On history, her last menstrual period was one week ago, and she was not sexually active. It was noted that she had consumed Kinder-SurpriseTM eggs in the last week, which had been recently recalled for Salmonella contamination. Pelvic exam initially showed multiple hemorrhagic vesicles involving the labia minora (Fig. 1A). On day one of admission, she developed two symmetric, well-delimited ulcers at the 5 o’clock and 7 o’clock positions on the labia minora, each 12 mm x 5 mm in size (Fig. 1B). By the second day of admission, she had dysuria requiring catheterizing. HSV, chlamydia and gonorrhea were not detected on vaginal swabs, and serologic testing for syphilis was negative. Salmonella montevideo was detected on her blood cultures and she was started on intravenous antibiotics. Her vulvar ulcers improved with a conservative regimen of local hygiene, wound care, and pain control. The final diagnosis was of Lipschütz’s ulceration (LU). Discussion LU is characterized by the sudden onset of necrotic and painful genital ulcers, and typically occurs in young (< 20 years old), sexually inactive females who have no history of genital ulcers, and were not previously affected by a chronic inflammatory condition. In LU, there are usually 1-3 lesions that are large (>10 mm), shallow and well-delimited. These tend to occur on the medial aspects of the labia minora and vestibule and have a symmetric “kissing” distribution on opposing surfaces. Voiding disorders, aphthous ulcers and enlarged inguinal lymph nodes have also been reported in LU. The pathophysiology of LU is not well understood. It often occurs concomitantly with an infectious disease and develops before its resolution. LU is most associated with Epstein-Barr virus, but has also been described with cytomegalovirus, mycoplasma species, Toxoplasma gondii, and influenza A and B. LU is self-limited and generally resolves without recurrence or long-term sequelae within 3 weeks. For analgesia, topical lidocaine is recommended, especially 5-10 minutes before micturition. Sitz baths, application of a bland barrier cream prior to and after voiding, avoidance of pads and underwear, and maintenance of good hydration to minimize irritation by concentrated urine also constitute important components of supportive care for LU. Conclusion Lipschütz’s ulcers are poorly understood and therefore under-diagnosed, or mis-diagnosed as herpes simplex infection, in which case they are treated with acyclovir without benefit. Identification of an etiologic trigger, such as a viral or bacterial infection, can aid in their appropriate detection. To date, there is one other reported case of LU associated with salmonella. This case report aims to describe LU as an uncommon but important entity in the paediatric population. Images