{
  "abstract": "Crohn’s Disease (CD) is a chronic condition characterised by transmural gastrointestinal inflammation from oropharynx to anus. 1Extra-intestinal manifestations of CD can occur in up to 40% of patients, spanning musculoskeletal, mucocutaneous, and ophthalmic involvement.Whilst perianal and recto-vaginal associations of disease have been well-described in the literature,2 rectolabial fistula is a rare extra-intestinal manifestation of CD, described only once prior in the literature, in the case of a 9-year-old pre-menarchal female.3 This report presents the unique diagnostic and management challenges in a previously fit and well 14-year-old Caucasian female with rectolabial fistula as first presentation of CD.The patient presented acutely with labial pain and swelling. She reported 2 kg weight loss and menorrhagia in the few months prior to presentation. This was on a background of chronic crampy abdominal pain, and loose bowel motions twice daily with blood on wiping and mixed with stool. She denied rectal mucus or other features of systemic involvement including fevers, arthralgia, eye, or skin involvement. She was not sexually active at any time prior to presentation.Initial blood tests including ESR, CRP and FBC were within normal limits. Faecal calprotectin was 447 ug/g.The patient was initially admitted under the gynaecology team for possible labial cyst. She underwent examination under anaesthesia (EUA), which excluded labial cysts but showed possible perianal fistula.She was transferred to a tertiary centre for further assessment and management.MRI perineum showed an area of oedema in the anterior right labia. A thin fistula at 11 o’clock was noted to pass posteriorly towards the anus (figure 1).Considering these findings, a multi-disciplinary investigative approach was undertaken incorporating GI endoscopies and rectal EUA.Endoscopic appearances were in keeping macroscopically with CD. Colonic biopsies confirmed moderate to mild active chronic colitis with granulomas.EUA rectum confirmed anterior trans-sphincteric fistula between right labium and rectum, involving 50% of internal sphincter without evidence of recto-vaginal fistula, abscess or collection. An anterior seton was inserted.Labial biopsy showed multiple dermal non-caseating granulomata associated with a predominantly chronic inflammatory cell infiltrate. No dysplasia or malignancy.After initial treatment with antibiotics, the patient commenced infliximab 4 weeks post-seton insertion. The patient completed a 3-month course of oral Metronidazole.Repeat EUA 6-months following the first confirmed persistence of the fistula involving 40% of the sphincter, with development of granulation tissue. Partial fistulectomy and seton change were performed.As well as evidence of clinical remission, MRI perineum at 1 year showed complete fistula resolution.Alongside clinical complexity, this patient suffered with the psychological burden of this chronic disease; she had significant school absence that correlated with the fluctuating severity of physical symptoms and psychological distress. She was offered psychological and educational support.Key Messages A presentation of labial swelling and tenderness may warrant further work-up for CD in a paediatric population.Effective clinical management requires close multi-specialty collaboration including paediatrics, gastroenterology, gynaecology, radiology and general surgery.The biopsychosocial burden of this chronic disease with gynaecological manifestations presents a unique challenge, mandating multi-modal support including psychologists, school and family.Abstract OC61 Figure 1MRI perineum showing fistulous tract and labial swelling, arrows in left image indicate the fistulaReferences Feuerstein JD, Cheifetz AS. Crohn disease: epidemiology, diagnosis, and management. Mayo Clin Proc. [Online]. 2017 [Accessed 2024 Oct 10];92(7):1088–1103. Available from: https://pubmed.ncbi.nlm.nih.gov/28601423 Bernstein CN, Blanchard JF, Rawsthorne P, Yu N. The prevalence of extraintestinal diseases in inflammatory bowel disease: a population-based study. Am J Gastroenterol. [Online]. 2001 [Accessed 2024 Oct 15];96:1116–1122. Available from: https://pubmed.ncbi.nlm.nih.gov/11316157/ Brioso XB, Banks D, Christison-Lagay E, Phatak UP, Vash-Margita A. Vulvovaginal complaints as the first manifestation of Crohn’s disease in children: case report. J Pediatr Adolesc Gynecol. [Online]. 2023 [Accessed 2024 Oct 13];36(5):494–496. Available from: https://pubmed.ncbi.nlm.nih.gov/37196754/",
  "authors": [
    {
      "affiliations": [
        "University Hospitals Sussex NHS Foundation Trust"
      ],
      "name": "Hadis Reyhani"
    },
    {
      "affiliations": [
        "University Hospitals Sussex NHS Foundation Trust"
      ],
      "name": "Alfredo Tonsi"
    },
    {
      "affiliations": [
        "University Hospitals Sussex NHS Foundation Trust"
      ],
      "name": "Jigna Sheth"
    },
    {
      "affiliations": [
        "University Hospitals Sussex NHS Foundation Trust"
      ],
      "name": "Kyriakos Iliadis"
    },
    {
      "affiliations": [
        "University Hospitals Sussex NHS Foundation Trust"
      ],
      "name": "Farah Barakat"
    }
  ],
  "title": "OC61 Rectolabial fistula as first presentation of Crohn’s disease in an adolescent",
  "uid": "9bfa6953-d8dc-5409-bdb6-4f942f54ae2e"
}