A 26-year-old black woman, born in Cape Verde and living in Portugal since the age of 3 years, presented with anal and vaginal suppuration. She had a known history of two deliveries, including a cesarean section in 2005 and a vaginal delivery in 2011 without episiotomy. During the second trimester of her second pregnancy, she had a bartholinitis requiring surgical drainage. She was using oral contraceptives and denied previous use of intrauterine devices. She was a smoker (6-7 cigarettes/day) and did not drink alcohol. She denied recent travels to Africa.
The patient was first admitted in 2013 for complaints of anal and vaginal suppuration lasting 2 weeks, without fever or other accompanying symptoms. Perianal inspection revealed the presence of an external fistulous orifice in the posterior perineum, close to the left labium majus. On rectal examination, there was a palpable induration of the left anterior rectal wall. Her gynecological examination was unremarkable.
A pelvic computed tomography excluded the presence of abscesses or other pelvic masses. The patient was put on antibiotics and underwent rectal examination under anesthesia, where no abscess was found. Two fistulous tracts were identified and treated with seton placement, including one tract involving the vagina. Biopsies of the indurated rectal wall were taken, and pus was collected for analysis. Pelvic magnetic resonance imaging 1 month after hospital discharge revealed multiple fistulous tracts, including trans-sphincteric and suprasphincteric tracts and a rectovaginal fistula (Figure 1).
An extensive work-up to exclude inflammatory bowel disease (IBD) was done, including an upper endoscopy, colonoscopy with ileoscopy, small bowel capsule endoscopy and computed tomography enterography, which were unremarkable. Endoscopic biopsies of the colon, terminal ileum, and stomach did not show any significant findings. Anti-Saccharomyces cerevisiae antibodies titer was also negative. Cytology and cultural exams of both rectal and vaginal exudates and biopsy of the rectum were negative for neoplastic cells and microorganisms, including Mycobacterium tuberculosis. Serologies of Chlamydia trachomatis and human immunodeficiency virus were also negative, as was a VDRL test for syphilis.
In the following 3 years, the patient underwent multiple abscess drainages, fistulae treatment by seton placement, and empiric antibiotic courses, with no overall improvement (Figure 2). This situation had a high impact on the patient's quality of life, eventually leading to clinical depression. Despite high suspicion of inflammatory bowel disease, a complete endoscopic investigation again was negative. In 2016, repeated anal cytology revealed the presence of bacterial colonies compatible with Actinomyces spp. (Figure 3). The patient was then hospitalized and completed a 6-week course of intravenous (IV) penicillin G, after which she was started on oral amoxicillin, which is to be maintained for 12 months. This resulted in a remarkable clinical improvement, allowing for the removal of setons (Figure 4). The reevaluation magnetic resonance imaging, performed after only 5 weeks of antibiotic therapy, showed almost complete resolution of the fistulous tracts (Figure 5). Currently, after 6 months of oral amoxicillin, the patient is clinically well, with no perianal suppuration, and reports a significant improvement in her quality of life. Although notably reduced, the vaginal fistula is still patent and will possibly require surgical correction.