To the Editors,

The anorectal region is the most frequent site of associated problems such as fissure, abscess, and fistula formation in patients with ulcerative colitis (UC).1 Anorectal complications of UC can cause difficult management problems and lead to further disability. Rectovaginal fistula is rare complication of UC that has been difficult to manage successfully. We report a case of UC patient for whom the rectovaginal fistula could be successfully managed by the administration of adalimumab.

A 42-year-old woman with a 3-year history of UC was admitted to our hospital due to abdominal pain, diarrhea, hematochezia (more than 6 times per day), skin erythema nodule, and the symptom of a spontaneous rectovaginal fistula for 3 months. Subsequent laboratory findings revealed that there was a significant increase in elevated white blood cells (WBC, 12.04x109/L), C-Reactive Protein (CRP, 76.2 mg/L), tumor necrosis factor-alpha (TNF-α, 7.48 pg/mL), interleukin-2 (IL-2, 99.82 pg/mL), IL-6 (157.73 pg/mL), IL-10 (61.01 pg/mL), and IL-17 (149.48 pg/mL). Albumin (ALB, 16 g/L) and hemoglobin (Hb 73 g/L) were significantly decreased. Stool culture and Clostridium difficile toxin were negative. The abdominal contrast-enhanced computed tomography (CT) showed that there was thickened intestinal wall in colorectum. Colonoscopy and intestinal histopathology suggested active severe extensive UC. Diffuse neutrophilic infiltration was observed in histopathology of skin biopsy. Abdominal magnetic resonance imaging (MRI) showed that the rectovaginal fistula was located in the anterior distal anorectal canal about 1.5 cm to the anorectal junction. The fistula orifice was approximately 4 to 5 mm in diameter (Figure 1).

Abdominal MRI showed the rectovaginal fistula before administration of adalimumab.
Figure 1.

Abdominal MRI showed the rectovaginal fistula before administration of adalimumab.

The diagnosis for this patient was acute severe UC associated with skin erythema nodule and rectovaginal fistula. After confirmed that the patient was negative for tuberculosis and informed consent was obtained, the adalimumab-based treatment was started (subcutaneous injection of adalimumab at a dose of 160 mg at day 0 and 80 mg at week 2, 40 mg at week 4, and then 40 mg every 2 weeks). The patient’s clinical symptoms improved during the follow-up period. Laboratory findings indicated that the clinical condition of this patient gradually improved: WBC 9.12x109/L, CRP 12.5 mg/L, ALB 31.4 g/L, and Hb 102 g/L. Ten weeks after the first injection, the drainage from the fistula was evidently reduced, and 4 weeks later, the fistula was completely closed. The closure of the fistula was confirmed by the abdominal MRI. Thereafter, we continued the administration of adalimumab at the outpatient clinic. And no recurrence of the fistula was observed for at least 12 weeks.

Author Contribution

C.D. and Y-h.H. had the original idea for the article and were in charge of treatment and management of the patient. C.D. wrote the article and incorporated the comments from other author. All authors reviewed and approved the final draft of the article.

Conflicts of Interest

The authors declare no conflicts of interest.

Data Availability

All data are incorporated into the article. The data underlying this article are available in the article.

References

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