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Anal Canal Adenocarcinoma in a Patient with Longstanding Crohn’s Disease Arising From Rectal Mucosa that Migrated From a Previously Treated Rectovaginal Fistula

Taku Maejima, Toru Kono, Fumika Orii, Atsuo Maemoto, Shigeru Furukawa, Wang Liming, Shoji Kasai, Susumu Fukahori, Nobutaka Mukai, Daitaro Yoshikawa, Hidenori Karasaki, Hiroya Saito, Kazuo Nagashima

(Department of Surgery, Sapporo Higashi Tokushukai Hospital, Sapporo, Japan)

Am J Case Rep 2016; 17:448-453

DOI: 10.12659/AJCR.897876


BACKGROUND: This study reports the pathogenesis of anal canal adenocarcinoma in a patient with longstanding Crohn’s disease (CD).
CASE REPORT: A 50-year-old woman with a 33-year history of CD presented with perianal pain of several months’ duration. She had been treated surgically for a rectovaginal fistula 26 years earlier and had been treated with infliximab (IFX) for the previous 4 years. A biopsy under anesthesia revealed an anal canal adenocarcinoma, which was removed by abdominoperineal resection. Pathological examination showed that a large part of the tumor consisted of mucinous adenocarcinoma at the same location as the rectovaginal fistula had been removed 26 years earlier. There was no evidence of recurrent rectovaginal fistula, but thick fibers surrounded the tumor, likely representing part of the previous rectovaginal fistula. Immunohistochemical analysis using antibodies against cytokeratins (CK20 and CK7) revealed that the adenocarcinoma arose from the rectal mucosa, not the anal glands.
CONCLUSIONS: Mucinous adenocarcinoma can arise in patients with CD, even in the absence of longstanding perianal disease, and may be associated with adenomatous transformation of the epithelial lining in a former fistula tract.

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