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

Patient: Female, 50 Final Diagnosis: Anal canal adenocarcinoma Symptoms: — Medication: — Clinical Procedure: CT • MRI • biopsy Specialty: Surgery Objective: Unknown ethiology 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.

[1]  I. Papaconstantinou,et al.  Anal adenocarcinoma complicating chronic Crohn’s disease , 2015, International journal of surgery case reports.

[2]  L. Guidi,et al.  Management of perianal fistulas in Crohn's disease: an up-to-date review. , 2015, World journal of gastroenterology.

[3]  G. Pellino Immunosuppression may exert a hypoxia-mediated carcinogenetic effect in long-standing fistulizing Crohn's disease. , 2014, European journal of gastroenterology & hepatology.

[4]  L. Peyrin-Biroulet,et al.  Microscopic features of colorectal neoplasia in inflammatory bowel diseases. , 2014, World journal of gastroenterology.

[5]  A. Ford,et al.  Systematic review with meta‐analysis: malignancies with anti‐tumour necrosis factor‐α therapy in inflammatory bowel disease , 2014, Alimentary pharmacology & therapeutics.

[6]  W. Sandborn,et al.  Drug Therapies and the Risk of Malignancy in Crohn's Disease: Results From the TREAT™ Registry , 2014, The American Journal of Gastroenterology.

[7]  G. Rogler,et al.  Epithelial-to-mesenchymal transition in a fistula-associated anal adenocarcinoma in a patient with long-standing Crohn’s disease , 2014, European journal of gastroenterology & hepatology.

[8]  T. Jess,et al.  Has the risk of colorectal cancer in inflammatory bowel disease decreased? , 2013, World journal of gastroenterology.

[9]  M. Unno,et al.  Adenocarcinoma associated with perianal fistulas in Crohn's disease. , 2013, Anticancer research.

[10]  H. Ikeuchi,et al.  Adenocarcinomas associated with perianal fistulae in Crohn's disease have a rectal, not an anal, immunophenotype , 2011, Pathology.

[11]  A. Agha,et al.  Fistula‐associated anal adenocarcinoma in Crohn's disease , 2010, Inflammatory bowel diseases.

[12]  A. Sobala,et al.  Colorectal carcinoma and preceding fistula in Crohn's disease. , 2010, Journal of Crohn's & colitis.

[13]  S. Itzkowitz,et al.  Colorectal cancer in inflammatory bowel disease. , 2008, Gut and liver.

[14]  H. Freeman Colorectal cancer risk in Crohn's disease. , 2008, World journal of gastroenterology.

[15]  A. Rajput,et al.  Adenocarcinoma Arising from Chronic Perianal Crohn's Disease: Case Report and Review of the Literature , 2008, The American surgeon.

[16]  K. Futami,et al.  Study of colorectal cancer with Crohn's disease. , 2007, Anticancer research.

[17]  H. Bouzourène,et al.  Adenocarcinoma in Crohn's disease , 2003, Histopathology.

[18]  B. Korelitz Carcinoma Arising in Crohn's Disease Fistulae: Another Concern Warranting Another Type of Surveillance , 1999, American Journal of Gastroenterology.

[19]  M. Kamm,et al.  Lower gastrointestinal malignancy in Crohn's disease. , 1994, Gut.

[20]  J. Domínguez,et al.  Carcinoma associated with fistula in ano. , 1973, International surgery.

[21]  I. Hales,et al.  Mucoid carcinoma supervening on fistula-in-ano, its surgical pathology and treatment. , 1953, Annals of Surgery.

[22]  I. Skir Mucinous carcinoma associated with fistulas of long-standing. , 1948, American journal of surgery.

[23]  B. Cagir,et al.  Malignant Transformation in Perianal Fistulas of Crohn’s Disease: a Systematic Review of Literature , 2009, Journal of Gastrointestinal Surgery.

[24]  I. Leodolter [Crohn's disease]. , 1967, Wiener Zeitschrift fur innere Medizin und ihre Grenzgebiete.