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Clin Colon Rectal Surg 2024; 37(01): 041-045
DOI: 10.1055/s-0043-1762928
Review Article

Cancer in Anal Fistulas

Sean Perez
1   Division of Colorectal Surgery, Department of Surgery, University of California San Diego, La Jolla, California
,
Samuel Eisenstein
1   Division of Colorectal Surgery, Department of Surgery, University of California San Diego, La Jolla, California
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Abstract

Fistula-associated anal cancer in Crohn's disease (CD) can be challenging to diagnose and treat. Patients with longstanding fistulas in the setting of CD who present with a sudden change in their symptoms should undergo biopsy under anesthesia with extensive sampling, followed by staging imaging. Pelvic magnetic resonance imaging (MRI) can be helpful in identifying the extent of the disease locally. Patients often present in the later stages due to the challenges associated with diagnosing these patients. Two subtypes of this disease include squamous cell carcinoma and adenocarcinoma, and treatment depends on diagnosis. Small sample size and lack of uniform data on treatments make it difficult to say which treatment modalities are optimal, but aggressive combined therapy is likely the best approach for survival. This will include chemotherapy and radiation and often radical resection as well. Despite this, survival is poor, although more recent data suggest that outcomes are improving.

Keywords

fistula-associate anal cancer - squamous cell carcinoma - adenocarcinoma - Crohn's disease - perianal disease


Publication History

Article published online:
29 March 2023

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  • References

  • 1 Wewer MD, Zhao M, Nordholm-Carstensen A, Weimers P, Seidelin JB, Burisch J. The incidence and disease course of perianal Crohn's disease: a Danish nationwide cohort study, 1997-2015. J Crohn's Colitis 2021; 15 (01) 5-13
    CrossrefPubMedGoogle Scholar
  • 2 Beaugerie L, Carrat F, Nahon S. et al; Cancers et Surrisque Associé aux Maladies Inflammatoires Intestinales En France Study Group. High risk of anal and rectal cancer in patients with anal and/or perianal Crohn's disease. Clin Gastroenterol Hepatol 2018; 16 (06) 892-899.e2
    CrossrefPubMedGoogle Scholar
  • 3 Shwaartz C, Munger JA, Deliz JR. et al. Fistula-associated anorectal cancer in the setting of Crohn's disease. Dis Colon Rectum 2016; 59 (12) 1168-1173
    CrossrefPubMedGoogle Scholar
  • 4 Ky A, Sohn N, Weinstein MA, Korelitz BI. Carcinoma arising in anorectal fistulas of Crohn's disease. Dis Colon Rectum 1998; 41 (08) 992-996
    CrossrefPubMedGoogle Scholar
  • 5 Connell WR, Sheffield JP, Kamm MA, Ritchie JK, Hawley PR, Lennard-Jones JE. Lower gastrointestinal malignancy in Crohn's disease. Gut 1994; 35 (03) 347-352
    CrossrefPubMedGoogle Scholar
  • 6 Thomas M, Bienkowski R, Vandermeer TJ, Trostle D, Cagir B. Malignant transformation in perianal fistulas of Crohn's disease: a systematic review of literature. J Gastrointest Surg 2010; 14 (01) 66-73
    CrossrefPubMedGoogle Scholar
  • 7 Nishigami T, Kataoka TR, Ikeuchi H. et al. Adenocarcinomas associated with perianal fistulae in Crohn's disease have a rectal, not an anal, immunophenotype. Pathology 2011; 43 (01) 36-39
    CrossrefPubMedGoogle Scholar
  • 8 Scharl M, Frei P, Frei SM, Biedermann L, Weber A, Rogler G. Epithelial-to-mesenchymal transition in a fistula-associated anal adenocarcinoma in a patient with long-standing Crohn's disease. Eur J Gastroenterol Hepatol 2014; 26 (01) 114-118
    CrossrefPubMedGoogle Scholar
  • 9 Kodama M, Kobayashi D, Iihara K. et al. Adenocarcinoma within anorectal fistulae: different clinicopathological characteristics between Crohn's disease-associated type and the usual type. Mod Pathol 2019; 32 (02) 314-325
    CrossrefPubMedGoogle Scholar
  • 10 Gaertner WB, Hagerman GF, Finne CO. et al. Fistula-associated anal adenocarcinoma: good results with aggressive therapy. Dis Colon Rectum 2008; 51 (07) 1061-1067
    CrossrefPubMedGoogle Scholar
  • 11 Maejima T, Kono T, Orii F. et al. Anal canal adenocarcinoma in a patient with longstanding Crohn's disease arising from rectal mucosa that migrated from a previously treated rectovaginal fistula. Am J Case Rep 2016; 17: 448-453
    CrossrefPubMedGoogle Scholar
  • 12 Kotsafti A, Scarpa M, Angriman I, Castagliuolo I, Caruso A. Fistula-related cancer in Crohn's disease: a systematic review. Cancers (Basel) 2021; 13 (06) 1445
    CrossrefPubMedGoogle Scholar
  • 13 Wisniewski A, Fléjou JF, Siproudhis L, Abramowitz L, Svrcek M, Beaugerie L. Anal neoplasia in inflammatory bowel disease: classification proposal, epidemiology, carcinogenesis, and risk management perspectives. J Crohn's Colitis 2017; 11 (08) 1011-1018
    CrossrefPubMedGoogle Scholar
  • 14 Baars JE, Kuipers EJ, Dijkstra G. et al; Initiative for Crohn and Colitis. Malignant transformation of perianal and enterocutaneous fistulas is rare: results of 17 years of follow-up from the Netherlands. Scand J Gastroenterol 2011; 46 (03) 319-325
    CrossrefPubMedGoogle Scholar
  • 15 Fornaro R, Frascio M, Caratto M. et al. Adenocarcinoma arising from perianal fistulizing Crohn's disease. Case Rep Gastroenterol 2018; 12 (02) 390-395
    CrossrefPubMedGoogle Scholar
  • 16 Palmieri C, Müller G, Kroesen AJ. et al. Perianal fistula-associated carcinoma in Crohn's disease: a multicentre retrospective case control study. J Crohn's Colitis 2021; 15 (10) 1686-1693
    CrossrefPubMedGoogle Scholar
  • 17 Annese V, Beaugerie L, Egan L. et al; ECCO. European evidence-based consensus: inflammatory bowel disease and malignancies. J Crohn's Colitis 2015; 9 (11) 945-965
    CrossrefPubMedGoogle Scholar
  • 18 Brochard C, Siproudhis L, Wallenhorst T. et al. Anorectal stricture in 102 patients with Crohn's disease: natural history in the era of biologics. Aliment Pharmacol Ther 2014; 40 (07) 796-803
    CrossrefPubMedGoogle Scholar
  • 19 Iesalnieks I, Gaertner WB, Glass H. et al. Fistula-associated anal adenocarcinoma in Crohn's disease. Inflamm Bowel Dis 2010; 16 (10) 1643-1648
    CrossrefPubMedGoogle Scholar
  • 20 Lichtenstein GR, Feagan BG, Cohen RD. et al. Drug therapies and the risk of malignancy in Crohn's disease: results from the TREAT™ Registry. Am J Gastroenterol 2014; 109 (02) 212-223
    CrossrefPubMedGoogle Scholar
  • 21 Ogawa H, Haneda S, Shibata C. et al. Adenocarcinoma associated with perianal fistulas in Crohn's disease. Anticancer Res 2013; 33 (02) 685-689
    PubMedGoogle Scholar
  • 22 Yamamoto T, Kotze PG, Spinelli A, Panaccione R. Fistula-associated anal carcinoma in Crohn's disease. Expert Rev Gastroenterol Hepatol 2018; 12 (09) 917-925
    CrossrefPubMedGoogle Scholar
  • 23 Benjelloun EB, Abkari M, Ousadden A, Taleb KA. Squamous cell carcinoma associated anal fistulas in Crohn's disease unique case report with literature review. J Crohns Colitis 2013; 7 (06) e232-e235
    CrossrefPubMedGoogle Scholar
  • 24 Lad SV, Haider MA, Brown CJ, Mcleod RS. MRI appearance of perianal carcinoma in Crohn's disease. J Magn Reson Imaging 2007; 26 (06) 1659-1662
    CrossrefPubMedGoogle Scholar
  • 25 Zhu X, Zhu TS, Ye DD, Liu SW. Magnetic resonance imaging findings of carcinoma arising from anal fistula: a retrospective study in a single institution. World J Clin Cases 2020; 8 (21) 5159-5171
    CrossrefPubMedGoogle Scholar
  • 26 Protyniak B, Shutt T, Farmer R. Squamous cell carcinoma originating from a Crohn's enterocutaneous fistula. Case Rep Surg 2017; 2017: 1929182
    PubMedGoogle Scholar
  • 27 Okada K, Shatari T, Sasaki T. et al. Is histopathological evidence really essential for making a surgical decision about mucinous carcinoma arising in a perianal fistula? Report of a case. Surg Today 2008; 38 (06) 555-558
    CrossrefPubMedGoogle Scholar
  • 28 Sobin L, Gospodarowicz M, Wittekind WC. TNM Classification of Malignant Tumors. 7th ed.. New York, NY: Wiley; 2009: 106-109
    Google Scholar
  • 29 An SH, Kim IY. Management of large and surrounding tissue infiltrative anal fistula cancer: two case reports. Int J Surg Case Rep 2020; 77: 643-646
    CrossrefPubMedGoogle Scholar
  • 30 Osone K, Ogawa H, Katayama C. et al. Outcomes of surgical treatment in patients with anorectal fistula cancer. Surg Case Rep 2021; 7 (01) 32
    CrossrefPubMedGoogle Scholar
  • 31 Sakanaka K, Fujii K, Hirashima H. et al. Chemoradiotherapy for fistula-related perianal squamous cell carcinoma with Crohn's disease. Int Cancer Conf J 2021; 10 (04) 305-311
    CrossrefPubMedGoogle Scholar