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 Table of Contents  
Year : 2017  |  Volume : 10  |  Issue : 2  |  Page : 225-227

Development of mucinous adenocarcinoma in chronic fistula-in-ano: A case study

1 Department of Shalyatantra, KLEU's Shri B.M.Kankanwadi Ayurveda Mahavidyalaya, Shahpur, Belagavi, Karnataka, India
2 Department of Rogavignan, KLEU's Shri B.M.Kankanwadi Ayurveda Mahavidyalaya, Shahpur, Belagavi, Karnataka, India

Date of Web Publication30-May-2017

Correspondence Address:
Pradeep S Shindhe
Department of Shalyatantra, KLEU's Shri B.M.Kankanwadi Ayurveda Mahavidyalaya, Shahpur, Belagavi, Karnataka
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/kleuhsj.ijhs_277_16

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Chronic perianal fistulas are a common clinical condition. However, their evolution to carcinoma is rare. We report a case of recurrent chronic perineal fistula which turned into malignant tumor. The diagnosis was established by clinical examination, repeated biopsy, and colonoscopy. Clinical examination features suggested malignancy, but initial biopsy report showed cellular atypia and colonoscopy followed with biopsy revealed as large ulcerated rectal growth suggesting Carcinoma (CA) rectum and histopathological report showed infiltrating mucinous adenocarcinoma. It is suggested that this rare complication of chronic fistula-in-ano may be prevented by prompt expert management of complex primary fistula.

Keywords: Biopsy, chronic fistula-in-ano, colonoscopy, mucinous adenocarcinoma

How to cite this article:
Shindhe PS, Killedar RS, Timmapur RY. Development of mucinous adenocarcinoma in chronic fistula-in-ano: A case study. Indian J Health Sci Biomed Res 2017;10:225-7

How to cite this URL:
Shindhe PS, Killedar RS, Timmapur RY. Development of mucinous adenocarcinoma in chronic fistula-in-ano: A case study. Indian J Health Sci Biomed Res [serial online] 2017 [cited 2022 Aug 13];10:225-7. Available from: https://www.ijournalhs.org/text.asp?2017/10/2/225/207250

  Introduction Top

The development of a carcinoma in a long-standing fistula-in-ano, however, is very rare.[1] Perianal mucinous adenocarcinoma is a rare disease often associated with a long-standing anal fistula, representing approximately 2%–3% of large bowel cancers.[2] The occurrence of a carcinoma arising in a fistula is probably due to chronic inflammation.[3] Like all other types of surgeries, a fistula surgery poses a risk for developing some complications which may range from minor to major complications although these are often rare and seldom serious, such as recurrent abscess, Fournier's gangrene, fecal incontinence, and fistula recurrence.[3] Detection is usually late as the symptoms often initially mimic benign inflammatory conditions of the anorectal region and biopsies fail to reveal the infiltrating carcinoma unknotted carcinoma of the rectum may coexist.[4],[5]

  Case Report Top

We report the case of a 54-year-old man with the history of recurrent perineal sepsis. History of twice surgery for fistula-in-ano on the right side of perineum 8 years back and on left side 2 years back, respectively. Local examination revealed that hypertrophied scar on either side of perineum [Figure 1] at 5'o clock and 7'o clock position of length 6 cm and 8 cm, respectively. Digital examination reveals normal sphincter tone and no tenderness, no mass inside the rectum, and anal canal. Proctoscopic findings were normal without revealing any internal pathology.
Figure 1: Scar of fistula on either side

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The patient frequently visited outpatient department for recurrent pain at the right side perineum. On examination, it usually revealed mild signs of inflammation and induration at hypertrophied scar on the right side perineum (7'o clock position). It used to be treated by Avagaha sweda (sitz bath) with Panchvalkala kashaya and tablet Nimbaadi Guggulu 2 bid after food. The patient was asymptomatic in the treatment interval, and he was advised for regular follow-up, but patient used to neglect and visit only at the time of perineal pain and fever. After 1 year because of repeated local perineal sepsis, there were changes observed in the perineum, i.e., hypertrophied scar and right perineum which was progressively hard in consistency, along with new complaints were encountered such as Recurrent fever, unable to sit for longer duration and feeling of difficulty in passing stools (flattening and deviation of stools toward left side perineum). On digital examination, there was tenderness with stony hard consistency on the right side anal canal and perineum, all the examination features were directed toward the suspicion of malignancy so for further assessment and management he was advised to undergo investigations (biopsy, magnetic resonance imaging [MRI], and colonoscopy) and to consult oncosurgeon for opinion. The MRI pelvis impression showed fistula-in-ano with collection in the right ischio anal, ischiorectal, and perineal region with inflammatory thickening of rectum and anal canal.

The patient came after a gap of 1 month with severe septicemia, and the cause was diagnosed to be perianal abscess on the same side (right perineum) and for this incision and drainage was performed as emergency management, meanwhile excisional biopsy [Figure 2] of hypertrophied perineal tissue was sent for histopathology study.
Figure 2: Biopsy of fistulous tract

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The patient recovered well and wound healed without any complications. The biopsy report revealed cellular atypia, further to confirm the diagnosis patient was advised for colonoscopic biopsy [Figure 3] to reconfirm the diagnosis. The report confirmed the pathology as large ulcerated rectal growth noted? CA rectum and histopathological report showed mucinous infiltrating mucinous adenocarcinoma.
Figure 3: Colonoscopy report

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  Discussion Top

The manifestation of a carcinoma in chronic fistula-in-ano is possibly due to chronic inflammation although the rarity of the condition precludes any definite assumption in regard to the etiologic relationship of the fistula and carcinoma.[6] A rectal carcinoma may present as a fistula, and it may be difficult to determine whether the tumor is a complication of a long-standing perianal fistula or whether the perianal fistula is merely a manifestation of the malignancy itself.[7] In the present case as the patient got operated for fistula-in-ano at right perineum (Fistulectomy 8 years before) followed by the left side partial fistulectomy with Kshara sutra ligation) during the second surgery, patient was screened to rule out any systemic and local causes for repeated fistula-in-ano by anorectal examination (inspection, digital, and proctoscopy) no local features of malignancy were found. The patient did well postoperatively and postoperative period was uneventful and wound healed completely with Kshara sutra ligation.

Patient repeatedly visited with perineal sepsis on the right side of perineum, but such features were not found on the fellow side, gradually the right perineal side became indurated and hard suggesting chronic fibrosis may be because of incomplete excision of the tract or remnant ramifications which tend to harbor recurrent infection/inflammation to the local area. Therefore, multiple investigations are necessary to rule out local and systemic causes (ulcerative colitis, chron's, tuberculosis, and malignancy) for repeated perineal sepsis and colonoscopy followed with biopsy found to be beneficial in this case which detected mucinous adenocarcinoma of rectum. Fistula-associated anal mucinous adenocarcinoma is an uncommon complication of chronic perianal fistula. The diagnosis is often unsuspected. Thus, tissue from chronic anal fistula tracts should be submitted for pathologic evaluation before the further management.[8]

  Conclusion Top

Carcinoma is rare complication of chronic fistula-in-ano. It is debatable question that fistula leading to carcinoma or carcinoma leads to fistula formation. In this case, recurrent chronic fistula-in-ano with repeated perineal sepsis has led to the formation of mucinous adenocarcinoma so any case of chronic fistula-in-ano and complex fistulas should be subjected for histopathological study before treating and regular follow-ups are necessary for operated cases of fistula-in-ano to rule out the conversion of fistula into malignancy.

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Conflicts of interest

There are no conflicts of interest.

  References Top

Kyzer S, Bayer I, Turani H, Chaimoff C, Verrucous squamous carcinoma as a complication of recurrent multiple perianal fistulae. Coloproctology 1985;7:104-6.  Back to cited text no. 1
Salat A, Trost A, Roka F, Roka S. Human papillomavirus (HPV)-associated proctologic disease. Eur Surg 2006;38:411-6.  Back to cited text no. 2
Heidenreich A, Collarini HA, Paladino AM, Fernandez JM, Calvo TO. Cancer in anal fistulas: Report of two cases. Dis Colon Rectum 1966;9:371-6.  Back to cited text no. 3
Okada KI, Shatari T, Sasaki T, Tamada T, Suwa T, Furuuchi 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;38:555-8.  Back to cited text no. 4
Baars JE, Kuipers EJ, Dijkstra G, Hommes DW, de Jong DJ, Stokkers PC, et al. Malignant transformation of perianal and enterocutaneous fistulas is rare: Results of 17 years of follow-up from the Netherlands. Scand J Gastroenterol 2011;46:319-25.  Back to cited text no. 5
Leal RF, Ayrizono ML, Coy CS, Fagundes JJ, Góes JR. Mucinous adenocarcinoma derived from chronic perianal fistulas: Report of a case and review of the literature. Tech Coloproctol 2007;11:155-7.  Back to cited text no. 6
Nazki S, Chowdari NA, Parray FQ, Wani RA, Samad LA. Adenocarcinom arising from fistula in ano. J Gastrointest Dig Syst 2015;5:370.  Back to cited text no. 7
Santos MD, Nogueira C, Lopes C. Mucinous adenocarcinoma arising in chronic perianal fistula: Good results with neoadjuvant chemoradiotherapy followed by surgery. Hindawi Publ Corp Case Rep Surg 2014;1:386140.  Back to cited text no. 8


  [Figure 1], [Figure 2], [Figure 3]


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