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Implantation metastasis of rectosigmoid cancer in an anal fistula.

Byline: Weifeng Lao, Jing Deng, Sameer Memon, Weifang Mao and Chao He

Implantation metastasis of colorectal cancer in an anal fistula is very rare. We report a case of a 61 years old male who underwent fistulectomy for an anal fistula. Histopathology unexpectedly revealed adenocarcinoma in the fistula track, however the patient refused further treatment. Sixteen months later he presented with an obstructing locally advanced rectosigmoid cancer found to be fixed into the pelvic wall. An ileosigmoid bypass was fashioned and he was treated with neoadjuvant chemoradiotherapy followed by high anterior resection. Histopathology confirmed a colorectal adenocarcinoma, Immunohistochemistry of the tumors from both sites was CK7-/CK20+. The patient died 34 months later with liver and lung metastasis however no perianal recurrence occurred. Local resection with or without radiotherapy, instead of abdominoperineal resection, was feasible for control of perianal metastatic lesion implanted from colorectal cancer if local extended resection was possible.

KEY WORDS: Implantation metastasis; Colorectal cancer; Anal fistula.

How to cite this article:

Lao W, Deng J, Memon S, Mao W, He C. Implantation metastasis of rectosigmoid cancer in an anal fistula. Pak J Med Sci 2012;28(1):209-212

INTRODUCTION

Adenocarcinoma in an anal fistula is rare. It can arise as a complication of long-standing chronic inflammation or by metastatic implantation from a colorectal cancer. We present a case of anal fistula implantation metastasis from a rectosigmoid adenocarcinoma successfully managed with local excision and review the literature. The evidence suggests local excision appears to provide good local control if negative margins can be achieved.

CASE REPORT

A 61-year-old man with a six month history of perianal pain and purulent discharge underwent fistulectomy in June 2006 for a subcutaneous anal fistula. The internal and external openings of the fistula were clearly identified at the operation and the track was completely excised. Histopathology revealed an unexpected diagnosis of moderately differentiated adenocarcinoma near the external opening (Fig.1). However the patient refused further investigation. The incision was completely healed five weeks later. Sixteen months later he presented with fecal urgency and PR bleeding. Colonoscopy revealed a circumferential ulcerating lesion 18cm from the anal verge, confirmed on biopsy to be a moderately differentiated adenocarcinoma. The perianal scar was soft with no palpable mass. Computerized tomography (CT) showed irregular thickening of rectosigmoid wall and a locally invasive mass (Fig.2).

Laparotomy revealed an irresectable 10x12cm mass fixed to the retroperitoneum, the terminal ileum and the right psoas muscle. Side-to-side ileo-ileostomy and sigmoid colostomy were fashioned and the patient was subsequently treated with seven cycles of sandwich FOLFOX4 chemotherapy and a total of 50-Gy of radiotherapy. Six weeks following completion of treatment CT showed downsizing of the mass (Fig.2) and the patient underwent anterior resection, small bowel resection with defunctioning ileostomy. Histopathology revealed a ypT3N0 moderately differentiated adenocarcinoma. Immunohistochemistry stained CK7-/CK20+ (Fig.1). The patient made a good recovery and was treated with five further cycles of FOLFOX4 chemotherapy. In July 2009, screening CT revealed widespread liver and lung metastases. However there was no evidence of recurrent perianal disease. The patient died from pulmonary failure 17 months later.

DISCUSSION

Adenocarcinoma can arise in an anal fistula though two mechanisms. Malignant change can occur secondary to longstanding chronic inflammation or malignant implantation of cancer cells may occur from a remote site. Ryall1 was the first to report implantation metastasis in 1907. This is now a well accepted mechanism for the metastatic recurrences observed in surgical wounds and port sites. Shed cancer cells are unable to implant on intact epithelial membranes, however, de-epithelialised surfaces such as hemorrhoidectomy wounds2,3, biopsy tracks4 and anastomotic site5 provide a more fertile environment for implantation. Implantation metastasis from colorectal adenocarcinoma to anal fistula is rare. Guiss6 reported the first case in

Cancer implantation in anal fistula

Table-I: Reported cases of implantation metastasis from colorectal cancer to an anal fistula.

Author, year###Age###Sex###Loc###Dukes###Dur###Histology###Operation###Status###LR###DM

Guiss,1954###47###M###S###A###2m###mod###APR###14m(A)###NA###NA

Killingback,1965###63###M###S###A###8y###well###APR###NA###NA###NA

Rollinson,1984###65###M###Rs###NA###20y###well###APR###10m(A)###NA###NA

Thomas,1992###68###M###S###B###1y###mod###APR###NA###NA###NA

Isbister,2000###47###M###Rs###C###2y###mod###NA###NA###NA###NA

###39###M###S###NA###1y###well###LoR###NA###NA###NA

Shinohara,2001###36###M###R###C###16y###mod###LAR,LoR###6m(A)###No###Liver

Kouraklis,2002###75###M###S###B###1y###mod###APR###NA###NA###NA

Hyman,2003###66###M###S###B###15y###mod###APR###1y(A)###No###No

Gupta,2005###44###M###L###C###NA###mod###LH+LoR###3y(A)###No###No

Madoka,2005###53###M###S###B###7y###well###AR, LoR###1y(A)###No###No

Satoshi,2006###53###M###R###C###20y###mod###LoR, LAR###10m(D)###No###Peritoneum

Sandiford,2006###72###M###Rs###B###2y###mod+muc###i AR+LoR###14m(A)###No###No

Wakatsuki,2008###57###M###Rs###C###7y###mod###AR, LoR###43m(A)###No###Liver

Gravante,2008###64###M###L###C###8m###mod###LR, APR###12m(A)###No###No

Our case###61###M###Rs###B###6m###mod###LoR, AR###54m(D)###No###Liver, lung

Loc, location; Dur, duration of anal fistula; Diff, differentiation; M, male; S, sigmoid colon; Rs, rectosigmoid region; R, rectal; L, left colon; m, month; y, year;mod, moderatedifferentiated adenocarcinoma; well, well-differentiated adenocarcinoma; muci, mucinous adenocarcinoma; APR, abdominoperineal resection; LoR, local resection; LAR, low anterior resection; AR, anterior resection; LH, left hemicolectomy; 'LAR, LoR', LAR followed by LoR; 'LH+LoR', homeochronous LH and LoR; A, alive; D, dead; NA, not available, LR, local recurrence; DM, distant metastasis.

1954. Only fifteen cases have been reported so far. All these cases occurred in patients with a history of anal fistula for two months 16 years duration and diagnosis was confirmed by confirming similar histopathological characteristics between the primary cancer and the perianal lesion. Considering the possibility of a primary adenocarcinoma developing in an anal gland, Satoshi7 defined the presence of a mucinous component on pathology as an exclusion criterion for diagnosing an implantation metastasis. However, Sandiford8 later reported a case of implantation metastasis from a moderatedifferentiated adenocarcinoma with extensive mucinous component. Immunohistochemical staining of CK7 and CK20 is helpful to differentiate cells of colorectal origin from anal orgin. Ranalingam9 reported that 87% of rectal adenocarcinomas had a CK7-/CK20+ immunophenotype, which is seen in colon adenocarcinomas. However anal gland and anal transitional mucosa has is CK7+/CK20-.

The similar immunophenotypes of the malignancies in the case we report confirms the metastatic nature of the malignancy in the fistula.

There are no recommendations for the management of colorectal cancer implantation metastases to anal fistulae. Although abdominoperineal resection was performed in 7/15 of the reported cases, more recent reports have reported successful manage-ment with sphincter sparing surgery and combined with local resection with or without radiotherapy to manage the implantation metastasis, without local recurrence. In our case, there was no evidence of perianal recurrence after four years following management with local excision and chemoradiotherapy. The evidence suggests local excision with or without radiotherapy appears to provide good local control for perianal metastatic colorectal adenocarcinoma if negative margins can be achieved. However, others have advocated a more aggressive approach. Gravante10 has suggested that even when a benign anal lesion was excised in the presence of a colorectal malignancy, abdominoperineal resection should be strongly considered.

Metastatic cancer has different biological characteristics and different surgical stratagem compared to its primary cancer in a certain organ. For example, the surgical margin of zero to four mm is acceptable for a hepatic lesion metastasized from colorectal cancer.11 However for primary hepatocellular carcinoma, surgical margin was advised more than 10mm.12 Local resection of liver metastatic lesions with generous margin has been achieved encouraging outcome, which highlight the feasibility of local resection, instead of APR, for implantation metastasis of anal fistula from colorectal adenocarcinoma, to avoid a colostomy.

REFERENCES

1. Ryall C. Cancer infection and cancer recurrence. Lancet 1907;2:1311-6.

2. Beahrs OH, Phillips JW, Dockerty MB. Implantation of tumor cells as a factor in recurrence of carcinoma of the rectosigmoid: report of four cases with implantation at dentate line. Cancer 1995;8:831-8.

3. Hsu TC, Lu IL. Implantation of adenocarcinoma on hemorrhoidectomy wound. Inter J Colorectal Dis 2007;22:1407-8.

4. Basha G, Ectors N, Penninckx F, Filez L, Geboes K. Tumor cell implantation after colonoscopy with biopsies in a patient with rectal cancer: report of a case. Dis Colon Rectum 1997;40:1508-10.

5. Norgren J, Svensson JO. Anal implantation metastasis from carcinoma of the sigmoid colon and rectum: a risk when performing anterior resection with EEA stapler. Br J Surg 1985;72:602.

6. Guiss RL. The implantation of cancer cells within a fistula in ano. Surgery 1954;3:136-9.

7. Satoshi I, Satoshi I, Katsutoshi K, Yoshifumi S, Noriaki K, Yoji Y, et al. Implantation of Rectal Cancer in an Anal Fistula: Report of a Case. Surg Today 2006;36:747-9.

8. Sandiford N, Prussia PR, Chiappa A, Zbar AP. Synchronous mucinous adenocarcinoma of the rectosigmoid seeding onto a pre-existing anal fistula. Int Semi Surg Oncol 2006;3:25.

9. Ramalingam P, Hart WR, Goldblum JR. Cytokeratin subset immunostaining in rectal adenocarcionoma and normal anal glands. Arch Pathol Lab Med 2001;125:1074-7.

10. Gravante G, Delogu D, Venditti D. Colosigmoid Adenocarcinoma Anastomotic Recurrence Seeding Into a Transsphincteric Fistula-in-ano: A Clinical Report and Literature Review. Surg Laparo Endo Per 2008;18:407-8.

11. Lochan R, White SA, Manas DM. Liver resection for colorectal liver metastasis. Surg Oncol 2007;16:33-45.

12. Tsim NC, Frampton AE, Habib NA, Jiao LR. Surgical treatment for liver cancer. World J Gastroenterol 2010;16:927-33.

1. Weifeng Lao, MD,

2. Jing Deng, MD, Department of Clinical Medicine, Zhejiang Medical College, Hangzhou, Zhejiang, China

3. Sameer Memon, MBChB, FRACS, Department of Cancer Surgery, Peter MacCallum Cancer Centre, Melbourne, Australia.

4. Weifang Mao, MD,

5. Chao He, MD, 1,4,5: Department of Colorectal Surgery,

Sir Run Run Shaw Institute of Clinical Medicine, Sir Run Run Shaw Hospital, Zhejiang University School of Medicine, Hangzhou, Zhejiang, China.

Correspondence: Chao He, Department of Colorectal Surgery, Sir Run Run Shaw Hospital, School of Medicine, Zhejiang University, 3 Qing Chun Road East, Hangzhou, Zhejiang, China 310016.
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Publication:Pakistan Journal of Medical Sciences
Article Type:Report
Geographic Code:9PAKI
Date:Mar 31, 2012
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