Japanese Journal of Clinical Oncology Advance Access originally published online on November 7, 2005
Japanese Journal of Clinical Oncology 2005 35(11):676-679; doi:10.1093/jjco/hyi181
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© 2005 Foundation for Promotion of Cancer Research
Case Report |
A Case of Rectosigmoid Cancer Metastasizing to a Fistula in ano
1 Department of Surgery and 2 Department of Pathology, Kochi Health Sciences Center, Kochi, Japan
For reprints and all correspondence: Madoka Hamada, Kochi Health Sciences Center, 52125-1 Ike, Kochi-city, Kochi 781-855, Japan. E-mail: KGH03145{at}nifty.com
Received April 11, 2005; accepted August 8, 2005
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Abstract |
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 TOP Abstract INTRODUCTIONCASE REPORTDISCUSSIONReferences |
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We herein report a case of rectosigmoid cancer metastasizing to a fistula in ano. A 53-year-old man complaining of anal bleeding consulted another hospital. He had been suffering from an anal fistula since 7 years. On the left upper side of the skin surface around the anus a fistula end was seen as a hole that tunneled down into the back passage, although no hard tumor was palpable on the hole. Complete colonoscopy revealed an ulcerative tumor in the rectosigmoid colon. On 5 February 2004, anterior resection and lymphadenectomy was performed. The post-operative pathological diagnosis was rectosigmoid cancer, Type 2, T2, N0, M0, stage II. The anal fistula was a simple type and mucinous discharge was not observed. On 23 February 2004, coring out the anal fistula was performed by the former hospital. Pathological diagnosis of the excised fistula revealed well-differentiated adenocarcinoma; identical to the colon tumor. Immunohistochemical staining of these two lesions were negative for (CK) 7 but staining with CK20 revealed some stained tumor cells in two lesions. We diagnosed this tumor as metastatic adenocarcinoma from a rectosigmoid cancer. Recurrent lesions were not seen during the first year after the first operation.
Key Words: implantation metastasis • colorectal cancer • fistula in ano • FDG-PET
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INTRODUCTION |
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TOPAbstractINTRODUCTIONCASE REPORTDISCUSSIONReferences |
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It is well-known that exfoliated colorectal cancer cells can implant on the staple line, resulting in tumor recurrence at the site (1). Implantation of colorectal cancer cells also had been seen at scars on anal mucosa. A similar mechanism is considered to be the cause of metastatic tumors of the anal fistula derived from oral-sided colorectal cancer (2). Sumikoshi, Rosser, Skir, McIntyre and Rundle have established the following diagnostic criteria for primary cancers of the anal fistula: (i) suffering from anal fistula for >10 years; (ii) indurations and severe pain at an anal fistula; (iii) mucus secretion; (iv) stoma aperture in the anal canal and anus crypt; and (v) no tumor at the oral side of an anal fistula. For diagnosis of implantation metastasis, oral-sided large bowel cancer must exist metachronously or synchronously, and the tumor also must not display these five criteria.
In cases of primary anal fistula cancer, abdominoperineal resection (APR) or total pelvic excenteration (TPE) have been adopted for curative surgery. Similarly, metastatic anal fistula cancers have also been treated with APR or TPE (3). However, the small number of these cases makes it difficult to determine the best treatment. For metastatic liver or lung tumors local resection is considered to be the standard treatment leading to the question: If curative resection is possible, is local resection for a metastatic tumor of the anal fistula similarly efficacious? Here, we present a case of metastatic tumor of the anal fistula that was resected locally and followed by adjuvant chemotherapy.
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CASE REPORT |
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TOPAbstractINTRODUCTIONCASE REPORTDISCUSSIONReferences |
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A 53-year-old man complaining of anal bleeding consulted another hospital. On the left upper side of the anus, a fistula end was seen as a hole, which tunneled down into the back passage, although no tumor was palpable on the hole. Total colonoscopy revealed an ulcerative lesion in the rectosigmoid colon, but no lesion on the dentate line. The patient was referred to our hospital for surgical removal of the rectosigmoid cancer. On 5 February 2004, anterior resection and lymphadenectomy was performed. The physical examination showed no specific problems. Levels of serum carcinoembryonic antigen (CEA) and carbohydrate antigen (CA) 19-9 were 1.7 ng/ml and 33.3 U/ml, respectively. The post-operative diagnosis was well-differentiated adenocarcinoma of the rectosigmoid colon, Type 2, T2, N0, M0, stage II (Fig. 1A). The anal fistula was a simple type and mucinous discharge was not observed (Fig. 1B). The post-operative course was uneventful and the patient was discharged on 5 February 2004.
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Coring out for the anal fistula was performed on 23 February 2004 at the former hospital. During the operation no liquid excretion from the stoma was seen, but a hard and well-demarcated tumor in the fistula was palpable. Pathological diagnosis of the tumor was well-differentiated adenocarcinoma; identical to the colon tumor (Fig. 1C and D). The tumor tissue was adjacent to the cut edge but not detected on the stump. Immunohistochemical staining of these two lesions were negative for cytokeratin (CK) 7 but staining with CK20 revealed some stained tumor cells in two lesions (Fig. 2). Despite our strong recommendation of additional surgery (APR or TPE), he refused the operation. He was administrated oral leucovorin (UZEL; Taiho pharmaceutical, Tokyo, Japan) and uracil plus futrafur (UFT; Taiho pharmaceutical) for adjuvant chemotherapy in another hospital.
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On 21 May 2004, FDG-PET scanning of the body revealed abnormal uptake at the rectum (Fig. 3A). On 20 August 2004, second FDG-PET scanning revealed an increased area of abnormal uptake around the rectum, but MRI did not pinpoint any local recurrent lesion. On 30 August 2004, pus discharge from the scar at the anal fistula removal site was observed. Additional excision of the abscess cavity was performed but pathological examination did not reveal any evidence of malignancy. He is currently an outpatient without adjuvant chemotherapy in a clinic, and additional FDG-PET scans taken on 14 December 2005 and 4 February 2005 revealed diminished uptake at the rectal wall (Fig. 3B).
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DISCUSSION |
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TOPAbstractINTRODUCTIONCASE REPORTDISCUSSIONReferences |
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In this case, a tumor of the anal fistula was detected unexpectedly during a requested operation to remove an anal fistula. Carcinomas originating from anal fistula are generally suggested when mucinous discharge, painful indurations, hyperplasia and hypertrophy are observed at the site of external openings (4). Although the patient had suffered with the anal fistula for 7 years, none of these conditions were observed at the time of the initial operation. It is well-known that exfoliated cells of colorectal carcinoma can develop into implantation metastases on injured mucosa, such as anastomosis, whereas they do not usually develop on normal mucosa. For this reason, operation of the anal fistula was avoided at the initial operation. His anal tumor was well confined to the canal of the fistula, and neither contact with large bowel mucosa nor duplication of the colon (5) was observed. For these reasons, the tumor of anal fistula in this case was diagnosed as a metastatic lesion of oral-sided adenocarcinoma of the rectosigmoid colon.
The diagnostic criteria for primary cancer of the anal fistula are based on results of epidemiological study, without consideration of biological evidence. Immunohistochemistry of CK7 and CK20 can be useful for distinguishing colonic tumor tissues from tumors originating from the anal glands. Ramalingam et al. have previously described that anal glands were strongly immunoreactive with antibodies to CK7 but not to CK20 (6). In contrast, rectal adenocarcinomas showed strong immunoreactivity for CK20, and in some cases of them only 20% of the tumor cells stained for CK20. Similarly, in our case, some tumor cells of two lesions were stained with CD20 but not with CK7. We considered the tumor of the anal fistula as metastases of the rectal cancer.
Since there are only 14 reported cases of metastatic tumors of anal fistula, standard treatment for these tumors has not been established (Table 1). In 11 of these cases, APR or TPE were performed with curative intent, as is recommended for primary tumors arising in chronic anal fistulas, although their biological behavior is quite different from metastatic colonic adenocarcinomas. In cases of liver or lung metastases, local resection has been generally adopted, and anatomical resection shows no prognostic advantages (7–10).
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It is thought that exfoliated colorectal carcinoma cells develop into tumors by implantation onto the injured wall and the anastomosis. Is the biological behavior of metastatic lesions of the anal fistula significantly different from that of the liver or lung? In this particular case, we were forced to adopt local resection of the fistula tumor. Nonetheless, FDG-PET images taken 12 months after coring out showed less uptake at the rectum than the previous examination, without adjuvant chemotherapy. Local resection of metastatic fistula cancer may be considered as a possible curative surgery in similar cases (11).
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References |
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1 Gertsch P, Baer HU, Kraft R, Maddern GJ, Altermatt HJ. Malignant cells are collected on circular staplers. Dis Colon Rectum 1992;35:238–41.[CrossRef][Web of Science][Medline]
2 Kouraklis G, Glinavou A, Kouvaraki M, Raftopoulos J, Karatzas G. Anal lesion resulting from implantation of viable tumor cells in a pre-existing anal fistula. A case report. Acta Chir Belg 2002;102:212–3.[Medline]
3 Hyman N, Kida M. Adenocarcinoma of the sigmoid colon seeding a chronic anal fistula. Dis Colon Rectum 2003;46:835–6.[Medline]
4 McIntire JM. Carcinoma associated with fistula-in-ano. Am J Surg 1952;84:610–3.[Medline]
5 Shinohara T, Hara H, Kato Y, Asano M, Nakazawa Y, Kato T, et al. Implantation of rectal cancer cells in a fistula in ano: report of a case. Surg Today 2001;31:1094–6.[Medline]
6 Ramalingam P, Hart WR, Goldblum JR. Cytokeratin subset immunostaining in rectal adenocarcionoma and normal anal glands. Arch Pathol Lab Med 2001;125:1074–7.[Medline]
7 Sugihara K, Hojo K, Moriya Y, Yamasaki S, Kosuge T, Takayama T. Pattern of recurrence after hepatic resection for colorectal metastases. Br J Surg 1993;80:1032–5.[Web of Science][Medline]
8 Girard P, Ducreux M, Baldeyrou P, Rougier P, Le Chevalier T, Bougaran J, et al. Surgery for lung metastases from colorectal cancer: analysis of prognostic factors. J Clin Oncol 1996;14:2047–53.
9 Sakamoto T, Tsubota N, Iwanaga K, Yuki T, Matsuoka H, Yoshimura M. Pulmonary resection for metastases from colorectal cancer. Chest 2001;119:1069–72.
10 Okumura S, Kondo H, Tsuboi M, Nakayama H, Asamura H, Tsuchiya R, et al. Pulmonary resection for metastatic colorectal cancer: experiences with 159 patients. J Thorac Cardiovasc Surg 1996;112:867–74.
11 Gupta R, Kay M, Birch DW. Implantation metastasis from adenocarcinoma of the colon into a fistula-in-ano: a case report. Can J Surg 2005;48:162–3.[Medline]
12 Guiss RL. The implantation of cancer cells within a fistula in ano: case report. Surgery 1954;36:136–9.[Medline]
13 Killingback M, Wilson E, Hughes ES. Anal metastases from carcinoma of the rectum and colon. Aust N Z J Surg 1965;34:178–87.[Medline]
14 Rollinson PD, Dundas SA. Adenocarcinoma of sigmoid colon seeding into pre-exsiting fistula in ano. Br J Surg 1984;71:664–5.[Medline]
15 Ueta K, Umekita N, Matsumine T. A case of metastatic carcinoma found at an anal fistula (in Japanese). J Jpn Surg Assoc 1991;52:1323–6.
16 Thomas DJ, Thompson MR. Implantation metastasis from adenocarcinoma of sigmoid colon in to fistula in ano. J R Soc Med 1992;85:361.[Medline]
17 Isbister WH. Unusual ‘recurrence’ sites for colorectal cancer. Dig Surg 2000;17:81–3.[Medline]
18 Tokuhara K, Yamanaka H, Ito D, Koshiba T, Sato M, Kogire M. A case of metastatic carcinoma at an anal fistula (in Japanese). Jpn J Gastroenterol Surg 2001;34:1690–4.
19 Yoshimura H, Ienaga T, Ueda S, Ota T. A case of metastatic carcinoma of anal fistula caused implantation from rectal cancer (in Japanese). Jpn J Gastroenterol Surg 2001;34:1363–6.
20 Yagihashi N, Ohsawa T, Narita J, Iwabuchi T, Tsuge T. A case of carcinoma metastatic to an anal fistula treated by pelvic exenteration (in Japanese). J Jpn Surg Assoc 2002;63:2224–8.
21 Shimoyama M, Suda T, Iiai T, Okamoto H, Hatakeyama K. A case of metastatic carcinoma of an anal fistula carsed by implantaion of rectal cancer (in Japanese). J Jpn Surg Assoc 2003;64:1434–8.
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