Adenocarcinoma of a Colostomy Following Abdominoperineal Resection for Squamous Cell Carcinoma of the Anal Canal: A Case Study

Abstract

  Malignant neoplasms presenting on a stoma, as well as the development of colorectal adenocarcinoma after previous treatment for squamous cell carcinoma (SCC) of the anal canal, are rare.

The unique case is presented of an 81-year-old woman with parastomal bleeding and ulceration found to have a primary colorectal adenocarcinoma arising de novo on a colostomy, formed after salvage abdominoperineal resection (APR) 3 years earlier for recurrent anal SCC. This is the first reported case of a colonic adenocarcinoma on a colostomy formed after an APR for anal SCC. Although stomal neoplasia is rare, the appearance of a friable bleeding lesion on the stoma should be investigated to exclude metastatic cancer or a second primary malignancy.

Potential Conflicts of Interest: none disclosed

  It is devastating for a cancer survivor to be diagnosed with a new second malignancy. Patients who have had a primary cancer are at higher risk of developing a further malignant neoplasm¹; second cancers accounted for 16% (or almost one in six) of incident cancers reported to the National Cancer Institute’s (NCI) Surveillance, Epidemiology and End Results Program in 2003.² Analysis of the Swedish Family Cancer Database³ revealed that, compared with the general population, cancer patients with an initial cancer at most sites ran a 10% to 50% increased risk for a new primary cancer at a different site. Certain cancers such as those of the lung and cervix⁴ are particularly prevalent after squamous cell carcinoma (SCC) of the anus and reflect shared etiologies such as smoking and human papilloma virus (HPV) infection; however, risk of developing a subsequent colorectal adenocarcinoma following a SCC is not increased.^2,3 Stomal malignancies are also uncommon. Fewer than 50 cases of adenocarcinoma occurring on an ileostomy have been reported,⁵ almost all of which occurred after proctocolectomy for inflammatory bowel disease or familial adenomatous polyposis, although the incidence is greater than that of spontaneous terminal ileal adenocarcinoma.⁶ Colostomy malignancy is even less common, with fewer than a dozen prior cases.⁷

  A case of primary colonic adenocarcinoma on a colostomy that occurred after a salvage abdominoperineal resection (APER) for an anal SCC is described.

Case Report

  When she was 78 years old, Ms. V received radical chemoradiotherapy for an anal canal SCC. She had a good response at first, but 10 months later the anal cancer had recurred and she underwent a salvage APER with complete excision of a pT3N0M0 recurrent squamous carcinoma (ie, tumor >5 cm in size, without lymph node or distant metastases). She made an uneventful recovery. Two years after the APER, Ms. V developed small bowel obstruction from a parastomal hernia and underwent a laparotomy and a resiting of the colostomy to the right iliac fossa (from the left iliac fossa) and repair of the hernia. Eight months later, she presented to her Stoma Nurse Specialist (who referred her to the authors’ facility) with a 1-month history of parastomal ulceration and bleeding from her colostomy. Results of a flexible sigmoidoscopy, performed just before the APER, were normal and other than the anal canal SCC, showed only diverticulosis. Examination of the stoma revealed an irregular 2-cm ulcerated lesion on the superior margin of the stoma. Biopsies confirmed a moderately differentiated adenocarcinoma with no evidence of metastatic disease on cross-sectional imaging; colonoscopic assessment of the remaining colon was normal. Due to Ms. V’s frailty, a limited excision of the stoma and peristomal skin was performed under general anesthetic. She had an uneventful postoperative recovery, requiring routine stoma care. The revised colostomy functioned normally on postoperative day 2. Histology of the resected specimen confirmed complete excision of a pT3N1M0 colonic adenocarcinoma (ie, the tumor had invaded into, but not through, the outermost layer of the colon, with nodal metastases present, but with no evidence of distant metastases). There was no evidence of pre-existing adenoma (see Figure 1). Adjuvant chemotherapy was not offered because of her frailty, but 8 months post procedure Ms. V remains well with no signs of recurrence of either cancer.

Discussion

  Synchronous colorectal adenocarcinomas have an approximately 5% reported incidence⁸; the risk of metachronous cancers is about 3%.⁹ The US National Cancer² database reports that overall almost one in six primary cancers are, in fact, second cancers; Swedish data suggest a slightly lower incidence of 8.5% with a strong association between anogenital SCC and subsequent second cancers.³ Lung and cervical carcinomas are particularly common after anal SCC,⁴ presumably reflecting the shared etiological risks of smoking and HPV infection. No association between anal SCC and subsequent colonic adenocarcinoma has been demonstrated. Only three previous cases of anal SCC and colonic adenocarcinoma have been reported,^10-12 all of which were synchronous lesions. Postmortem and screening colonoscopy studies estimate the prevalence of colonic adenomas in the general population to be up to 40% at age 60 years.¹³ In a cross-sectional study, colonic evaluation of 68 patients with anal SCC revealed only eight adenomas in eight individuals10 and a similar study reported an adenomatous polyp rate of 7% in a cohort of 58 anal SCC patients.¹⁴

  Stomal malignancy in an end ileostomy is reported to be more common than terminal ileal adenocarcinoma in an in situ small bowel⁶; this usually occurs after panproctocolectomy for either inflammatory bowel disease or familial adenomatous polyposis. It is still uncommon; only 45 cases of primary ileostomy adenocarcinoma have been reported⁵ since the first case was described in 1969.¹⁵ Three cases of SCC¹⁶ and two lymphomas¹⁷ also have been reported. Colostomy malignancy is less commonly described — only 11 other cases of colorectal malignancy presenting on an existing colostomy are described in the literature^7,18-20 Of these 11, only one occurred within 5 years of original rectal resection and thus all 11 cases are likely to represent true second primary (metachronous) cancers rather than metastases from the original tumor.⁷ Single cases of basal cell carcinoma,²¹ leimyosarcoma,²² and a Desmoid tumor²³ arising at colostomies formed after rectal carcinoma resection also have been reported.

  The etiology of stomal neoplasms is unclear but prolonged bile acid contact in the stool within the stoma bag¹⁸ or repeated mechanical trauma from repeated application of the stomal appliance have been suggested as relevant.⁷ If either of those theories is true, the risk of neoplasia increases over time and, considering the high numbers of stomas created each year (10,952 abdominoperineal or Hartmanns resections of rectal cancer were performed over a 6-year period in England alone²⁴), more stomal malignancies can be expected.

  The patient in the case reported here received pelvic and perineal radiotherapy as her initial treatment for an anal SCC. It is possible the colonic segment subsequently used to form her end colostomy was included in the radiotherapy field, raising the possibility her second cancer was a late effect of her first treatment, especially in light of the fact she had a clear left-sided colon 32 months earlier and her adenocarcinoma developed de novo without pre-existing adenoma formation. Most of the data for secondary cancers after irradiation come from childhood populations, but there appears to be only a slight increase in risk.²⁵ Retrospective analysis²⁶ of more than 20,000 cases of rectal cancer showed no increased risk of subsequent second primary cancers with previous pelvic irradiation.

  Once they are competent to manage their own stoma, most patients will not have their stoma examined by their clinician unless problems occur. Reports of stomal bleeding, peristomal growths or ulceration, or difficulty fitting the stoma appliance should prompt a close inspection of the uncovered stoma. Educating the patient to ensure prompt assessment if the above signs and symptoms occur is key; early recognition of a stomal malignancy, as with all cancer, will improve outcome. Optimal treatment is by en bloc resection of the stoma, mesentery, and surrounding abdominal wall, with resiting of the colostomy²⁷; however, considering the general frailty and age of many of these patients, they may be better served by a local resection.

Conclusion

  This is the first reported case of a colonic adenocarcinoma on a colostomy almost 3 years following an APER for anal SCC. The stomal lesion arose de novo without adenomatous change and may have been related to the patient’s previous radiotherapy. No association was noted between anal SCC and the subsequent development of a colorectal adenocarcinoma but the appearance of a friable bleeding lesion on the stoma should be investigated to exclude metastatic cancer or a second primary malignancy.

 Mr. Wild is a General Surgical Registrar, Sheffield Colorectal Unit, Northern General Hospital, UK. Mr. Garner is a Consultant Colorectal Surgeon, Rotherham District General Hospital, UK. Mr. Skinner is a Consultant Colorectal Surgeon, Sheffield Colorectal Unit, Northern General Hospital. Please address correspondence to: Jonathan RL Wild, 35 Whirlow Court Road, Whirlow, Sheffield S11 9NS, UK; email: jonnywild@doctors.org.uk.

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