Adenocarcinoma occurring from a sigmoid colostomy 20 years after Hartmann's procedure for rectal cancer: A case report

Abstract

Introduction

Cancer arising from the stoma is relatively rare. There is no established surgical procedure for stomal cancer. Furthermore, when a subcutaneous lymphovascular invasion occurs, there is no consensus on whether lymph node dissection along the lymph flow is required. We diagnosed colorectal cancer 20 years after radical resection of rectal cancer.

Presentation of case

We encountered a 70-year-old man who had undergone Hartmann's procedure for rectal cancer 20 years before consultation. Colonoscopy revealed a 30-mm-sized sub-pedunculated polyp with a base at the stoma, and a well-differentiated adenocarcinoma was detected. Approximately 30 mm of the intestinal tract, including the stoma and skin in contact with the tumor, was resected. Pathological examination revealed submucosal invasive cancer with infiltration into the resected skin dermis and invasion of lymphatic vessels under the mucosa. Surgical margins were negative.

Discussion

It is thought that several causes overlap for stomal cancer, although a clear cause of occurrence is yet to be identified. However, as no established surgical procedure exists, the necessity for resection of the lymph nodes without exposure appears indisputable. Although it was reported that skin or subcutaneous metastasis in colorectal cancer is generally regarded as a symptom of systemic metastasis, opinions on the subcutaneous dissection margin of stomal cancer are rarely discussed.

Conclusion

Stomal cancer can be observed macroscopically without colonoscopy. Patients and staff engaged in stoma care should be fully aware that continuous observation of the stoma is necessary even after rectal cancer surveillance is complete.

Highlights

• •We diagnosed stomal cancer 20 years after radical resection of rectal cancer.
• •Colonoscopy revealed a 30 mm subpedunculated polyp with a base at the stoma.
• •Biopsy revealed a well-differentiated adenocarcinoma and radical resection was undergone.
• •Cancer arising from stoma is relatively rare.
• •Staff engaged in stoma care should be fully aware that continuous observation of the stoma is necessary even after rectal cancer surveillance is complete.

1. Introduction

Early and late complications are known to arise after ileostomy or colostomy [1]. A stomal tumor is a relatively rare condition that is classified as a late complication. Among them, there are very few reports of metachronous colorectal cancer at colostomy that have been reported more than 5 years after radical resection [2]^, [3]^, [4]^, [5]^, [6]^, [7]. At diagnosis, invasion into the skin and subcutaneous tissue may already be observed, and extensive resection with reconstruction may be required [4]. In addition, although there is no established surgical procedure for stomal cancer, there has been a case report of lymph node metastasis [6]. Therefore, early diagnosis is important. Herein, we report a case of adenocarcinoma occurring from a sigmoid colostomy diagnosed as bleeding 20 years after radical resection and could be treated with minimal reconstruction. This work has been reported in line with the SCARE 2020 criteria [8].

2. Presentation of case

A 70-year-old man had undergone Hartmann's procedure for rectal cancer 20 years before consultation. He had a history of diabetes and hypertension, but no family history of any cancer. He had completed surgical surveillance and did not have recurrence after the primary surgery. He continued to follow up for diabetes and hypertension treatment. Colonoscopy was performed due to a complaint of bleeding from the mucosal-skin boundary during stoma care. Colonoscopy revealed a 30 mm sub-pedunculated polyp with a base at the stoma (Fig. 1). Biopsy revealed a well-differentiated adenocarcinoma. Tumor markers, including carcinoembryonic antigen and carbohydrate antigen 19–9, were within normal limits. Computed tomography showed no distant metastasis and no subcutaneous infiltration; therefore, primary resection was performed. The surgery was performed under general anesthesia. When the stoma was closed, the skin around the stoma was incised in a ring shape to raise the subcutaneous intestine. During the primary surgery, a colostomy was constructed using the retroperitoneal route, and adhesions were observed between the abdominal wall fat and mesenteric fat. The intestinal tract of approximately 30 mm, including the stoma and skin in contact with the tumor, was resected together. The stoma was reconstructed after the intestinal resection. Although subcutaneous dissection was required because the stoma was created by the retroperitoneal route during the initial surgery, the stoma could be reconstructed at the same site (Fig. 2). After the postoperative period, the patient was discharged without complications. No skin problems were observed around the stoma. Pathological examination revealed submucosal invasive cancer with infiltration into the resected skin dermis and invasion of lymphatic vessels under the mucosa (Fig. 3). Surgical margins were negative. One year after the surgery, the patient survived without recurrence.

Fig. 1.

Endoscopic findings

a) A 30-mm-sized sub-pedunculated polyp with a base at the stoma.

b) The surface of the tumor was rough and bleeding easily. The biopsy result was adenocarcinoma.

Fig. 2.

Surgical findings

a) Macroscopic findings of the tumor

b) The tumor was accompanied by mucus production, and direct infiltration into the skin was suspected.

c) Resected specimen

d) Wound at the end of surgery.

Fig. 3.

Microscopic findings of resected specimen with Hematoxylin & Eosin staining

a, b) Well-differentiated adenocarcinoma with infiltration into the submucosa was observed.

c) Lymphatic infiltration of cancer cells (black arrow) was detected.

d) The tumor infiltrated the dermis.

3. Discussion

Cancer arising from a stoma is relatively rare. In previous reports, adenoma-carcinoma sequence [9], de novo development [2], and suspected implantation [3] have been reported. Some of the proposed mechanisms for the occurrence of stoma tumors include: 1) adenoma-carcinoma sequence, 2) exposure to enterobacteria and bile acids in stool, 3) de novo metaplasia, 4) cancer family syndrome, 5) physical mucosal damage from persistent compression by clothing or stenosis, 6) direct extension of the disease, 7) hematogenous or lymphatic spread, and 8) implantation of exfoliated tumor cells. Stress on the mucous membrane peculiar to the stoma called physical mucosal damage is also thought to contribute. It is thought that several causes overlap, althoguh none of them have been able to explain the rarity of stomal tumors and a clear cause of occurrence is yet to be identified. In particular, in cases such as this case, which are more than 5 years after the initial surgery, it appears that the first surgery has little to do with the development of stomal cancer. In fact, only six cases, including this case, have been reported since 2000, in which radical resection for rectal cancer was performed with no recurrence for 5 years or more and stomal cancer occurred (Table 1).

Table 1.

Stomal cancer reported after 2000, which occurred more than 5 years after radical resection.

No	Author	Year	DFS (y)	Chief complain	Extended resection	Lymph nodes dissection	Metastasis
1	Shibuya	2002	8	Stenosis	−	+	−
2	Chintamani	2007	6	Stenosis	PALN	+	Lymph nodes
3	Vijayasekar	2008	14	Subcutaneous mass	Subcutaneous tissue	+	−
4	Okamoto	2009	20	Constipation	−	N/A	N/A
5	McEntee	2020	14	Nothing	−	+	Lymph nodes
6	Our case	2021	20	Bleeding	−	+	−

DFS: disease-free survival, PALN: paraaortic lymph node, N/A: not available.

The principle of surgery for colorectal cancer with lymph node dissection is complete mesocolonic excision or total mesorectal excision with ligation of the artery. However, there is no established surgical procedure for stomal cancer, and it is necessary to examine the resection range for each case. The presence of lymph node metastasis in the resected specimen is described in the Table 1; however, the necessity for resection of the paraintestinal lymph nodes without exposure seems indisputable. Skin or subcutaneous metastasis in colorectal cancer is generally regarded as a symptom of systemic metastasis [10]. However, the spread of tumors under the skin in stomal cancer has not been investigated, and opinions on the subcutaneous dissection margin are rarely discussed. In addition, in cases of extensive subcutaneous infiltration, such as the case reported by Vijayasekar et al., subcutaneous combined resection and reconstruction are required [4]. In our patient, invasion into the dermis occurred, although mesenteric and subcutaneous lymphovascular invasion was not observed. At present, there is no consensus on whether lymph node dissection along the lymph flow is required when a subcutaneous lymphovascular invasion occurs.

As a reflection of this case, it may have been possible to detect a stomal tumor early by performing colonoscopy and monitoring the stoma. In Japanese colorectal cancer treatment guidelines, surveillance for colorectal cancer that has undergone curative resection has been recommended for five years [11]. In this case, colonoscopy was performed 5 years after the surgery, although no endoscopy was performed until bleeding was reported. The National Comprehensive Cancer Network guideline for rectal cancer recommends repeating colonoscopy every 5 years after rectal cancer surgery, even in cases without adenomas. From the perspective of surveillance for intestinal adenomas, the positions of Europe, the United States, and Japan appear to differ. Western guidelines state that all adenomatous polyps should be resected, and the colonoscopy frequency is set according to these guidelines [12]^, [13]. However, in Japan, it is possible to follow up because of the low cancer-bearing rate of polyps that are 5 mm in diameter or less [14]^, [15]. Therefore, it is arguable to apply the endoscopy intervals followed by Europe and the United States in clinical practice in Japan for better monitoring.

4. Conclusion

Cancer arising from the stoma is relatively rare. Although there are various possible causes, none of them have been able to explain the rarity of stomal tumors. Stomal cancer can be observed macroscopically without colonoscopy. Patients and staff engaged in stoma care should be fully aware that continuous observation of the stoma is necessary even after rectal cancer surveillance is complete.

Sources of funding

Nothing to declare.

Ethical approval

This research was approved by the Local Ethics Committee of the Chofu Touzan Hospital (No. 2021-1028-1) and was conducted according to the guidelines put forth in the Declaration of Helsinki.

Consent

This case presentation was approved by the Local Ethics Committee of the Chofu Touzan Hospital (No. 2021-1028-1). Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.

Research registration

Researchregistry7321 available at: https://www.researchregistry.com/register-now#home/registrationdetails/617f6fe33f935f00228a8282/

Guarantor

Yusuke Kitagawa

Provenance and peer review

Not commissioned, externally peer-reviewed.

CRediT authorship contribution statement

YK made substantial contributions to the study conception and design, acquisition of data, and analysis and interpretation of data. MB was involved in drafting the manuscript and revising it critically for important intellectual content. SH and MB participated in discussions about this study. All the authors read and approved the final manuscript.

Declaration of competing interest

The authors report no declarations of interest.