Case Report
Copyright ©The Author(s) 2019. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Jul 6, 2019; 7(13): 1643-1651
Published online Jul 6, 2019. doi: 10.12998/wjcc.v7.i13.1643
Colon cancer arising from colonic diverticulum: A case report
Hajime Kayano, Yusuhiko Ueda, Takashi Machida, Shinichiro Hiraiwa, Hidenori Zakoji, Takuma Tajiri, Masaya Mukai, Eiji Nomura
Hajime Kayano, Yusuhiko Ueda, Takashi Machida, Masaya Mukai, Eiji Nomura, Department of Gastroenterological and General Surgery, Tokai University Hachioji Hospital, Tokyo, Hachioji 192-0032, Japan
Shinichiro Hiraiwa, Takuma Tajiri, Departments of Pathology, Tokai University Hachioji Hospital, Tokyo, Hachioji 192-0032, Japan
Hidenori Zakoji, Departments of Urology, Tokai University Hachioji Hospital, Tokyo, Hachioji 192-0032, Japan
ORCID number: Hajime Kayano (0000-0002-8304-1136); Yasuhiko Ueda (0000-0003-1130-9209); Takashi Machida (0000-0002-4122-1016); Shinichiro Hiraiwa (0000-0003-4946-9144); Hidenori Zakoji (0000-0002-9886-5429); Takuma Tajiri (0000-0002-3051-4489); Masaya Mukai (0000-0001-6120-0012); Eiji Nomura (0000-0002-7365-115X).
Author contributions: All authors contributed to the acquisition of data, writing, and revision of this manuscript.
Informed consent statement: Consent was obtained from the patient for publication of this report and any accompanying images.
Conflict-of-interest statement: All the authors declare that they have no conflict of interest.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
Corresponding author: Hajime Kayano, MD, PhD, Assistant Professor, Doctor, Surgeon, Department of Gastroenterological and General Surgery, Tokai University Hachioji Hospital, 1838 Ishikawa-machi, Tokyo, Hachioji 192-0032, Japan. h.kayano@tsc.u-tokai.ac.jp
Telephone: +81-42-6391111 Fax: +81-42-6391144
Received: February 26, 2019
Peer-review started: February 27, 2019
First decision: April 18, 2019
Revised: April 28, 2019
Accepted: May 1, 2019
Article in press: May 1, 2019
Published online: July 6, 2019

Abstract
BACKGROUND

Colonic diverticulosis is a common disease, and the coexistence of colonic diverticulosis and colorectal cancer is often seen clinically. It is very rare that colon cancer arises from the mucosa of a colonic diverticulum. When colon cancer arises in a diverticulum and then tends to develop outside the wall, without developing within the lumen, the differential diagnosis from complicating lesions due to colonic diverticulitis is difficult.

CASE SUMMARY

A 76-year-old man was admitted to a nearby clinic with a chief complaint of discomfort and urinary frequency. Since a vesicosigmoidal fistula was seen on abdominal computed tomography, he was referred to our hospital. Laparoscopic sigmoidectomy was performed because the various diagnostic findings were diagnosed as a vesicosigmoidal fistula with diverticulitis of the sigmoid colon. However, on histopathological examination, it was diagnosed as a vesicosigmoidal fistula due to colon cancer arising in the diverticulum. Laparoscopic partial resection of the bladder was performed because local recurrence was observed in the bladder wall one and a half years after surgery. It is currently one year after reoperation, but there has been no recurrence or metastasis.

CONCLUSION

Colon cancer arising in a diverticulum of the colon should be considered when diverticulitis with complications is observed.

Key Words: Colon cancer, Diverticulum, Colovesical fistula, Laparoscopic surgery, Diverticulitis, Case report

Core tip: Cancers arising from a diverticulum of the colon are extremely rare, and few cases have been reported. Cancer of a colonic diverticulum has difficulty progressing into the lumen, and it is often difficult to identify by imaging examinations. In addition, due to the anatomical characteristics of the diverticulum, it is easy for the cancer to progress outside the colon wall. Thus, it is often in an advanced stage at the time of discovery, and careful attention is needed.



INTRODUCTION

The co-existence of colorectal diverticulosis and colorectal cancer is seen clinically, and so far there have been many research reports and case reports[1-4]. However, it is extremely rare for colon cancer to develop from within a diverticulum of the colon. In this case, laparoscopic surgery was performed for a vesicosigmoidal fistula with diverticulitis of the sigmoid colon, and the postoperative histopathological examina-tion showed colon cancer arising in the diverticulum. The clinical features of colon cancer arising in the diverticulum are reviewed, along with past reports[5-20] and including this case.

CASE PRESENTATION
Chief complaints

Discomfort during urination and frequent urination.

History of present illness

A 76-year-old man became conscious of urgency when urinating three months earlier and developed frequent urination. He also recognized food residue in the urine three days earlier and consulted a nearby clinic. Since a vesicosigmoidal fistula was found on abdominal computed tomography (CT), he was referred to our hospital and admit-ted for the purpose of intensive examination and treatment.

History of past illness

The patient had a history of spinal posterior fusion surgery for disc herniation. There was no history of trauma, abdominal disease, abdominal surgery, or urological disease. Moreover, there was no history of colonoscopy or upper esophagogastro-duodenoscopy.

Personal and family history

The patient was taking Olmetec OD tablets (olmesartan medoxomil) for hypertension, Allopurinol tablets (Allopurinol) for hyperuricaemia, and Urief tablets (Silodosin) and Cernilton tablets (cernitin pollen extract) for prostate enlargement. His family medical history was unremarkable.

Physical examination upon admission

The patient’s abdomen was smooth and soft, with no tenderness and no palpable tumor.

Laboratory examinations

The results of hematological examinations were: White blood cell count 8200/μL; C-reactive protein 4.635 mg/dL; Hb 11.8 g/dL; Plt 15.3 × 104/μL; BUN 19 mg/dL; Cre 1.00 mg/dL; and HbA1c 6.1%. Thus, there was a mild inflammatory response and anemia. The tumor markers were not elevated (CEA 1 ng/mL, CA19-9 15.3 U/mL). On urinalysis, the leukocyte reaction was 3+, and occult blood was 1+.

Imaging examinations

Enhanced abdominal CT showed multiple diverticula in the sigmoid colon, and an increased CT value of panniculitis was observed around the large intestinal tract. Inflammatory changes around the large intestinal tract continued to the left bladder wall, and the bladder wall was slightly thickened. Neither a tumorous lesion nor swelling of the para colic lymph nodes was seen (Figure 1A). Similarly, enhanced abdominal magnetic resonance imaging (MRI) showed a penetrating portion continuous with the sigmoid colon on the upper left side wall of the bladder. However, tumorous lesions were not seen (Figure 1B). With an enema of a water-soluble contrast agent (Figure 1C) and colonoscopy, only multiple diverticula were found in the sigmoid colon. Cystoscopy showed inflammatory changes on the mucosal surface of the bladder, but there was no fistula; no intestinal fluid flowed into the bladder, and no tumorous lesion was seen.

Figure 1
Figure 1 Preoperative computed tomography, magnetic resonance imaging, and gastrografin enema examination. A: The inflammatory changes around the intestinal tract continue to the left bladder wall, and the bladder wall is slightly thickened. Tumor cannot be seen; B: A penetrating portion continuous with the sigmoid colon is visible on the upper left side wall of the bladder; C: Only a diverticulum is found in the sigmoid colon.
Preoperative diagnosis

Vesicosigmoidal fistula with diverticulitis of the sigmoid colon.

TREATMENT

Laparoscopic sigmoidectomy and fistulectomy were performed. At surgery, fistula formation was observed in the sigmoid colon and the upper left side of the bladder wall (Figure 2). To resect the fistula, blunt dissection was first performed using a suction tube followed by sharp dissection using bipolar scissors. Although the fistula was somewhat firm, it was relatively easily separated from the surrounding tissue during the dissection. The bladder wall was preserved because no tumorous lesions that suggested cancer were observed. Finally, sigmoidectomy was performed. Anastomosis was completed with a double stapling technique, and the operation was completed. A colostomy was not created.

Figure 2
Figure 2 Intraoperative findings. A portion of the fistula with the sigmoid colon is observed on the upper left side of the bladder wall.
FINAL DIAGNOSIS

In the resected specimen, macroscopic findings only showed diffuse diverticula in the sigmoid colon lumen. A diverticulum was found in the area of fistula formation, but no tumorous lesions were observed (Figure 3A, 3B). On microscopic examination, a protruding lesion appeared toward the diverticular lumen, and well-differentiated cylindrical cancer cells proliferated in a papillary or tubular pattern, indicating a diagnosis of well-differentiated adenocarcinoma (Figure 3C). The depth of invasion was diagnosed as exceeding the serosal surface, and the resection margin was positive. Metastasis was not observed in the para colic lymph nodes near the lesions. The patient was offered additional resection, but refused. Thus, strict observation was performed. For follow-up, the patient’s serum carcinoembryonic antigen level was tested at a 3-mo interval, chest and abdomino-pelvic CT were performed at a 6-mo interval, and colonoscopy was performed at a 1-year interval.

Figure 3
Figure 3 Histopathological examination findings. A, B: Macroscopically, several diverticulum are visible in the colonic lumen, but tumorous lesions around the fistula are not observed; C: Histologically, there is tumor characterized by mild exophytic papillary growth (arrowheads) along the diverticular lumen (arrows). It was diagnosed as well-differentiated adenocarcinoma (HE staining, × 100).
OUTCOME AND FOLLOW-UP

A tumorous lesion about 1-cm in size was noted in the bladder wall on enhanced abdominal CT (Figure 4) one and a half years after the operation, and tumor biopsy was performed transurethrally by urological surgery. On histopathological examination, well-differentiated adenocarcinoma was recognized, which was similar in appearance to the cancer in the diverticulum operated previously, and it was diagnosed as recurrence in the bladder wall. The condition of the disease was again explained to the patient and family, and informed consent was obtained for laparoscopic partial resection of the bladder. On follow-up after the surgery, there has been no recurrence or metastasis one year after re-operation.

Figure 4
Figure 4 Computed tomography one and a half years after surgery. A tumor lesion about 1 cm in size is seen in the bladder wall.
DISCUSSION

The co-existence of colonic diverticulosis, colon cancer, and adenoma is often seen clinically. However, there have been various reports as to whether there is a difference in the incidence of colorectal cancer due to the presence or absence of colonic diverticula, and the relationship is unclear[1-4]. As for the effect of the presence of diverticula on carcinogenesis, it has been reported that carcinogenesis may be caused by repeated chronic inflammation due to diverticulitis and changes in the intestinal bacterial flora caused by multiple diverticula[5]. On the other hand, colorectal cancer that arises in the diverticulum of the colon is extremely rare. The relevant English literature was retrieved from the PubMed database from 1990 to 2018 using the keywords “colon cancer”, “diverticulum”, “diverticulosis”, and “diverti-culitis”. The clinical features of cancers in diverticula were considered based on the 17 cases identified and the present case[5-20] (Table 1). The cancer was detected due to the appearance of various symptoms, such as abdominal pain, urination pain due to abscess formation, and bleeding from a tumor. Abdominal examination was performed, but only 47% (8/17) of cases were diagnosed with colorectal cancer by colonoscopy or enema before surgery, and in abdominal CT, only 37% (4/7) of cases were suspected colorectal cancer. Suspected colorectal cancer/colorectal cancer was diagnosed in about 50% (9/18) of cases preoperatively. About half of the cases were operated for complications due to colonic diverticulitis, and in many cases the diagnosis of colorectal cancer arising in the diverticulum was made for the first time after surgery. Even in the present case, it was not possible to identify the colorectal cancer in all examinations performed, including enhanced abdominal CT, enhanced abdominal MRI, and enema and colonoscopy. Therefore, it was decided to perform surgery based on the diagnosis of a vesicosigmoidal fistula caused by sigmoid colon diverticulitis. On pathological examination, no tumorous lesion was observed on macroscopic observation. Microscopic findings showed that the tumor developed slightly in the mucous membrane of the luminal side, but progressed mainly from the inside of the diverticulum in which the muscularis propria was missing. Colorectal cancer generally grows and progresses along the mucous membrane direction (horizontal development) and the membrane direction (vertical development). Compared with common colorectal cancer, the colon cancer that develops from within a diverticulum has less mucosal direction toward the luminal side (horizontal development), despite the fact that the cancer propagates easily from the diverticulum in the direction of the serosa (vertical development) out the wall. This is due to the anatomical characteristics of the diverticular lumen, which spreads widely in the colon diverticulum, but the diverticular orifice area is narrower than the diverticular lumen. Thus, it seems that it is difficult for the cancer to develop in the direction of the luminal side. Unlike usual colorectal cancer, due to such anatomical features that restrict development, it is difficult to identify lesions on colonoscopy with an enema of the lower gastrointestinal tract. Furthermore, another anatomical characteristic of the colonic diverticulum is that it is a pseudo diverticulum in which the muscularis propria is deficient, and compared with usual colorectal cancer, colon cancer that arises from within the diverticulum progresses more in the direction of the membrane towards the mucosal surface than in the direction of the mucosal surface. Therefore, at the time of detection, the depth of invasion is already above T3 in over 80% (15/18) of cases. Furthermore, infiltration into other organs is not uncommon in T4a cases (4/18 22.2%). As to the histopathological type, there were 5 cases of mucinous carcinoma (5/18, 27%). Since it is generally reported that mucinous carcinoma is seen in 3.5% to 5.1% of colon cancer cases[21-23], this is a higher rate than in normal colon cancer. Since the histopathological feature of mucinous carcinoma is development to the center of the submucosa, and it is difficult for it to be exposed on the mucosal surface, early detection is difficult. In the case of multiple diverticula, there are cases where the intestinal wall hardens or shortens and the lumen narrows due to chronic inflamma-tion; therefore, detailed observation of mucous membranes is often difficult in colonoscopy. From the above, it was thought that cancers arising from the inside of the diverticulum of the colon are difficult to identify by various examinations and are difficult to detect at an early stage. As a result, it is necessary to always keep in mind the coexistence of colon cancer in patients with colon diverticulosis/diverticulitis.

Table 1 Clinical characteristics of reported cases of colon cancer arising in a colonic diverticulum.
CaseRef.YrAge (yr)/ SexSymptomSeen on CTSeen on CS/CEPreoperativediagnosisTumor siteOperationTNM (UICC)Differentiation
1Tolley et al[6]196759/MConstipationND2YesDiverticulitisCecumRight hemicolectomyT4N0M0Mucinous
2Drut et al[7]197484/MAbdominal painNDNoDiverticulitisSigmoidLeft hemicolectomyT4N3M0Adenosqua-mous
3Hines et al[8]197555/FAbdominal painNDNoDiverticulitisDescendingLeft hemicolectomyT4N0M0Mucinous
4McCrow et al[5]197680/MHematocheziaNDNoDiverticulitisSigmoidSigmoidectomyT3N0M0Mucinous
5Prescott et al[9]199289/FAbdominal painNDNoGiant diverticulumSigmoidSigmoidectomyT3N0M0Well
6Cohn et al[10]199380/MAbdominal painNDNDIleusSigmoidHartmann operationT3N0M0Mucinous
7Cohn et al[10]199361/MHematocheziaNDYesBulky tumorSigmoidSigmoidectomyT2N0M0Well
8Kajiwara et al[11]199667/MHematocheziaNoYesCancerAscendingRight hemicolectomyT3N0M0Mucinous
9Kikuchi et al[12]199958/FNo symptomNDYesTumorCecumIleocecal resectionTisNxM0Moderate
10Bellows et al[13]200263/MHematocheziaNoNoVesicosigmoidal fistulaSigmoidSigmoidectomy Partial resection of the urinary bladderT4aN0M0Moderate
11Van Beyrden et al[14]200854/MNDNoNoDiverticulitisSigmoidSigmoidectomyT4aN2M0Moderate
12Fu et al[15]201071/MHematocheziaNDYesTumorDescendingEMR with the assistance of laparoscopyTisNxM0Well
13Merkow et al[16]201160/MAbdominal painYesNoBulky tumorSigmoidSigmoidectomyT4bN2M0Poorly
14Parsyan et al[18]201360/MNDYesYesCancerSigmoidLeft hemicolectomyT3N1M0Moderate
15Yagi et al[19]201473/MHematuriaYesYesCancerSigmoidSigmoidectomy, total resection of the urinary bladderT4bN0M0Well
16Nomi et al[20]201464/FAnemiaYesNoBulky tumorTransverseRight hemicolectomyT4bN0M0Well
17Our case-76/MDysuriaNoNoVesicosigmoidal fistulaSigmoidLaparoscopic sigmoidectomyT4bN0M0Well

Colon diverticulosis/diverticulitis is usually diagnosed by first performing abdominal CT examination in most cases. If there is no inflammation and only diverticula exist, careful examination such as colonoscopy cannot be performed. On the other hand, as to whether colonoscopy is necessary for colon diverticulitis found on abdominal CT, some studies[24-26] have reported that colonoscopy is not necessary for colon diverticulitis without complications, but in cases with complications such as abscess formation and penetration, the possibility of finding colon cancer or polyps increases. Moreover, it has been reported that CT findings of a wall thickness > 6 mm, abscess, lymphadenopathy, localized mass, and obstruction are predictive of malignancy[27]. One report also noted that patients with CT findings of local perfora-tion, abscess, and fistula have higher odds of malignancy than patients with diverticulitis without complications[25].

Based on the present case, it was considered important to conduct a detailed examination at the time of colonoscopy. Even when a preoperative diagnosis is made, many cases present with T4 invasion, so treatment requires combined resection. In cases where early detection was possible, even for Tis cases, which are oncologically indicated for endoscopic resection, because it is difficult to safely and completely excise tumorous lesions in diverticula without endoscopically causing perforation, some cases[15] are treated with combined laparoscopic surgery. In this case, it seems that recurrence could be prevented by performing partial resection of the bladder in addition to fistula resection as initial surgery. However, in the present case, no lesions were seen before surgery, and the presence of cancer was not considered. Moreover, surgery for a colonic urinary bladder fistula due to colon diverticulitis has been performed previously with colon resection and partial resection of the bladder, but recently, partial resection of the bladder has not been considered necessary[28,29]. From the above two perspectives, partial resection of the bladder was not performed. However, in cases of complications of abscess formation and fistula formation of colonic diverticulitis, given the possibility of cancer developing in a colonic diverti-culum as in the present case, even in the absence of mass lesions on the luminal side, it is considered necessary to add intraoperative rapid pathologic diagnosis for the fistula.

CONCLUSION

It is difficult to detect cancer arising in a diverticulum of the colon, and it is not easy to differentiate it from complications caused by diverticulitis. Therefore, with this in mind, imaging examinations and surgery are needed.

Footnotes

Manuscript source: Unsolicited manuscript

Specialty type: Medicine, Research and Experimental

Country of origin: Japan

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