Chagasic megacolon and large bowel neoplasms: case series and literature review

CASE REPORT

Chagasic megacolon and large bowel neoplasms: case series and literature review

Maxwel Capsy Boga Ribeiro^I; Raquel Franco Leal^II; Cláudio Saddy Rodrigues Coy^III; Priscilla de Sene Portel oliveira^IV; Débora Helena Gonçalves Rossi^V; João José Fagundes^VI; Maria De Lourdes Setsuko Ayrizono^II

^IResident of Digestive System Surgery in the School of Medical Sciences of Universidade Estadual de Campinas (UNICAMP) - Campinas (SP), Brazil

^IIDoctor Professor of the Coloproctology Group in the subject of diseases of the digestive system in the School of Medical Sciences of UNICAMP - Campinas (SP), Brazil; Permanent at the Brazilian Society of Coloproctology

^IIIAssociate Professor and Coordinator of the subject of diseases of the Digestive System in the School of Medical Sciences of UNICAMP - Campinas (SP), Brazil; Permanente in the Brazilian Society of Coloproctology

^IVMedical Assistant of the Coloproctology Group in the subject of diseases of the digestive system in the school of Medical Sciences of UNICAMP - Campinas (SP), Brazil; Associated to the Brazilian Coloproctology Society

^VMedical Assistant of the Coloproctology Group in the subject of diseases of the digestive system in the school of Medical Sciences of UNICAMP - Campinas (SP), Brazil

^VIAssociate Professor of the Coloproctology Group in the subject of diseases of the digestive system in the School of Medical Sciences of UNICAMP Campinas (SP), Brazil; Permanent at the Brazilian Society of Coloproctology

^{Correspondence to}

ABSTRACT

There is a clear association between chagasic megaesophagus and the esophageal cancer. On the other hand, the association between chagasic megacolon and intestinal neoplasm is uncommon. There are only a few cases described in literature. We selected two cases of colorectal adenocarcinoma associated with adenoma from 2000 to 2011, which are added to the four patients already described by this group. The mean age of the patients, was 68.5 years. Both had been submitted to surgical resection of the neoplasm. Survival rates ranged and were directly related to tumor staging at the time of diagnosis. In this context, we report our case series and reviwed the corresponding literature, especially the clinical and epidemiological aspects of this rare association.

Keywords: megacolon; Chagas disease; adenocarcinoma.

RESUMO

Há uma clara associação entre megaesôfago por doença de Chagas e o câncer esofágico. Ao contrário, tal relação, entre megacólon chagásico e neoplasias do intestino grosso é, reconhecidamente, incomum. Existem poucos casos relatados na literatura. Destacamos, entre 2000 e 2011, dois casos, sendo ambos adenocarcinomas colorretais e associados a adenomas, que se somam aos outros quatro já descritos por este grupo. A média de idade dos pacientes, foi de 68,5 anos. Todos foram submetidos à ressecção cirúrgica da neoplasia. A sobrevida foi variável e diretamente relacionada ao estádio do tumor no momento do diagnóstico. Dentro desse contexto, relatamos essa série de casos e revisamos a literatura correlata, com relação aos aspectos clínicos e epidemiológicos dessa rara associação.

Palavras-chave: megacólon; doença de Chagas; adenocarcinoma.

INTRODUCTION

The association between esophageal cancer and chagasic megaesophagus is very clear, indexes that range from 22.4 to 9.2%^1-10. Chronic esophagitis secondary to eating cholestasis and the prolonged exposure of the esophageal mucosa to carcinogens that are present in the diet would be predisposing factors for this relation¹¹.

Such association seems to be invalid, and even negative, for large intestine neoplasm and chagasic colopathy, due to the few described cases^5,7,10,12. In endemic regions for Chagas disease, the incidence of colorectal cancer is about 0.1%¹. From 1984 to 2011, in the Coloproctology Service of Hospital de Clínicas of Universidade Estadual de Campinas (UNICAMP), six cases of this rare association were described, the largest case series known, and four had already been reported¹². We bring two cases that occurred in the last decade, their clinical and epidemiological aspects, besides the corresponding literature review.

CASE REPORT

Patient n° 1

A 58 year-old black woman with positive serology and medical record for Chagas disease reported history of intestinal constipation for 15 years, with many episodes of fecaloma. Thirty six months before, she complained of intermittent and painless enterorrhagia. She underwent enema, which showed megarectum and megasigmoid (Figure 1). Colonoscopy showed many polyps, and six were resected (Figure 2). The anatomopathological test (AP) showed intramucosal adenocarcinoma in one of the polyps, located in the rectosigmoid transition. The patient was submitted to total colectomy. The AP of the surgical piece showed 20 polyps distributed in the colon with no malignancy (Figure 3). She presented with postoperative evolution without intercurrences, and remains in outpatient follow-up, without intestinal complaints.

Patient n° 2

A 79 year-old white man with positive serology and medical record for Chagas disease reported history of intestinal constipation for a year, associated with recurring abdominal pain and poorly characterized. He also reported losing 11 pounds in this period. The colonoscopy showed ulcerative lesions in descending colon. The biopsy showed a moderately differentiated adenocarcinoma. The abdominal computed tomography showed dilated rectum and sigmoid, with a great amount of feces and image suggesting descending colon neoplasm, besides multiple hepatic lesions compatible with metastasis. Due to the bowel subocclusion, he was submitted to a segmental colectomy and primary anastomosis (Figures 4 and 5). He had a mixed shock (cardiogenic and pulmonary sepsis), leading to death on the 34^th postoperative day due to multiple organ failure.

Cases reported in literature concerning the association between chagasic megacolon and large intestine neoplasm are demonstrated in Table 1.

DISCUSSION

The relation between chagasic megacolon and large intestine neoplasm is a paradox and also intriguing. First, fecal stasis would lead to chronic bowel irritation and to the prolonged exposure of the mucosa to the carcinogenic factors in the diet. On the other hand, the incidence of chagasic megacolon cancer in endemic regions for Chagas disease is only 0.1%¹.

Garcia and Garcia et al.^13,14 experimentally induced megacolon with the topic application of benzalkonium chloride in the intestinal serosa of Wistar rats, and observed that these animals presented a lower incidence of tumors after being exposed to dimethylhydrazine. Oliveira and Oliveira et al.8,15 observed that Wistar rats chronically infected by Tripanossoma cruzi presented a lower frequency of benign and malignant chemically induced neoplastic colics.

Studies with necropsies and surgical specimens confirmed these findings. Meneses et al.¹⁶ found only one case of colorectal cancer in 198 necropsies of patients with chagasic megacolon, and no cases in 129 surgical pieces from patients with chagasic colopathy. Garcia¹³, on the other hand, found one rectal adenocarcinoma out of the 802 studied surgical specimens.

Changes in the bacterial flora and intestinal pH, as well as in the composition of the affected intestinal wall that would lead to changes in the neurotransmitter and neuropeptide levels, have been studied as protective factors to the occurrence of neoplasms in these patients^13,14,17,18. Concerning the described case series, mean age of our patients was 68.5 years, very close to that observed by Fagundes et al.¹², in 2002; it is also close to the age group of colorectal cancer incidence in non-chagasic patients.

Men were prevalent in the sample of the service.

As to location, we observed that the distal large bowel was mostly affected, and one case was in descending colon, and the other in the sigmoid. Both described adenocarcinomas presented concomitant adenomas. A literature review showed a 36% incidence of adenoma in patients with chagasic megacolon and colorectal neoplasm¹². Thus, adenoma seems to be also a risk factor for colorectal cancer in these patients.

The surgery was related to the tumor location and staging. For patient 1, due to the high number of polyps distributed in the colon, the choice was total colectomy. In case 2, because of the presence of disseminated hepatic metastasis, as well as the clinical performance of the patient, the choice was a palliative partial colectomy.

Generally, literature relates the worst prognosis to intestinal neoplasms that are incident in the chagasic megacolon. This would be related to the late diagnosis in these patients, once the colic dilatation could slow the appearance of obstructive symptoms. We still question if such tumors could not present more aggressive genetics, once they occur in an apparently inhospitable environment for the development of neoplasms.

CONCLUSION

The rarity of large intestine neoplasms in patients with chagasic megacolon does not justify the additional colonoscopic monitoring or the performance of enema in patients whose symptomatology does not indicate it. A high level of suspicion is demanded with the occurrence of any change in the clinical picture of patients with chagasic colopathy.

Finally, more studies are necessary to clear up the factors that are really involved in the relation between chagasic megacolon and colorectal carcinogenesis.

Submitted on: 07/27/2011

Approved on: 09/16/2011

Financing source: none.

Conflict of interest: nothing to declare.

Study carried out at the Coloproctology Service in the subject of Diseases of the Digestive System in the Department of Surgery of the School of Medical Sciences of Universidade Estadual de Campinas (UNICAMP) - Campinas (SP), Brazil.

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Publication Dates

• Publication in this collection
  07 May 2012
• Date of issue
  Dec 2011

History

• Received
  27 July 2011
• Accepted
  16 Sept 2011