Case Report Open Access
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World J Gastroenterol. Mar 7, 2015; 21(9): 2862-2864
Published online Mar 7, 2015. doi: 10.3748/wjg.v21.i9.2862
Perforation of small bowel caused by Schistosoma japonicum: A case report
Jin-Jian Xiang, Ben-Jing Cheng, Fu Tian, Mi Li, Xue-Feng Jiang, Xiao-Miao Hu, Bao-Lai Xiao, Jian-Ping Xie, Arpana Shrestha, Department of General Surgery, the First Hospital affiliated to Yangtze University, Jingzhou 434000, Hubei Province, China
Hui-Chuan Zhao, Department of Pathology, the First Hospital Affiliated to Yangtze University, Jingzhou 434000, Hubei Province, China
Author contributions: Xiang JJ and Cheng BJ designed the report; Xiang JJ, Tian F, Li M and Jiang XF performed surgical operation; Hu XM, Xiao BL and Xie JP were attending doctors for the patient; Cheng BJ wrote paper; Zhao HC performed pathological examinations and imaging diagnosis; and Shrestha A polished the article.
Ethics approval: The study was reviewed and approved by the First Hospital affiliated to Yangtze University Institutional Review Board.
Informed consent: All study participants, or their legal guardian, provided informed written consent prior to study enrollment.
Conflict-of-interest: No potential conflicts of interest relevant to this article were reported.
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/
Correspondence to: Ben-Jing Cheng, Nurse-in-Charge, Department of General Surgery, the First Hospital affiliated to Yangtze University, No. 8, Hangkong Road, Shashi District, Jingzhou 434000, Hubei Province, China. 309802903@qq.com
Telephone: +86-716-8115071 Fax: +86-716-8118888
Received: September 22, 2014
Peer-review started: September 23, 2014
First decision: October 29, 2014
Revised: November 26, 2014
Accepted: December 13, 2014
Article in press: December 16, 2014
Published online: March 7, 2015

Abstract

A 67-year-old man from Jingzhou was admitted to the First Hospital Affiliated to Yangtze University in July 2013 with sudden onset of abdominal pain with dizziness for 12 h. The patient had sign of peritoneal irritation. Ultrasonography of the abdomen and pelvis showed hepatic fibrosis due to schistosomiasis. Computed tomography showed free gas in the peritoneal cavity. Plain abdominal radiography showed bilateral subdiaphragmatic accumulation of gas, perforation of the viscus, and radio-opacity in the left renal area. The patient underwent emergency exploratory laparotomy. At laparotomy, a moderate amount of muddy yellow pus was found in the intra-abdominal cavity. At the junction of the jejunum and ileum, about 250 cm from Treitz’s ligament, there was an about 10-cm length of inflamed small bowel with perforation (3 mm in diameter) along the mesenteric border at the middle of the lesion. The patient underwent resection of the affected intestinal segment, along with end-to-end intestinal anastomosis. Histopathological examination revealed mucosal necrosis and hemorrhage with a large number of infiltrating eosinophils and neutrophils, and acute submucosal inflammation with a large number of infiltrating eosinophils and neutrophils associated with Schistosoma japonicum (S. japonicum) eggs. No intravascular adult parasite was found. Postoperatively, the patient was treated with praziquantel (30 mg/kg daily) for 4 d. The patient progressed well. To the best of our knowledge, this is the first case of small bowel perforation associated with eggs of S. japonicum.

Key Words: Schistosoma japonicum, Intestinal perforation, Small bowel

Core tip: There are rare cases of intestinal perforation caused by schistosomiasis. When patients living in an endemic area for schistosomiasis develop intestinal perforation, the condition may be caused by Schistosoma. We present a case report of a patient from Jingzhou, China with small bowel perforation with peritonitis secondary to infection with S. japonicum.



INTRODUCTION

There are 76 countries and regions with schistosomiasis[1]. Among them, the distribution of Schistosoma japonicum (S. japonicum) is mainly in Asia, including Japan, the Philippines, Indonesia and China. This schistosome was first discovered in Japan, hence its species name. Schistosoma haematobium infection is most prevalent in Africa and Western Asia. Schistosoma mansoni is most prevalent in certain tropical and subtropical areas of sub-Saharan Africa, the Middle East, South America, and the Caribbean. China is only endemic for S. japonicum. In 1981, Biomphalaria straminea, an intermediate host of S. mansoni was first discovered in Southern China as an invasive snail species, and its habitation has extended considerably[2]. There are rare reports of intestinal perforation caused by schistosomiasis. We present a case report of a patient from Jingzhou, China with small bowel perforation with peritonitis secondary to infection with S. japonicum.

CASE REPORT

A 67-year-old man from Jingzhou was admitted to the First Hospital Affiliated to Yangtze University in July 2013 with sudden onset of abdominal pain and dizziness for 12 h. The patient had sign of peritoneal irritation. Ultrasonography of the abdomen and pelvis showed hepatic fibrosis due to schistosomiasis. Computed tomography showed free gas in the peritoneal cavity. Plain abdominal radiography showed bilateral subdiaphragmatic accumulation of gas, perforation of the viscus, and radio-opacity in the left renal area. The patient underwent emergency exploratory laparotomy. At laparotomy, a moderate amount of muddy yellow pus was found in the intra-abdominal cavity. At the junction of the jejunum and ileum, about 250 cm from Treitz’s ligament, there was an about 10-cm length of inflamed small intestine with perforation (3 mm in diameter) along the mesenteric border at the middle of the lesion. The patient underwent resection of the affected intestinal segment, along with end-to-end intestinal anastomosis. Histopathological examination revealed mucosal necrosis and hemorrhage with a large number of infiltrating eosinophils and neutrophils (Figure 1A), and submucosal acute inflammation with a large number of infiltrating eosinophils and neutrophils associated with Schistosoma eggs (Figure 1B). No intravascular adult parasite was found. IgG for schistosomiasis was positive. Postoperatively, the patient was treated with praziquantel (30 mg/kg daily) for 4 d. The patient progressed well.

Figure 1
Figure 1 Microscopic images. A: Mucosal necrosis and hemorrhage with large numbers of infiltrating eosinophils and neutrophils (magnification × 400); B: Submucosal acute inflammation with a large number of infiltrating eosinophils and neutrophils associated with Schistosoma eggs (magnification × 400).
DISCUSSION

A review of the literature found four cases of intestinal perforation (excluding the appendix) associated with schistosomiasis: (1) colon perforation associated with S. japonicum[3]; (2) sigmoid perforation associated with S. mansoni[4]; (3) rectal perforation associated with S. haematobium[5]; and (4) jejunal perforation associated with adult worm of Schistosoma[6]. To the best of our knowledge, this is the first case report of small bowel perforation associated with eggs of S. japonicum. No intravascular adult parasite was found in this case.

Among the 81 national surveillance sites in China, the average positive rate of residents with serum examinations and average infection rate of the residents were 7.78% and 0.54% respectively and the infection rate of snails was 0.14% in 2011[7]. From the 64 monitoring sites, in Jingzhou, a city in Central China, the average schistosomal infection rate of residents was 1.48% and the infection rate of snails was 0.08% in 2011[8]. The snail Oncomelania hupensis is the only intermediate host of the parasite S. japonicum, which causes human schistosomiasis in East and Southeast Asia. Humans acquire schistosomiasis via contact with freshwater containing infectious, free-living, cercarial larvae. The worms then migrate through the circulation (mainly in the mesenteric vein) affecting the portal system. The main pathological changes are egg granuloma caused by ova, and deposition of eggs in the intestine or liver tissue. Patients develop fever, abdominal pain, bloody diarrhea, hepatomegaly and eosinophilia in the acute phase; chronic diarrhea and hepatosplenomegaly as the main features in the chronic phase; and liver periportal fibrosis with splenomegaly and ascites in the advanced phase. Digestive lesions are most frequently caused by rectal or large bowel involvement.

Some studies have shown that Schistosoma is related to appendicitis, and even rupture of the appendix with peritonitis[9-11]. We consider that the intestinal pathological changes of schistosomiasis are similar to those of appendicitis associated with schistosomiasis. Two pathogenetic pathways[5] have been described to explain this occurrence with appendicitis. The first pathway considers the role of feces, with their passage through inflammatory and fibrotic mucosa infected with the parasite. The second pathway is a direct inflammatory lesion of the appendix induced by an immunological granulomatous reaction around the parasite eggs, leading to tissue destruction and appendicitis.

In our patient, several elements directed us to a diagnosis of small bowel perforation caused by acute infection with Schistosoma. The first was the presence of schistosomal eggs without granuloma formation, which indicated active infection. The second was the presence of acute inflammation of the mucosa and submucosa. The third was the presence of mucosal necrosis and hemorrhage. The patient should have a long history of infection with schistosomiasis. The reason was the presence of hepatic fibrosis caused by schistosomiasis shown by ultrasonography.

COMMENTS
Case characteristics

A 67-year-old man presented with abdominal pain and dizziness.

Clinical diagnosis

Upon physical examination, the patient had a sign of peritoneal irritation.

Differential diagnosis

Acute pancreatitis, tuberculous peritonitis, acute ileus, and acute appendicitis.

Laboratory diagnosis

IgG positive for schistosomiasis.

Imaging diagnosis

Computed tomography and plain abdominal radiography showed free gas in the peritoneal cavity.

Pathological diagnosis

Histopathological examination revealed mucosal necrosis and hemorrhage with large numbers of infiltrating eosinophils and neutrophils, and submucosa acute inflammation with a large number of infiltrating eosinophils and neutrophils associated with Schistosoma eggs.

Treatment

The patient underwent emergency exploratory laparotomy and resection of the affected intestinal segment along with end-to-end intestinal anastomosis.

Related reports

A review of the literature only found four cases of intestinal perforation (excluding the appendix) associated with schistosomiasis. To the best of our knowledge, this is the first case report of small bowel perforation associated with eggs of Schistosoma japonicum.

Term explanation

Schistosoma japonicum (S. japonicum) is currently only prevalent in Japan, the Philippines, Indonesia and China. This worm was first discovered in Japan, hence the species name.

Experiences and lessons

In our case, small bowel perforation was caused by S. japonicum. In schistosomiasis endemic areas, this rare etiology needs to be considered in cases of intestinal perforation.

Peer-review

The authors have described a case of perforation of small intestine caused by Schistosoma japonicum. The article highlights this rare etiology which should be considered in cases of intestinal perforation in schistosomiasis endemic area.

Footnotes

P- Reviewer: He SH, Kamocki ZK S- Editor: Qi Y L- Editor: A E- Editor: Wang CH

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