Hollow viscus perforation secondary to brucellosis: a rare case report

Abstract

Introduction:

Brucellosis, a zoonotic infection, rarely causes hollow viscus perforation. This case highlights ileal perforation in a patient with pre-diagnosed brucellosis, which reinforces the importance of early surgical evaluation in an acute abdomen.

Presentation of Case:

A young male with confirmed brucellosis (positive IgM serology) presented with fever and arthralgia. On day 2 of admission, he developed sudden severe abdominal pain, guarding, and rigidity. Imaging revealed pneumoperitoneum, prompting emergency laparotomy, which identified an ileal perforation. Wedge resection and anastomosis was performed, followed by combined antibiotics. Histopathology showed transmural inflammation with ulceration. The patient recovered fully postoperatively.

Discussion:

Brucellosis typically manifests with systemic symptoms; gastrointestinal involvement is uncommon. This case demonstrates ileal perforation without prior Gastrointestinal (GI) symptoms, likely due to ulceration. Early imaging and surgical intervention were crucial. In endemic regions, brucellosis should be considered in an acute abdomen with prolonged fever associated with arthralgia.

Conclusion:

Although rare, brucellosis can lead to life-threatening perforation. A high index of suspicion, prompt imaging, and timely surgery are vital for optimal outcomes, alongside prolonged antibiotic therapy.

Introduction

Brucellosis is a zoonotic disease caused by Gram-negative bacteria of the Brucella genus. It primarily affects both livestock and wildlife. The genus encompasses 10 recognized species, including three species that are of major public health and economic importance. These are B. melitensis, which predominantly infects sheep and goats; B. abortus, which affects cattle; and B. suis, which affects swine. The bacteria are transmitted from animals to humans by ingestion of infected raw dairy products, direct contact with an infected animal, or inhalation of aerosols^[1]. The clinical features vary from high-grade fever, anorexia, and malaise to osteo-articular, gastrointestinal, hepato-biliary, cardiovascular, and neurological symptoms. Although gastrointestinal complications are rare, if present, they can vary with diverse presentations ranging from milder forms like diarrhea, anorexia, abdominal pain, and constipation to more serious conditions like acute abdomen mimicking acute appendicitis, ileitis, infective colitis, acute cholecystitis, acute pancreatitis, mesenteric lymphadenitis, and intestinal obstruction and perforation^[2–9].

HIGHLIGHTS

• Terminal ileal perforation in a patient with brucellosis, emphasizing that gastrointestinal perforation can occur even without prior GI symptoms.

• Brucellosis should be considered in acute abdomen presentations in endemic regions, especially when accompanied by prolonged fever and arthralgia, even in patients without obvious risk factors.

• Early imaging and emergency laparotomy were lifesaving, highlighting the importance of prompt surgical evaluation in hollow viscus perforation.

• Integrated approach from diagnosis to treatment improves prognosis in these complex cases.

This report presents a rare case of a young male patient with terminal ileum perforation with brucellosis with no risk for the disease. This case has been reported in line with the Revised Surgical CAse REport (SCARE) guideline, 2025 criteria^[10].

Case presentation

A 27-year-old male presented to the emergency department with a 9-day history of high-grade fever (38.8°C). He also had chills, rigor, and generalized myalgia, without respiratory, urinary, or gastrointestinal symptoms. He had no past history of surgery or any illness or comorbidity. He is a student and had already completed a course of amoxicillin prior to his visit to our hospital. On evaluation, his temperature was 103°C, with elevated liver enzymes and a positive rapid immunochromatographic test for Brucella M via B. abortus antigen rapid test cassette, suggesting Brucella infection with a normal chest X-ray (Fig. 1a). Initial treatment included Tab doxycycline 100 mg twice a day, Inj. gentamicin 80 mg twice a day, and other supportive care.

Figure 1.

(a) Chest X-ray on the day of presentation showing normal lung fields with no gas under the diaphragm. (b) Repeat chest X-ray (day 2) demonstrating gas under the right diaphragm (arrow) suggestive of pneumoperitoneum secondary to hollow viscus perforation.

On the second day, he developed sudden severe abdominal pain with diffuse tenderness, guarding, and rigidity. Repeat imaging revealed gas under the right diaphragm (Fig. 1b).

Contrast-enhanced computed tomography of the abdomen showed loculated free air noted adjacent to the terminal ileum, suggestive of localized terminal ileal perforation with intra-abdominal air in perihepatic regions and ileal loops. Significant perienteric fat stranding is noted, along with multiple enlarged mesenteric nodes, the largest measuring 14*8 mm. Hepatomegaly (measuring 20 cm) and splenomegaly (measuring 14 cm) were also observed (Fig. 2a–c).

Figure 1.

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Emergency laparotomy under general anesthesia in supine position with midline incision confirmed a 0.5 cm ileal perforation, which is located around 35 cm proximal to the ileocecal valve with surrounding inflammation, purulent as well as bile collection and edematous bowel loops (Fig. 3). A wedge resection of the perforated margin with handsewn Lambert’s seromuscular suturing in a double layer was performed. Since there was a mild edematous appendix and the patient was living in a rural region of the country with no easy access to surgical care, after counselling with the patient’s family, prophylactic appendectomy was performed in order to reduce the risk of appendicitis and potential perforation in the future. A thorough peritoneal lavage was performed with warm saline, and a silicon abdominal drainage tube of size 26Fr was kept in the pelvic cavity and fixed at the right iliac region. The abdominal wall was closed with absorbable suture and the skin with nonabsorbable suture.

Figure 3.

Intraoperative picture demonstrating a perforated segment of the terminal ileum with surrounding inflamed and edematous bowel loops.

Figure 2.

(a) Axial section of contrast-enhanced computed tomography (CECT) scan of abdomen showing free intra-abdominal air in perihepatic regions. A crescent-shaped area of free air (arrow) is visualized beneath the diaphragm indicative of pneumoperitoneum suggestive of hollow viscus perforation. (b) Axial section of CECT scan of the abdomen demonstrating loculated extraluminal air adjacent to the terminal ileum, reflecting perforation with thickening of the ileal wall. (c) Axial section of CECT scan of the abdomen demonstrating free intra-abdominal air and ascites.

Postoperatively, antibiotics were upgraded to intravenous piperacillin/tazobactam (4000 mg/500 mg) three times a day and intravenous metronidazole 500 mg three times a day for 1 week. Whereas preoperatively used antibiotics, intravenous gentamicin 80 mg two times a day was completed for 2 weeks and oral tablet doxycycline 100 mg two times a day was asked to complete for 6 weeks. Enteral feeding was initiated on day 3, starting with a liquid diet as tolerated, and fever was completely resolved since the third postoperative day. Patient’s postoperative recovery was uneventful with Clavien–Dindo classification observed as Grade 1. Histopathology of wedge resection of edges at the perforation site revealed ulceration, transmural inflammation, and fibrinopurulent exudate (Fig. 4). However, the histopathology of vermiform appendix revealed normal mucosa with no infiltration of inflammatory cells. The patient was discharged on the eighth postoperative day with continued follow-up, showing good recovery. Upon follow-up, we observed an excellent recovery in the patient, who was able to resume his studies, and at 1 month following the surgery, he reported feeling well with no residual symptoms. All his laboratory reports were within normal ranges, and no side effects from the medication were observed. He was grateful for the care received and emphasized the importance of considering unusual causes for common symptoms like fever and abdominal pain.

Figure 4.

Histopathological section of the terminal ileum showing tissue devoid of surface epithelium with ulceration and overlying fibrinopurulent exudate. The underlying stroma shows edema, mixed acute and chronic inflammatory infiltrates, and fibroblastic proliferation. Inflammation extends into the scant underlying muscularis propria (hematoxylin and eosin stain).

Figure 2.

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The chronological summary of the patient’s clinical course, from presentation to follow-up, is summarized in Table 1.

Table 1.

Chronological summary of the patient’s management

Day 1 (Admission): Patient admitted with a 9-day history of fever. Brucellosis diagnosed (positive IgM). Treatment started with IV doxycycline and gentamicin.

Day 2: Acute onset of severe abdominal pain with signs of peritonitis.

Imaging 1: Initially erect chest X-ray confirmed pneumoperitoneum.

Imaging 2: Further CECT abdomen localized the perforation to the terminal ileum.

Emergency laparotomy performed. A 0.5 cm ileal perforation was found and managed with wedge resection and anastomosis with prophylactic appendectomy.

Postoperative day 1: Antibiotic regimen was broadened to cover peritonitis (piperacillin–tazobactam, metronidazole) while continuing anti-Brucella therapy (doxycycline, gentamicin).

Postoperative day 3: Enteral feeding was successfully initiated.

Postoperative day 8: Patient was discharged in stable condition on oral doxycycline for 6 weeks and gentamycin for 2 weeks.

One-month follow-up: Full recovery was confirmed with no signs of relapse and no adverse effects from the medications used.

Discussion

Brucellosis is the second most important zoonotic disease in the world after rabies, according to the World Organization for Animal Health^[11]. At present, the global burden of brucellosis ranges from 1 to 200 new human cases per 10 million individuals per year. According to the available data, the incidence of human brucellosis is increasing in Eastern Mediterranean countries and is endemic in some Asian countries, including Sri Lanka, India, China, Pakistan, Mongolia, and Nepal^[12,13]. Three major species of Brucella (B. abortus, B. melitensis, and B. suis) have been reported to be present in Nepal, although the precise distribution and prevalence of human brucellosis are still not available. Although brucellosis is reportedly endemic in Nepal, neither the distribution nor the economic and public health impacts of the disease are well characterized. In Nepal, approximately 90% of people live in villages and come into direct contact with domestic animals on a daily basis. As a consequence, the local people of the villages are at a high risk of acquiring these types of zoonotic diseases^[14]. In one of the retrospective studies comprising 1028 patients, 42.3% had a history of raising livestock, and 55.2% were found to have no occupational risk for brucellosis, as in our case^[15]. In our case, the patient is also a student with no history of involvement in livestock farming, animal husbandry, or handling of raw animal products.

Brucella species are aerobic Gram-negative coccobacilli that possess a unique ability to invade both phagocytic and nonphagocytic cells and to survive in an intracellular environment by finding ways to avoid the immune system. This ability explains why brucellosis is a systemic disease and can involve almost every organ system^[16]. The main complaints of infected patients are fever, chills, night sweats, myalgia, anorexia, headache, and joint pain. Out of all the features, fever and arthralgia are reported to be the most common symptoms and signs in brucellosis^[17]. In the present case, the major presenting complaints were also fever and myalgia. Gastrointestinal symptoms are common in patients with brucellosis and can manifest as decreased appetite, nausea, vomiting, abdominal pain, diarrhea, constipation, and hepatosplenomegaly in about 50% of patients with brucellosis. Among the digestive system, the liver is the most commonly affected and presents in the form of hepatitis^[18]. Complications of brucellosis are a major medical problem in countries where the infection is still endemic. The prevalence of focal involvement has been reported to range from 20 to 40% in many studies^[19]. According to a multicenter cross-sectional study conducted in China from December 2019 to August 2021, the most common complications involved osteoarticular involvement, followed by the genitourinary system, respiratory system, and hematological system, respectively^[20]. Although our patient did not present with gastrointestinal symptoms initially, the disease course was later complicated by gastrointestinal perforations. Notably a hepatosplenomegaly – a common finding in other reported cases – was also observed in our patient.

The precise mechanism of Brucella-induced intestinal perforation remains incompletely defined; however, it is hypothesized to result from severe, localized damage to the intestinal wall following bacterial invasion and a dysregulated host immune response. The process is initiated by Brucella’s ability to invade host cells, facilitated by surface proteins like U-Omp, which allow it to evade initial defenses and infiltrate the intestinal mucosa^[21]. Following uptake, the bacterium’s intracellular survival within macrophages disrupts normal cellular functions and triggers a complex immune response. It is proposed that an imbalance in this response leads to excessive tissue damage, compounded by localized vascular injury and ischemia. This cascade of events – bacterial invasion, dysregulated immunity, and tissue necrosis – ultimately compromises intestinal integrity, resulting in perforation^[9].

Brucella species are intracellular pathogens that infect immune cells and are therefore difficult to diagnose and treat. Diagnosis of intracellular pathogens like Brucella can be challenging and relies on a combination of clinical presentation and confirmatory serological or molecular tests. Various laboratory test is used across different regions as confirmatory diagnostic tools for Brucella infection. In many developed countries, the Standard Tube Agglutination (STA) test and ELISA remain a confirmatory serological test. A 10-year retrospective analysis of 1028 brucellosis cases admitted to the Department of Infectious Diseases and Clinical Microbiology found the STA test positive in 98.8% (1016 cases)^[15]. Brucella ELISA offers greater sensitivity and specificity, allowing differentiation between acute (IgM) and chronic (IgG) infection^[22]. More recently, PCR-based detection has enabled direct identification of Brucella DNA in human blood samples, even in culture-negative cases^[23]. The diagnosis in this case was based on a positive rapid immunochromatographic test (B. abortus antigen rapid test cassette). While this method offers high specificity and sensitivity in endemic, resource-limited settings^[24], it is non-species-specific. In our setting, confirmatory tests like the STA test, ELISA, or PCR were not available. Therefore, the attribution to B. abortus is presumptive, based on the rapid test and epidemiological context, and we acknowledge this as a limitation.

Diagnosis of this intracellular pathogen can be challenging. While culture is definitive, it is difficult due to slow growth and prior antibiotic use^[25].

Brucellosis is both preventable and curable. Avoiding the ingestion of under-cooked animal products, refraining from consuming unpasteurized dairy products or those without quality assurance, and using appropriate protective equipment while handling the animal and its waste are essential. Once brucellosis is confirmed, combination therapy is started in order to avoid relapse. Previously, the preferred regimen included doxycycline in combination with rifampicin and streptomycin or gentamicin in uncomplicated cases for individuals greater than 8 years of age, or rifampicin and streptomycin or gentamicin for those under 8 years. Trimethoprim-sulfamethoxazole replaces tetracycline in children under 8 years of age^[26]. However, recent studies have shown the use of doxycycline plus gentamycin to be more effective in treating brucellosis with higher safety efficacy and lower relapse rates^[27,28]. In our case, intravenous piperacillin and tazobactam with metronidazole were also used to cover for hospital-acquired and anaerobic pathogens in peritonitis settings and Tab doxycycline and Tab gentamycin as specific anti-Brucella therapy. Studies indicate that certain antibiotics during the course of the disease may lead to Clostridium difficile colitis, potentially causing complications such as toxic megacolon and perforation. However, in our case, the antibiotics administered were unlikely to directly cause C. difficile colitis or perforation^[29,30].

While our case is rare, it is not without precedent. A review of the modern literature reveals several documented instances of brucellosis leading to life-threatening intestinal perforation, as summarized in Table 2. These cases, along with our own, share several common features. The terminal ileum appears to be the most frequently involved site.

Table 2.

Summary of reported cases of intestinal perforation secondary to brucellosis

Reference (source)	Country	Patient age/sex	Perforation site	Surgical management	Antibiotics regimen	Outcome
Fenandez Marcial Delgado et al (2004)	Spain	15 years Male	Appendix	Laparatomy with appendectomy	Intravenous imipenem and cilastin (1 gm QID for 9 days)	Improved
Wang et al (2024)	China	69 years Male	Terminal ileum	Exploratory laparoscopy + resection and anastomosis	Oral doxycycline 0.1 gm OD + oral rifampicin 150 mg OD for 4 weeks	Improved
Noureen et al (2020)	Pakistan	32 years Male	Terminal ileum	Right hemicolectomy and ileocolostomy	Tab doxycycline 100 mg BD and Tab rifampicin 600 mg OD for 6 weeks + Tab metronidazole 400 mg TDS for 7 days	Improved

Conclusion

Brucellosis is an infection with multiple presentations requiring high clinical suspicion, especially in endemic regions or with livestock exposure. However, it can also present in patients without obvious risk factors, as seen in our case. This case highlights that brucellosis may present with gastrointestinal symptoms and should be considered in cases of fever with an acute abdomen. Though rare, if it presents, it can be serious and life-threatening, warranting prompt intervention.

Unlike most reported cases associated with gastrointestinal complications, where brucellosis is diagnosed postoperatively due to persistent fever, our patient was identified early, with symptoms developing subsequently. This underscores the importance of including brucellosis in the differential diagnosis of acute abdominal conditions.

Ethical approval

Ethical approval was exempted by the Institutional Review Board (IRB) of this institution because it was a single case report and did not require formal ethics review.

Consent

Written informed consent was obtained from the patient for the 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 upon request.

Sources of funding

The study has no funding source.

Author contributions

R.A.K.: First author with study concept design and manuscript writing. D.K.: Co-author who helped prepare the manuscript, collect the patients’ information, and radiological images. A.K.C., S.S., and C.S.: contributors who prepared the images, analyzed and described the images, and helped prepare the manuscript. R.R.K.: Corresponding author responsible for editing, reviewing, and concluding the manuscript and following up on the patient’s condition.

Conflicts of interest disclosure

No conflict of interest.

Guarantor

Dr Rukma Raj Kafle.

Research registration unique identifying number (UIN)

Not applicable.

Provenance and peer review.

The data supporting the findings of this study are available from the corresponding author upon reasonable request.

Acknowledgements

Not applicable.