Published online Jan 7, 2027. doi: 10.3748/wjg.123843
Revised: July 1, 2026
Accepted: July 20, 2026
Published online: January 7, 2027
Processing time: 163 Days and 19.5 Hours
Upper gastrointestinal bleeding is most often caused by mucosal, variceal, or malignant gastrointestinal disease. Vascular etiologies are uncommon but po
A 78-year-old woman was admitted with acute hematemesis and subsequently developed recurrent bleeding with a decline in hemoglobin. Urgent upper endo
Aortic pseudoaneurysm should be excluded with CTA when atypical bleeding esophageal submucosal lesions are encountered.
Core Tip: This case highlights a rare suspected Brucella-associated descending thoracic aortic pseudoaneurysm presenting with upper gastrointestinal bleeding. The lesion initially mimicked an ulcerated esophageal submucosal tumor, which created potential diagnostic confusion. Posterior esophageal protrusion with ulceration, adherent clot, dysphagia, or unexplained fever may indicate an underlying vascular etiology; therefore, contrast-enhanced computed tomography/computed tomography angiography should be prioritized before endoscopic biopsy or therapeutic intervention. Recognition of relevant zoonotic exposure may support early consideration of infectious aortitis.
- Citation: Xue Y, Yun XJ, Liu QY, Qi YT, Zhang LH, Jiang K, Li BD, Zhang JX. Suspected Brucella-associated descending thoracic aortic pseudoaneurysm presenting as upper gastrointestinal bleeding: A case report and review of literature. World J Gastroenterol 2027; 33(1): 123843
- URL: https://www.wjgnet.com/1007-9327/full/v33/i1/123843.htm
- DOI: https://dx.doi.org/10.3748/wjg.123843
Upper gastrointestinal bleeding (UGIB) is a common medical emergency, but the source of bleeding is not always gastrointestinal. Current UGIB guidelines recommend early risk assessment, hemodynamic stabilization, and prompt endoscopic evaluation[1]. Although peptic ulcer disease, variceal bleeding, erosive disease, and malignancy account for most cases, vascular etiologies should be considered when the endoscopic appearance is atypical or when bleeding arises from the esophagus without an obvious mucosal or variceal source[2].
Among vascular etiologies, aortoesophageal fistula (AEF) is rare but life-threa
Brucellosis is a zoonotic infection transmitted mainly through contact with infected animals or contaminated animal products[6]. Clinical manifestations are diverse and may include prolonged or recurrent fever, fatigue, sweating, osteoarticular disease, endocarditis, and other focal complications[6-8]. Cardiovascular involvement is uncommon but po
We present a rare case of suspected Brucella-associated descending thoracic aortic pseudoaneurysm that presented with dysphagia and UGIB. The lesion initially mimicked an ulcerated esophageal submucosal tumor on endoscopy. This case emphasizes the importance of integrating endoscopic, radiologic, infectious disease, and vascular surgical perspectives in the evaluation of atypical esophageal bleeding.
A 78-year-old rural woman was admitted to the Department of Gastroenterology with acute hematemesis lasting more than one hour.
The patient reported three episodes of fresh hematemesis shortly before admission, accompanied by diaphoresis and fatigue. She denied melena, nausea, vomiting, chest pain, and respiratory symptoms. Initial supportive care, including fluid resuscitation, acid suppression, and hemostatic therapy, achieved temporary hemodynamic stabilization.
Approximately 28 hours after admission, she had another episode of fresh hematemesis, estimated at 20 mL, followed by a decline in hemoglobin to 82 g/L, indicating ongoing UGIB and prompting urgent evaluation. Urgent upper en
Her medical history included chronic headache, intermittently managed with antipyretics, and coronary artery disease controlled with metoprolol and compound Danshen dripping pills, a traditional Chinese medicine preparation. She had no history of hepatitis, tuberculosis, surgery, trauma, blood transfusion, or known allergies.
Further history-taking revealed close contact with sheep raised by her son. She did not smoke, drink alcohol, or use illicit drugs. The family history was unremarkable.
On admission, her temperature was 36.0 °C, pulse was 74 beats/minute, respiratory rate was 17 breaths/minute, and blood pressure was 106/71 mmHg. She was conscious and cooperative, with a pale appearance and conjunctival pallor. Breath sounds were clear bilaterally. Cardiac examination revealed a regular rhythm, normal heart sounds, and no murmurs. The abdomen was soft, with mild epigastric tenderness. The liver and spleen were not palpable, and no lower-limb edema was present.
On admission, laboratory testing revealed a hemoglobin level of 107 g/L. Renal function, electrolytes, and coagulation parameters were within normal limits. After recurrent hematemesis, hemoglobin declined to 82 g/L. Because the patient had a history of unexplained fever and close contact with sheep, Brucella serology and blood culture were performed. The Rose Bengal plate agglutination test (RBT) showed marked visible agglutination (+++), corresponding to a strongly positive screening result according to the visual grading criteria provided by the manufacturer[11]. In this patient, standard tube agglutination test titers were unavailable; therefore, serological interpretation was based on RBT in combination with clinical and epidemiological findings.
Blood culture was performed using the BACT/ALERT 3D automated blood culture system (bioMérieux, Shanghai, China). The blood culture bottle generated a positive signal after 71.5 hours of incubation. Subculture on blood agar yielded small, circular, gray-white colonies after 24 hours of incubation, and the isolate was identified as Brucella spp. using the VITEK 2 Compact system with a Gram-negative identification card. These findings supported a diagnosis of Brucella spp. bloodstream infection.
Upper endoscopy revealed a large submucosal protrusion on the posterior esophageal wall, 26-32 cm from the incisors, with apical ulceration and an adherent dark-red clot. The esophageal lumen was mildly narrowed but passable, and no gastric or duodenal ulcer was identified. The lesion was initially considered a ruptured and bleeding esophageal submucosal protrusion, possibly a leiomyoma (Figure 1).
Contrast-enhanced CT and CTA of the chest and abdomen were performed to clarify the nature of the esophageal protrusion. The images revealed a focal saccular contrast-filled outpouching arising from the descending thoracic aorta, with peripheral low-attenuation components and compression of the adjacent esophagus. Central enhancement indicated communication with the aortic lumen, whereas the peripheral low-attenuation component was considered compatible with mural thrombus, perianeurysmal inflammatory tissue, or hematoma (Figures 2 and 3). Three-dimensional CTA reconstruction further demonstrated a localized saccular outpouching arising from the descending thoracic aorta distal to the origin of the left subclavian artery (LSA) (Figure 4). Given the focal saccular morphology and local mass effect, the lesion was considered more consistent with a pseudoaneurysm than with a typical fusiform degenerative true aneurysm. The imaging findings raised suspicion of an infectious etiology.
Retrospective review of the outside non-contrast chest CT images showed subtle abnormalities of the descending thoracic aorta, including inward displacement of aortic wall calcification and blurred periaortic fat planes, raising the possibility of early intramural hematoma, mural thrombus, or inflammatory change (Supplementary Figure 1). In the context of prior unexplained febrile illness, sheep exposure, a suspected early aortic wall abnormality on prior CT, and the current pseudoaneurysmal lesion, these findings raised suspicion for infection-related aortic wall injury, particularly suspected Brucella-associated vascular involvement or ulcerative aortic wall injury.
The cardiovascular surgery team assessed the lesion as having a high risk of rupture or catastrophic hemorrhage and recommended emergency intervention. Infectious disease consultation recommended targeted antimicrobial therapy with doxycycline plus rifampicin, with adjunctive ceftriaxone during the acute phase if clinically indicated.
The final diagnosis was suspected Brucella-associated descending thoracic aortic pseudoaneurysm presenting with esophageal compression and UGIB.
The patient underwent emergency thoracic endovascular aortic repair (TEVAR) under general anesthesia. Femoral arterial access was obtained, and intraoperative aortography delineated the pseudoaneurysm and defined the proximal and distal landing zones. A thoracic covered stent-graft was successfully deployed, with the proximal landing zone distal to the LSA origin and the distal end positioned within the descending thoracic aorta. Completion angiography confirmed accurate stent-graft deployment, preserved LSA perfusion, and absence of endoleak. Estimated blood loss was 50 mL, and the procedure was completed without complications. This emergent TEVAR effectively excluded the pseudoaneurysm and reduced the immediate risk of rupture and catastrophic hemorrhage (Figure 5). After the procedure, the patient received supportive care, transfusion therapy, and blood pressure control.
Given the positive RBT result and clinical suspicion of Brucella infection with vascular involvement, targeted anti-Brucella therapy was initiated after infectious disease consultation. The core regimen consisted of doxycycline 100 mg orally twice daily plus rifampicin 600 mg orally once daily. Ceftriaxone 2 g intravenously every 12 hours was added as adjunctive acute-phase therapy for a planned duration of 4 weeks.
Postoperatively, the patient developed a fever up to 38.5 °C, which responded to symptomatic antipyretic treatment. She subsequently had intermittent low-grade fever, which gradually resolved. At the latest follow-up, no further fever had been recorded since June 1, 2026, and no recurrent hematemesis or melena had occurred.
Two postoperative aortic CTA examinations were performed before discharge planning. CTA on postoperative day 9 demonstrated satisfactory early post-TEVAR findings, with maintained pseudoaneurysm exclusion and no obvious endoleak or acute stent-graft-related complication (Supplementary Figure 2). Repeat CTA performed approximately 25 days after TEVAR showed stable postoperative findings, with persistent pseudoaneurysm exclusion and no obvious endoleak or acute stent-graft-related complication (Figure 6). Bilateral pleural effusion showed interval absorption on the later scan. Together with the absence of recurrent fever, hematemesis, or melena, these imaging findings supported discharge planning with continued antimicrobial therapy and structured clinical and imaging surveillance.
Doxycycline plus rifampicin was planned for at least 12 weeks, with possible extension to 6 months according to the clinical course, inflammatory markers, drug tolerance, and serial CTA findings. Further CTA surveillance was scheduled at approximately 2 months, 6 months, and 12 months after TEVAR and annually thereafter if the findings remained stable, to evaluate pseudoaneurysm exclusion, detect potential endoleaks, monitor periaortic inflammatory changes, assess graft integrity, and rule out delayed AEF. Clinical and laboratory follow-up was planned at regular intervals during antimicrobial therapy to monitor recurrent fever, dysphagia, hematemesis, melena, chest or back pain, inflammatory markers, liver and renal function, and antimicrobial-related adverse events.
The novelty of this case lies in the presentation of a suspected Brucella-associated descending thoracic aortic pseudoaneurysm as UGIB that initially mimicked an ulcerated esophageal submucosal tumor on endoscopy. This rare presentation created a high-risk diagnostic challenge. The patient presented with hematemesis, and upper endoscopy revealed an ulcerated posterior esophageal submucosal protrusion with an adherent clot. This appearance can mimic a primary esophageal submucosal tumor, such as a leiomyoma. However, the posterior location of the lesion, progressive dys
Previous reports have described atypical endoscopic findings in AEF or impending fistulization, including submucosal tumor-like protrusion, pulsatile bulging, submucosal hematoma, ulceration, and adherent clot[3,4]. Recognizing this distinction is clinically important because AEF is a rare but potentially fatal source of hematemesis, and delayed re
Dysphagia in this patient was mechanistically important. Dysphagia aortica refers to difficulty swallowing caused by external esophageal compression from an ectatic, tortuous, or aneurysmal thoracic aorta, typically in elderly patients[16-19]. In this case, dysphagia preceded hematemesis, suggesting that early mechanical compression by the descending thoracic aortic pseudoaneurysm may have caused mucosal ischemia, followed by ulceration, clot formation, and bleeding. Thus, dysphagia represented an early clinical indicator of progressive aortic pathology.
The suspected infectious etiology further highlights the clinical importance of this case. Brucellosis is a zoonotic infection with diverse manifestations, including prolonged or relapsing fever, fatigue, and focal complications[6-8]. Contemporary reviews emphasize that vascular involvement is uncommon but life-threatening. Willems et al[9] reviewed aortic and iliac involvement in brucellosis and concluded that, although rare, it carries a high risk of arterial rupture and secondary complications; open surgery combined with antibiotics has historically been considered a therapeutic cornerstone, whereas the role of endovascular therapy requires careful individualized assessment. A more recent review by Li et al[10] similarly reported that aortic and iliac involvement in brucellosis is associated with older age and arteriosclerosis and that early diagnosis and therapeutic intervention are crucial.
To contextualize the novelty of this case, we compared it with published individual cases of Brucella-associated thoracic aortic aneurysmal lesions with extractable patient-level data (Table 1)[20-26]. This comparison focused on thoracic rather than abdominal aortic or iliac disease because the key clinical issue in this case was esophageal compression and UGIB. Unlike most reported thoracic cases, which presented with fever, chest or back pain, hemoptysis, hoarseness, or established fistulization, our patient initially presented with hematemesis and an endoscopic appearance mimicking an ulcerated esophageal submucosal tumor. This finding emphasizes the need to exclude a vascular or extrinsic mediastinal cause before biopsy or therapeutic endoscopic manipulation.
| Ref. | Age/sex | Aortic lesion | Presentation | Diagnostic evidence | Treatment | Outcome |
| Fuchs et al[22], 2017 | 69/M | Descending thoracic aorta; 82-mm false aneurysm | Dysphagia; abdominal pain; weight loss; hemoptysis/possible hematemesis; mid-esophageal ulcerative/external compression | Blood culture positive for Brucella melitensis; Brucella tube agglutination test IgM and IgG 1:80 | Emergency TEVAR; gentamicin + doxycycline; doxycycline for 1 year | Asymptomatic; 3-year CTA showed good graft position and near-complete/full involution |
| Wang et al[23], 2017 | 65/M | Descending thoracic aortic pseudoaneurysm; 54 mm | Intermittent fever; chest pain; mild hemoptysis | Positive blood culture for Brucella; SAT 1:400; CTA | Rifampicin + doxycycline; covered stent-graft | Symptoms resolved; SAT negative; asymptomatic at 26 weeks |
| Chen et al[24], 2017 | 71/M | Descending thoracic rupture near celiac trunk + abdominal bifurcation rupture | Dyspnea; malaise; hypotension/acute rupture | Prior and postoperativeblood cultures confirmed Brucella melitensis | Thoracic and abdominal stent-graft exclusion; coil embolization; doxycycline + gentamicin + rifampin | Acute event controlled; discharged to rehabilitation |
| Alsheef et al[25], 2019 | 52/M | Descending thoracic aorta; saccular mycotic aneurysm with AEF | Chest/back pain; fever; weight loss; dysphagia; massive hematemesis | Two blood cultures: Brucella melitensis; CTA; TTE/TEE negative for endocarditis | Rifampin + doxycycline; urgent endovascular repair; gentamicin; planned 6-month oral therapy | Recurrent massive hematemesis; death after deterioration |
| Wu et al[20], 2019 | 65/M | Thoracic saccular aneurysm with aortobronchial fistula | Hemoptysis; feral swine hunting | Blood and aortic wall tissue cultures positive for Brucella suis; granulomatous aortitis | Open repair and fistula closure | Recurrent hemoptysis after prior TEVAR; uneventful recovery after open repair |
| Li and Wang[21], 2022 | 67/M | Thoracic penetrating ulcers with partial aneurysm formation | Left waist/buttock pain | Positive Brucella blood culture; CTA; prior Brucella abdominal pseudoaneurysm | Thoracic covered stent-graft + antimicrobial therapy | No discomfort at 6-week follow-up |
| Guo et al[26], 2023 | 65/M | Proximal descending thoracic pseudoaneurysm involving LSA/distal arch; 58 mm × 56 mm | Fever; hoarseness; previous brucellosis | Positive Brucella agglutination test; tissue mNGS: Brucella melitensis | Open arch/proximal descending aortic replacement + LSA reconstruction; antibiotics | Uneventful recovery; no recurrent fever |
| Present case | 78/F | Descending thoracic aortic pseudoaneurysm compressing esophagus | Hematemesis; progressive dysphagia; ulcerated esophageal submucosal tumor-like lesion | RBT positive (+++); blood culture identifying Brucella spp.; sheep exposure; CTA; no tissue confirmation | Emergency TEVAR; doxycycline + rifampicin; adjunctive ceftriaxone | No recurrent bleeding or fever; POD 9/POD 25 CTA showed persistent exclusion without obvious endoleak |
Because direct aneurysmal wall or periaortic tissue was unavailable, the diagnosis required integration of clinical features, epidemiologic exposures, radiologic findings, serologic testing, and blood culture results. The available evidence was stratified according to diagnostic strength and anatomical specificity. First, Brucella spp. bloodstream infection was strongly supported by the positive RBT result, a time-to-positivity of 71.5 hours in automated blood culture, subculture findings, and VITEK 2 Compact phenotypic identification of Brucella spp. Second, Brucella-associated vascular in
The pathogenesis in this patient was interpreted as a probable multifactorial process rather than a single proven pathway. Advanced age and atherosclerotic calcification may have created a vulnerable vascular substrate. Super
Given the high risk of rupture associated with infectious pseudoaneurysms, timely endovascular intervention was imperative. TEVAR effectively excluded the pseudoaneurysm from systemic arterial pressure, thereby reducing the immediate risk of rupture and catastrophic hemorrhage. Completion angiography confirmed stent-graft deployment without endoleak, indicating immediate procedural success. According to the 2022 American College of Cardiology/American Heart Association aortic disease guideline, CT imaging is central to the diagnosis, procedural planning, and surveillance of thoracic aortic disease, whereas endovascular repair is increasingly preferred in selected thoracic aortic conditions[28]. Recent studies have also suggested that endovascular repair, when combined with appropriate antimicrobial therapy, is feasible in selected patients with infected aortic aneurysms, including Brucella-associated aortic or aorto-iliac lesions, although long-term surveillance remains essential[29-31]. However, infection changes the risk profile of endovascular repair because retained prosthetic material may remain vulnerable to persistent infection, endoleak, recurrent pseudoaneurysm, graft infection, or delayed fistula formation[32].
The choice between open surgery and endovascular repair in Brucella-associated aortic lesions should therefore be individualized[9,10,32]. Open surgery offers the advantages of aneurysm resection, debridement of infected tissue, reconstruction, and tissue acquisition for microbiological and histopathological confirmation. These advantages are particularly relevant when infection is uncontrolled, abscesses are present, anatomy is unsuitable for endovascular sealing, or fistulization requires definitive surgical repair. However, open thoracic aortic surgery may involve substantial perioperative risk, especially in elderly patients, unstable patients, or patients with acute bleeding or impending rupture. In such settings, TEVAR may serve as a lifesaving strategy for rapid hemorrhage control and pseudoaneurysm exclusion. However, it should not be regarded as a stand-alone treatment for suspected infection-associated disease. When TEVAR is selected, prolonged targeted antimicrobial therapy and structured imaging surveillance are essential to monitor for persistent infection, endoleak, recurrent pseudoaneurysm, graft infection, or delayed fistulization[22,23,32].
The antimicrobial strategy also deserves emphasis. For uncomplicated brucellosis, doxycycline-based combination therapy remains widely used. Recent reviews and meta-analyses suggest that doxycycline plus rifampicin remains an accepted regimen, although aminoglycoside-containing regimens or selected triple-therapy regimens may reduce relapse in some settings[7,8]. In suspected or confirmed vascular brucellosis, antimicrobial treatment should generally be longer and more closely monitored than in uncomplicated disease, particularly after endovascular graft implantation[9,10,33]. Accordingly, doxycycline plus rifampicin was selected as the core anti-Brucella regimen in this patient, with treatment duration guided by clinical response, inflammatory markers, drug tolerance, and serial CTA findings. Ceftriaxone was used only as adjunctive acute-phase therapy rather than as a substitute for doxycycline-based and rifampicin-based treatment. Although ceftriaxone is not standard monotherapy for brucellosis, limited clinical evidence suggests that it may be used as an adjunct in selected complicated cases when combined with an anti-Brucella regimen[34].
This case has several limitations. First, although Brucella spp. bloodstream infection was supported by positive blood culture with phenotypic identification and a strongly positive RBT result, direct microbiological confirmation from the aneurysmal wall, mural thrombus, or periaortic tissue was unavailable because emergency TEVAR did not allow tissue sampling. Therefore, the causal relationship between Brucella infection and the descending thoracic aortic pseudoaneurysm should be interpreted as suspected rather than definitively proven. Polymerase chain reaction, MALDI-TOF MS, 16S rRNA sequencing, metagenomic next-generation sequencing, or tissue-based microbiological examination would have provided stronger etiological evidence, but these tests were not performed in this case. Second, no biopsy was obtained from the esophageal lesion, which was clinically appropriate to avoid catastrophic bleeding in a potential vascular lesion; however, this limited histopathological exclusion of a primary esophageal submucosal tumor. Finally, although the short-term clinical and imaging outcomes were favorable, longer-term follow-up beyond the early postoperative period remains unavailable. Continued surveillance is therefore required to assess the durability of pseudoaneurysm exclusion, infection control, endograft-related complications, and the risk of delayed AEF.
This case highlights several key clinical lessons. A posterior mid-esophageal submucosal protrusion with ulceration and an adherent clot should be recognized as a potential vascular warning sign rather than a primary tumor-like lesion. In patients with dysphagia, unexplained fever, anemia, or atypical esophageal bleeding, these features should prompt contrast-enhanced CT/CTA evaluation before biopsy, endoscopic ultrasound-guided tissue acquisition, or therapeutic endoscopic manipulation. When prolonged unexplained fever coincides with a saccular or pseudoaneurysmal aortic lesion and routine evaluation fails to identify an alternative cause, Brucella serology and prolonged blood culture should be considered to support diagnosis. A suspected infected aortic pseudoaneurysm with esophageal compression warrants urgent multidisciplinary management.
A suspected Brucella-associated descending thoracic aortic pseudoaneurysm can mimic an ulcerated esophageal submucosal tumor and present with dysphagia and UGIB. Posterior mid-esophageal protrusion with ulceration, adherent clot, dysphagia, or unexplained fever should raise suspicion for an extrinsic mediastinal or vascular lesion. Contrast-enhanced CT/CTA should be performed before biopsy, endoscopic ultrasound-guided tissue acquisition, or therapeutic endoscopic manipulation. In patients with prolonged unexplained fever, relevant animal exposure, and a saccular or pseudoaneurysmal aortic lesion, Brucella serology and blood culture with adequate incubation should be considered. TEVAR may enable rapid pseudoaneurysm exclusion and reduce the risk of fatal hemorrhage. However, suspected vascular brucellosis requires targeted antimicrobial therapy and structured imaging follow-up.
The authors thank the radiology team for assistance with computed tomography angiography image review and three-dimensional reconstruction, and the clinical pharmacy team for advice on antimicrobial therapy. The authors also thank the nursing staff involved in peri-procedural care. In addition, the authors sincerely acknowledge the Microbiology Laboratory for assistance in interpreting the microbiological findings.
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