Ruptured celiac artery aneurysm mimicking Boerhaave syndrome

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Ruptured Celiac Artery Aneurysm Mimicking Boerhaave's Syndrome

Chien-Yu Liu

^a,b

, M.D., Kai-Wei Yang

^a,b

, M.D., Wei-Kung Chen

^a,b

, M.D., and Chen- Hsiung Huang

^a,b

, M.D.

a

Department of Emergency Medicine, China Medical University Hospital, Taichung, Taiwan

b

School of Medicine, College of Medicine, China Medical University, Taichung, Taiwan

Correspondence:

Dr. Chen-Hsiung Huang, Department of Emergency Medicine, China Medical University Hospital, #2, Yuh-Der Road, 404 Taichung, Taiwan

Tel: +886-4-2205-2121 ext. 5431 Fax: +886-4-2234-4055

Key Words: Celiac Artery Aneurysm; Boerhaave's syndrome; pleural effusion

Word count: 664

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Abstract

Ruptured celiac artery aneurysm is a rare cause for epigastric pain and is usually detected incidentally. Atypical presentation with postemetic epigastralgia and pleural effusion usually lead physicians to make the diagnosis of Boerhaave's syndrome.

Herein we report a 32-year-old woman who was diagnosed with Boerhaave's syndrome initially after presenting with acute postemetic epigastralgia and

predominant left-side pleural effusion. Left thoracentesis yielded bloody fluid with similar amylase level to serum. Esophageal repair was withheld because the chest computed tomography scan showed no evidence of esophageal rupture. However, a ruptured celiac artery aneurysm with retroperitoneal hematoma extending to the posterior mediastinum and bilateral pleural space was found incidentally. Although ruptured celiac artery aneurysm is an uncommon cause for postemetic epigastralgia, acute vascular events such as the previously stated cause should be the first

impression rather than Boerhaave's syndrome if the patient also presents with isolated pleural effusion containing unelevated amylase.

Ruptured celiac artery aneurysm is a rare cause for epigastric pain and is usually detected incidentally. Atypical presentation with postemetic epigastralgia and pleural effusion often lead physicians to make diagnosis of Boerhaave's syndrome. Herein we report a 32-year-old woman who was diagnosed with Boerhaave's syndrome initially after presenting with acute postemetic epigastralgia and predominant left-side pleural effusion. However, the chest computed tomography (CT) scan revealed a ruptured celiac artery aneurysm with retroperitoneal hematoma extending cephalad to the posterior mediastinum and bilateral pleural space. Instead of esophageal repair, she received transcatheter coil embolization for the ruptured aneurysm and had an uneventful recovery.

A 32-year-old female presented to our emergency department because of severe epigastric pain after vomiting several times. Her consciousness was clear but she was quite irritable due to epigastric and central pleuritic chest pain. She also complained of dyspnea, palpitation, and dizziness. She had blood pressure of 112/78 mmHg, pulse rate of 110 beats/minute, respiration rate of 20 times/minute, and body temperature of 37.6℃. She had no personal or family history of vascular disease. She also denied any history of trauma or infection (e.g., endocarditis, tuberculosis, or syphilis). Blood tests revealed a decreased hemoglobin of 9.7gm/dL (normal range 12.0-16.0 gm/dL) and normal amylase of 30U/L (range 28-100 U/L). Chest radiograph showed

predominant left-side pleural effusion without pneumothorax or pneumomediastinum

(Fig. 1). Spontaneous esophageal rupture was considered first based on her clinical

presentation. Diagnostic thoracentesis and chest CT scan were performed to confirm

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the diagnosis. However, neither oral contrast leakage outside the esophagus, pneumothorax nor pneumomediastinum was found. But the subsequent scan with intravenous contrast medium showed a ruptured aneurysm at the celiac trunk, with retroperitoneal hematoma extending cephalad through the aortic hiatus into posterior mediastinum (Fig. 2) and bilateral pleural space. Left thoracentesis yielded bloody fluid with an amylase of 29U/L and a pH of 8.0. Esophageal repair was withheld due to no evidence of esophageal rupture disclosed by the CT scan and the pleural fluid analysis. Transcatheter coil embolization for the ruptured celiac artery aneurysm, followed by aortic stenting with superior mesenteric artery chimney graft were performed successfully. The patient had an uneventful recovery.

Spontaneous esophageal rupture, or Boerhaave’s syndrome, is a rare but life- threatening disease requiring urgent surgical management. Classical symptoms are sudden chest or epigastric pain, dysphagia and dyspnea following severe emesis or retching. Because the left distal third of the esophagus is most vulnerable, pleural effusion predominantly on the left side is the most common finding on chest

radiograph or CT scan [1]. On the other hand, isolated pleural effusion without other pathologic findings like pneumothorax or pneumomediastinum was only observed in 25% of Boerhaave’s syndrome [2]. In a patient suffering from postemetic chest or epigastric pain with pleural effusion, other diseases such as acute myocardial infarction with heart failure, or dissecting aneurysm should also be considered.

Pleural fluid analysis may help to differentiate between Boerhaave’s syndrome and the other diseases. If the level of pleural fluid amylase is higher than the level of serum amylase, pleural effusion secondary to esophageal rupture is more likely, because it is mixed with swallowed salivary amylase.

In our patient, acute vascular event should be considered first because the presentation of isolated pleural effusion with similar amylase level to serum. The tentative diagnosis was further elucidated by the subsequent CT scan. The pleural effusion resulted from the celiac artery aneurysm rupturing into not only the

retroperitoneal cavity but also the thoracic cavity [3]. To the best of our knowledge, only a few cases of a ruptured celiac artery aneurysm presenting with hemothorax had been reported [3, 4]. However, all of them were diagnosed by autopsy after death.

This case emphasizes the importance of evaluating the entire aorta and its branches if a patient presents with sudden onset epigastric pain and isolated pleural effusion.

Although ruptured celiac artery aneurysm is an uncommon cause for postemetic

epigastralgia, it should be considered if the patient presents simultaneously with

unusual isolated pleural effusion containing unelevated amylase.

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References

[1] Ghanem N, Altehoefer C, Springer O, et al. Radiological findings in Boerhaave's syndrome. Emerg Radiol 2003;10:8-13.

[2] Hegenbarth R, Birkenfeld P, Beyer R. [Roentgen findings in spontaneous esophageal perforation (Boerhaave syndrome)]. Aktuelle Radiol 1994;4:337-338.

[3] Carrel D, Cohle SD, Chapman AJ. Fatal hemothorax from mycotic celiac artery aneurysm. Am J Forensic Med Pathol 1992;13:233-237.

[4] Naiken VS, Shapiro JH, Tellem M. Celiac axis aneurysm. Report of a fatal case with rupture into the right pleural cavity. Angiology 1962;13:138-143.

Figure legend

Fig. 1 Chest radiograph showed predominant left side pleural effusion. No pneumothorax or pneumomediastinum was found.

Fig. 2 Sagittal contrast-enhanced CT revealed a ruptured celiac artery aneurysm

(arrow) with retroperitoneal hematoma extending to the posterior mediastinum

(arrowhead).