Elevated tumour markers in the pleural effusion of a patient with spontaneous esophageal rupture: a rare case report

Abstract

Background

Esophageal rupture is a rare but life-threatening condition. Esophageal tumours do not usually cause esophageal ruptures, and elevated tumour markers have rarely been detected in pleural effusion after esophageal rupture with no detectable tumour. The presence of elevated tumor markers in pleural effusion can lead to a misdiagnosis of esophageal cancer in patients with esophageal rupture, resulting in inappropriate treatment.

Case presentation

The 65-year-old male patient who was admitted to the emergency department with left chest pain and dyspnoea after severe vomiting. Chest computed tomography (CT) indicated left hydropneumothorax and mediastinal emphysema. The patient underwent bedside closed thoracic drainage. The drainage fluid was coffee-coloured and turbid, with significantly elevated CA199, CA125 and CEA levels. After transferring the patient to the emergency operating room, the esophageal defect was repaired, and a jejunostomy was performed. No tumours were detected in the thoracic cavity during surgery. The patient recovered and was discharged from the hospital.

Conclusion

Esophageal tumours should be suspected in patients with elevated pleural effusion CA199, CA125 and CEA levels. The findings from chest CT and oesophagography did not support the diagnosis of a thoracic tumor.These tumor markers may be concomitant changes during esophageal rupture.

Supplementary Information

The online version contains supplementary material available at 10.1186/s13019-025-03539-y.

Background

Esophageal rupture is a rare but life-threatening condition that often presents with complications such as pneumothorax or mediastinal emphysema. Esophageal rupture, which has a mortality rate of more than 20%, is usually caused by foreign body perforation, endoscopic manipulation, vomiting or coughing [1]. We report a rare case of elevated pleural effusion tumour markers after esophageal rupture with no detectable tumour. The presence of elevated tumor markers in pleural effusion can lead to a misdiagnosis of esophageal cancer in patients with esophageal rupture, resulting in inappropriate treatment.

Case presentation

The 65-year-old male, who worked for the government before retirement, had a history of atrial fibrillation and underwent radiofrequency ablation one year ago. The atrial fibrillation recurred six months ago, and the patient was treated with rivaroxaban 15 mg daily since then. The patient had no known allergies or other chronic diseases. The patient received no regular medications or supplements besides rivaroxaban. The patient had normal diet and exercise habits and no history of travel, exposure to environmental toxins, smoking, alcohol abuse or drug use. No family history of esophageal cancer or other malignancies was noted. The patient did not exhibit any preexisting psychosocial concerns.

The patient presented with chest pain, dyspnoea and vomiting after dinner. The physical exam revealed fever, tachycardia, and reduced breath sounds on the left side. The patient’s abdomen presented a flat and soft appearance, without any tenderness or rebound pain, and no palpable masses were detected; digital rectal examination was not conducted. No signs of peritonitis, abdominal organ injury, lymphadenopathy or skin lesions were detected. Laboratory tests showed normal liver and kidney function, normal coagulation function, normal stool and normal urine. The patient’s pleural effusion exhibited a brown turbid appearance and tested positive on the Rivalta test. Laboratory analysis of the pleural effusion revealed a white blood cell count of 74,210 × 10^6/L, with neutrophils comprising 81.7% and lymphocytes accounting for 9.1%. The total protein concentration was 46.2 g/L, adenosine deaminase (ADA) activity was 82 U/L, lactate dehydrogenase (LDH) level was 4295 U/L, and glucose concentration was 0.69 mmol/L. Microbiological culture of the pleural effusion demonstrated the presence of Staphylococcus epidermidis and Streptococcus salivarius. Blood leukocytes, neutrophil percentage, haemoglobin and platelets were within normal range. Blood tumour markers (AFP, CA153, CA199, CA125 and CEA) were normal (Table 1). Significantly elevated levels of CA199, CA125 and CEA were detected in the pleural effusion (Table 1). Chest computed tomography (CT) showed left hydropneumothorax and mediastinal emphysema (Fig. 1). Oesophagography showed no abnormalities (Fig. 2). Thoracoscopy showed food debris in the left thoracic cavity (Fig. 3).

Table 1.

Tumor marker results

Tumor Marker	Result	Reference Value
Group A
AFP	< 2.00 ng / ml	0–14
CEA	164.85 ng / ml	0–5
CA125	397.22 U / mL	0–35
CA199	233.14 U / ml	0–27
CA153	3.31 U / ml	0–25
Group B
AFP	< 2.00 ng / ml	0–14
CEA	329.18 ng / ml	0–5
CA125	228.06 U / mL	0–35
CA199	232.46 U / ml	0–27
CA153	6.50 U / ml	0–25
Group C
AFP	2.21 ng / ml	1–9
CEA	< 1.73 ng / ml	0–5
CA125	9.91 U / mL	0–35
CA199	5.74 U / ml	0–37
CA153	6.21 U / ml	0–31
Group D
AFP	2.67 ng / ml	1–9
CEA	1.79 ng / ml	0–5
CA125	6.68 U / mL	0–35
CA199	7.62 U / ml	0–37
CA153	4.80 U / ml	0–31
Ca724	0.88 U / ml	0–7

*Group A: Pleural effusion 5 h before surgery; Group B: Pleural effusion 36 days after surgery

Group C: Blood 5 h before surgery; Group D: Blood 1 year before surgery

Fig. 1.

Chest CT, left hydropneumothorax and mediastinal emphysema

Fig. 2.

The oesophagography

Fig. 3.

The food debris in the left thoracic cavity

The patient was diagnosed with esophageal rupture based on his clinical presentation, chest CT scan and thoracoscopic findings. Left hydropneumothorax and mediastinal emphysema, which were shown on the chest CT, are typical signs of esophageal rupture. The thoracoscopy showed food debris in the left thoracic cavity, confirming the diagnosis of esophageal rupture.

The patient underwent emergency surgery for esophageal rupture, including a thoracotomy, esophageal defect repair, food removal, irrigation and thoracic drainage tube placement. The patient also underwent a jejunostomy for nutritional support. The patient received conservative postoperative treatment with antibiotics, proton pump inhibitors, analgesics and antiemetics. In addition to the jejunostomy tube, the patient received intravenous nutritional support. The patient was not treated for a tumour, as no tumour was found. The patient was advised to have regular follow-ups and tests to monitor his condition and exclude the possibility of a tumour.

The patient adhered well to the therapeutic intervention and stayed in the hospital for 53 days due to a postoperative oesophago-thoracic fistula. The chest and abdomen underwent CT scans and esophagography, which revealed no detectable abnormalities. Subsequently, all drainage tubes were successfully removed. The patient recovered and resumed oral feeding at discharge.

Discussion and conclusion

Herein, we report a rare case of elevated tumour markers in the pleural effusion after esophageal rupture but no detectable tumour. To our knowledge, this phenomenon has not been previously reported. Elevated tumour markers in pleural effusion are associated with tumours. Hackbarth et al. reported that CEA and CA199 levels were significantly higher in malignant effusions compared to non-malignant effusions, particularly in lung cancer and other cancers associated with elevated serum CEA and/or CA199 levels [2]. Choi et al. demonstrated that the CA125 levels were significantly higher in a malignant group compared with levels in a benign group [3].

Esophageal tumours should be suspected in patients with elevated pleural effusion CA199, CA125 and CEA levels. In our patient, the thoracic cavity was contaminated with chyme, and the tissue structure was difficult to identify. Thus, small tumour lesions could have been missed during surgery. Even if a malignant esophageal tumour has been detected, performing radical surgery for esophageal cancer in an emergency is unrealistic. Tumour marker elevation is not always associated with a tumour, i.e. tumour markers are not 100% specific for diagnosing tumours [4–6]. Abnormal levels of tumour markers before surgery make accurate diagnoses difficult. Esophageal tumours should be suspected in patients with elevated pleural effusion CA199, CA125 and CEA levels. The findings from chest CT and oesophagography did not support the diagnosis of a thoracic tumor. These tumor markers may be concomitant changes during esophageal rupture. Therefore, the treatment was not influenced by the elevated tumour markers in the pleural effusion. However, regular follow-up is strongly recommended.

We report a rare case of elevated tumour markers in the pleural effusion after esophageal rupture, but no tumour was detected. This phenomenon was rarely reported in the literature. The cause of tumour markers in the pleural effusion is unclear but may be a combination of multiple factors. Further research is needed to elucidate the mechanism and clinical significance of this rare phenomenon.

The limitations of this case report are related to the rarity and complexity of this phenomenon. We did not perform other tests such as pleural biopsy, cytology, immunohistochemistry or molecular analysis to confirm or exclude the presence of tumour cells in the pleural effusion or tissue. In addition, we did not perform a long-term follow-up to observe the changes and outcomes of tumour markers and pleural effusion in this patient.

Electronic supplementary material

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Author contributions

ZW conceived and designed the study. XL and HL wrote the manuscript. All authors have read and approved the final manuscript.

Funding

The authors declare that they have not received any funding to support the writing and publication of this case report.

Data availability

No datasets were generated or analysed during the current study.

Declarations

Ethics approval and consent to participate

The patient involved in this case report agreed to participate in this study and signed an informed consent form. This study was approved by the ethics committee of the institution where it was conducted and complied with the principles of the Declaration of Helsinki.

Consent for publication

Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.

Informed consent

The patient was fully informed about the diagnosis, treatment and prognosis of his condition. The patient was also informed about the risks, benefits and possible complications of the surgery. The patient was given enough time to ask questions and express his concerns. The patient agreed to undergo the surgery and signed the informed consent form. He was also informed about the purpose and process of this case report. The patient was informed that his personal information would be anonymised and protected. He agreed to participate in this case report and signed the informed consent form.

Competing interests

The authors declare no competing interests.