Rare case of high amylase pleural effusion without pancreatitis, oesophageal rupture or malignancy
- Nyrvan Baishya ,
- Ruchi Dua ,
- Randeep Singh and
- Ajeesh Krishnadas Padmanabhan
- Pulmonary, Critical care and Sleep medicine, AIIMS Rishikesh, Rishikesh, Uttarakhand, India
- Correspondence to Dr Nyrvan Baishya; nyrvanab@gmail.com
Abstract
High amylase pleural effusion remains an entity which includes a wide variety of differentials, with pancreatitis, oesophageal rupture or malignant pleural effusion being most commonly encountered in clinical practice. Keeping the clinical picture (suggestive of pain in abdomen preceding any respiratory complaints) and differentials at hand, the case was evaluated with contrast-enhanced CT of the thorax and abdomen which revealed normal pancreatic architecture and no abnormal communication was noted between the pancreas and pleural space. A contrast oesophagogram, done when pancreatitis was ruled out, showed no evidence of any leak. The patient underwent upper gastrointestinal endoscopy which was suggestive of an ulcer with fistulous communication with the pleural space. Following nasojejunal feeding and clipping of the fistulous tract the patient’s symptoms improved.
Background
High amylase effusions due to gastropleural fistula (GPF) are rarely encountered. GPF is uncommon in patients presenting as pyopneumothorax. They are uncommon complications of perforated peptic ulcer, traumatic diaphragmatic hernia, malignancy, postsplenectomy, postlobectomy and other conditions.1–3 Clinical presentations include chest pain, shortness of breath and abdominal pain. Untreated cases can prove fatal and surgical management is mostly needed,4 although endoscopic repair of GPF has been reported.5
Case presentation
A man in his 30s, who was a chronic alcoholic presented to our emergency department with abdominal pain involving the left hypochondrium and epigastrium for 2 years. The pain was intermittent, radiated towards the back, aggravated with food intake and had increased in severity over the last 15 days. It was associated with bilious vomiting (two to three episodes per week), loss of appetite and loss of weight (5 kg over the last 15 days).
During the last 15 days he also experienced sudden-onset left-sided chest pain not associated with palpitation and syncope which aggravated on inspiration and coughing. Chest pain was gradually progressive and subsided after taking oral analgesics. After 2 days of initiation of chest pain, there was breathlessness which progressed initially modified medical research council grade I to grade III. He also presentted with cough associated with mucoid expectoration about 10–15 cc/day, aggravated by lying in the left lateral position.
On examination, the patient was afebrile, tachypnoeic, tachycardic, with normal oxygen saturation. On respiratory system examination, there was decreased air entry, tactile vocal fremitus and vocal resonance on the left side of the chest.
Investigations
On routine blood examination he was found to be anaemic (haemoglobin: 75.3 g/L) with raised total leucocyte count (table 1). A chest X-ray was performed which showed left-sided hydropneumothorax (figure 1) following which an intercostal drainage tube was inserted (figure 2). Serum amylase and lipase were sent which were within normal range. Pleural fluid investigations were suggestive of exudative, neutrophilic effusion with high pleural fluid amylase levels (749 IU/mL) (table 2). Pleural fluid cartridge-based nucleic acid amplification test for tubercular aetiology was negative. After initiation of treatment there was a decrease in the total leucocyte count. On evaluation of the cause of anaemia, iron deficiency anaemia was noted.
Depicting routine blood counts
| Investigation | Before antibiotic initiation | After 3 days of antibiotics |
| Haemoglobin | 75.3 g/L | 68.07 g/L |
| White cell count | 63.53 × 109/L | 16.37 × 109/L |
| Platelet count | 305 × 109/L | 209 × 109/L |
Depicting pleural fluid reports
| Pleural fluid | Amylase | Adenosine deaminase | Gram stain | Total white cell count | Lactate dehydrogenase |
| 749 IU/mL | 70.5 IU/mL | Gram-positive cocci with pus cells | 47 000 | 1349 IU/mL |
Chest X-ray on initial presentation.
Chest X-ray after ICD insertion.
A contrast-enhanced CT (CECT) of the abdomen was performed, with clinical suspicion of pancreatitis, which demonstrated hepatomegaly with normal pancreatic architecture (figure 3). In view of high suspicion of pancreatitis, magnetic resonance cholangiopancreatography (MRCP) was performed which was normal. The patient continued to have high volume output with thick yellowish-white pus with debris from the chest tube which ranged from 750 mL to 1100 mL daily despite medical management (figure 4).
Contrast-enhanced CT (CECT) of the thorax and abdomen.
ICD drain showing thick yellowish pus.
Pleural fluid culture was suggestive of Escherichia coli sensitive to only tigecycline and colistin.
Oral contrast study was then performed to look for oesophageal perforation which revealed no perforation. In view of persistently elevated pleural fluid amylase levels and high volume drain, an upper gastrointestinal endoscopy was performed which showing a large ulcer 8 cm × 6 cm in size with a fistulous opening (figure 5). A 1 cm guidewire could be passed across the ulcer base to the thoracic cavity (figure 6).
Upper gastrointestinal endoscopy showing rent in the gastric fundal mucosa.
Fluoroscopic visualisation of the guidewire from the fundus to the pleural space.
Differential diagnosis
Pancreatitis
The patient was initially evaluated in view of history of alcoholism and presence of abdominal pain radiating to the back. Serum amylase and lipase were normal. A CECT of the abdomen and MRCP was done to rule out the above diagnosis.
Oesophageal rupture
An oral contrast oesophagogram showed no evidence of leak.
Tubercular pyopneumothorax
Pleural fluid investigations were neutrophilic and cartridge-based nucleic acid amplification test was negative.
Malignancy presenting with high amylase pleural effusion
A CECT of the abdomen and thorax showed no evidence of mass.
Treatment
A nasojejunal tube was then inserted which reduced the amount and turbidity of intercostal drain.
The fistulous communication was clipped via upper gastrointestinal endoscopy and the patient improved symptomatically. Patient has been planned for surgical repair of the GPF once the inflammation subsides and general condition improves. The surgical management was deferred in view of poor general condition and low albumin. The patient had acquired sepsis during the course of hospital stay and passed away due to septic shock.
Outcome and follow-up
The amount of drain had reduced following nasojejunal feeding and clipping of the fistula. The amount of ICD drain and respiratory symptoms had improved. Surgical closure of the fistulous tract was planned but the patient had acquired sepsis during his hospital stay and expired due to septic shock.
Discussion
GPF is an abnormal communication between the stomach and the pleural cavity. It was first described by Markowitz and Herter in 19606 as a complication of oesophageal hiatal hernia. GPFs are uncommon complications of abdominal and thoracic surgical procedures. Multiple aetiologies reported in the literature include perforated gastric ulcer, gastric bypass surgery, erosion of subphrenic abscess, splenectomy, diaphragmatic injury, pulmonary resection, gastric lymphoma, sleeve resection, Ewing’s sarcoma.7 8 GPF has also been reported as a complication of concomitant chemoradiotherapy.9 There have also been case reports of GPF following bevacizumab chemotherapy.10 11 Most patients present with insidious onset of left-sided chest pain, breathlessness, cough and abdominal pain. Patients may present with gastrointestinal symptoms like pain in the abdomen and haematemesis. In our case the patient presented with pain in the abdomen. Upper gastrointestinal endoscopy revealed an ulcer which probably had perforated leading to fistulous connection. Chronic alcoholism could have predisposed him for ulcer. He had none of the common risk factors like history of surgery/malignancy/chemotherapy/radiotherapy or hiatus hernia . Chest X-ray in these patients may show pleural effusion, pneumothorax, hydropneumothorax, pneumonia or lung abscess. In this case the patient presented with hydropneumothorax which on intercostal drain insertion turned out to be pyopneumothorax.
Water-soluble contrast-based fluoroscopy shows leaking of contrast into the pleural space which was not seen in our case because of the anatomical location of the fistula (fundus of the stomach). CT with oral contrast shows pooling of the contrast medium in the pleural space. Upper gastrointestinal endoscopy also helps in identifying the leak site. The presence of food debris in the intercostal chest drainage also raises the suspicion of GPF. In this case the pleural fluid had debris which led to clogging of the ICD multiple times but the patient never reported passage of food particles via ICD. Contrast-based fluoroscopy also did not reveal the aetiology. CT was unremarkable and did not reveal the aetiology but direct visualisation by UGIE proved to be diagnostic.
Guidelines regarding management of GPFs are lacking, and most of the evidence is from anecdotal case reports. Medical management includes intercostal chest tube drainage for pleural effusion, feeding through jejunostomy or total parenteral nutrition. Definite management of GPF is the surgical closure of the leak site, although endoscopic closures have also been reported in the literature. Endoscopic interventions include fistula closure using stents, argon plasma coagulation,12 clips,13 vicryl and fibrin glue, and suturing.
In our case surgical closure had been planned after improvement of general condition. Delayed diagnosis owing to unusual location of the fistula in the cardia of the stomachs, hypoalbuminemia attributable to high volume GPF and ongoing pleural sepsis had contributed to high preoperative risk in our patient. As part of shared decision making, the endoscopic procedure was opted by the patient and his relatives before proceeding for any surgical intervention carrying higher risk. In the case report of postradiation GPF by Baka et al,4 sequential endoscopic procedures failed twice and finally surgical repair of the fistula was successful. However, the authors feel that early surgical intervention in spite of the high preoperative risk might have potentially led to a different outcome.
Learning points
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Pyopneumothorax with high amylase needs an in-depth search to rule out abdominal causes which includes history of any abdominal complaints followed by adequate imaging to come up with appropriate diagnosis.
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Any leaks with communication from the fundus to the pleura can be missed in contrast oesophagogram.
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Patients with pyopneumothorax irrespective of the cause needs to be evaluated and monitored for signs of sepsis as any deleterious events may be seen.
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Early surgical closure of gastropleural fistulas may prevent associated morbidity and mortality.
Ethics statements
Patient consent for publication
Footnotes
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Contributors NB: main contributing author; RD: drafting the work or revising it. RS and AKP: manuscript conception and design.
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Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.
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Case reports provide a valuable learning resource for the scientific community and can indicate areas of interest for future research. They should not be used in isolation to guide treatment choices or public health policy.
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Competing interests None declared.
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Provenance and peer review Not commissioned; externally peer reviewed.
- © BMJ Publishing Group Limited 2022. No commercial re-use. See rights and permissions. Published by BMJ.
References
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