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A rare cause of esophagopleural fistula due to intensity-modulated proton therapy: a case report and review of literature

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Abstract

Esophagopleural fistula (EPF), initially described in 1960, is an abnormal communication between the esophagus and the pleural cavity which can occur due to congenital malformation or acquired due to malignancy or iatrogenic treatment. The most common presenting symptoms are of a respiratory infection, such as fever, chest tenderness, cough and imaging findings consistent with pleural fluid consolidation. In this report, we present a 59-year-old man who exhibited shortness of breath, productive cough, and significant weight loss for 2 weeks. His medical history was significant for smoking-related lung disease and pulmonary squamous cell carcinoma (SCC). His SCC (T4N0) was diagnosed 6 years prior to this presentation and was treated with chemoradiotherapy. The cancer recurred a year ago and he was treated with intensity-modulated proton therapy (IMPT) and consolidation chemotherapy. During admission, he was found to have an EPF by CT scan after initially failing antibiotic treatment for suspected complicated pneumonia and pleural effusion. Multiple attempts of esophagopleural fistula closure were made using endoscopic self-expandable metallic stents and placement of an esophageal vacuum-assisted closure device. However, these measures ultimately failed and, therefore, he required an iliocostalis muscle flap (Clagett window) procedure for closure. Esophageal pulmonary fistulae should be suspected whenever patients have undergone thoracic IMPT and may present with acute pulmonary complications, particularly pneumonia refractory to antibiotic treatment. This case reviews the current literature, potential complications, and treatment options for esophagopleural fistulas.

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Abbreviations

ERF:

Esophago-respiratory fistula

EPF:

Esophagopleural fistula

SCC:

Squamous cell carcinoma

IMPT:

Intensity modulated proton therapy

EGD:

Esophagogastroduodenoscopy

SEMS:

Self-expandable metallic stents

EVAC:

Endoscopic vacuum-assisted closure

IMN:

Internal mammary lymph nodes

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Funding

Author Osman Ali is a gastroenterology and hepatology fellow at the University of Maryland Medical Center and his work efforts are entirely funded by the National Institutes of Health / National Institute of Diabetes and Digestive and Kidney Diseases under T32 DK067872 grant. As a part of the grant agreement, all efforts and time spent on his role in collecting data, writing and editing this manuscript requires disclosure of grant funding.

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Authors and Affiliations

  1. University of Maryland Medical Center Midtown Campus, Baltimore, MD, 21201, USA

    Osman Ali, Suryanarayana Reddy Challa & Raymond E. Kim

  2. American University of Antigua College of Medicine, Coolidge, WI, Antigua and Barbuda

    Sukaina Ali

  3. University of Maryland Medical Center, Baltimore, MD, 21201, USA

    Osman M. Siddiqui & Raymond E. Kim

  4. Department of Gastroenterology and Hepatology, University of Maryland School of Medicine, Baltimore, MD, 21201, USA

    Raymond E. Kim

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  1. Osman Ali
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Contributions

OA collected data, wrote and edited the manuscript. SC collected data and wrote the manuscript. SA wrote and edited the manuscript. RK collected procedural and patient data, wrote and edited manuscript, and is the article guarantor. All authors read and approved the final manuscript.

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Correspondence to Raymond E. Kim.

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The authors report no relevant competing interests.

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All procedures followed have been performed in accordance with the ethical standards laid down in the 1964 Declaration of Helsinki and its later amendments.

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Informed consent was obtained from all patients for being included in the study.

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Written consent for publication was received from the patient on 09/03/2019 during initial hospitalization. A copy of the consent is available from the patient’s medical record upon request by the Editor.

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Ali, O., Challa, S.R., Siddiqui, O.M. et al. A rare cause of esophagopleural fistula due to intensity-modulated proton therapy: a case report and review of literature. Clin J Gastroenterol 14, 955–960 (2021). https://doi.org/10.1007/s12328-021-01388-5

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