Ann Thorac Surg 2010;90:e28-e29. doi:10.1016/j.athoracsur.2010.05.020
© 2010 The Society of Thoracic Surgeons
Case Reports
Esophageal Mucocele With Compression of the Right Recurrent Laryngeal Nerve 20 Years After Surgical Intervention for Caustic Esophagitis
Yu-Jen Cheng, MDa,
Kang-Hung Wang, MDa,
Hung-Chi Chen, MDb,
Kun-Chou Hsieh, MDa,
Po-Chih Chang, MDa,*
a Division of Thoracic Surgery, Department of Surgery, E-DA Hospital/I-Shou University, Kaohsiung County, Taiwan
b Division of Plastic and Reconstructive Surgery, Department of Surgery, E-DA Hospital/I-Shou University, Kaohsiung County, Taiwan
Accepted for publication May 6, 2010.
* Address correspondence to Dr PC Chang, 1, Yi-Da Road, Jiau-shu Tsuen, Yan-chau Shiang, Kaohsiung County 824, Taiwan (Email: dyno910076{at}hotmail.com).
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Abstract
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A 48-year-old woman presented with 3 days of hoarseness and 1 week of unrelenting back pain. Her medical history was remarkable for an esophageal operation 20 years before to repair damage from caustic esophagitis. The computed tomography scan identified a large esophageal mucocele. Resection of the remaining intrathoracic esophagus was required for complete resolution of the patient's symptoms.
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Introduction
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Esophagectomy with simultaneous reconstruction remains the standard of care in the treatment of severe esophageal stricture secondary to caustic injury. Complete excision can prevent malignant transformation in esophageal remnants; however, some surgeons may perform esophageal exclusion instead due to the simplicity of the procedure and its lower complication rate [1]. The latter procedure may result in the formation of an esophageal mucocele, with an increased risk of infection from poor drainage of impacted mucus.
Symptomatic compression from large esophageal mucoceles is not uncommon, and respiratory distress related to tracheobronchial compression has been reported; however, recurrent laryngeal nerve involvement is rarely seen [2, 3]. This case report involves a patient with hoarseness and back pain secondary to esophageal mucocele as a complication of esophageal exclusion and reconstruction with a colon flap in the treatment of caustic esophagitis.
A 48-year-old woman with a history of caustic esophagitis after the ingestion of sodium hydroxide was successfully treated by subcutaneous colon interposition and esophageal exclusion. She presented to the emergency department 20 years later with persistent, intractable back pain for 1 week and unexplained hoarseness for 3 days.
The physical examination was remarkable for a flaccid neoesophagus visible within the right anterior chest wall and prominent engorgement of the right jugular vein. Stridor was noticeably absent. The laboratory evaluation revealed neutrophilia and elevation of C-reactive protein (139.0 mg/dL). A chest roentgenogram demonstrated mediastinal widening that suggested the presence of a mediastinal mass (Fig 1A). A computed tomography scan identified a very large esophageal mucocele with a diameter of almost 7 cm (Fig 1B). Surgical excision was expedited due to underlying concerns of rupture and subsequent infection.
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Fig 1. (A) A chest roentgenogram shows widening of the mediastinum. (B) A chest computed tomography scan identified a large esophageal mucocele at the right, middle mediastinum with tracheal compression (arrowhead) and partial obliteration of the right brachiocephalic vein (arrow).
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Right posterolateral thoracotomy was performed to visualize the enlarged esophagus and surrounding dense mediastinal pleura (Fig 2). More than 300 mL of dirty discharge was initially evacuated. After a longitudinal incision was created in the mediastinal pleura, the mucocele, along with the intrathoracic esophagus, was resected. Both ends of the esophageal mucocele were reinforced with hand sutures.
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Fig 2. Intraoperative finding: The distended esophageal mucocele (arrowhead) was covered with thick mediastinal pleura (arrow).
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The distended jugular vein disappeared almost immediately after the operation. The patient experienced complete resolution of her back pain and an uneventful postoperative course. She was discharged 13 days after the operation, and her hoarse voice disappeared 1 month later. The final pathology report confirmed a large esophageal mucocele with normal inner mucosa.
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Comment
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Esophageal mucoceles are rare and result from the surgical exclusion of a diseased or damaged esophagus, as seen in cases of caustic injury, perforation, or bleeding varices [2, 4]. Although esophageal mucoceles are usually asymptomatic, they can enlarge, resulting in tracheobronchial compression with chest pain or back pain due to their mass effect [5]. Interestingly, this patient presented not only with back pain but also with hoarseness and jugular vein distention on examination. This enormous mucocele created significant compression over the right thoracic inlet and resulted in compression of the right recurrent laryngeal nerve and right brachiocephalic vein. Laryngoscopic examination was not performed due to concerns of rupturing the infected mucocele. The hoarse voice improved relatively soon after mucocele resection, which suggests the mucocele compression resulted in neurapraxia or nerve palsy to the right recurrent laryngeal nerve.
Cardiovascular pathology, such as ruptured or distended aortic aneurysm, aortic dissection, enlarged left atrium, or mitral stenosis, can also cause hoarseness [6]. Cardio-vocal hoarseness is associated more commonly with left recurrent laryngeal nerve involvement than right [7]. Only a few case reports have demonstrated recurrent laryngeal nerve palsy secondary to compression from a distended esophagus, such as achalasia [3]. This patient presented with esophageal mucocele complicated with hoarseness that was successfully relieved after surgical intervention.
Symptomatic mucocele within the remaining intrathoracic esophagus is best treated with resection by thoracotomy, and this intervention generally provides a satisfactory result [2, 5]. Some clinicians advocate cervical exploration with tube drainage or laparotomy with internal drainage; these techniques could be considered for those patients unsuited to undergo an extensive surgical intervention [4, 8]. Esophageal exclusion should be used judiciously and is indicated for patients with a limited life span [2]. Maintaining a high degree of suspicion for esophageal mucocele in patients whose medical histories involve esophageal operations will facilitate early recognition and prompt treatment.
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References
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- Mulpuru SK, Vasavada BC, Punukollu GK, Patel AG. Cardiovocal syndrome: a systematic review Heart Lung Circ 2008;17:1-4.[Medline]
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- van Till JW, van Sandick JW, Cardozo ML, Obertop H. Symptomatic mucocele of a surgically excluded esophagus Dis Esophagus 2002;15:96-98.[Medline]
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Abstract
Introduction
