Ann Thorac Surg 2002;73:1294-1296
© 2002 The Society of Thoracic Surgeons
Case report
Spontaneous esophageal perforation presenting as meningitis
Carlo C. Jurani, MD*a,
Gerald L. Early, MDb,
Shauna R. Roberts, MDb
a Department of General Surgery, University of Kansas, Kansas City, Kansas, USA
b Department of Cardiothoracic Surgery, Genesis Heart Institute, Davenport, Iowa, USA
Accepted for publication August 17, 2001.
* Address reprint requests to Dr Jurani, Department of General Surgery, University of Kansas, 4002 Murphy Administration Building, 3901 Rainbow Blvd, Kansas City, KS 66160-7119 USA
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Abstract
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This report describes a unique case of spontaneous esophageal perforation (Boerhaave’s syndrome) presenting as meningitis. After a delay in diagnosis (16 days), the patient was successfully treated with debridement, primary closure, and drainage. Although rare, central nervous system infections have been reported in association with esophageal perforation caused by instrumentation, trauma, and malignancy. We report this case of spontaneous esophageal perforation giving rise to meningitis.
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Introduction
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The deposition of oral aerobic and anaerobic bacteria into the mediastinum after esophageal disruption leads to a rapidly spreading and often lethal infection. Although uncommon, the infection can extend to the central nervous system (CNS) and manifest as meningitis, brain abscess, or vertebral osteomyelitis. The following case report is a description of spontaneous esophageal perforation leading to meningitis.
A 42-year-old woman presented to an outside hospital with a 2-week history of progressive upper back and neck pain that began after straining to lift an object at work. She complained of confusion, blurred vision, weakness, and fever. She admitted to severe coughing spells, but no emesis. Initial studies showed a white blood cell count of 36,000/mm3 and a urinary tract infection for which she was started on ciprofloxacin. Blood cultures grew Staphylococcus aureus. Chest roentgenogram uncovered a right apical lesion, and computed tomography scan of the chest revealed a right apical lung mass extending into the mediastinum and right neck. Computed tomography of the head demonstrated marked pneumocephalus. Because of uncertainty of the primary diagnosis and her deteriorating clinical status, the patient was transferred to our institution.
She was resuscitated in the intensive care unit, and broad-spectrum antibiotics were started. Chest computed tomography demonstrated a pneumomediastinum, and magnetic resonance imaging characterized the right paraspinous mass as containing gas and necrotic debris and showed air in the spinal canal (Fig 1).
A lumbar puncture was discussed; however, an esophagram revealed extravasation of contrast from the posterolateral aspect of the proximal esophagus with accumulation in the right apex (Fig 2),
and the patient was taken immediately to the operating room.
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Fig 1. Magnetic resonance imaging, sagittal view, T1-weighted image demonstrating air in the thecal sac (arrowheads) and paraesophageal abscess (arrow).
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Through a right thoracotomy incision the esophagus was exposed and found to have a perforation at the level of the second thoracic vertebral body. Purulent material from the area subsequently grew out Staphylococcus aureus, Streptococcus organisms, and Bacteroides organisms. The area was debrided, exposing the vertebrae, but no gross communication with the spinal canal was identified. The esophagus was repaired primarily in two layers with a pleural flap, and the mediastinum was widely incised and irrigated. The chest was closed with three thoracostomy tubes draining the area, and esophagoscopy was performed, demonstrating no masses or strictures. The right neck was then drained, a pharyngostomy was created, and a gastrostomy was performed to establish drainage from above and below.
The patient remained septic and ventilator dependent in the early postoperative period. An esophagram 10 days postoperatively demonstrated a small leak at the area of the repair, and a percutaneous drain was placed into the fluid collection to provide a controlled esophagocutaneous fistula. During the next several weeks the patient’s clinical status waxed and waned but gradually improved. An esophagram 11 weeks after the repair demonstrated no leak, and after 13 weeks of hospitalization she was discharged home on a full liquid diet with no drains, and no neurologic deficits.
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Comment
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Central nervous system infections are rare complications of esophageal perforation. They have been reported after stenting for benign stricture [1], after blunt traumatic esophageal rupture [2], in a patient with esophageal cancer [3], and as a delayed complication of dilatation of lye strictures in children [4]. The spontaneous origin of the esophageal rupture and the apparent esophagosubarachnoid fistula make this case unique.
In our case, mental status changes, fever, chest mass, and bacteremia were all present without a unifying primary diagnosis, and until pneumomediastinum was manifest on computed tomography of the chest, the integrity of the esophagus was not questioned. The location (proximal) and cause (spontaneous without emesis) in the present patient are atypical for Boerhaave’s syndrome, which is usually associated with a distal esophageal perforation after forceful emesis. Contrast study of the esophagus, which is both highly sensitive and specific, was diagnostic.
Although unusual under any circumstances, CNS infections seem to have been more frequently reported with proximal esophageal perforations [1–3]. The posterior cricopharyngeal region is the most common site of iatrogenic perforation, and at this area the outer longitudinal esophageal muscle layer diverges as two fasciculi exposing a V-shaped area [5]. The proximity of the cervical esophagus to the spinal column may play a role in the development of CNS infections, and this may help explain the rare occurrence of CNS infections after distal perforations.
The management of patients with CNS infections associated with esophageal perforation varies according to the clinical circumstance. Kotler and associates [4] have reported CNS infections 4 to 9 months after the occurrence of esophageal perforation in which brain abscesses were drained operatively and meningitis was treated with antibiotics. When CNS infection is identified while there is ongoing contamination from the esophageal perforation, the management is not straightforward. One patient with an esophageal subarachnoid fistula initially improved with antibiotic treatment, then died when meningitis recurred [3]. Subarachnoid fistulas if identified intraoperatively can be repaired by simple ligation. Like others [6], we favor primary esophageal repair for Boerhaave’s syndrome even if it is more than 24 hours since perforation. This approach, combined with mediastinal debridement and drainage plus a pharyngostomy and gastrostomy, resulted in a good outcome in this desperately ill woman.
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References
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Abstract
Introduction
