Management of hypopharyngeal and esophageal perforations in children: three case reports and a review of the literature.

Abstract

We report 2 cases of pediatric hypopharyngeal perforation that occurred during endoscopy and 1 case of esophageal perforation that developed during nasogastric tube insertion at a tertiary care academic medical center. These cases were identified during a retrospective chart review. All 3 patients were treated with intravenous antibiotics and nasogastric tube feedings, and none experienced further sequelae. Perorations of the hypopharynx and esophagus in children during endoscopy or insertion of endotracheal and nasogastric tubes are not uncommon. Many affected children can be managed conservatively without surgical drainage, depending on the cause and specific location of the perforation and the timing of the diagnosis. We discuss the clinical criteria for various management options, and we offer an algorithm that outlines important clinical considerations in the decision-making process. Our aim in presenting these cases is to increase awareness of the management options for children with hypopharyngeal and esophageal peoCorations and to demonstrate the effectiveness of nonsurgical management in selected cases.

Introduction

The severity of the sequelae of hypopharyngeal and esophageal perforations varies greatly, and consistent guidelines on management are lacking. The choice of treatment varies depending on the cause and specific location of the perforation and the time to recognition. We report the successful conservative management of 3 children who experienced an iatrogenic perforation, and we discuss the current literature. We also provide an algorithm that highlights the most important considerations in the decision-making process. When treating such a case, the surgeon should pay particular attention to each patient's specific clinical conditions because they can help determine whether conservative management may be safely pursued.

Case reports

Patient 1. An infant boy with a history of hypoplastic left heart syndrome was injured during placement of a nasoenteral feeding tube. Following insertion, a small amount of blood was aspirated from the lumen. An immediate chest x-ray revealed that the tube had exited the aerodigestive tract at the level of the hypopharynx, tracked inferiorly in the mediastinum, and followed the course of the right hemidiaphragm laterally.

The tube was removed uneventfully before it had been used, and the patient was placed on a 1-week course of ampicillin. Three days later, a nasojejunal tube was placed under fluoroscopic guidance without difficulty. Although the patient died soon thereafter, a postmortem examination confirmed that the congenital heart disease had been responsible; no significant contribution by any other factor was observed.

Patient 2. A healthy 2-year-old boy presented to the emergency room choking and gagging after he had ingested a small metal toy. Physical examination revealed no abnormality except mild drooling. Chest radiography located the toy in the esophagus at the level of the thoracic inlet. The object was removed via rigid esophagoscopy. Following removal, a small superficial abrasion of the esophageal mucosa was noted.

Approximately 12 hours later, the patient developed a fever of 38.5[degrees]C. He exhibited no other signs or symptoms, and findings on a repeat chest x-ray were normal. However, computed tomography (CT) of the chest revealed the presence of periesophageal air in the neck and mediastinum.The patient's oral diet was halted for 36 hours, and intravenous ampicillin/sulbactam was administered. The results of a barium swallow examination suggested a small mucosal tear, although no extravasation of the contrast material was seen. A nasogastric tube was placed; it was removed after the fever resolved. The patient had no other symptoms. He resumed an oral diet and finished a 10-day course of amoxicillin/clavulanate at home. No sequelae were noted.

Patient 3. A 6-day-old boy with Pierre Robin syndrome underwent rigid bronchoscopy for evaluation of the lower airway. Intubation proved difficult, however, and the bronchoscope perforated the lower pharynx and esophagus. An endotracheal tube and nasogastric tube were immediately inserted under direct vision. A chest x-ray demonstrated pneumomediastinum. The results of the physical examination were notable for neck crepitation. The patient's white blood cell count and body temperature remained normal. He was kept on intravenous antibiotics until extubation a few days later, and he was discharged without any problems.

Discussion

Most esophageal perforations are iatrogenic, and most occur in the thoracic or cervical segments. 1.2 Patients with these types of perforations generally have a better outcome than do those whose perforations (1) are caused by spontaneous rupture or (2) occur after surgical anastomosis. (1) Overall, esophageal perforations are fatal in as many as 29% of cases. (3)

Diagnosis. The most important prognostic factor is the amount of time that has passed between the acquisition of the injury and the initiation of therapy (3) Signs suggestive of esophageal perforation may be absent or delayed; even when they are evident, they may be nonspecific. The classic presenting signs include pain in the neck or substernal area, subcutaneous or mediastinal emphysema, and fever. Patients may also exhibit other signs suggestive of upper aerodigestive tract infection, including trismus, dysphagia, odynophagia, drooling, and/or dyspnea. (1,4)

A suspicion of esophageal injury demands rapid diagnostic testing, usually beginning with a chest and upright abdominal film. (1) X-rays often show signs of pneumothorax or pneumomediastinum, mediastinal widening, subcutaneous emphysema, or pleural effusion. One study of radiographic findings in patients with esophageal perforations found abnormalities in 88% of cases, so while plain x-rays are usually positive, a negative x-ray alone is insufficient to rule out a tear (5) Confirmation of the injury and information about its location and extent can be obtained from contrast x-ray studies. Although barium is more sensitive than water-soluble agents, especially in the case of small tears, (6) it is more damaging when there is extravasation. Therefore, barium should be reserved for cases in which an initial water-soluble contrast study is negative. (7)

CT offers the advantage of precise localization, which can aid in treatment. It can also aid in the diagnosis of atypical presentations, (8) and it is helpful in cases in which clinical suspicion of perforation remains despite negative contrast studies. (9) CT is also a useful modality for reevaluation after the initiation of therapy. (10)

Treatment. Criteria to help select the appropriate level of treatment are still evolving. The classic management strategy for esophageal perforations is aggressive surgical debridement and drainage. In rare cases, esophagectomy may be necessary. At the other end of the management spectrum, however, is an emerging understanding that many patients may be safely treated via much less morbid and less invasive means. (11) Nonsurgical management in children with iatrogenic esophageal perforations may be justified on a variety of grounds: These perforations are generally small, located in the cervical esophagus, discovered promptly, and associated with little soilage. In addition, most children with an esophageal perforation lack many of the risk factors for complications that are often seen in adults: unrelievable distal obstruction, malignancy, repeated dilation procedures, and chronic systemic corticosteroid treatment. (10) Finally, the availability of broad-spectrum antibiotics and nonoral nutritional support (both enteral and parenteral) further expands nonsurgical treatment options.

A variety of authors have reported successful cases of conservative management, although the criteria for what actually constitutes "conservative" management of an esophageal perforation is not universally agreed upon. (2,11-16) What emerges from reviewing these many reports and small case reviews are a few basic guidelines for successful conservative management:

* Conservative management can be attempted when the patient is diagnosed early and is hemodynamically stable, although possibly symptomatic.

* Use high-dose and/or broad-spectrum antibiotics.

* Confirm the healing progress with follow-up radiologic studies.

Surprisingly, these reports have shown that many patients were successfully treated conservatively despite distal perforations, high fever, leukocytosis, and even continuation of an oral diet. Endicott et al recommend that patients eligible for conservative treatment are those (1) whose perforation was quickly discovered, (2) whose injury was asymptomatic, (3) who had not taken anything orally since the insult, and (4) who had no esophageal obstruction distal to the perforation. (10) Such a description fits most children with iatrogenic esophageal perforation. Other research suggests that nonsurgical therapy is best considered only in selected patients, including those with cervical esophageal perforations caused by instrumentation. (2,12,16)

We have developed an algorithm outlining the basic components of conservative management of esophageal perforation (figure). The first step is the initiation of a "nothing by mouth" regimen for at least 1 week; nutrition is generally supplied via a nasogastric tube. Patients are given a broad-spectrum antibiotic for 1 to 2 weeks. Any extraesophageal fluid collections must be drained promptly. A patient's failure to improve within 24 hours should trigger a prompt reassessment, generally with a chest x-ray. If the child's condition worsens, CT and directed surgical intervention may become necessary. There is little agreement on the role of nasogastric drainage; successful cases with it and without it have been reported. (14)

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In conclusion, the traditional management of esophageal perforation has been driven by our experience with spontaneous intrathoracic rupture in adults. In these cases, which generally involve massive chest soilage and significant comorbidities and barriers to healing, highly aggressive surgical intervention represents the only significant chance for survival. In recent years, however, advances in endoscopy and neonatology have led to a marked increase in the incidence of iatrogenic high-esophageal perforations in young patients. Differences in the etiology of esophageal perforations and the general and specific health problems of young patients have led to an emerging protocol for nonsurgical management. The ideal candidates are those with no history of esophageal disease or obstruction in whom iatrogenic cervical esophageal perforations (often caused by alimentary tube misplacement or other instrumentation) are discovered quickly. With appropriate nutritional alternatives, antibiotics, and radiologic studies, the long-term outcomes in these cases can be excellent.

Eric D. Baum MD; Lisa M. Elden, MD; Steven D. Handler, MD; Lawrence W.C. Tom, MD

From the Division of Otolaryngology, Children's Hospital of Philadelphia, and the Department of Otorhinolaryngology-Head and Neck Surgery, University of Pennsylvania School of Medicine, Philadelphia.

Corresponding author: Lawrence W.C. Tom, MD, ORL/1 Wood, Children's Hospital of Philadelphia, 34th and Civic Center Blvd., Philadelphia, PA 19104. Phone: [phone]; fax: [phone]; e-mail: [e-mail]

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