Recognition and Management of Nonaccidental Esophageal Perforation

A 5-week-old previously healthy girl presented to the emergency department at our tertiary care children’s hospital 1 hour after developing sudden onset respiratory difficulty. On initial presentation, she was hypothermic, tachycardic, and apneic. Positive pressure ventilation was initiated, followed by an atraumatic intubation on the first attempt. Chest radiography revealed subcutaneous emphysema tracking to the neck (Fig 1), and further examination revealed bruising to the child’s abdomen, medial thighs, lower flank, and perineum. These injuries raised suspicion for nonaccidental trauma, and a consult was placed to the Child Assessment Team. This team, consisting of a child abuse pediatrician and social worker, initiated a NAT workup on the day of admission, including head computed tomography (CT), abdominal CT, and laboratories to evaluate for occult injuries or an underlying bleeding disorder. Further questioning revealed that the child lived at home with both parents and had recently missed her 1-month newborn appointment with her pediatrician. When probed about the child’s injuries, the history provided by the child’s mother was disjointed and conflicting, although she did not express any concern for intentional harm. Dilated eye examination was negative for retinal hemorrhages, and skeletal survey was deferred until the patient was stabilized. The child was subsequently admitted to the PICU.

The patient was also assessed by the otolaryngology department on the day of admission. Neck and chest CT to assess for laryngotracheal injury was obtained, with findings concerning for pharyngeal wall or cervical esophageal injury. The patient was then taken to the operating room for direct laryngoscopy, bronchoscopy, and esophagoscopy, which revealed a 1 cm transmural defect in the left lateral wall of the proximal esophagus, just distal to the cricopharyngeus muscle. Granulation tissue was seen at the left lateral hypopharyngeal wall, and the upper esophageal sphincter was intact (Fig 2). Under direct endoscopic visualization, a nasogastric tube was placed distal to the tear to facilitate enteric feeding (Fig 3). Broad-spectrum antibiotics were initiated, and a Replogle tube was placed in the right pharynx to divert saliva away from the defect.

The patient was slowly weaned off the ventilator after 12 days, and antibiotics were continued for a total of 20 days. An esophagram to evaluate the status of the injury was obtained on hospital day 13 that revealed persistent esophageal extravasation (Fig 4). Esophagoscopy the following day revealed the 1 cm defect in the left superior esophagus appeared to be healing appropriately. A repeat esophagram obtained just >3 weeks after the initial insult revealed resolution of the esophageal perforation (Fig 5).

After stabilization in the PICU, a skeletal survey was performed on hospital day 12. This revealed multiple subacute fractures of the right sixth rib and the left sixth-eighth ribs. Although the mother of the patient had initially denied any history of trauma, she later reported that the father had admitted to sticking his finger in the child’s mouth because he “thought she was choking.” When questioned about the bruising and fractures, she stated that the father was “heavy handed.” It was also noted that the patient’s 2-year-old sibling was in custody of Child Protective Services (CPS) because of an inflicted burn injury that occurred 1 year previous. Reports were made to CPS and law enforcement during the admission, and the patient was discharged into foster care after a 24-day hospital course. The child was seen in clinic 1 week after discharge, and, with a repeat esophagram, closure of the defect was confirmed.

Esophageal perforation is a rare occurrence in the pediatric population. Between 71% to 84% of cases are iatrogenic in nature, typically resulting from stricture dilation or nasogastric tube placement.³  Regardless of the etiology, the most common presenting symptom is dysphagia, followed by dyspnea and fever. Perforation of the cervical esophagus, as seen in this case, generally presents less severely because the attachment of the esophagus to the prevertebral fascia prevents the spread of pathogens to the mediastinum.³  Serious complications of esophageal perforation include acute mediastinitis, sepsis, pneumothorax, and death. Because of the high risk of mortality, the timeliness of establishing the diagnosis of esophageal perforation is critical. Prompt diagnosis is of even greater importance in an infant presenting with esophageal injury, which may indicate NAT, because of the high incidence of recurrent and escalating abuse. We advocate for emergent workup of patients presenting with symptoms consistent with esophageal injury, including CT of the neck and chest, which may reveal periesophageal air or fluid, or an esophagram, which can be used to confirm leak. However, because of the low sensitivity of CT in detecting esophageal insult, esophagoscopy may be necessary to directly visualize the extent of the injury.

At present, there are few published reports of esophageal perforation resulting from nonaccidental trauma. In one such report, the authors describe a 15-day-old infant found to have an extensive posterior midline pharyngeal tear from the uvula to the cricopharyngeus muscle. Medical management with parenteral nutrition was employed, and barium swallow and endoscopy was used to monitor for resolution of the injury. A NAT workup was completed, and a report was made to social services, although the county did not assign a case worker and determined that the case did not require any further investigation. The patient was discharged from the hospital under the care of her parents. At 3-months of age, the child presented to the emergency department for apnea and was pronounced dead as a result of abusive head trauma.⁴  In another report, the authors describe a 3-month-old girl presenting with a large 3.5 cm hypopharyngeal laceration as well as a 1 cm esophageal perforation resulting from a caregiver intentionally sticking an object in the patient’s throat. The patient was successfully treated nonoperatively with intravenous antibiotics and nasogastric feeds for 2 weeks before being transitioned to oral feeds.⁵  Although nonoperative therapy was successful in these cases, the appropriate management of these patients has not been well defined because of the rare nature of this condition.

In general, treatment of esophageal perforation involves either surgical repair or medical management. The decision to manage operatively versus nonoperatively should be based on the extent of injury as well as the patient’s clinical status. Medical management can be considered for previously healthy, hemodynamically stable pediatric patients with no evidence of mediastinitis. In these cases, we recommend initiation of broad-spectrum intravenous antibiotics to prevent infection, the insertion of a nasogastric tube under direct visualization to allow for enteric feeding, and consideration for placement of a Replogle tube to divert saliva away from the defect. Interval esophagrams should be obtained to assess for appropriate healing. Operative management is indicated for patients who fail medical treatment.

The successful outcome of this case supports the use of nonoperative management for select pediatric victims of nonaccidental esophageal injury. This is consistent with previous reports in which the authors suggest medical management for cervical esophageal injuries unless they are >2 cm, diagnosis is delayed, or the patient is septic.⁶  In 1 study of a neonatal population, researchers reported 96% of patients demonstrated resolution of esophageal perforation in a median of 7 days.⁷  Thus, with our case, we highlight that nonaccidental esophageal injuries in pediatric patients can be managed in a similar manner, with the expectation of eventual resolution.

Perhaps most importantly, a high index of suspicion for NAT should be maintained for children and, especially, infants presenting with esophageal perforation in the absence of a clear etiology. Although involvement of CPS is imperative to the safety of these patients, their involvement does not always guarantee safe outcomes, as evidenced by the earlier referenced case of a child who ultimately succumbed to subsequent abusive head trauma. That case highlights the potential for catastrophe when there is a failure to intervene, and it reinforces the importance of advocating for these children. As medical providers, we do not control the actions taken by CPS, but we have the opportunity to explain the medical reasoning underlying our level of concern to nonmedically trained CPS caseworkers. In our case, the esophageal perforation and other injuries were communicated to CPS and law enforcement, and the patient was removed from the presumed abusive environment. At her most recent follow-up, the patient remained well healed from her esophageal injury. Her follow-up skeletal survey identified additional healing rib fractures presumed to have been inflicted at the same time as the previously identified fractures but less apparent on initial radiographs. At the time of submission, the patient remains in foster care, with no additional cutaneous or bony injuries noted at any follow-up appointments.

In conclusion, with this case, we illustrate the importance of prompt recognition of abusive injuries to facilitate child protection. In particular, esophageal injury in an infant may indicate possible abuse and should be immediately reported to CPS and law enforcement to prevent the possibility of further harm to that child or others in the home. Our case may help empower future medical providers who are faced with unexplained esophageal injuries in young children to communicate a high concern for NAT.

CT

computed tomography

CPS

Child Protective Services

NAT

nonaccidental trauma