The Laryngoscope C 2014 The American Laryngological, V

Rhinological and Otological Society, Inc.

Systematic Review

Esophageal Perforation Caused by Edible Foreign Bodies: A Systematic Review of the Literature Ryan M. Aronberg, MS; Salman R. Punekar, MD; Stewart I. Adam, MD; Benjamin L Judson, MD; Saral Mehra, MD, MBA; Wendell G. Yarbrough, MD, MMHC Objectives/Hypothesis: Presentation of a case of esophageal perforation caused by ingestion of a foreign body that was difficult to visualize endoscopically, and systematic review of the current literature with a focus on available diagnostic modalities and missed diagnoses. Data Sources: MEDLINE (1946–2014) Review Methods: We conducted a systematic review of MEDLINE for cases and studies of esophageal perforation due to foreign bodies. Results: A systematic review of esophageal perforations caused by foreign bodies revealed 40 studies that included 168 patients. Neck/chest pain, odynophagia, and dysphagia were the most common presenting symptoms—each occurring in the majority of patients. Where reported, diagnostic sensitivity of lateral neck X-rays was 56%, and computed tomography (CT) scan was 100%. Nine of 168 cases specifically mentioned a missed diagnosis. Very few studies reported a failure to visualize the foreign object at the time of primary rigid or flexible esophagoscopy. No studies reported nonvisualization of the foreign body at the time of open surgery. Conclusion: Esophageal perforations due to foreign bodies are rare but carry potentially devastating complications. Delays in diagnosis are common, and no standard diagnostic algorithm exists. Lateral neck X-rays are an easy but insensitive screening test. Laryngoscopy is an excellent screening test but not always diagnostic. CT is a more sensitive diagnostic method, which can also help guide management based on the detection of complications. Although seldom described, if an object is not visualized at the time of endoscopic or surgical intervention, follow-up CT may be useful in determining the persistence of effects of the perforation or the foreign body. Key Words: Esophageal perforation, esophagoscopy, foreign body, computed tomography. Laryngoscope, 00:000–000, 2014

INTRODUCTION Foreign body ingestion is a relatively common occurrence in the United States. Esophageal foreign bodies account for roughly 1,500 deaths annually in the United States.1 Whereas 80% to 90% of ingested objects pass through the gastrointestinal tract inconsequentially, 10% to 20% will require nonoperative intervention, and < 1% will require surgery.2 Complications due to the ingestion may include ulcers (21%), laceration (15%), erosion (12%), perforation (2%), or migration.3 The likelihood of complications has been linked to the type and size of the object ingested, the time to presentation, and host factors such as age.

From the Department of Surgery, Section of Otolaryngology, Yale University School of Medicine, New Haven, Connecticut, U.S.A. Editor’s Note: This Manuscript was accepted for publication July 30, 2014. The authors have no funding, financial relationships, or conflicts of interest to disclose. Send correspondence to Wendell Yarbrough MD, MMHC, Section Chief, Otolaryngology, PO Box 208041, New Haven, CT 06520–8041. E-mail: [email protected] DOI: 10.1002/lary.24899

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One of the most clinically significant consequences, esophageal perforation, is a potentially devastating event that can be difficult to diagnose and treat properly. Delays in diagnosis and time to treatment are highly associated with morbidity and mortality.4 The challenge of diagnosis may be attributed to the rarity of the condition, its nonspecific symptoms, and its ability to mimic other conditions that present similarly (e.g., acute coronary syndrome). Additionally, no standard protocol exists for diagnostic evaluation, and “screening” diagnostics such as lateral X-rays are not as reliable as previously thought. Here, we present a case in which a patient presented with esophageal perforation due to an impacted foreign body that escaped detection during esophagoscopy and surgery, complicating management. We also systematically review the literature to examine the prevalence and success of various diagnostic modalities used during the workup of foreign body-induced esophageal perforation.

MATERIALS AND METHODS Search Strategy and Selection Criteria A MEDLINE search using Ovid was conducted (1946– May 2013 and updated in March 2014) using the terms “esophageal

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CASE REPORT A 61-year-old female with no significant past medical history presented to her local emergency department complaining of severe pain in her throat that worsened by swallowing. She had been eating red snapper the night before, when she experienced sudden severe pain. Upon presentation, the patient was afebrile with normal vital signs, and examination of the neck revealed some tenderness over the cricoid area but no subcutaneous emphysema. Her white blood cells count was 15,000/ mm3 with 86% neutrophils. Fiberoptic laryngoscopy revealed neither evidence of foreign body nor any signs of hypopharyngeal mucosal trauma. A lateral neck X-ray showed a periesophageal linear radiopaque focus at the T1 level, concerning a fish bone or other foreign object, and evidence of precervical air indicative of esophageal perforation. A CT scan confirmed a 2.5-cm radiopaque foreign body oriented transversely in the cervical esophagus, as well as air in the surrounding soft tissues (Fig. 2). The patient was transferred to our hospital for definitive management. The patient was taken to the operating room (OR) for rigid endoscopy, neck exploration, and primary repair of the esophageal perforation. Thorough endoscopic

Fig. 1. Literature search and selection. perforation” and “foreign bodies.” All subheadings were included in the search. The search design is illustrated in Figure 1. Additional studies were identified by searching the references of the studies found during the search process.

Study Selection Studies were limited to adult cases in which esophageal perforation was a direct result of ingestion of an edible foreign body. Cases in which perforation was iatrogenic or was due to a nonedible foreign body were excluded. Excluding nonedible objects such as dentures allowed for a maximally homogenous population; nonedible objects are unlikely to be overlooked on initial radiographs, endoscopy, or surgery (as in the index case). Studies were further excluded if the foreign body was not confirmed either by endoscopy, surgery, or autopsy. Lastly, we utilized a multi-institutional intralibrary search; and following this, studies for which full text was not available were not included in this review. Included studies were all English language and provided data regarding the diagnosis and management of patients.

Data Collection and Analysis For the purposes of this study, two reviewers (S.P. and A.R.) independently conducted the literature review and evaluated relevant articles. Data collected included, when available, demographic information, details of presentation, diagnostic modalities employed, management decisions, and morbidity and mortality data. Data collected was tabulated, and the following calculations were performed: average age of patients, range of age, geographic distribution, relative frequency of types of foreign bodies, presenting symptoms, management options employed, complications, mortality, median time to presentation, and sensitivity of diagnostics (including X-ray, computed tomography [CT], endoscopy, and surgery).

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Fig. 2. Axial and coronal computed tomography at initial presentation, showing a fishbone penetrating the right cervical esophagus transversely.

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patient’s foreign body sensation subsided, but she had worsening odynophagia and purulence from her JacksonPratt drain. A third CT scan showed a multi-loculated collection suggestive of an abscess. On postoperative day 8, external drainage of retropharyngeal abscess was performed, and the neck was left open with suction irrigation catheters. The patient subsequently improved with intravenous antibiotics and antibiotic irrigations, and was discharged home in good condition 15 days after her initial presentation.

RESULTS

Fig. 3. Postsurgical axial computed tomography of the cervical esophagus, showing persisting fish bone oriented anteroposteriorly, adjacent to a nasogastric tube.

inspection revealed esophageal inflammation and a moderate amount of pus, but no foreign body was identified. An anterior transcervical approach to the upper esophagus was carried out by retracting the carotid sheath laterally, exposing and allowing direct visualization of the esophagus. Small amounts of pus circumscribed an approximately 1-cm lateral perforation in the right esophagus just distal to the upper esophageal sphincter. Careful inspection and palpation of this area of the esophagus failed to reveal a foreign body. The deep neck space was irrigated, and the esophagus was repaired with a running-locking 4-0 Vicryl suture. The patient was admitted for observation, given nothing by mouth, and placed on broad spectrum intravenous antibiotics. In the postoperative period, the patient complained of persistent foreign body sensation and odynophagia. She continued to be afebrile, but her white blood cells count rose to 17,000 /mm3. A repeat CT scan of the neck performed on postoperative day 2 showed a radio-opaque foreign body in the same location, although the orientation had changed (Fig. 3). The patient was returned to the OR for rigid esophagoscopy. This time, a fish bone was identified below the level of the cricopharyngeus muscle (Fig. 4), although there was no esophageal perforation noted. Postoperatively, the Laryngoscope 00: Month 2014

Forty manuscripts reporting esophageal perforations in adults due to foreign bodies, totaling 168 patients identified in the literature, consisting of 29 individual case reports, nine case series, and two large retrospective studies (Table I). One large study of 89 patients60 was not used for tabulation of diagnostic modalities or treatments due to lack of case-by-case data and inclusion of nonedible objects (e.g., dentures). The average age was 59 years old, with a range of 27 to 86 years old. Most reported cases (122/168, 72.6%) occurred in Asia, where the incidence of esophageal perforation is presumably higher for dietary reasons (fish consumption). The most common edible foreign objects associated with perforation were fish and chicken bones, accounting for 54% and 29%, respectively. Although nonedible objects were excluded from this review, dentures were the most common such object associated with esophageal perforation. The median time to presentation was 118 hours. Clinical presentations varied widely, but neck pain, odynophagia, and dysphagia were most common symptoms, each occurring in the majority of patients (Table II). Other forms of pain, such as retrosternal or epigastric pain, were less common and were more likely to occur in thoracic perforations. Rarely, symptoms included hematemesis, dyspnea, or melena. The most common diagnostic studies performed were X-Ray, CT, and flexible laryngoscopy. X-ray results were reported in 25 patients, with a sensitivity of 56%. For cases in which CT was reported, it was positive in

Fig. 4. Operative picture of the fish bone. [Color figure can be viewed in the online issue, which is available at www.laryngoscope.com.]

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TABLE I. Characteristics of Included Studies. Author, Year

Study Site

N

Study Type

Missed Diagnosis

Primary Aim

Donnelly and Deverall, 196824

United Kingdom

1

Case study

Management

Wychulis et al.,196925 Hardy et al., 197326

United States United States

9 2

Case series Case series

Descriptive Descriptive

McCormack and Monroe, 197727

United States

1

Case study

Descriptive

Triggiani and Belsey, 197728

United Kingdom

2

Case series

Vernon and Carmichael Jr, 197829

United States

1

Case study

Diagnosis, management Descriptive

Hardaway, 198130

United States

1

Case study

Descriptive

Russo et al., 198631 Wilson, Dean, and Lewis, 198732

United States United States

1 1

Case study Case study

Descriptive Descriptive

Radford and Wells, 198833

United Kingdom

1

Case study

Descriptive

Nashef et al., 199234 Gougoutas, Levine, and Laufer, 199835 Kamath et al., 199836

France United States

10 1

Case series Case study

Descriptive Descriptive

India

1

Case study

Descriptive

Litzlbauer et al., 199937

Germany

1

Case study

Descriptive

Shimamoto et al., 200038 Nishimaki et al., 200139

Japan Japan

1 1

Case study Case study

Treatment Treatment

Chung, 200340

Hong Kong

1

Case study

Patient sent home after negative XR.

Descriptive

D’Costa et al., 200322

United Kingdom

1

Case study

Patient sent home after negative XR.

Descriptive

Hinojar et al., 200341

Spain

3

Case series

Katsetos et al., 200342

United States

1

Case study

Lam, Woo, and van Hasselt, 200343

Hong Kong

1

Case series

Marco De Lucas et al., 200444

Spain

1

Case study

Medina et al., 200445

United States

1

Case study

Sica et al., 200446

United Kingdom

1

Case study

Ng, 200547

Hong Kong

1

Case study/ Correspondence

Petrou and Wilson, 200548

Australia

1

Case study

Descriptive

Mawdsley et al., 200649 Metz et al., 200650

United Kingdom Netherlands

1 1

Case study Case study

Descriptive Management

Kunishige et al., 200851

Japan

1

Case Study

Descriptive

Sng et al., 200852

Singapore

9

Case Series

Incidence, Diagnosis, Management

Blanco Ramos et al., 200953 Huang, Shen, and Tseng, 201054

Spain Taiwan

1 1

Case Study Case Study

Descriptive Descriptive

M. Chen, Ling, and Yang, 201055

China

8

Case Series

Swan et al., 201056 A. Chen et al., 201157

Australia China

1 2

Case Study Case Series

Ihama et al., 201158

Japan

1

Case Study

Descriptive

Sung et al., 20113

Korea

4

Large Retrospective Study

Risk Stratification

Li, Manetta, and Iqbal, 201259 Peng et al., 201260

United States China

1 89

Case Study Large Retrospective Study

Management Management

Ko et al., 201361

Taiwan

1

Case Study

Management Negative laryngoscopy

Descriptive Management

Negative initial/repeat flexible esophagoscopy, found on CT

Diagnosis

Descriptive Descriptive No imaging initially

Negative rigid esophagoscopy (n 5 3)

Descriptive

Management

Management Descriptive

Negative rigid esophagoscopy, found on CT

Descriptive

CT 5computed tomography; N 5 number of cases in each study; XR 5 X-ray.

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TABLE II. Common Clinical Presentations of Esophageal Perforation Due to a Foreign Body. Presenting Signs/Symptoms

Number

% (N 5 162)

Odynophagia/neck pain

134

82.7%

Dysphagia Polysialia

119 91

73.5% 56.2%

Other Pain

28

17.3%

Retrosternal Epigastric

20 2

12.3% 1.2%

Other/unspecified

complications were mentioned in 48 cases. The most commonly reported complications were: mediastinitis (17 cases) and arterioesophageal fistula and subsequent hemorrhage (12 cases). Others included pneumomediastinum, sepsis, and pneumothorax. Although not included in our tabulations, in the largest study found, mortality was < 1% (1/121 cases), and frequent complications included cervical abscess (79 cases), mediastinal abscess (26 cases), and mediastinitis (15 cases).

6

3.7%

DISCUSSION

Hematemesis Dyspnea

8 5

4.9% 3.1%

Pathogenesis

Melena

2

1.2%

N 5 number of patients in which presenting symptoms were reported (162).

18/18 patients (100% sensitivity)—either showing a foreign body (n 5 10), signs of the esophageal perforation, or related complications. The method used for definitive diagnosis and/or management was reported in 55 cases. Esophagoscopy (rigid and flexible) (40/55, 73%) was most commonly utilized, followed by open surgery (13/55, 24%) and autopsy (2/55, 4%). A missed diagnosis was defined as a patient who, after the preliminary workup, was deemed not to have a perforation; and nine out of 168 cases met these criteria for a missed diagnosis. Six diagnoses were reported as missed on initial esophagoscopy; four were caught on a subsequent esophagoscopy and two were found on follow-up CT. To our knowledge, our case is the first reported in which the foreign body was not visualized or retrieved during initial surgery after radiographic diagnosis of a perforation caused by a foreign body. Additionally, no study specifically looked at the utility of second-look radiography after esophagoscopy and/or surgery. Treatment modality was reported in 74 cases. Treatment consisted of endoscopic removal in 53% (39/ 74), open surgery in 42% (31/74). The remaining 5% (4/74) of patients received no intervention (neither removal nor repair), usually because they were not deemed candidates for surgery. Endoscopic removal with conservative treatment was most common in patients with uncomplicated perforation, whereas open surgery was largely reserved for those with complications such as cervical abscess or difficult-to-retrieve foreign bodies. Mortality rates for the three treatment groups (endoscopic removal, surgery, none) were 5%, 6%, and 50%, respectively. Complications were common and were a focus of many of the case reports, suggesting that complication and mortality rates may be subject to reporting bias. Reliable follow-up and mortality data were available in 78 cases. The overall mortality rate among these studies was 10% (8/78 cases). Excluding cervical abscess (found to be present in most patients with a delayed diagnosis), Laryngoscope 00: Month 2014

Following a compromise in the mucosa of the esophagus, leakage of luminal digestive contents quickly leads to an inflammatory process in the mediastinum; and contamination from the flora of the upper GI and alimentary tract can quickly lead to infection, sepsis, and death if not recognized early. Lodged esophageal foreign bodies can quickly impale or slowly erode through the mucosa, muscle, and serosa, possibly aided by esophageal contraction and inflammation. The longer the object remains, the greater the level of pressure necrosis and the higher the likelihood of perforation—thus the importance of timely diagnosis and treatment. A recent large retrospective review reported mortality of esophageal perforation (all types) as 18%.4 Although iatrogenic injuries were the most common etiology, foreign body ingestions were the cause in 12% of cases.4 Trauma and spontaneous rupture are the other most common etiologies. Esophageal perforations due to foreign body are most likely to occur in the cervical esophagus,5 whereas spontaneous or iatrogenic injuries are more likely to occur distally. Eighty percent of all cervical esophageal perforations are due to foreign bodies, which tend to impact or lodge in areas of natural anatomic narrowing such as the cricopharyngeus, aortic notch, and gastroesophageal junction. In one study, 76% of cervical esophageal perforations occurred at the level of the cricopharyngeus,6 which is also vulnerable due to the lack of muscle covering Killian’s triangle posteriorly. Cervical perforations have been associated with lower mortality than thoracic perforations, likely due to their greater accessibility for intervention and the anatomic planes in the neck that may limit the spread of infection and separate it from the mediastinal and pleural cavities.7 In addition to inflammatory and infectious complications, foreign bodies can elicit immune reactions of their own or migrate into nearby structures.

Presentation Esophageal perforation is uncommon following an ingested foreign body, and signs and symptoms are nonspecific and variable; thus, a high degree of clinical suspicion and an aggressive diagnostic approach are important for timely diagnosis and management. Patients with a history of ingestion presenting with any degree of neck (cervical esophagus) or chest (thoracic) pain, dysphagia, odynophagia, difficulty breathing, Aronberg et al.: Esophageal Perforation Due to Foreign Bodies

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vomiting, drooling, or globus pharyngeus should have diagnostic evaluation. A delay in the occurrence of symptoms even up to several days should not discourage evaluation because some objects may quickly impale, whereas others may slowly erode the esophageal wall. In the current study, patients most commonly present for medical attention more than 24 hours after ingestion. Although symptoms may be limited to mild or moderate pain, some may be in significant distress, with tachycardia, tachypnea, and/or fever. Examination of a stable patient includes a thorough HEENT exam, examining the oral cavity carefully for any mucosal damage or objects, palpating the neck for subcutaneous emphysema (present in up to 80% of cases in one study), and auscultating for Hamman’s crunch (usually in thoracic esophageal injuries)—while also performing a thorough exam of heart, lungs, and abdomen.

Diagnosis In appropriate patients, diagnostic studies should not be delayed. Fiberoptic laryngoscopy or upright anteroposterior and lateral chest and neck radiography, due to their rapid availability and noninvasive nature, can be performed and are occasionally diagnostic. X-rays may reveal cervical or mediastinal emphysema, pleural effusion, or the presence of a radiopaque object. However, X-rays may often be falsely negative due to varying radio-densities of fish/animal bones8 and the size and location of the object. Sensitivity of X-ray detection of ingested fish bones ranges from 25% to 57%9 and is especially low in more proximal impactions. Contrast esophagogram may show extravasation extraluminally, confirming the existence of a perforation, whereas a filling defect may reveal larger impacted foreign bodies. Although a mainstay in the diagnosis of abdominal and thoracic perforations (where it detects 90% of tears), it is not as useful in cervical perforations because false negatives rates are as high as 60%.4,10 Barium studies may not be the best study for initial foreign body detection because contrast dye may interfere with subsequent surgical or radiographic visualization of the foreign object. Additionally, the use of Barium contrast should be restricted when perforation is suspected because extravasation is associated with soft tissue inflammation.11 If esophagography is used, a water soluble contrast, considered more delicate on tissues than barium contrast, should be used first. Barium esophagogram, although a more sensitive test for esophageal perforation than water-soluble examinations, should be performed only after a negative water-soluble exam and when there are no indications of perforation that would require surgery.12 CT scans are widely available, and they effectively detect even early soft tissue emphysema, fluid collections, abscesses, and locating the foreign object itself.13 CT has been reported to detect esophageal bone impactions with 90% to 100% sensitivity, and with a positive predictive value as high as 99%.14 Esophagoscopy is the gold standard for diagnosis and is the most common procedure for removal of ingested objects; however, CT Laryngoscope 00: Month 2014

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imaging provides useful information about the presence, nature, and location of ingested objects, as well as signs of esophageal injury. Given the high sensitivity and low false negative rates, CT imaging of stable patients with nonspecific physical signs can add to diagnostic accuracy and assist with decisions to either proceed with or avoid operative procedures. Interestingly, in the case presented, CT demonstrated the impacted fish bone, whereas subsequent rigid esophagoscopy and open surgery failed to identify the foreign body. It is not clear from the literature how commonly this occurs because this had not been previously described. Rigid esophagoscopy or flexible endoscopy is currently considered the gold standard for confirming and visualizing the foreign body and/or perforation. Although false negatives have been rarely known to occur,15,16 it is difficult to determine the true incidence. One study described two of 96 fish bones being missed on endoscopy, attributed to location (one each in hypopharynx and oropharynx) and local tissue swelling.17 In the case presented, it was a combination of the location and orientation of the foreign body, soft tissue inflammation, and the presence of excessive purulent and luminal contents that hampered identification of the fish bone. Follow-up radiography has not been well studied in the context of foreign body removal, but it can be considered an option, along with repeat endoscopy when there is a question of persistent foreign body.

Treatment Treatment of a perforation can be either nonoperative (conservative) or operative, depending on the clinical situation and severity. Nonoperative measures include broad spectrum antibiotics, nothing by mouth status, nasogastric tube placement, proton pump inhibitors, and percutaneous drainage. Repeat CT imaging can help to track the status of the perforation and/or complications. In the proper candidates, nonoperative approaches are emerging as the preferred treatment.18 Likewise, esophageal stents recently have also been used with some success, but is more challenging in the proximal esophagus where many foreign bodies lodge.19,20 The type and extent of operative treatment depends on the severity and location of the perforation, but for cervical perforation, primary closure of the defect is preferred. In cases where primary closure is not possible, drainage may be a temporizing measure before repairing with local, regional, or free flaps to allow closure without stricture. When the perforation is caused by a foreign body, immediate removal of the foreign object is a necessity. In cases such as the one presented, if the foreign body is not found at the time of endoscopy/surgery, re-evaluation should be considered and may include repeat endoscopy and repeat imaging by CT. If the foreign body was observed on the previous scan and is not detected on repeat imaging, further unnecessary intervention can be avoided. If the foreign body is identified on the repeat scan, anatomic location and orientation of the foreign body will help with intraoperative localization. Aronberg et al.: Esophageal Perforation Due to Foreign Bodies

Limitations There were several limitations in this study. First, given that most studies were case reports, publication bias is probable. Case reports are more often reported and published when they represent rare or interesting clinical scenarios. Thus, it is not appropriate to generalize the outcomes or characteristics to the wider population due to the inherent biased sample. Next, our results are dependent on the quality of the literature search. Completeness of the search was maximized by having two reviewers perform this task independently; however, some articles were not able to be retrieved in full text or English form, and thus were not included in our study. The review was also limited because many of the reports did not contain a comprehensive set of data, depending on the specific aims of each study. One study (Peng et al.60) described perforations following ingestion of both edible (70%) and nonedible (30%) objects, but did not delineate between the two groups when reporting aggregate data. Additionally, despite the large sample size, this study was not useful for analyses that required case-by-case data.

CONCLUSION Foreign body ingestion is common but results in esophageal perforation in only approximately 1% to 2% of the cases. Perforations are most commonly located in the cervical esophagus; and presentations vary widely, often with vague and nonspecific complaints. Whereas flexible laryngoscopy or X-rays may be helpful in some cases, CT imaging is an effective means to diagnose and guide management. Management can be conservative or surgical depending on the clinical situation, although the foreign body must be removed as soon as possible. When the foreign body is not able to be visualized during initial endoscopy, our experience shows that repeat imaging is helpful to determine if the object persists and to guide endoscopic localization of impacted foreign bodies. The majority of the literature consists of case reports describing ingested bones causing perforation or migrating into the mediastinum, aorta, lungs, heart, and other nearby structures.21–23 Few studies examine the effectiveness of diagnostic modalities and the frequency and risk factors for missed diagnoses. The rarity of the diagnosis makes it difficult to perform such a study at an individual institution, particularly in North America. Further investigation is necessary to examine the factors associated with difficulty identifying ingested objects in the setting of esophageal perforation.

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Aronberg et al.: Esophageal Perforation Due to Foreign Bodies

Esophageal perforation caused by edible foreign bodies: a systematic review of the literature.

Presentation of a case of esophageal perforation caused by ingestion of a foreign body that was difficult to visualize endoscopically, and systematic ...
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