DOI: 10.1177/0003489414521145 Annals of Otology, Rhinology & Laryngology 123(1):19-24. © The Author(s) 2014
Chronic Cervical Esophageal Foreign Bodies in Children: Surgical Approach After Unsuccessful Endoscopic Management Jordan C. Schramm, MD, MS; Ryan K. Sewell, MD, JD; Kenneth S. Azarow, MD; Stephen C. Raynor, MD; Shahab F. Abdessalam, MD Objectives: We reviewed the surgical management of chronic cervical esophageal foreign bodies (CCEFBs) in a pediatric population after failed endoscopic retrieval. Methods: A descriptive analysis via a retrospective chart review of patients with CCEFBs who failed initial endoscopic management was performed between 2008 and 2013. Details were recorded regarding presenting symptoms, time from symptom onset to diagnosis of the CCEFB, surgical approach, and complications. Results: Three patients with CCEFBs unsuccessfully managed with endoscopy were identified. The range of ages at diagnosis was 14 months to 4.5 years. The foreign bodies (FBs) were present for at least 1 month before diagnosis (range, 1 to 10 months). Respiratory symptoms were predominant in all cases. Neck exploration with removal of the FB was performed in each case. Complications included esophageal stricture necessitating serial dilations (patient 1), left true vocal fold paresis that resolved spontaneously (patient 3), and tracheoesophageal fistula with successful endoscopic closure (patient 3). No long-term sequelae were experienced. Conclusions: A high index of suspicion is required to recognize CCEFBs in children with respiratory distress. Although endoscopic management remains the first-line treatment, it may fail or may not be possible because of transmural FB migration. In this setting, neck exploration with FB removal is a safe and effective alternative. Key Words: chronic illness, foreign body, infant, ingestion, injury, neck, preschool child.
abnormalities, such as esophageal strictures or prior surgery (eg, fundoplication, tracheoesophageal fistula repair), younger age, unwitnessed ingestions, and mental disability.1,3
INTRODUCTION
Pediatric foreign body (FB) ingestion is a relatively common occurrence, with 125,000 instances per year reported to poison control centers in the United States. There is an equal distribution between male patients and female patients. The peak incidence is between 6 months and 3 years of age.1 Coins are the most commonly ingested FB, and have been reported in 4% of all children.2 However, smooth objects, such as coins, are less likely to become “chronic,” because they are more apt to pass spontaneously. In contrast, irregularly shaped objects or objects with sharp edges are more likely to become lodged within the wall of the esophagus and become chronic esophageal FBs. Despite the common occurrence of FB ingestion, chronic esophageal FBs are relatively rare, accounting for 5% to 8% of all esophageal FBs in pediatric patients. The predisposing factors for an FB ingestion to become chronic include underlying anatomic
Endoscopic retrieval of both acute and chronic esophageal FBs is preferred and is usually successful. However, when the FB is lodged in the wall of the esophagus, an open surgical approach may be necessary. Transcervical3-6 and transthoracic3,5-13 approaches have been employed, depending on the location of the FB. We present a case series of chronic cervical esophageal foreign bodies (CCEFBs) in children who underwent attempted endoscopic FB removal but ultimately required open transcervical FB removal. METHODS
After approval was obtained from our Joint Pediatric Institutional Review Board (Children’s Hospital & Medical Center and the University of Ne-
From the Department of Otolaryngology–Head and Neck Surgery (Schramm, Sewell) and the Division of Pediatric Surgery, Department of Surgery (Azarow, Raynor, Abdessalam), College of Medicine, University of Nebraska Medical Center; the Ear, Nose, and Throat Clinic (Schramm, Sewell) and Children’s Specialty Physicians (Azarow, Raynor, Abdessalam), Children’s Hospital & Medical Center; and ENT Specialists, PC (Sewell); Omaha, Nebraska. Presented as a poster at the meeting of the American Broncho-Esophagological Association, Orlando, Florida, April 10-11, 2013. Correspondence: Jordan C. Schramm, MD, MS, Dept of Otolaryngology–Head and Neck Surgery, 981225 Nebraska Medical Center, Omaha, NE 68198-1225.
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A
C Fig 1. (Patient 1) A) Axial and B) sagittal computed tomograms of chest with contrast demonstrate retained radiolucent esophageal foreign body (arrowheads). C) Endoscopic and D) intraoperative views of foreign body. E) Specimen after removal.
D
B
E
braska Medical Center), a retrospective review of the electronic medical record at Children’s Hospital & Medical Center was carried out. Patients with CCEFBs who required neck exploration for FB removal between 2008 and 2013 were identified. The patients’ inpatient and outpatient records were reviewed, and their demographics and medical history were recorded. Details were compiled regarding their clinical course, including presenting symptoms, duration of symptoms prior to diagnosis of the CCEFB, and complications. RESULTS
6 months before the CCEFB diagnosis. Endoscopic management was first attempted in each patient before they underwent surgical removal of the FB. Each FB was irregularly shaped, lending itself to lodging within the wall of the esophagus and transmural migration. One patient’s recovery was complicated by esophageal stricture that resolved after 6 months of serial dilations. Another patient experienced left true vocal fold paresis with spontaneous resolution, and tracheoesophageal fistula that was successfully closed endoscopically. None of the patients experienced long-term sequelae.
Three cases of CCEFBs in pediatric patients were identified. Their ages at diagnosis ranged from 14 to 56 months. All patients presented primarily with respiratory symptoms. Two patients also had choking and aspiration of food. The times from symptom onset to FB removal ranged from 1 to 10 months. In 2 patients, the FB was radiolucent on computed tomography (CT), but was identified subsequently on endoscopy. The FB was radiopaque on CT in the third patient. The radiopaque FB was retrospectively identified on a lateral neck radiograph obtained
There were only minor variations in surgical technique for each case. Neck exploration was performed by making an incision along the sternocleidomastoid muscle and dissecting to the tracheoesophageal groove, with care taken to identify and preserve the recurrent laryngeal nerve. Depending on the surgeon’s preference, either a Jackson-Pratt or a Penrose surgical drain was placed in each case. Two of the FBs spanned the area from the esophageal lumen to the superior mediastinum, and required esophagotomy for removal. For these cases,
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Fig 2. (Patient 2) A) Axial and B) coronal computed tomograms of chest with contrast demonstrate retained radiolucent esophageal foreign body (arrowheads). C) Endoscopic view of foreign body. D) Specimen after removal.
Schramm et al, Chronic Cervical Esophageal Foreign Bodies 21
A
C
B
D
the patient was kept on nil per os (NPO) status until 1 week after the operation, at which time an esophagram was obtained. Because the esophageal mucosa did not appear to be violated after esophagomyotomy in the third case, an esophagram was obtained earlier, on postoperative day (POD) 1. Further details of the cases are included below. Patient 1. A girl 4 years 8 months of age presented with a 1-month history of wheezing, stridor, and cough. Her past medical history was pertinent for hypoplastic left heart syndrome, which had been surgically treated in two stages within the first 4 months of life, with the sequelae of left true vocal fold paralysis and left hemidiaphragm paralysis. Her past surgical history was also significant for gastrostomy tube placement, fundoplication, and left hemidiaphragm plication at 6 months of age. A CT scan of the chest with contrast showed moderate tracheal narrowing at the level of the thoracic inlet secondary to an esophageal abnormality (Fig 1A,B). An FB partially lodged within the esophageal wall was identified on upper gastrointestinal endoscopy (Fig 1C), and multiple attempts at removal were unsuccessful. The patient was noted to have a large amount of granulation tissue and swelling, which prevented the scope from passing beyond the FB. Right neck exploration and esophagotomy was performed with removal of the FB (Fig 1D), which
appeared to be the “zipper” tab from a plastic bag (Fig 1E). The patient was kept intubated overnight in the pediatric intensive care unit. Ampicillin and clindamycin were given by intravenous administration during the operation. Intravenous antimicrobial treatment was then given for 1 week in the form of ampicillin-sulbactam. After successful extubation, the patient was transferred to a regular pediatric hospital bed on POD 1. She was kept on NPO status until an esophagram confirmed no leak on POD 7. Her diet was advanced, and there was no evidence of leakage of formula in the right-neck JacksonPratt drain. The drain was removed on POD 9, and she was discharged to home on POD 10. She experienced postoperative esophageal stricture. She underwent 3 esophageal dilations over the subsequent 6 months before the stricture was resolved. The dilations were all performed under general anesthesia and in the same manner: the area of esophageal stricture was visualized with a pediatric endoscope, balloon dilation was performed up to 15 mm in diameter at 8 atmospheres of pressure for 2 minutes, and no further area of stricture was seen at the end of each dilation. The patient experienced no longterm sequelae. Patient 2. A 14-month-old, otherwise healthy boy presented initially at 4 months of age with noisy breathing and aspiration of thin liquids. Over the
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A
C Fig 3. (Patient 3) A) Axial and B) coronal computed tomograms of chest with contrast demonstrate retained radiopaque esophageal foreign body (arrowheads). C) Intraoperative endoscopy was used to identify location of transmurally migrated foreign body by placing 25-gauge needles in surgical field through esophagus to mark its location. D) Intraoperative view of foreign body. E) Specimen after removal.
D
B
E
subsequent 10 months, he received diagnoses of and treatment for respiratory syncytial virus infection, asthma, recurrent upper respiratory tract infections, and gastroesophageal reflux. He underwent microdirect laryngoscopy and rigid bronchoscopy that showed a type 1 laryngeal cleft and extrinsic compression of the middle trachea. A CT scan of the chest with contrast was performed during the same anesthesia and demonstrated a thickened esophageal wall without contrast enhancement and dextroanterior deviation of the trachea (Fig 2A,B). Initial diagnostic considerations included a foregut duplication cyst and an infiltrating esophageal neoplasm. Flexible esophagoscopy revealed an irregularly shaped FB eroding the left esophageal wall (Fig 2C). Left-sided neck exploration and esophagotomy was performed under the same anesthesia with the flexible scope still in place to help localize the FB. A chewed plastic half-cylinder, possibly from a pen, was removed from the esophageal wall (Fig 2D). A Penrose neck drain and a nasogastric (NG) tube were placed. The patient was kept intubated and se-
dated in the pediatric intensive care unit. Intravenous administration of ampicillin-sulbactam was initiated before the operation, intravenous administration of clindamycin was initiated after the operation, and both regimens were continued until discharge. On POD 5, repeat microdirect laryngoscopy and bronchoscopy showed significant improvement of tracheal compression, and the patient was extubated. On POD 7, he was transferred to a regular pediatric hospital bed. An esophagram confirmed no leak on POD 8, the NG tube and Penrose drain were removed, and the patient’s diet was advanced. He was discharged to home on POD 10, and experienced no subsequent complications. Patient 3. A 16-month-old girl with a history of failure to thrive presented with a 9-month history of stridor and coughing and choking on milk. She was born at 32 weeks’ gestation, was small for her gestational age, and had been intubated for the first week of life. A deglutition study at 14 months of age showed penetration but no aspiration. Microdirect laryngoscopy and bronchoscopy showed extrinsic
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Schramm et al, Chronic Cervical Esophageal Foreign Bodies 23
status. She was discharged to home on POD 3 on NG tube feeding. The NG tube was removed on POD 7, after a deglutition study showed no evidence of a TEF. However, the patient continued to have dysphagia. She was noted to have left true vocal fold paresis on POD 10, which resolved spontaneously by 3 months after surgery. She was ultimately found to have persistence of a small TEF at 4 months after surgery. Endoscopic closure was attempted by cannulating the fistulous tract from the tracheal side, applying electrocautery with a flexible 3F Bugbee cautery electrode, and applying fibrin sealant with a 4F pigtail ureteral catheter. A retained Prolene suture was also identified and removed from the tract during this process. The TEF persisted, and repeat endoscopic closure was attempted 2 weeks later in a similar manner, but with cautery applied from both the tracheal and the esophageal sides of the fistula. There was no evidence of a persistent TEF on a repeat esophagram 2 weeks later. Fig 4. (Patient 3) Lateral neck radiograph obtained 6 months before diagnosis of chronic cervical esophageal foreign body shows radiopaque object (arrowhead) that had been initially mistaken for artifact.
tracheal compression and a possible tracheoesophageal fistula (TEF). However, esophagogastroduod enoscopy under the same anesthesia did not show any abnormalities. A CT scan of the chest with contrast was performed, and a radiopaque FB was identified within the anterior esophageal wall and superior mediastinum (Fig 3A,B). On retrospective review of a lateral neck radiograph taken 6 months before presentation, the FB was appreciated (Fig 4). Left neck exploration was performed with intraoperative flexible esophagoscopy, 25-gauge needles were placed transmurally to localize the FB within the wall of the esophagus (Fig 3C), and an FB was confirmed by intraoperative ultrasonography. It was found by endoscopy to correlate with an area of remodeled esophageal mucosa. Dissection was carried through the inflammatory mass, esophagomy otomy was performed to reveal the FB (Fig 3D), and a plastic flower-shaped button was retrieved (Fig 3E). Repeat intraoperative esophagoscopy showed no obvious mucosal violation. A Jackson-Pratt drain was placed. The patient was admitted to a regular pediatric hospital bed after the operation. Intravenous administration of ampicillin-sulbactam, initiated before the operation, was continued through her hospitalization. This regimen was converted to oral intake of amoxicillin–clavulanic acid at discharge for a total of 10 days of antibiotic coverage. An NG tube was required to facilitate obtaining an esophagram on POD 1. This showed a TEF, so the NG tube was kept in place and the patient was kept on NPO
DISCUSSION
Endoscopic removal of esophageal FBs is still preferred and is usually successful even in the setting of a chronic FB. Our literature search on PubMed, limited to English-language articles, revealed 23 specifically addressing chronic esophageal FBs in pediatric patients,3-25 for a total of 85 patients identified. We reviewed the mode of retrieval and complications in these cases plus the 3 presented herein, for a total of 88 patients. The FB was successfully removed endoscopically in 62 patients (70.5%).3,9,10,13,15-25 All but 1 of the remaining patients underwent open surgical removal of the FB; 17 (19.3%) underwent thoracotomy3,5-13,18 and 9 (10.2%) underwent a transcervical approach.3-6,18 The FB was identified postmortem in 1 patient (1.1%) who died of sudden infant death syndrome.14 Although complications were rare in this select group of patients, they were often serious, including esophageal perforation in 8 patients (9.1%), mediastinitis in 8 (9.1%), TEF in 6 (6.8%), esophageal stricture in 3 (3.4%), aortoesophageal fistula in 3 (3.4%), true vocal fold paralysis in 2 (2.3%), death in 2 (2.3%), and postobstructive pulmonary edema in 1 (1.1%). Up to 75% of pediatric patients with chronic esophageal FBs present primarily with respiratory complaints such as cough, stridor, wheezing, diffuse ronchi, or elevated respiratory rate,3 and this finding is consistent with our series. It makes the diagnosis of a CCEFB elusive, as esophageal disease is not necessarily at the forefront of the clinician’s mind with such a presentation. Hence, an initial misdiagnosis of asthma, reflux, viral upper
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respiratory tract infection, or pharyngitis is not uncommon. Radiolucent FBs are particularly difficult to diagnose, and thus are especially likely to become chronic.4,8,10,13,19 These may appear similar to foregut duplication cysts on CT, because the lack of signal by the radiolucent FB makes it appear similar to air,8,13 as in our second case. Foreign bodies can be missed or dismissed as an artifact on imaging,13 particularly when they are flat, as in our third case. Hence, a high index of suspicion and clinical correlation of the patient’s symptoms with radiographic findings is necessary for accurate diagnosis. When present, gastrointestinal symptoms, either in isolation or in conjunction with respiratory symptoms, often lead to the correct diagnosis of an FB.
These may include dysphagia, nausea, emesis, and decreased oral intake. When infection ensues, intermittent fever may develop. Failure to thrive is also common as a result of decreased oral intake and/or increased work of breathing. CONCLUSIONS
A CCEFB should be included in the differential diagnosis for respiratory distress in children. A high index of suspicion is required to recognize this problem. Although endoscopic management remains first-line and is usually successful, some patients may require open surgical removal. Neck exploration with removal of the FB is safe and effective when endoscopic management fails or is not possible because of transmural FB migration.
REFERENCES 1. Hesham A-Kader H. Foreign body ingestion: children like to put objects in their mouth. World J Pediatr 2010;6:301-10. 2. Conners GP, Chamberlain JM, Weiner PR. Pediatric coin ingestion: a home-based survey. Am J Emerg Med 1995;13:63840. 3. Miller RS, Willging JP, Rutter MJ, Rookkapan K. Chronic esophageal foreign bodies in pediatric patients: a retrospective review. Int J Pediatr Otorhinolaryngol 2004;68:265-72. 4. Katz KR, Emmens RW, Wood BP. Radiological case of the month. Esophageal obstruction and abscess formation secondary to impacted, eroding tiddlywink. Am J Dis Child 1989; 143:961-2. 5. Shepherd RL, Raffensperger JG, Goldstein R. Pediatric esophageal perforation. J Thorac Cardiovasc Surg 1977;74:2617. 6. Tucker JG, Kim HH, Lucas GW. Esophageal perforation caused by coin ingestion. South Med J 1994;87:269-72. 7. Gilchrist BF, Valerie EP, Nguyen M, Coren C, Klotz D, Ramenofsky ML. Pearls and perils in the management of prolonged, peculiar, penetrating esophageal foreign bodies in children. J Pediatr Surg 1997;32:1429-31. 8. Haegen TW, Wojtczak HA, Tomita SS. Chronic inspiratory stridor secondary to a retained penetrating radiolucent esophageal foreign body. J Pediatr Surg 2003;38:e6. 9. Naidoo RR, Reddi AA. Chronic retained foreign bodies in the esophagus. Ann Thorac Surg 2004;77:2218-20. 10. Newman DE. The radiolucent esophageal foreign body: an often-forgotten cause of respiratory symptoms. J Pediatr 1978;92:60-3. 11. Persaud RA, Sudhakaran N, Ong CC, Bowdler DA, Dykes E. Extraluminal migration of a coin in the oesophagus of a child misdiagnosed as asthma. Emerg Med J 2001;18:312-3. 12. Stuth EA, Stucke AG, Cohen RD, Jaquiss RD, Kugathasan S, Litwin SB. Successful resuscitation of a child after exsanguination due to aortoesophageal fistula from undiagnosed foreign body. Anesthesiology 2001;95:1025-6. 13. Warner BW, Plecha FM, Torres AM, Myer CM III, Ryckman FC, Garcia VF. Chronic respiratory distress caused by
radiolucent esophageal foreign body. Am J Otolaryngol 1992;13: 181-4. 14. Byard RW, Moore L, Bourne AJ. Sudden and unexpected death — a late effect of occult intraesophageal foreign body. Pediatr Pathol 1990;10:837-41. 15. Cole S, Kearns D, Magit A. Chronic esophageal foreign bodies and secondary mediastinitis in children. Ann Otol Rhinol Laryngol 2011;120:542-5. 16. Doolin EJ. Esophageal stricture: an uncommon complication of foreign bodies. Ann Otol Rhinol Laryngol 1993;102:863-6. 17. Heiss NM, Baker KG, Martin FA, Bredfeldt RC. Esophageal foreign body: a case presentation. J Fam Pract 1995;41:48991. 18. Kerschner JE, Beste DJ, Conley SF, Kenna MA, Lee D. Mediastinitis associated with foreign body erosion of the esophagus in children. Int J Pediatr Otorhinolaryngol 2001;59: 89-97. 19. Kim N, Atkinson N, Manicone P. Esophageal foreign body: a case of a neonate with stridor. Pediatr Emerg Care 2008; 24:849-51. 20. Mohiuddin S, Siddiqui MS, Mayhew JF. Esophageal foreign body aspiration presenting as asthma in the pediatric patient. South Med J 2004;97:93-5. 21. Narasimhappa GM, Baranwal AK, Singhi SC. Impacted esophageal foreign body mistaken for chronic pharyngitis. Indian J Pediatr 2009;76:862. 22. Rao CC, McNiece WL, Krishna G. Acute pulmonary edema after removal of an esophageal foreign body in an infant. Crit Care Med 1986;14:988-9. 23. Sapru A, bin Seif Ali Elbualy M, Nayyar PM. Esophageal foreign body causing recurrent respiratory symptoms. Gastrointest Endosc 1998;48:218-9. 24. Savitt DL, Wason S. Delayed diagnosis of coin ingestion in children. Am J Emerg Med 1988;6:378-81. 25. Tauscher JW. Esophageal foreign body: an uncommon cause of stridor. Pediatrics 1978;61:657-8.
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Chronic Cervical Esophageal Foreign Bodies in Children: Surgical Approach After Unsuccessful Endoscopic Management Jordan C. Schramm, MD, MS; Ryan K. Sewell, MD, JD; Kenneth S. Azarow, MD; Stephen C. Raynor, MD; Shahab F. Abdessalam, MD Objectives: We reviewed the surgical management of chronic cervical esophageal foreign bodies (CCEFBs) in a pediatric population after failed endoscopic retrieval. Methods: A descriptive analysis via a retrospective chart review of patients with CCEFBs who failed initial endoscopic management was performed between 2008 and 2013. Details were recorded regarding presenting symptoms, time from symptom onset to diagnosis of the CCEFB, surgical approach, and complications. Results: Three patients with CCEFBs unsuccessfully managed with endoscopy were identified. The range of ages at diagnosis was 14 months to 4.5 years. The foreign bodies (FBs) were present for at least 1 month before diagnosis (range, 1 to 10 months). Respiratory symptoms were predominant in all cases. Neck exploration with removal of the FB was performed in each case. Complications included esophageal stricture necessitating serial dilations (patient 1), left true vocal fold paresis that resolved spontaneously (patient 3), and tracheoesophageal fistula with successful endoscopic closure (patient 3). No long-term sequelae were experienced. Conclusions: A high index of suspicion is required to recognize CCEFBs in children with respiratory distress. Although endoscopic management remains the first-line treatment, it may fail or may not be possible because of transmural FB migration. In this setting, neck exploration with FB removal is a safe and effective alternative. Key Words: chronic illness, foreign body, infant, ingestion, injury, neck, preschool child.
abnormalities, such as esophageal strictures or prior surgery (eg, fundoplication, tracheoesophageal fistula repair), younger age, unwitnessed ingestions, and mental disability.1,3
INTRODUCTION
Pediatric foreign body (FB) ingestion is a relatively common occurrence, with 125,000 instances per year reported to poison control centers in the United States. There is an equal distribution between male patients and female patients. The peak incidence is between 6 months and 3 years of age.1 Coins are the most commonly ingested FB, and have been reported in 4% of all children.2 However, smooth objects, such as coins, are less likely to become “chronic,” because they are more apt to pass spontaneously. In contrast, irregularly shaped objects or objects with sharp edges are more likely to become lodged within the wall of the esophagus and become chronic esophageal FBs. Despite the common occurrence of FB ingestion, chronic esophageal FBs are relatively rare, accounting for 5% to 8% of all esophageal FBs in pediatric patients. The predisposing factors for an FB ingestion to become chronic include underlying anatomic
Endoscopic retrieval of both acute and chronic esophageal FBs is preferred and is usually successful. However, when the FB is lodged in the wall of the esophagus, an open surgical approach may be necessary. Transcervical3-6 and transthoracic3,5-13 approaches have been employed, depending on the location of the FB. We present a case series of chronic cervical esophageal foreign bodies (CCEFBs) in children who underwent attempted endoscopic FB removal but ultimately required open transcervical FB removal. METHODS
After approval was obtained from our Joint Pediatric Institutional Review Board (Children’s Hospital & Medical Center and the University of Ne-
From the Department of Otolaryngology–Head and Neck Surgery (Schramm, Sewell) and the Division of Pediatric Surgery, Department of Surgery (Azarow, Raynor, Abdessalam), College of Medicine, University of Nebraska Medical Center; the Ear, Nose, and Throat Clinic (Schramm, Sewell) and Children’s Specialty Physicians (Azarow, Raynor, Abdessalam), Children’s Hospital & Medical Center; and ENT Specialists, PC (Sewell); Omaha, Nebraska. Presented as a poster at the meeting of the American Broncho-Esophagological Association, Orlando, Florida, April 10-11, 2013. Correspondence: Jordan C. Schramm, MD, MS, Dept of Otolaryngology–Head and Neck Surgery, 981225 Nebraska Medical Center, Omaha, NE 68198-1225.
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A
C Fig 1. (Patient 1) A) Axial and B) sagittal computed tomograms of chest with contrast demonstrate retained radiolucent esophageal foreign body (arrowheads). C) Endoscopic and D) intraoperative views of foreign body. E) Specimen after removal.
D
B
E
braska Medical Center), a retrospective review of the electronic medical record at Children’s Hospital & Medical Center was carried out. Patients with CCEFBs who required neck exploration for FB removal between 2008 and 2013 were identified. The patients’ inpatient and outpatient records were reviewed, and their demographics and medical history were recorded. Details were compiled regarding their clinical course, including presenting symptoms, duration of symptoms prior to diagnosis of the CCEFB, and complications. RESULTS
6 months before the CCEFB diagnosis. Endoscopic management was first attempted in each patient before they underwent surgical removal of the FB. Each FB was irregularly shaped, lending itself to lodging within the wall of the esophagus and transmural migration. One patient’s recovery was complicated by esophageal stricture that resolved after 6 months of serial dilations. Another patient experienced left true vocal fold paresis with spontaneous resolution, and tracheoesophageal fistula that was successfully closed endoscopically. None of the patients experienced long-term sequelae.
Three cases of CCEFBs in pediatric patients were identified. Their ages at diagnosis ranged from 14 to 56 months. All patients presented primarily with respiratory symptoms. Two patients also had choking and aspiration of food. The times from symptom onset to FB removal ranged from 1 to 10 months. In 2 patients, the FB was radiolucent on computed tomography (CT), but was identified subsequently on endoscopy. The FB was radiopaque on CT in the third patient. The radiopaque FB was retrospectively identified on a lateral neck radiograph obtained
There were only minor variations in surgical technique for each case. Neck exploration was performed by making an incision along the sternocleidomastoid muscle and dissecting to the tracheoesophageal groove, with care taken to identify and preserve the recurrent laryngeal nerve. Depending on the surgeon’s preference, either a Jackson-Pratt or a Penrose surgical drain was placed in each case. Two of the FBs spanned the area from the esophageal lumen to the superior mediastinum, and required esophagotomy for removal. For these cases,
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Fig 2. (Patient 2) A) Axial and B) coronal computed tomograms of chest with contrast demonstrate retained radiolucent esophageal foreign body (arrowheads). C) Endoscopic view of foreign body. D) Specimen after removal.
Schramm et al, Chronic Cervical Esophageal Foreign Bodies 21
A
C
B
D
the patient was kept on nil per os (NPO) status until 1 week after the operation, at which time an esophagram was obtained. Because the esophageal mucosa did not appear to be violated after esophagomyotomy in the third case, an esophagram was obtained earlier, on postoperative day (POD) 1. Further details of the cases are included below. Patient 1. A girl 4 years 8 months of age presented with a 1-month history of wheezing, stridor, and cough. Her past medical history was pertinent for hypoplastic left heart syndrome, which had been surgically treated in two stages within the first 4 months of life, with the sequelae of left true vocal fold paralysis and left hemidiaphragm paralysis. Her past surgical history was also significant for gastrostomy tube placement, fundoplication, and left hemidiaphragm plication at 6 months of age. A CT scan of the chest with contrast showed moderate tracheal narrowing at the level of the thoracic inlet secondary to an esophageal abnormality (Fig 1A,B). An FB partially lodged within the esophageal wall was identified on upper gastrointestinal endoscopy (Fig 1C), and multiple attempts at removal were unsuccessful. The patient was noted to have a large amount of granulation tissue and swelling, which prevented the scope from passing beyond the FB. Right neck exploration and esophagotomy was performed with removal of the FB (Fig 1D), which
appeared to be the “zipper” tab from a plastic bag (Fig 1E). The patient was kept intubated overnight in the pediatric intensive care unit. Ampicillin and clindamycin were given by intravenous administration during the operation. Intravenous antimicrobial treatment was then given for 1 week in the form of ampicillin-sulbactam. After successful extubation, the patient was transferred to a regular pediatric hospital bed on POD 1. She was kept on NPO status until an esophagram confirmed no leak on POD 7. Her diet was advanced, and there was no evidence of leakage of formula in the right-neck JacksonPratt drain. The drain was removed on POD 9, and she was discharged to home on POD 10. She experienced postoperative esophageal stricture. She underwent 3 esophageal dilations over the subsequent 6 months before the stricture was resolved. The dilations were all performed under general anesthesia and in the same manner: the area of esophageal stricture was visualized with a pediatric endoscope, balloon dilation was performed up to 15 mm in diameter at 8 atmospheres of pressure for 2 minutes, and no further area of stricture was seen at the end of each dilation. The patient experienced no longterm sequelae. Patient 2. A 14-month-old, otherwise healthy boy presented initially at 4 months of age with noisy breathing and aspiration of thin liquids. Over the
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A
C Fig 3. (Patient 3) A) Axial and B) coronal computed tomograms of chest with contrast demonstrate retained radiopaque esophageal foreign body (arrowheads). C) Intraoperative endoscopy was used to identify location of transmurally migrated foreign body by placing 25-gauge needles in surgical field through esophagus to mark its location. D) Intraoperative view of foreign body. E) Specimen after removal.
D
B
E
subsequent 10 months, he received diagnoses of and treatment for respiratory syncytial virus infection, asthma, recurrent upper respiratory tract infections, and gastroesophageal reflux. He underwent microdirect laryngoscopy and rigid bronchoscopy that showed a type 1 laryngeal cleft and extrinsic compression of the middle trachea. A CT scan of the chest with contrast was performed during the same anesthesia and demonstrated a thickened esophageal wall without contrast enhancement and dextroanterior deviation of the trachea (Fig 2A,B). Initial diagnostic considerations included a foregut duplication cyst and an infiltrating esophageal neoplasm. Flexible esophagoscopy revealed an irregularly shaped FB eroding the left esophageal wall (Fig 2C). Left-sided neck exploration and esophagotomy was performed under the same anesthesia with the flexible scope still in place to help localize the FB. A chewed plastic half-cylinder, possibly from a pen, was removed from the esophageal wall (Fig 2D). A Penrose neck drain and a nasogastric (NG) tube were placed. The patient was kept intubated and se-
dated in the pediatric intensive care unit. Intravenous administration of ampicillin-sulbactam was initiated before the operation, intravenous administration of clindamycin was initiated after the operation, and both regimens were continued until discharge. On POD 5, repeat microdirect laryngoscopy and bronchoscopy showed significant improvement of tracheal compression, and the patient was extubated. On POD 7, he was transferred to a regular pediatric hospital bed. An esophagram confirmed no leak on POD 8, the NG tube and Penrose drain were removed, and the patient’s diet was advanced. He was discharged to home on POD 10, and experienced no subsequent complications. Patient 3. A 16-month-old girl with a history of failure to thrive presented with a 9-month history of stridor and coughing and choking on milk. She was born at 32 weeks’ gestation, was small for her gestational age, and had been intubated for the first week of life. A deglutition study at 14 months of age showed penetration but no aspiration. Microdirect laryngoscopy and bronchoscopy showed extrinsic
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Schramm et al, Chronic Cervical Esophageal Foreign Bodies 23
status. She was discharged to home on POD 3 on NG tube feeding. The NG tube was removed on POD 7, after a deglutition study showed no evidence of a TEF. However, the patient continued to have dysphagia. She was noted to have left true vocal fold paresis on POD 10, which resolved spontaneously by 3 months after surgery. She was ultimately found to have persistence of a small TEF at 4 months after surgery. Endoscopic closure was attempted by cannulating the fistulous tract from the tracheal side, applying electrocautery with a flexible 3F Bugbee cautery electrode, and applying fibrin sealant with a 4F pigtail ureteral catheter. A retained Prolene suture was also identified and removed from the tract during this process. The TEF persisted, and repeat endoscopic closure was attempted 2 weeks later in a similar manner, but with cautery applied from both the tracheal and the esophageal sides of the fistula. There was no evidence of a persistent TEF on a repeat esophagram 2 weeks later. Fig 4. (Patient 3) Lateral neck radiograph obtained 6 months before diagnosis of chronic cervical esophageal foreign body shows radiopaque object (arrowhead) that had been initially mistaken for artifact.
tracheal compression and a possible tracheoesophageal fistula (TEF). However, esophagogastroduod enoscopy under the same anesthesia did not show any abnormalities. A CT scan of the chest with contrast was performed, and a radiopaque FB was identified within the anterior esophageal wall and superior mediastinum (Fig 3A,B). On retrospective review of a lateral neck radiograph taken 6 months before presentation, the FB was appreciated (Fig 4). Left neck exploration was performed with intraoperative flexible esophagoscopy, 25-gauge needles were placed transmurally to localize the FB within the wall of the esophagus (Fig 3C), and an FB was confirmed by intraoperative ultrasonography. It was found by endoscopy to correlate with an area of remodeled esophageal mucosa. Dissection was carried through the inflammatory mass, esophagomy otomy was performed to reveal the FB (Fig 3D), and a plastic flower-shaped button was retrieved (Fig 3E). Repeat intraoperative esophagoscopy showed no obvious mucosal violation. A Jackson-Pratt drain was placed. The patient was admitted to a regular pediatric hospital bed after the operation. Intravenous administration of ampicillin-sulbactam, initiated before the operation, was continued through her hospitalization. This regimen was converted to oral intake of amoxicillin–clavulanic acid at discharge for a total of 10 days of antibiotic coverage. An NG tube was required to facilitate obtaining an esophagram on POD 1. This showed a TEF, so the NG tube was kept in place and the patient was kept on NPO
DISCUSSION
Endoscopic removal of esophageal FBs is still preferred and is usually successful even in the setting of a chronic FB. Our literature search on PubMed, limited to English-language articles, revealed 23 specifically addressing chronic esophageal FBs in pediatric patients,3-25 for a total of 85 patients identified. We reviewed the mode of retrieval and complications in these cases plus the 3 presented herein, for a total of 88 patients. The FB was successfully removed endoscopically in 62 patients (70.5%).3,9,10,13,15-25 All but 1 of the remaining patients underwent open surgical removal of the FB; 17 (19.3%) underwent thoracotomy3,5-13,18 and 9 (10.2%) underwent a transcervical approach.3-6,18 The FB was identified postmortem in 1 patient (1.1%) who died of sudden infant death syndrome.14 Although complications were rare in this select group of patients, they were often serious, including esophageal perforation in 8 patients (9.1%), mediastinitis in 8 (9.1%), TEF in 6 (6.8%), esophageal stricture in 3 (3.4%), aortoesophageal fistula in 3 (3.4%), true vocal fold paralysis in 2 (2.3%), death in 2 (2.3%), and postobstructive pulmonary edema in 1 (1.1%). Up to 75% of pediatric patients with chronic esophageal FBs present primarily with respiratory complaints such as cough, stridor, wheezing, diffuse ronchi, or elevated respiratory rate,3 and this finding is consistent with our series. It makes the diagnosis of a CCEFB elusive, as esophageal disease is not necessarily at the forefront of the clinician’s mind with such a presentation. Hence, an initial misdiagnosis of asthma, reflux, viral upper
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Schramm et al, Chronic Cervical Esophageal Foreign Bodies
respiratory tract infection, or pharyngitis is not uncommon. Radiolucent FBs are particularly difficult to diagnose, and thus are especially likely to become chronic.4,8,10,13,19 These may appear similar to foregut duplication cysts on CT, because the lack of signal by the radiolucent FB makes it appear similar to air,8,13 as in our second case. Foreign bodies can be missed or dismissed as an artifact on imaging,13 particularly when they are flat, as in our third case. Hence, a high index of suspicion and clinical correlation of the patient’s symptoms with radiographic findings is necessary for accurate diagnosis. When present, gastrointestinal symptoms, either in isolation or in conjunction with respiratory symptoms, often lead to the correct diagnosis of an FB.
These may include dysphagia, nausea, emesis, and decreased oral intake. When infection ensues, intermittent fever may develop. Failure to thrive is also common as a result of decreased oral intake and/or increased work of breathing. CONCLUSIONS
A CCEFB should be included in the differential diagnosis for respiratory distress in children. A high index of suspicion is required to recognize this problem. Although endoscopic management remains first-line and is usually successful, some patients may require open surgical removal. Neck exploration with removal of the FB is safe and effective when endoscopic management fails or is not possible because of transmural FB migration.
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