Clin J Gastroenterol (2012) 5:146–149 DOI 10.1007/s12328-012-0283-8
CASE REPORT
Esophageal foreign body causing sustained stridor in an infant Eitaro Hiejima • Hiroshi Nakase • Shinji Uemoto Toshio Heike
•
Received: 28 September 2011 / Accepted: 19 December 2011 / Published online: 11 February 2012 Ó Springer 2012
Abstract The patient was an 8-month-old boy with a 2-month history of stridor. Initially, he had been diagnosed with bronchitis and/or asthma and treated with antibiotics, a b-antagonist and a leukotriene antagonist, but his symptoms were not relieved. An enhanced computed tomography scan showed a gas filled mass and tracheal compression above the level of the aortic arch, and barium swallow revealed an approximately 3 cm mass with irregular surface. Endoscopic examination showed a yellow foreign body (FB) with surrounding granulation tissue. The FB was removed surgically. Postoperative bronchoscopy showed improvement of tracheal compression. The patient had an uneventful recovery with disappearance of his stridor. Keywords
Esophagus Foreign bodies Stridor Infant
Introduction Though foreign body (FB) ingestion is common in infants and young children, chronic stridor caused by esophageal E. Hiejima T. Heike Department of Pediatrics, Graduate School of Medicine, Kyoto University, Kyoto, Japan H. Nakase (&) Department of Gastroenterology and Hepatology, Graduate School of Medicine, Kyoto University, 54 Shogoin Kawahara-cho, Sakyo-ku, Kyoto 606-8507, Japan e-mail: [email protected] S. Uemoto Department of Hepato-Biliary-Pancreatic and Transplant Surgery, Graduate School of Medicine, Kyoto University, Kyoto, Japan
123
FB is rare, and the frequency is unknown. Here, we describe a case of esophageal FB causing sustained stridor in an infant. This case warrants gastroenterologists that esophageal FB should be taken into consideration as a differential diagnosis in infantile cases with sustained stridor.
Case report An 8-month-old boy was admitted with a 2-month history of stridor. He was born at 41 weeks gestation with uncomplicated pregnancy and delivery. At the age of 6 months, he presented with postprandial vomiting and stridor. His stridor gradually increased in loudness until 8 months of age, and got worse on crying, but was not influenced by body position or feeding. There was no episode of feed refusal nor swallowing difficulties. Initially, he had been diagnosed with bronchitis and/or asthma and was treated with antibiotics, a b-agonist and a leukotriene antagonist by medical practitioners, but the symptoms were not relieved. His father had a past history of asthma in childhood. On physical examination, he was well looking with normal growth. He was afebrile with a normal heart rate, respiratory rate, and blood pressure. His oxygen saturation was 97% on room air. Biphasic stridor was slightly auscultated and was unaffected by position. Laboratory studies showed elevated white blood count of 15.8 9 109/l with eosinophilia of 11.0% and IgE 91 IU/ml (normal range for age \10 IU/ml), suggesting his allergic condition. C-reactive protein was within the normal range. Radiograph of the chest and the neck did not show any abnormities, such as tracheal deviation, although chest X-ray was taken twice. Laryngoscopy also revealed no obvious abnormality. An enhanced computed tomography
Clin J Gastroenterol (2012) 5:146–149
(CT) scan demonstrated tracheal compression and gas filled mass above the level of the aortic arch (Fig. 1). Magnetic resonance imaging (MRI) showed a mass in the wall of the esophagus, but no evidence of FB. Barium swallow revealed an approximately 3 cm mass with irregular surface above the level of the aortic arch (Fig. 2). The barium was injected through a nasogastric tube placed in the proximal esophagus without sedation. Endoscopic examination showed a yellow FB with surrounding granulation tissue (Fig. 3). We managed to remove the FB with an endoscopic grasping forceps and tip of hood, but couldn’t remove it because the FB was firmly attached to
147
esophageal wall. Also, bleeding from esophageal wall was observed during these endoscopic procedures. Therefore, we gave up retrieving it endoscopically. The pediatric surgeon then proceeded with a right thoracotomy. Adhesion between esophagus and trachea, indicating chronic
Fig. 1 An enhanced computed tomography (CT) scan demonstrated a gas filled mass and tracheal compression above the level of the aortic arch
Fig. 3 Endoscopic examination showed a yellow foreign body (FB) with surrounding granulation tissue
Fig. 2 Barium swallow revealed the approximately 3 cm mass with irregular surface (arrow) above the level of aortic arch
Fig. 4 The FB was packing tape which was a folded multiple layer and had two sharp tips like a crab claw
123
148
Clin J Gastroenterol (2012) 5:146–149
inflammation in the upper mediastinum, was observed. After removal of the adhesion the surgeon pushed the FB toward the esophageal lumen from the adventitial side. FB was successfully detached from the esophageal wall and was removed endoscopically without esophagotomy. The FB was packing tape with a folded multiple layer (Fig. 4). It had two sharp tips like a crab claw and was stuck in the esophagus, which caused ulcers (Fig. 5). The patient had an uneventful recovery with disappearance of his stridor and could eat diets normally. Endoscopic examination at 2 months after removing FB revealed the healing of ulcers without esophageal stricture.
Fig. 5 The FB which was stuck in the esophagus caused ulcers
Discussion Complications from prolonged retention of a FB can be very serious and occasionally lethal. They include bronchopulmonary fistula, tracheoesophageal fistula, mediastinitis, esophageal diverticulum, granulomas, and aortoesophageal fistulas [1]. The incidence of complications is related to the duration of FB impaction, especially in cases presenting more than 24 h after the onset of symptoms [2]. Prolonged retention may evoke inflammatory changes resulting in those complications [1]. In our case, an esophageal FB unrecognized for 2 months might evoke esophageal ulcers and adhesion to esophageal wall, for which endoscopic removal was difficult and surgical assist was required. In this case, reasons on delayed diagnosis were considered as follows: firstly, sustained stridor as a patient’s chief complaint possibly made the diagnosis difficult. Common causes of stridor or wheeze in infants are viral infection and asthma, while esophageal FB is rare. Generally, FB ingestion is common in children, especially between the ages of 6 months and 3 years [3]. The common symptoms are gastrointestinal manifestations such as vomiting, poor feeding, and dysphagia; however, respiratory manifestations such as stridor are uncommon. Our patient presented not only stridor but also postprandial vomiting with solid foods; however, vomiting was not observed when the patient took liquid diets. Pediatric cases of esophageal FB causing stridor have been rarely reported in the literatures which were published in English over the past 20 years [4–12] (Table 1). Secondly, nobody noticed
Table 1 A review of pediatric cases of esophageal foreign body causing stridor from MEDLINE articles published in English over the past 20 years Author
Age (months old)
Durations of symptoms
Foreign body
Diagnosis
Treatment
References [4]
Hamilton JM
19
Sudden onset
Button battery
Xp
Endoscopic removal
Mohiuddin S
11
A few weeks
Coin
Xp
Endoscopic removal
[5]
Mohiuddin S
10
1 month
Coin
Xp
Endoscopic removal
[5]
Mohiuddin S
10
1 month
No data
Xp
Endoscopic removal
[5]
Takahashi T
18
2 months
Button battery
Xp
Endoscopic removal
[6] [7]
3 weeks
Decorative button
CT
Operation
Parray T
Kim N
36
1
Several months
Plastic penny
Endoscopy
Operation
[8]
Warner BW
15
3 months
Plastic object
Endoscopy
Operation
[9]
Warner BW
11
No data
No data
Endoscopy
Operation
[9]
Gilchrist BF
36
2 months
Clothespin spring
Xp and barium study
Operation
[10]
Gilchrist BF Gilchrist BF
30 42
3 months 3 months
Coin Heart pendant
Xp and barium study Xp and barium study
Operation Operation
[10] [10]
Gilchrist BF
24
6 months
Coin
Xp and barium study
Operation
[10]
Gilchrist BF
42
24 months
Toy soldier
Xp and barium study
Operation
[10]
Sherrington CA Haegen TW
123
7
5 months
Plastic disc
CT
Operation
[11]
17
6 months
Pistachio shell
MRI
Operation
[12]
Clin J Gastroenterol (2012) 5:146–149
his ingestion of FB. In a retrospective analysis of 255 pediatric patients with esophageal FB ingestion, 84% of cases were observed events [13]. Thus, most cases of esophageal FB are witnessed soon after ingestion of it and are extracted. Therefore, in a not witnessed case, it would be difficult to diagnose ingestion of FB as a cause of stridor. Thirdly, the FB in our case was radiolucent and was not identified definitely even with barium esophagram and MRI. The mass detected in MRI and barium esophagram seemed to be a pathological change of the esophageal wall, and was not suggestive of FB. In the majority of pediatric patients with ingestion of FB, the plain chest X-ray confirms the diagnosis, because most ingested FBs are radiopaque objects such as metallic coins and batteries. In a study by Singh et al. [2] of 205 pediatric patients with ingested FB, 91.7% of patients showed abnormal findings on radiographic examination. In cases of radiolucent FB, barium esophagram or MRI might be helpful for detection. However, in a case report of two young children with esophageal FB, MRI was not useful for detecting the FB which was surrounded by inflammatory tissues as was our case [9]. Therefore, based on our experience, we recommend the following protocol of workup for stridor in infant. Unexplained stridor after chest and neck X-ray, and laryngoscopy should be evaluated with enhanced CT and MRI. If the etiology remains unclear, bronchoscopy and esophagoscopy should be performed under general anesthesia. In conclusion, when we encounter an infant who complains of sustained stridor, FB should be considered as one of the differential diagnoses. Delay in diagnosis can lead to prolonged impaction of esophagus and sometimes lethal complication. Conflict of interest of interest.
149
References 1. Newman DE. The radiolucent esophageal foreign body: an oftenforgotten cause of respiratory symptoms. J Pediatr. 1978;92:60–3. 2. Singh B, Kantu M, Har-El G, Lucente FE. Complications associated with 327 foreign bodies of the pharynx, larynx and esophagus. Ann Otol Rhinol Laryngol. 1997;106:301–4. 3. Cheng W, Tam PK. Foreign-body ingestion in children: experience with 1,265 cases. J Pediatr Surg. 1999;34:1472–6. 4. Hamilton JM, Schraff SA, Notrica DM. Severe injuries from coin cell battery ingestions: 2 case reports. J Pediatr Surg. 2009;44: 644–7. 5. Mohiuddin S, Siddiqui MS, Mayhew JF. Esophageal foreign body aspiration presenting as asthma in the pediatric patient. South Med J. 2004;97:93–5. 6. Takahashi T, Teramoto Y, Aoyama T, Sakakibara M, Hara M, Maseki M, et al. Anesthetic management of a child with an esophageal foreign body which was misdiagnosed as asthma attack. Masui. 2009;5:199–201. 7. Kim N, Atkinson N, Manicone P. Esophageal foreign body: a case of a neonate with stridor. Pediatr Emerg Care. 2008;24: 849–51. 8. Parray T, Shah S, Apuya JS, Shah S. Atypical presentation of an impacted radiolucent esophageal foreign body. J Anesth. 2010; 24:793–6. 9. Warner BW, Plecha FM, Torres AM, Myer CM 3rd, Ryckman FC, Garcia VF. Chronic respiratory distress caused by radiolucent esophageal foreign body. Am J Otolaryngol. 1992;13:181–4. 10. 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. 11. Sherrington CA, Crameri JA, Coleman LT, Sawyer SM. Stridor in an infant. Eur Respir J. 1999;14:717–9. 12. Haegen TW, Wojtczak HA, Tomita SS. Chronic inspiratory stridor secondary to a retained penetrating radiolucent esophageal foreign body. J Pediatr Surg. 2003;38:1–3. 13. Louie JP, Alpern ER, Windreich RM. Witnessed and unwitnessed esophageal foreign bodies in children. Pediatr Emerg Care. 2005; 21:582–5.
The authors declare that they have no conflict
123
CASE REPORT
Esophageal foreign body causing sustained stridor in an infant Eitaro Hiejima • Hiroshi Nakase • Shinji Uemoto Toshio Heike
•
Received: 28 September 2011 / Accepted: 19 December 2011 / Published online: 11 February 2012 Ó Springer 2012
Abstract The patient was an 8-month-old boy with a 2-month history of stridor. Initially, he had been diagnosed with bronchitis and/or asthma and treated with antibiotics, a b-antagonist and a leukotriene antagonist, but his symptoms were not relieved. An enhanced computed tomography scan showed a gas filled mass and tracheal compression above the level of the aortic arch, and barium swallow revealed an approximately 3 cm mass with irregular surface. Endoscopic examination showed a yellow foreign body (FB) with surrounding granulation tissue. The FB was removed surgically. Postoperative bronchoscopy showed improvement of tracheal compression. The patient had an uneventful recovery with disappearance of his stridor. Keywords
Esophagus Foreign bodies Stridor Infant
Introduction Though foreign body (FB) ingestion is common in infants and young children, chronic stridor caused by esophageal E. Hiejima T. Heike Department of Pediatrics, Graduate School of Medicine, Kyoto University, Kyoto, Japan H. Nakase (&) Department of Gastroenterology and Hepatology, Graduate School of Medicine, Kyoto University, 54 Shogoin Kawahara-cho, Sakyo-ku, Kyoto 606-8507, Japan e-mail: [email protected] S. Uemoto Department of Hepato-Biliary-Pancreatic and Transplant Surgery, Graduate School of Medicine, Kyoto University, Kyoto, Japan
123
FB is rare, and the frequency is unknown. Here, we describe a case of esophageal FB causing sustained stridor in an infant. This case warrants gastroenterologists that esophageal FB should be taken into consideration as a differential diagnosis in infantile cases with sustained stridor.
Case report An 8-month-old boy was admitted with a 2-month history of stridor. He was born at 41 weeks gestation with uncomplicated pregnancy and delivery. At the age of 6 months, he presented with postprandial vomiting and stridor. His stridor gradually increased in loudness until 8 months of age, and got worse on crying, but was not influenced by body position or feeding. There was no episode of feed refusal nor swallowing difficulties. Initially, he had been diagnosed with bronchitis and/or asthma and was treated with antibiotics, a b-agonist and a leukotriene antagonist by medical practitioners, but the symptoms were not relieved. His father had a past history of asthma in childhood. On physical examination, he was well looking with normal growth. He was afebrile with a normal heart rate, respiratory rate, and blood pressure. His oxygen saturation was 97% on room air. Biphasic stridor was slightly auscultated and was unaffected by position. Laboratory studies showed elevated white blood count of 15.8 9 109/l with eosinophilia of 11.0% and IgE 91 IU/ml (normal range for age \10 IU/ml), suggesting his allergic condition. C-reactive protein was within the normal range. Radiograph of the chest and the neck did not show any abnormities, such as tracheal deviation, although chest X-ray was taken twice. Laryngoscopy also revealed no obvious abnormality. An enhanced computed tomography
Clin J Gastroenterol (2012) 5:146–149
(CT) scan demonstrated tracheal compression and gas filled mass above the level of the aortic arch (Fig. 1). Magnetic resonance imaging (MRI) showed a mass in the wall of the esophagus, but no evidence of FB. Barium swallow revealed an approximately 3 cm mass with irregular surface above the level of the aortic arch (Fig. 2). The barium was injected through a nasogastric tube placed in the proximal esophagus without sedation. Endoscopic examination showed a yellow FB with surrounding granulation tissue (Fig. 3). We managed to remove the FB with an endoscopic grasping forceps and tip of hood, but couldn’t remove it because the FB was firmly attached to
147
esophageal wall. Also, bleeding from esophageal wall was observed during these endoscopic procedures. Therefore, we gave up retrieving it endoscopically. The pediatric surgeon then proceeded with a right thoracotomy. Adhesion between esophagus and trachea, indicating chronic
Fig. 1 An enhanced computed tomography (CT) scan demonstrated a gas filled mass and tracheal compression above the level of the aortic arch
Fig. 3 Endoscopic examination showed a yellow foreign body (FB) with surrounding granulation tissue
Fig. 2 Barium swallow revealed the approximately 3 cm mass with irregular surface (arrow) above the level of aortic arch
Fig. 4 The FB was packing tape which was a folded multiple layer and had two sharp tips like a crab claw
123
148
Clin J Gastroenterol (2012) 5:146–149
inflammation in the upper mediastinum, was observed. After removal of the adhesion the surgeon pushed the FB toward the esophageal lumen from the adventitial side. FB was successfully detached from the esophageal wall and was removed endoscopically without esophagotomy. The FB was packing tape with a folded multiple layer (Fig. 4). It had two sharp tips like a crab claw and was stuck in the esophagus, which caused ulcers (Fig. 5). The patient had an uneventful recovery with disappearance of his stridor and could eat diets normally. Endoscopic examination at 2 months after removing FB revealed the healing of ulcers without esophageal stricture.
Fig. 5 The FB which was stuck in the esophagus caused ulcers
Discussion Complications from prolonged retention of a FB can be very serious and occasionally lethal. They include bronchopulmonary fistula, tracheoesophageal fistula, mediastinitis, esophageal diverticulum, granulomas, and aortoesophageal fistulas [1]. The incidence of complications is related to the duration of FB impaction, especially in cases presenting more than 24 h after the onset of symptoms [2]. Prolonged retention may evoke inflammatory changes resulting in those complications [1]. In our case, an esophageal FB unrecognized for 2 months might evoke esophageal ulcers and adhesion to esophageal wall, for which endoscopic removal was difficult and surgical assist was required. In this case, reasons on delayed diagnosis were considered as follows: firstly, sustained stridor as a patient’s chief complaint possibly made the diagnosis difficult. Common causes of stridor or wheeze in infants are viral infection and asthma, while esophageal FB is rare. Generally, FB ingestion is common in children, especially between the ages of 6 months and 3 years [3]. The common symptoms are gastrointestinal manifestations such as vomiting, poor feeding, and dysphagia; however, respiratory manifestations such as stridor are uncommon. Our patient presented not only stridor but also postprandial vomiting with solid foods; however, vomiting was not observed when the patient took liquid diets. Pediatric cases of esophageal FB causing stridor have been rarely reported in the literatures which were published in English over the past 20 years [4–12] (Table 1). Secondly, nobody noticed
Table 1 A review of pediatric cases of esophageal foreign body causing stridor from MEDLINE articles published in English over the past 20 years Author
Age (months old)
Durations of symptoms
Foreign body
Diagnosis
Treatment
References [4]
Hamilton JM
19
Sudden onset
Button battery
Xp
Endoscopic removal
Mohiuddin S
11
A few weeks
Coin
Xp
Endoscopic removal
[5]
Mohiuddin S
10
1 month
Coin
Xp
Endoscopic removal
[5]
Mohiuddin S
10
1 month
No data
Xp
Endoscopic removal
[5]
Takahashi T
18
2 months
Button battery
Xp
Endoscopic removal
[6] [7]
3 weeks
Decorative button
CT
Operation
Parray T
Kim N
36
1
Several months
Plastic penny
Endoscopy
Operation
[8]
Warner BW
15
3 months
Plastic object
Endoscopy
Operation
[9]
Warner BW
11
No data
No data
Endoscopy
Operation
[9]
Gilchrist BF
36
2 months
Clothespin spring
Xp and barium study
Operation
[10]
Gilchrist BF Gilchrist BF
30 42
3 months 3 months
Coin Heart pendant
Xp and barium study Xp and barium study
Operation Operation
[10] [10]
Gilchrist BF
24
6 months
Coin
Xp and barium study
Operation
[10]
Gilchrist BF
42
24 months
Toy soldier
Xp and barium study
Operation
[10]
Sherrington CA Haegen TW
123
7
5 months
Plastic disc
CT
Operation
[11]
17
6 months
Pistachio shell
MRI
Operation
[12]
Clin J Gastroenterol (2012) 5:146–149
his ingestion of FB. In a retrospective analysis of 255 pediatric patients with esophageal FB ingestion, 84% of cases were observed events [13]. Thus, most cases of esophageal FB are witnessed soon after ingestion of it and are extracted. Therefore, in a not witnessed case, it would be difficult to diagnose ingestion of FB as a cause of stridor. Thirdly, the FB in our case was radiolucent and was not identified definitely even with barium esophagram and MRI. The mass detected in MRI and barium esophagram seemed to be a pathological change of the esophageal wall, and was not suggestive of FB. In the majority of pediatric patients with ingestion of FB, the plain chest X-ray confirms the diagnosis, because most ingested FBs are radiopaque objects such as metallic coins and batteries. In a study by Singh et al. [2] of 205 pediatric patients with ingested FB, 91.7% of patients showed abnormal findings on radiographic examination. In cases of radiolucent FB, barium esophagram or MRI might be helpful for detection. However, in a case report of two young children with esophageal FB, MRI was not useful for detecting the FB which was surrounded by inflammatory tissues as was our case [9]. Therefore, based on our experience, we recommend the following protocol of workup for stridor in infant. Unexplained stridor after chest and neck X-ray, and laryngoscopy should be evaluated with enhanced CT and MRI. If the etiology remains unclear, bronchoscopy and esophagoscopy should be performed under general anesthesia. In conclusion, when we encounter an infant who complains of sustained stridor, FB should be considered as one of the differential diagnoses. Delay in diagnosis can lead to prolonged impaction of esophagus and sometimes lethal complication. Conflict of interest of interest.
149
References 1. Newman DE. The radiolucent esophageal foreign body: an oftenforgotten cause of respiratory symptoms. J Pediatr. 1978;92:60–3. 2. Singh B, Kantu M, Har-El G, Lucente FE. Complications associated with 327 foreign bodies of the pharynx, larynx and esophagus. Ann Otol Rhinol Laryngol. 1997;106:301–4. 3. Cheng W, Tam PK. Foreign-body ingestion in children: experience with 1,265 cases. J Pediatr Surg. 1999;34:1472–6. 4. Hamilton JM, Schraff SA, Notrica DM. Severe injuries from coin cell battery ingestions: 2 case reports. J Pediatr Surg. 2009;44: 644–7. 5. Mohiuddin S, Siddiqui MS, Mayhew JF. Esophageal foreign body aspiration presenting as asthma in the pediatric patient. South Med J. 2004;97:93–5. 6. Takahashi T, Teramoto Y, Aoyama T, Sakakibara M, Hara M, Maseki M, et al. Anesthetic management of a child with an esophageal foreign body which was misdiagnosed as asthma attack. Masui. 2009;5:199–201. 7. Kim N, Atkinson N, Manicone P. Esophageal foreign body: a case of a neonate with stridor. Pediatr Emerg Care. 2008;24: 849–51. 8. Parray T, Shah S, Apuya JS, Shah S. Atypical presentation of an impacted radiolucent esophageal foreign body. J Anesth. 2010; 24:793–6. 9. Warner BW, Plecha FM, Torres AM, Myer CM 3rd, Ryckman FC, Garcia VF. Chronic respiratory distress caused by radiolucent esophageal foreign body. Am J Otolaryngol. 1992;13:181–4. 10. 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. 11. Sherrington CA, Crameri JA, Coleman LT, Sawyer SM. Stridor in an infant. Eur Respir J. 1999;14:717–9. 12. Haegen TW, Wojtczak HA, Tomita SS. Chronic inspiratory stridor secondary to a retained penetrating radiolucent esophageal foreign body. J Pediatr Surg. 2003;38:1–3. 13. Louie JP, Alpern ER, Windreich RM. Witnessed and unwitnessed esophageal foreign bodies in children. Pediatr Emerg Care. 2005; 21:582–5.
The authors declare that they have no conflict
123
