Tracheo-oesophageal fistula: a delayed complication of missed inhaled magnetic toys

Tracheo-oesophageal fistula: a delayed complication of missed inhaled magnetic toys Delayed diagnosis of tracheobronchial foreign body (FB) aspiration is not uncommon in children. It occurs when symptoms are underappreciated and/or radiological findings are overlooked. In such cases serious complications can arise, which make the diagnosis and removal of the FB much more difficult. Here, we present a case where FB aspiration was misdiagnosed as asthma after a radio-opaque FB on the chest radiograph was missed, leading to formation of a tracheo-oesophageal fistula as a rare delayed complication. x-ray (CXR) (Fig. 1). It was reported that the left lung was translu- INTRODUCTION cent with reduced peripheral lung markings: finding consistent Tracheobronchial foreign body (FB) aspiration is a leading domes- with asthma with bronchial plugging or a post infective bronchio- tic accident in children, under the age of 4 [1]. Delayed diagnosis litis. It also showed a round radio-opaque FB over the left main or misdiagnosis can occur if children present with no history of bronchus. However, this was labelled as ‘Clothing Artefact’ on the aspiration or with atypical history of non-specific symptoms. actual radiograph and therefore did not alarm the GP or the This is associated with increased morbidity, such as recurrent reporting radiologist. A repeated PA CXR (Fig. 2)was undertaken pulmonary infection, atelectasis and bronchiectasis [2]. in the asthma clinic where all clothes and hair were removed Here, we present a rare case of tracheo-oesophageal fistula from the thorax. This confirmed thepresenceofametallic FB in as a delayed complication of aspiration of two small magnetic the left main bronchus and the patient was immediately admit- balls in a 3-year old girl. Her initial presentation was misdiag- ted for bronchoscopy and removal of the FB. nosed as asthma after the radio-opaque FB in her first chest A ventilating bronchoscopy was performed by the otolaryn- radiograph was mislabelled as a clothing artefact. gology team where the FB was found to be stuck to the poster- ior wall of the left main bronchus. The FB was left in-situ. After the operation, a lateral CXR (Fig. 3) illustrated the metallic FB CASE REPORT appeared to be two spherical balls, with the anterior one in the A 3-year-old girl was referred to the asthma clinic with a 3-month left main bronchus and the posterior one in the oesophagus. A subsequent further history from the parents suggested these history of morning cough and wheeze. She was seen multiple times by her general practitioner (GP) who initially diagnosed her were from the older sibling’s toys. Patient underwent another rigid bronchoscopy under the with upper respiratory tract infection. Her symptoms persisted despite salbutamol inhaler treatment so the GP organised a chest cardiothoracic team, which confirmed a tracheo-oesophageal Received: December 29, 2017. Accepted: January 27, 2018 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2018. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/ licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy022/4877014 by Ed 'DeepDyve' Gillespie user on 16 March 2018 2 W. Yang et al. fistula that had formed likely due to the slow migration of the A history of initial choking and coughing episode with subsequent metallic FB. The team proceeded to convert the operation to respiratory symptoms such as stridor and wheeze are immedi- right thoracotomy where two pieces of stuck-together magnetic ately suggestive of the diagnosis. However, symptoms can subside balls were found and removed. The tracheo-oesophageal fistula spontaneously and quickly even when a FB remains [2]. Danger was repaired with a lateral costal flap. Water soluble contrast occurs especially when children fail to present with any his- swallow post-operation showed no evidence of any residual fis- tory of aspiration. Instead, they may display symptoms and tula nor any contrast leak. The patient made a full recovery signs of unresolved or recurrent pneumonia as a consequence and was discharged 16 days after her initial admission. of chronic inflammation and tissue reactions caused by a long- standing FB [2]. Therefore, it is not uncommon for patients to be treated for other disorders, such as persistent fever, asthma or DISCUSSION recurrent pneumonia for a long period of time before the diagno- Children of young age have a significant risk of FB aspiration as sis of FB aspiration is established. In our case, aspiration of the FB they tend to explore and place objects in their mouths. It fre- by the patient and the possible subsequent choking and/or quently occurs before the fourth year of life, mostly likely as a coughing episode were not witnessed. She was conservatively result of inadequate dentition, the relatively high position of treated with inhaler for a presumed upper respiratory tract infec- the epiglottis and the immature coordination of swallowing [1]. tion for 3 months by her GP before the diagnosis became appar- FB aspiration is considered a life-threatening emergency that ent on a repeated CXR. requires prompt removal, but sometimes it may remain Chest radiography as the first line investigation for sus- undetected because of atypical history or misleading clinical pected FB aspiration can prove the existence of radio-opaque and radiological findings. FB and evokes the likelihood of FB aspiratory if it demonstrates A convincing history and plain chest radiograph are the two hyperinflation, obstructive emphysema, atelectasis and/or con- main criteria for the diagnosis of FB aspiration [2]. Although FB solidation [2]. However, these signs are inconsistent. In a series aspiration may cause sudden airway obstruction and subse- of 189 children with proven FB aspiration, 90 cases (48%) had quent death, quite often symptoms are mild and non-specific. normal chest radiographs [3]. What is striking in our case is that, in addition to the finding of hyperinflation of the left lung, the initial CXR demonstrated a round radio-opaque FB over the left main bronchus. Unfortunately, this was overlooked by both the GP and the reporting radiologist due to the incorrect label- ling of ‘Clothing Artefact’ on the radiograph. As a result of delayed diagnosis, a tracheo-oesophageal fistula occurred for which the patient required open thoracotomy. In conclusion, clinicians should always maintain a high index of suspicion for FB aspiration when patients of this age group Figure 1: Initial posterior–anterior chest x-ray (CXR) demonstrated a translucent left lung with reduced peripheral lung markings. There is also a dense rounded foreign body projected over the left main bronchus. A label of ‘CLOTHING ARTEFACT’ was placed in the left upper corner of this CXR. Figure 2: The repeated posterior–anterior CXR taken with all clothes and hair Figure 3: Lateral CXR showed the metallic foreign body to be two spherical removed from the thorax of the patient. It confirmed the presence of a metallic balls, with the anterior one in the left main bronchus and the posterior one in foreign body in the left main bronchus. the oesophagus. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy022/4877014 by Ed 'DeepDyve' Gillespie user on 16 March 2018 Tracheo-oesophageal fistula: a delayed complication 3 present with acute or chronic respiratory symptoms and signs. REFERENCES Although labels such as ‘artefacts’ on radiographs are often useful 1. Heyer CM, Bollmeier ME, Rossler L, Nuesslein TG, Stephan V, to aid the interpretation of the image, suspicious artefacts should Bauer TT, et al. Evaluation of clinical, radiologic, and labora- be explored and clarified early to prevent any obvious pathologies tory prebronchoscopy findings in children with suspected being missed during the diagnostic process. foreign body aspiration. J Pediatr Surg 2006;41:1882–8. Toys with magnetic parts have become popular among the 2. Sersar SI, Rizk WH, Bilal M, El Diasty MM, Eltantawy TA, paediatric population, and they are accessible to children Abdelhakam BB, et al. Inhaled foreign bodies: presentation, because parents are not aware of their potential risks. The management and value of history and plain chest radiog- alarm has been raised previously about the risk of ingestion of raphy in delayed presentation. Otolaryngol Head Neck Surg magnetic toys causing intestinal obstruction and perforation 2006;134:92–9. [4]. We emphasize again the need for manufactures to highlight 3. Hilliard T, Sim R, Saunders M, Hewer S, Henderson J. Delayed the hazards of magnetic toys on package to prevent the appear- diagnosis of foreign body aspiration in children. Emerg Med J ance of new cases. 2003;20:100–1. 4. Hernandez Anselmi E, Gutierrez San Roman C, Barrios Fontoba JE, Ayuso Gonzalez L, Valdes Diequez E, Lluna CONFLICT OF INTEREST STATEMENT Gonzalez J, et al. Intestinal perforation caused by magnetic toys. J Pediatr Surg 2007;42:E13–6. None declared. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy022/4877014 by Ed 'DeepDyve' Gillespie user on 16 March 2018 http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Journal of Surgical Case Reports Oxford University Press

Tracheo-oesophageal fistula: a delayed complication of missed inhaled magnetic toys

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Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2018.
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Abstract

Delayed diagnosis of tracheobronchial foreign body (FB) aspiration is not uncommon in children. It occurs when symptoms are underappreciated and/or radiological findings are overlooked. In such cases serious complications can arise, which make the diagnosis and removal of the FB much more difficult. Here, we present a case where FB aspiration was misdiagnosed as asthma after a radio-opaque FB on the chest radiograph was missed, leading to formation of a tracheo-oesophageal fistula as a rare delayed complication. x-ray (CXR) (Fig. 1). It was reported that the left lung was translu- INTRODUCTION cent with reduced peripheral lung markings: finding consistent Tracheobronchial foreign body (FB) aspiration is a leading domes- with asthma with bronchial plugging or a post infective bronchio- tic accident in children, under the age of 4 [1]. Delayed diagnosis litis. It also showed a round radio-opaque FB over the left main or misdiagnosis can occur if children present with no history of bronchus. However, this was labelled as ‘Clothing Artefact’ on the aspiration or with atypical history of non-specific symptoms. actual radiograph and therefore did not alarm the GP or the This is associated with increased morbidity, such as recurrent reporting radiologist. A repeated PA CXR (Fig. 2)was undertaken pulmonary infection, atelectasis and bronchiectasis [2]. in the asthma clinic where all clothes and hair were removed Here, we present a rare case of tracheo-oesophageal fistula from the thorax. This confirmed thepresenceofametallic FB in as a delayed complication of aspiration of two small magnetic the left main bronchus and the patient was immediately admit- balls in a 3-year old girl. Her initial presentation was misdiag- ted for bronchoscopy and removal of the FB. nosed as asthma after the radio-opaque FB in her first chest A ventilating bronchoscopy was performed by the otolaryn- radiograph was mislabelled as a clothing artefact. gology team where the FB was found to be stuck to the poster- ior wall of the left main bronchus. The FB was left in-situ. After the operation, a lateral CXR (Fig. 3) illustrated the metallic FB CASE REPORT appeared to be two spherical balls, with the anterior one in the A 3-year-old girl was referred to the asthma clinic with a 3-month left main bronchus and the posterior one in the oesophagus. A subsequent further history from the parents suggested these history of morning cough and wheeze. She was seen multiple times by her general practitioner (GP) who initially diagnosed her were from the older sibling’s toys. Patient underwent another rigid bronchoscopy under the with upper respiratory tract infection. Her symptoms persisted despite salbutamol inhaler treatment so the GP organised a chest cardiothoracic team, which confirmed a tracheo-oesophageal Received: December 29, 2017. Accepted: January 27, 2018 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2018. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/ licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy022/4877014 by Ed 'DeepDyve' Gillespie user on 16 March 2018 2 W. Yang et al. fistula that had formed likely due to the slow migration of the A history of initial choking and coughing episode with subsequent metallic FB. The team proceeded to convert the operation to respiratory symptoms such as stridor and wheeze are immedi- right thoracotomy where two pieces of stuck-together magnetic ately suggestive of the diagnosis. However, symptoms can subside balls were found and removed. The tracheo-oesophageal fistula spontaneously and quickly even when a FB remains [2]. Danger was repaired with a lateral costal flap. Water soluble contrast occurs especially when children fail to present with any his- swallow post-operation showed no evidence of any residual fis- tory of aspiration. Instead, they may display symptoms and tula nor any contrast leak. The patient made a full recovery signs of unresolved or recurrent pneumonia as a consequence and was discharged 16 days after her initial admission. of chronic inflammation and tissue reactions caused by a long- standing FB [2]. Therefore, it is not uncommon for patients to be treated for other disorders, such as persistent fever, asthma or DISCUSSION recurrent pneumonia for a long period of time before the diagno- Children of young age have a significant risk of FB aspiration as sis of FB aspiration is established. In our case, aspiration of the FB they tend to explore and place objects in their mouths. It fre- by the patient and the possible subsequent choking and/or quently occurs before the fourth year of life, mostly likely as a coughing episode were not witnessed. She was conservatively result of inadequate dentition, the relatively high position of treated with inhaler for a presumed upper respiratory tract infec- the epiglottis and the immature coordination of swallowing [1]. tion for 3 months by her GP before the diagnosis became appar- FB aspiration is considered a life-threatening emergency that ent on a repeated CXR. requires prompt removal, but sometimes it may remain Chest radiography as the first line investigation for sus- undetected because of atypical history or misleading clinical pected FB aspiration can prove the existence of radio-opaque and radiological findings. FB and evokes the likelihood of FB aspiratory if it demonstrates A convincing history and plain chest radiograph are the two hyperinflation, obstructive emphysema, atelectasis and/or con- main criteria for the diagnosis of FB aspiration [2]. Although FB solidation [2]. However, these signs are inconsistent. In a series aspiration may cause sudden airway obstruction and subse- of 189 children with proven FB aspiration, 90 cases (48%) had quent death, quite often symptoms are mild and non-specific. normal chest radiographs [3]. What is striking in our case is that, in addition to the finding of hyperinflation of the left lung, the initial CXR demonstrated a round radio-opaque FB over the left main bronchus. Unfortunately, this was overlooked by both the GP and the reporting radiologist due to the incorrect label- ling of ‘Clothing Artefact’ on the radiograph. As a result of delayed diagnosis, a tracheo-oesophageal fistula occurred for which the patient required open thoracotomy. In conclusion, clinicians should always maintain a high index of suspicion for FB aspiration when patients of this age group Figure 1: Initial posterior–anterior chest x-ray (CXR) demonstrated a translucent left lung with reduced peripheral lung markings. There is also a dense rounded foreign body projected over the left main bronchus. A label of ‘CLOTHING ARTEFACT’ was placed in the left upper corner of this CXR. Figure 2: The repeated posterior–anterior CXR taken with all clothes and hair Figure 3: Lateral CXR showed the metallic foreign body to be two spherical removed from the thorax of the patient. It confirmed the presence of a metallic balls, with the anterior one in the left main bronchus and the posterior one in foreign body in the left main bronchus. the oesophagus. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy022/4877014 by Ed 'DeepDyve' Gillespie user on 16 March 2018 Tracheo-oesophageal fistula: a delayed complication 3 present with acute or chronic respiratory symptoms and signs. REFERENCES Although labels such as ‘artefacts’ on radiographs are often useful 1. Heyer CM, Bollmeier ME, Rossler L, Nuesslein TG, Stephan V, to aid the interpretation of the image, suspicious artefacts should Bauer TT, et al. Evaluation of clinical, radiologic, and labora- be explored and clarified early to prevent any obvious pathologies tory prebronchoscopy findings in children with suspected being missed during the diagnostic process. foreign body aspiration. J Pediatr Surg 2006;41:1882–8. Toys with magnetic parts have become popular among the 2. Sersar SI, Rizk WH, Bilal M, El Diasty MM, Eltantawy TA, paediatric population, and they are accessible to children Abdelhakam BB, et al. Inhaled foreign bodies: presentation, because parents are not aware of their potential risks. The management and value of history and plain chest radiog- alarm has been raised previously about the risk of ingestion of raphy in delayed presentation. Otolaryngol Head Neck Surg magnetic toys causing intestinal obstruction and perforation 2006;134:92–9. [4]. We emphasize again the need for manufactures to highlight 3. Hilliard T, Sim R, Saunders M, Hewer S, Henderson J. Delayed the hazards of magnetic toys on package to prevent the appear- diagnosis of foreign body aspiration in children. Emerg Med J ance of new cases. 2003;20:100–1. 4. Hernandez Anselmi E, Gutierrez San Roman C, Barrios Fontoba JE, Ayuso Gonzalez L, Valdes Diequez E, Lluna CONFLICT OF INTEREST STATEMENT Gonzalez J, et al. Intestinal perforation caused by magnetic toys. J Pediatr Surg 2007;42:E13–6. None declared. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy022/4877014 by Ed 'DeepDyve' Gillespie user on 16 March 2018

Journal

Journal of Surgical Case ReportsOxford University Press

Published: Feb 1, 2018

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