International Journal of Pediatric Otorhinolaryngology 79 (2015) 1140–1144
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Case Report
Plastic bronchitis arising from solitary influenza B infection: A report of two cases in children§ Stephen J. Nogan a, Nathan D. Cass c, Gregory J. Wiet a,b, James M. Ruda a,b,* a
Department of Otolaryngology-Head & Neck Surgery, Ohio State University-Wexner Medical Center, Columbus, OH 43212, USA Department of Pediatric Otolaryngology, Nationwide Children’s Hospital, Columbus, OH 43205, USA c Department of Otolaryngology, University of Colorado Anschutz Medical Campus, Aurora, CO 80045, USA b
A R T I C L E I N F O
A B S T R A C T
Article history: Received 28 January 2015 Received in revised form 22 March 2015 Accepted 28 March 2015 Available online 7 April 2015
Plastic bronchitis (PB) is characterized by thick, inspissated, tracheobronchial casts. It is classified as either inflammatory or acellular based on the content of the endobronchial casts. PB has never been reported in a healthy child with solitary influenza B infection. This study is a retrospective case series of two children who presented to our institution in acute respiratory distress. Emergency rigid bronchoscopy was performed with extraction of casts from the L mainstem bronchus in both patients. Influenza B was the only isolate identified. In otherwise healthy children with respiratory distress, influenza B-mediated inflammatory PB must be considered in the differential diagnosis. ß 2015 Elsevier Ireland Ltd. All rights reserved.
Keywords: Plastic bronchitis Children Influenza B
1. Introduction Plastic bronchitis (PB) is a rare entity characterized by the development of thick, obstructing mucoid/gelatinous casts in the tracheobronchial airway. It has been historically categorized as either cellular (type I) vs. acellular (type II) as well as inflammatory vs. non-inflammatory based on the pathologic consistency of the cast and underlying etiology [1]. It is frequently described in infants and children with congenital heart disease and following palliative cardiac surgery, i.e. (Fontan) secondary to their surgically altered cardiac anatomy and physiology that favors transudation or exudation of fluid into the airway. With solidification of these transudates and exudates, obstruction of the large and small airways occurs. This leads to the development of acute respiratory distress, hypoxia, tachycardia, tachypnea, and cough. Radiographically, PB can present as pulmonary opacification, mediastinal shift, atelectasis, and pneumonia. Treatment frequently requires bronchoscopic evacuation of the mucoid casts from the tracheobronchial tree and remedy of any underlying abnormal cardiac physiology or medical problem, when possible or applicable. PB has also been described in patients with associated inflammatory pulmonary conditions such as cystic fibrosis, acute chest syndrome
§
Presented at SENTAC, December 5–8th, 2013, Long Beach, California. * Corresponding author. Tel.: +1 614 722 6804; fax: +1 614 722 6609. E-mail addresses: [email protected] (S.J. Nogan), [email protected] (N.D. Cass), [email protected] (G.J. Wiet), [email protected] (J.M. Ruda). http://dx.doi.org/10.1016/j.ijporl.2015.03.028 0165-5876/ß 2015 Elsevier Ireland Ltd. All rights reserved.
of sickle-cell disease, allergic bronchopulmonary aspergillosis, asthma as well as in those with a personal or family history of atopy [2,3]. Influenza A has recently been identified for its association with PB, especially during the recent 2009 outbreak of the H1N1 influenza A pandemic in Asia and Mexico [4–11]. Children with influenza A infection can frequently develop acute pulmonary complications and sequelae such as pneumonia and acute respiratory failure when acutely infected. PB has been previously reported in one child who was co-infected with influenza A and B as well as in a child with underlying congenital heart disease and was also infected with influenza B [4,10]. To our knowledge, no reports of solitary influenza B infection in a healthy child who developed PB has ever been reported. We present a report of two cases of solitary influenza B-mediated inflammatory PB in otherwise healthy children with no known medical comorbidities during the winter of 2012–2013. For this study, institutional IRB approval was obtained. 2. Case 1 The first child, an 8 y/o Caucasian male, was transferred from a community hospital to our institution in acute respiratory distress in December 2012. He had no known medical history or comorbidities such as asthma or atopy but was not up-to-date on his annual influenza vaccination. He possessed a 4-day history of flu-like symptoms including fever to 38 8C, rhinorrhea, tachypnea, violent cough, and malaise/fatigue. On the morning
S.J. Nogan et al. / International Journal of Pediatric Otorhinolaryngology 79 (2015) 1140–1144
of admission, he was noted to have acute, diffuse chest/neck swelling by his family. Upon presentation to the community hospital later that day, he was found to be hypoxic, febrile to 38.1 8C, with labored breathing, and with extensive subcutaneous emphysema over his neck/chest. A CT chest scan was performed showing a small L pneumothorax and pneumomediastinum with a hyperinflated R lung, opacified L lung, but midline trachea (Images 2 and 3). He was immediately started on a high-flow oxygen non-rebreather mask as well as empiric vancomycin and ceftriaxone, and transferred to our institution. Upon arrival to our institution, the patient was persistently tachypneic, hypoxic, and febrile, but stable on high-flow nonrebreather mask oxygen. A confirmatory CXR showed persistent left-ward mediastinal shift and L lung collapse as seen on Image 1. He was started on preoperative empiric oseltamivir and azithromycin while in the emergency room (ER). He was urgently taken to the operating room (OR) for rigid bronchoscopy given the concern for a compressive endobronchial mass vs. mucus plug vs. airway foreign body. Of note, there was no preoperative history of a witnessed foreign body (FB) aspiration or ingestion event reported by the patient’s parents. Intraoperative examination in the OR revealed severe airway inflammation and a large inflammatory mucus cast occupying the entire left mainstem bronchus and completely obstructing the distal airways. The cast was removed via rigid bronchoscopy with optical bronchoscopic instrumentation and aspiration with immediate improvement in his L lung aeration and hypoxia. In both this child and the second child, normal bronchial anatomy was noted during the rigid bronchoscopy. A left thoracostomy tube was subsequently placed intraoperatively. Comprehensive infectious workup via a respiratory viral panel PCR and standard blood and BAL culture was positive only for influenza B but no other viral or bacterial isolates. The exact influenza B viral strain however was not further subtyped for either patient. Additionally, in both patients, extracted casts from the mainstem bronchi were not cultured or sent for surgical pathologic analysis. The patient’s respiratory status stabilized and rapidly improved over the subsequent few days. He did not require repeat bronchoscopy during his hospitalization but did require intubation/ventilator assistance postoperatively for 3 days. His thoracostomy tube removal and hospital discharge occurred
Image 1. Patient 1 preoperative CXR showing L lung opacification.
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Image 2. Patient 1 with extensive preoperative subcutaneous emphysema of neck.
respectively at 5 and 6 days postoperatively. A repeat postoperative chest CT at 6 weeks was normal and without evidence of any pulmonary sequelae. No bronchopleural fistula or bronchial tear was ever discovered at the time of his rigid bronchoscopy to explain his pneumothorax/pneumomediastinum. 3. Case 2 The second child, 5 y/o Indian male, presented to our emergency department (ER) in February 2013 in acute respiratory distress following 4–5 days of flu-like symptoms (fever to 38 8C, tachypnea, violent cough, labored breathing, wheezing). He also had no known medical comorbidities and also had not received his annual influenza vaccination. The patient had previously seen his primary care physician 2 days earlier who initiated albuterol, prednisone, and azithromycin for a presumed community-acquired pneumonia
Image 3. Patient 1 with preoperative L pneumothorax, subcutaneous emphysema, and L lung opacification, R lung hyperinflation.
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Image 4. Patient 2 preoperative CXR showing L lung opacification, L pneumothorax.
vs. viral URI. The patient was subsequently seen in an urgent care center 2 days later with hypoxia (pulse ox 70s), labored breathing, and respiratory distress. Subcutaneous emphysema was noted at this time on exam as well as radiologic evidence of R lung hyperinflation and pneumomediastinum (Image 4). The patient was immediately started on high-flow non-rebreather mask, and transferred to our institution’s ER. At this time, the patient was noted to be febrile to 38.1 8C, pursed lip breathing, tachypneic (RR 60 s), demonstrating moderate subcostal retractions, and with expiratory wheezing. Empiric vancomycin, ceftriaxone, azithromycin, and oseltamivir was immediately started and the patient was urgently transported to the OR for rigid bronchoscopy, intubation, and thoracostomy tube placement. In the OR, a thick mucus cast was identified in the left mainstem bronchus extending to the secondary bronchioles. There was severe airway erythema and edema of the trachea and L mainstem bronchus (Images 5 and 6). The cast was removed in a similar manner bronchoscopically but did require the use of a rigid larger
Image 6. Patient 2 intra-operative L mainstem bronchus after PB mucus cast removal.
esophageal suction placed through the bronchoscope given the tenacity and viscosity of the mucus plug. Immediately upon mucus plug removal, improvement in his respiratory status occurred. At the time of thoracostomy tube placement, 20 mL of serous fluid was obtained from his left chest cavity. Complete infectious workup (blood cx. and BAL cx.) was positive only for influenza B in this child. Extubation and chest tube removal were both accomplished on day 4 postoperatively, and hospital discharge by postoperative day 7. No repeat rigid bronchoscopy was required during this patient’s hospital stay. A surveillance CT chest imaging at 6 weeks postoperatively was normal. 4. Discussion
Image 5. Patient 2 intra-operative L mainstem bronchus with presence of PB mucus cast.
PB is a rare condition wherein casts are formed in the tracheobronchial airway. As the transudative or exudative material solidifies, it assumes the shape of the respiratory tree. Although the rarity of the disease precludes large observational cohorts, multiple small case studies and case series agree that the pathophysiological and histological etiologies of PB seem to fit into two categories. The first is associated with inflammatory processes of the lungs, including asthma, acute chest syndrome, and acute influenza infection. In these patients, the tracheobronchial casts consist of fibrin, neutrophils, and eosinophils and are therefore hypercellular. Airway inflammation, hypersecretion, and decreased mucociliary clearance contribute to cast formation. The second patient cohort, type II, includes patients with congenital cyanotic heart disease who have undergone a Fontan procedure or other palliative cardiac surgery. This presentation of PB has been well-described in the literature, with cast formation being more mucinous and hypocellular/acellular, and bronchial lymphatic leakage thought to play a leading role although no etiology has yet been proven. Cast formation is suspected secondary to the elevated central venous pressure gradient favoring lymphatic leakage into the central airways. Aggressive bronchoscopic removal of the casts is the most effective treatment for all etiologies, but this can be required frequently in children with congenital structural heart disease. In these patients, cardiac
S.J. Nogan et al. / International Journal of Pediatric Otorhinolaryngology 79 (2015) 1140–1144
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Table 1 Reported cases of PB associated with Influenza infections*. Author
Location
Age
Sex
Kim et al. [10] Sun et al. [5] Zheng et al. [6]
Korea China China
7 5 3–6
M M M
Uchimura et al. [9] Deng et al. [11]
Japan China
Hasegawa et al. [8] Terano et al. [7]
Japan Japan
4 3 3 7 6 2
F M M M M M
5 6
M F
Kunder et al. [4]
USA
6
M
Ruda
USA
Zhang and Kang [15]
China
5 8 1–4
? ? M
Casts
Notes **
‘‘Type I’’ ‘‘Type 1’’** Fibrinous material + eos, neuts, lymphs (x7). Fibrinous + necrotic material (no inflammatory cells) (x1) Not listed Fibrinous + neuts, eos ++eos, neuts, lymphs Fibrinous + necrotic material (no cells) Mucoid substance + eos infiltrate Fibrinous + mod inflam cells (70% neuts, 20% MØ, 10% lymphs) Fibrinous, ++eos 95%, Fibrinous and few cells (40% neut, 40% eos, 15% MØ, 5% lymphs) Mixture of hypocellular fibrinous casts and inflammatory casts w/abundant eos ‘‘Type I’’** ‘‘Type I’’** Fibrinous exudates (eos, neut, lymphs)
+ Influenza B 8 total patients
Influenza B. Also had d-TGA (fixed) and asthma Influenza B Influenza B 14 total patients; Influenza A or B (authors do not specify)
*
Unless otherwise listed, all influenza cases were influenza A (H1N1). Type I PB = cellular/inflammatory PB. PB, plastic bronchitis; TGA, transposition of the great arteries; neuts, neutrophils; eos, eosinophils; MØ, macrophages; lymphs, lymphocytes. **
transplantation, Fontan circuit fenestration, and extra-corporal membrane oxygenation have all been attempted as treatment for this disease [12–14]. Also, morbidity and mortality have been reported to be higher than that of type I PB patients [1,5]. Other adjunctive medical treatments also exist in the treatment of this disease including broad spectrum antibiotics, inhaled corticosteroids, bronchodilators, mucolytics, hypertonic saline, and fibrinolytics in addition to aggressive chest physiotherapy. Our report of two patients with solitary influenza B infections and no known medical comorbidities is a presentation previously unreported in the literature. Influenza A-associated PB has been described in a number of case studies that are listed in Table 1. Only two previous cases of influenza B-associated PB are known to us, one in the setting of congenital heart disease and the other with concurrent influenza A infection [4,10]. Influenza B is generally considered to be a less common and virulent viral strain than influenza A, and many of the cases of influenza A-associated PB were due to the novel H1N1 strain in 2009 in countries such as Mexico, Japan, and China. PB is also more commonly found in males more so than females. In our case series, both patients were males. However, both patients were atypical as they possessed no history of allergy, atopy, or asthma. Postoperatively, neither were ever found upon surveillance clinical examination to have developed asthma or reactive airway disease. We suspect that in the winter of 2012–2013, a more virulent strain of influenza B existed within our region of the country (Midwest) and state (Ohio) that contributed to the development of PB in our two patients. Unfortunately, the exact strain of influenza B for either child was never further characterized/typed at our institution. However, a review of Ohio Department of Health (DOH) Surveillance data was performed of state records of reportable cases of Influenza B infection for the dates when both our patients were diagnosed with their Influenza B-mediated PB (December 2012, February 2013). During the winter of 2012–2013, 60 reported cases of Influenza B were reported through February 28, 2013 to our state’s DOH. Ten cases of these Influenza-B cases were antigenically characterized and found to represent Influenza B/Brisbane/60/2008-like virus in 7 patients and Influenza B/ Wisconsin/1/2010-like virus in 3 patients. For both of our patients,
however, it is uncertain if either patient was reported to our state’s DOH and which exact Influenza B strain was responsible for their infection and associated PB. Thus, we were unable to discern the antigenic strain of Influenza B that was associated with PB in our 2 patients. To our knowledge, both patients had no prior physical contact, familial relation, or geographic proximity. Both cases were also chronologically separated by more than 2 months. We also suspect that their pneumothoraces and pneumomediastinum developed preoperatively from their associated vigorous cough as they attempted to mobilize and unknowingly expectorate their endobronchial casts. While aspirated foreign bodies are frequently noted in younger children and responsible for acute obstructive symptoms in this age cohort, in the wintertime months, other etiologies must be considered. In children with existing viral prodromal symptoms and any combination of pneumothorax, pneumomediastinum, mediastinal shift, asymmetric pulmonary hyperinflation, respiratory distress, and/or radiographic lung white-out, one should be suspicious for etiologies other than an airway foreign body. In these patients, an underlying infectious etiology as the source of a child’s acute airway obstruction with these findings should be entertained. Therefore, preoperative management for both patients included broad-spectrum IV antibiotics and anti-viral therapy until diagnostic/therapeutic rigid bronchoscopy was performed. Regardless of whether the etiology of the upper airway obstruction for both patients involved a bacterial tracheitis or virally mediated inflammatory plastic bronchitis, the preoperative management was identical. It was not until rigid bronchoscopy was performed that we were able to confirm the putative diagnosis of plastic bronchitis as the airway casts were identified and subsequently removed. In our two reported children with no witnessed or self-reported airway FB ingestion/aspiration, an airway FB was never discovered intra-operatively. The preoperative history suggested an acute infectious/inflammatory etiology for both patients’ airway obstruction which was confirmed intra-operatively as PB for both children. While uncommon, solitary influenza-B or any viral, inflammatory PB should be considered in the differential diagnosis of any child presenting during the winter months with acute respiratory distress and a preceding viral prodromal history.
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Conflicts of interest We have no personal or financial conflicts of interest to report as related to this publication.
References [1] P. Madsen, S.A. Shah, B.K. Rubin, Plastic bronchitis: new insights and a classification scheme, Paediatr. Respir. Rev. 6 (4) (2005) 292–300. [2] P. Do, I. Randhawa, T. Chin, K. Parsapour, E. Nussbaum, Successful management of plastic bronchitis in a child post Fontan: case report and literature review, Lung 190 (4) (2012) 463–468. [3] D. Preciado, S. Verghese, S. Choi, Aggressive bronchoscopic management of plastic bronchitis, Int. J. Pediatr. Otorhinolaryngol. 74 (7) (2010) 820–822. [4] R. Kunder, C. Kunder, H.Y. Sun, G. Berry, A. Messner, J. Frankovich, et al., Pediatric plastic bronchitis: case report and retrospective comparative analysis of epidemiology and pathology, Case Rep. Pulmonol. 2013 (2) (2013) 1–8. [5] D.J. Sun, Y.S. Yang, B.C. Wang, Plastic bronchitis associated with severe influenza A (H1N1) in children: a case report and review of the literature, Chin. J. Tuberc. Respir. Dis. 33 (November (11)) (2010) 837–839. [6] Y.J. Zheng, J.K. Deng, Z.W. Lu, H.L. Ma, J. Li, L. Wang, Clinical analysis of 8 children with plastic bronchitis associated with influenza A virus (H1N1) infection, Chin. J. Pediatr. 50 (July (7)) (2012) 521–524.
[7] C. Terano, M. Miura, R. Fukuzawa, Y. Saito, H. Arai, M. Sasaki, et al., Three children with plastic bronchitis associated with 2009 H1N1 influenza virus infection, Pediatr. Infect. Dis. J. 30 (January (1)) (2011) 80–82. [8] S. Hasegawa, R. Hirano, K. Hashimoto, Characteristics of atopic children with pandemic H1N1 influenza viral infection: pandemic H1N1 influenza reveals ‘occult’ asthma of childhood, Pediatr. Allergy Immunol. 22 (February (1)) (2011) e119–e123. [9] T. Uchimura, M. Mori, A. Nariai, S. Yokota, Analysis of cases of severe respiratory failure in children with influenza (H1N1) 2009 infection in Japan, J. Infect. Chemother. 18 (1) (2012) 59–65. [10] S. Kim, H.J. Cho, D.K. Han, Y.D. Choi, E.S. Yang, Y.K. Cho, et al., Recurrent plastic bronchitis in a child with 2009 influenza A (H1N1) and influenza B virus infection, J. Korean Med. Sci. 27 (September (9)) (2012) 1114–1119. [11] J. Deng, Y. Zheng, C. Li, Z. Ma, H. Wang, B.K. Rubin, Plastic bronchitis in three children associated with 2009 influenza A(H1N1) virus infection, Chest 138 (December (6)) (2010) 1486–1488. [12] J.E. Laubisch, D.M. Green, P.J. Mogayzel, W.R. Thompson, Treatment of plastic bronchitis by orthotopic heart transplantation, Pediatr. Cardiol. 32 (8) (2011) 1193–1195. [13] M. Tonan, S. Hashimoto, A. Kimura, H. Matsuyama, H. Kinose, M. Sawada, et al., Successful treatment of severe asthma-associated plastic bronchitis with extracorporeal membrane oxygenation, J. Anesth. 26 (2) (2012) 265–268. [14] J. Wilson, J. Russell, W. Williams, L. Benson, Fenestration of the Fontan circuit as treatment for plastic bronchitis, Pediatr. Cardiol. 26 (5) (2005) 717–719. [15] J. Zhang, X. Kang, Plastic bronchitis associated with influenza virus infection in children: a report of 14 cases, Int. J. Pediatr. Otorhinolaryngol. 79 (April) (2015) 481–486.
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Case Report
Plastic bronchitis arising from solitary influenza B infection: A report of two cases in children§ Stephen J. Nogan a, Nathan D. Cass c, Gregory J. Wiet a,b, James M. Ruda a,b,* a
Department of Otolaryngology-Head & Neck Surgery, Ohio State University-Wexner Medical Center, Columbus, OH 43212, USA Department of Pediatric Otolaryngology, Nationwide Children’s Hospital, Columbus, OH 43205, USA c Department of Otolaryngology, University of Colorado Anschutz Medical Campus, Aurora, CO 80045, USA b
A R T I C L E I N F O
A B S T R A C T
Article history: Received 28 January 2015 Received in revised form 22 March 2015 Accepted 28 March 2015 Available online 7 April 2015
Plastic bronchitis (PB) is characterized by thick, inspissated, tracheobronchial casts. It is classified as either inflammatory or acellular based on the content of the endobronchial casts. PB has never been reported in a healthy child with solitary influenza B infection. This study is a retrospective case series of two children who presented to our institution in acute respiratory distress. Emergency rigid bronchoscopy was performed with extraction of casts from the L mainstem bronchus in both patients. Influenza B was the only isolate identified. In otherwise healthy children with respiratory distress, influenza B-mediated inflammatory PB must be considered in the differential diagnosis. ß 2015 Elsevier Ireland Ltd. All rights reserved.
Keywords: Plastic bronchitis Children Influenza B
1. Introduction Plastic bronchitis (PB) is a rare entity characterized by the development of thick, obstructing mucoid/gelatinous casts in the tracheobronchial airway. It has been historically categorized as either cellular (type I) vs. acellular (type II) as well as inflammatory vs. non-inflammatory based on the pathologic consistency of the cast and underlying etiology [1]. It is frequently described in infants and children with congenital heart disease and following palliative cardiac surgery, i.e. (Fontan) secondary to their surgically altered cardiac anatomy and physiology that favors transudation or exudation of fluid into the airway. With solidification of these transudates and exudates, obstruction of the large and small airways occurs. This leads to the development of acute respiratory distress, hypoxia, tachycardia, tachypnea, and cough. Radiographically, PB can present as pulmonary opacification, mediastinal shift, atelectasis, and pneumonia. Treatment frequently requires bronchoscopic evacuation of the mucoid casts from the tracheobronchial tree and remedy of any underlying abnormal cardiac physiology or medical problem, when possible or applicable. PB has also been described in patients with associated inflammatory pulmonary conditions such as cystic fibrosis, acute chest syndrome
§
Presented at SENTAC, December 5–8th, 2013, Long Beach, California. * Corresponding author. Tel.: +1 614 722 6804; fax: +1 614 722 6609. E-mail addresses: [email protected] (S.J. Nogan), [email protected] (N.D. Cass), [email protected] (G.J. Wiet), [email protected] (J.M. Ruda). http://dx.doi.org/10.1016/j.ijporl.2015.03.028 0165-5876/ß 2015 Elsevier Ireland Ltd. All rights reserved.
of sickle-cell disease, allergic bronchopulmonary aspergillosis, asthma as well as in those with a personal or family history of atopy [2,3]. Influenza A has recently been identified for its association with PB, especially during the recent 2009 outbreak of the H1N1 influenza A pandemic in Asia and Mexico [4–11]. Children with influenza A infection can frequently develop acute pulmonary complications and sequelae such as pneumonia and acute respiratory failure when acutely infected. PB has been previously reported in one child who was co-infected with influenza A and B as well as in a child with underlying congenital heart disease and was also infected with influenza B [4,10]. To our knowledge, no reports of solitary influenza B infection in a healthy child who developed PB has ever been reported. We present a report of two cases of solitary influenza B-mediated inflammatory PB in otherwise healthy children with no known medical comorbidities during the winter of 2012–2013. For this study, institutional IRB approval was obtained. 2. Case 1 The first child, an 8 y/o Caucasian male, was transferred from a community hospital to our institution in acute respiratory distress in December 2012. He had no known medical history or comorbidities such as asthma or atopy but was not up-to-date on his annual influenza vaccination. He possessed a 4-day history of flu-like symptoms including fever to 38 8C, rhinorrhea, tachypnea, violent cough, and malaise/fatigue. On the morning
S.J. Nogan et al. / International Journal of Pediatric Otorhinolaryngology 79 (2015) 1140–1144
of admission, he was noted to have acute, diffuse chest/neck swelling by his family. Upon presentation to the community hospital later that day, he was found to be hypoxic, febrile to 38.1 8C, with labored breathing, and with extensive subcutaneous emphysema over his neck/chest. A CT chest scan was performed showing a small L pneumothorax and pneumomediastinum with a hyperinflated R lung, opacified L lung, but midline trachea (Images 2 and 3). He was immediately started on a high-flow oxygen non-rebreather mask as well as empiric vancomycin and ceftriaxone, and transferred to our institution. Upon arrival to our institution, the patient was persistently tachypneic, hypoxic, and febrile, but stable on high-flow nonrebreather mask oxygen. A confirmatory CXR showed persistent left-ward mediastinal shift and L lung collapse as seen on Image 1. He was started on preoperative empiric oseltamivir and azithromycin while in the emergency room (ER). He was urgently taken to the operating room (OR) for rigid bronchoscopy given the concern for a compressive endobronchial mass vs. mucus plug vs. airway foreign body. Of note, there was no preoperative history of a witnessed foreign body (FB) aspiration or ingestion event reported by the patient’s parents. Intraoperative examination in the OR revealed severe airway inflammation and a large inflammatory mucus cast occupying the entire left mainstem bronchus and completely obstructing the distal airways. The cast was removed via rigid bronchoscopy with optical bronchoscopic instrumentation and aspiration with immediate improvement in his L lung aeration and hypoxia. In both this child and the second child, normal bronchial anatomy was noted during the rigid bronchoscopy. A left thoracostomy tube was subsequently placed intraoperatively. Comprehensive infectious workup via a respiratory viral panel PCR and standard blood and BAL culture was positive only for influenza B but no other viral or bacterial isolates. The exact influenza B viral strain however was not further subtyped for either patient. Additionally, in both patients, extracted casts from the mainstem bronchi were not cultured or sent for surgical pathologic analysis. The patient’s respiratory status stabilized and rapidly improved over the subsequent few days. He did not require repeat bronchoscopy during his hospitalization but did require intubation/ventilator assistance postoperatively for 3 days. His thoracostomy tube removal and hospital discharge occurred
Image 1. Patient 1 preoperative CXR showing L lung opacification.
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Image 2. Patient 1 with extensive preoperative subcutaneous emphysema of neck.
respectively at 5 and 6 days postoperatively. A repeat postoperative chest CT at 6 weeks was normal and without evidence of any pulmonary sequelae. No bronchopleural fistula or bronchial tear was ever discovered at the time of his rigid bronchoscopy to explain his pneumothorax/pneumomediastinum. 3. Case 2 The second child, 5 y/o Indian male, presented to our emergency department (ER) in February 2013 in acute respiratory distress following 4–5 days of flu-like symptoms (fever to 38 8C, tachypnea, violent cough, labored breathing, wheezing). He also had no known medical comorbidities and also had not received his annual influenza vaccination. The patient had previously seen his primary care physician 2 days earlier who initiated albuterol, prednisone, and azithromycin for a presumed community-acquired pneumonia
Image 3. Patient 1 with preoperative L pneumothorax, subcutaneous emphysema, and L lung opacification, R lung hyperinflation.
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Image 4. Patient 2 preoperative CXR showing L lung opacification, L pneumothorax.
vs. viral URI. The patient was subsequently seen in an urgent care center 2 days later with hypoxia (pulse ox 70s), labored breathing, and respiratory distress. Subcutaneous emphysema was noted at this time on exam as well as radiologic evidence of R lung hyperinflation and pneumomediastinum (Image 4). The patient was immediately started on high-flow non-rebreather mask, and transferred to our institution’s ER. At this time, the patient was noted to be febrile to 38.1 8C, pursed lip breathing, tachypneic (RR 60 s), demonstrating moderate subcostal retractions, and with expiratory wheezing. Empiric vancomycin, ceftriaxone, azithromycin, and oseltamivir was immediately started and the patient was urgently transported to the OR for rigid bronchoscopy, intubation, and thoracostomy tube placement. In the OR, a thick mucus cast was identified in the left mainstem bronchus extending to the secondary bronchioles. There was severe airway erythema and edema of the trachea and L mainstem bronchus (Images 5 and 6). The cast was removed in a similar manner bronchoscopically but did require the use of a rigid larger
Image 6. Patient 2 intra-operative L mainstem bronchus after PB mucus cast removal.
esophageal suction placed through the bronchoscope given the tenacity and viscosity of the mucus plug. Immediately upon mucus plug removal, improvement in his respiratory status occurred. At the time of thoracostomy tube placement, 20 mL of serous fluid was obtained from his left chest cavity. Complete infectious workup (blood cx. and BAL cx.) was positive only for influenza B in this child. Extubation and chest tube removal were both accomplished on day 4 postoperatively, and hospital discharge by postoperative day 7. No repeat rigid bronchoscopy was required during this patient’s hospital stay. A surveillance CT chest imaging at 6 weeks postoperatively was normal. 4. Discussion
Image 5. Patient 2 intra-operative L mainstem bronchus with presence of PB mucus cast.
PB is a rare condition wherein casts are formed in the tracheobronchial airway. As the transudative or exudative material solidifies, it assumes the shape of the respiratory tree. Although the rarity of the disease precludes large observational cohorts, multiple small case studies and case series agree that the pathophysiological and histological etiologies of PB seem to fit into two categories. The first is associated with inflammatory processes of the lungs, including asthma, acute chest syndrome, and acute influenza infection. In these patients, the tracheobronchial casts consist of fibrin, neutrophils, and eosinophils and are therefore hypercellular. Airway inflammation, hypersecretion, and decreased mucociliary clearance contribute to cast formation. The second patient cohort, type II, includes patients with congenital cyanotic heart disease who have undergone a Fontan procedure or other palliative cardiac surgery. This presentation of PB has been well-described in the literature, with cast formation being more mucinous and hypocellular/acellular, and bronchial lymphatic leakage thought to play a leading role although no etiology has yet been proven. Cast formation is suspected secondary to the elevated central venous pressure gradient favoring lymphatic leakage into the central airways. Aggressive bronchoscopic removal of the casts is the most effective treatment for all etiologies, but this can be required frequently in children with congenital structural heart disease. In these patients, cardiac
S.J. Nogan et al. / International Journal of Pediatric Otorhinolaryngology 79 (2015) 1140–1144
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Table 1 Reported cases of PB associated with Influenza infections*. Author
Location
Age
Sex
Kim et al. [10] Sun et al. [5] Zheng et al. [6]
Korea China China
7 5 3–6
M M M
Uchimura et al. [9] Deng et al. [11]
Japan China
Hasegawa et al. [8] Terano et al. [7]
Japan Japan
4 3 3 7 6 2
F M M M M M
5 6
M F
Kunder et al. [4]
USA
6
M
Ruda
USA
Zhang and Kang [15]
China
5 8 1–4
? ? M
Casts
Notes **
‘‘Type I’’ ‘‘Type 1’’** Fibrinous material + eos, neuts, lymphs (x7). Fibrinous + necrotic material (no inflammatory cells) (x1) Not listed Fibrinous + neuts, eos ++eos, neuts, lymphs Fibrinous + necrotic material (no cells) Mucoid substance + eos infiltrate Fibrinous + mod inflam cells (70% neuts, 20% MØ, 10% lymphs) Fibrinous, ++eos 95%, Fibrinous and few cells (40% neut, 40% eos, 15% MØ, 5% lymphs) Mixture of hypocellular fibrinous casts and inflammatory casts w/abundant eos ‘‘Type I’’** ‘‘Type I’’** Fibrinous exudates (eos, neut, lymphs)
+ Influenza B 8 total patients
Influenza B. Also had d-TGA (fixed) and asthma Influenza B Influenza B 14 total patients; Influenza A or B (authors do not specify)
*
Unless otherwise listed, all influenza cases were influenza A (H1N1). Type I PB = cellular/inflammatory PB. PB, plastic bronchitis; TGA, transposition of the great arteries; neuts, neutrophils; eos, eosinophils; MØ, macrophages; lymphs, lymphocytes. **
transplantation, Fontan circuit fenestration, and extra-corporal membrane oxygenation have all been attempted as treatment for this disease [12–14]. Also, morbidity and mortality have been reported to be higher than that of type I PB patients [1,5]. Other adjunctive medical treatments also exist in the treatment of this disease including broad spectrum antibiotics, inhaled corticosteroids, bronchodilators, mucolytics, hypertonic saline, and fibrinolytics in addition to aggressive chest physiotherapy. Our report of two patients with solitary influenza B infections and no known medical comorbidities is a presentation previously unreported in the literature. Influenza A-associated PB has been described in a number of case studies that are listed in Table 1. Only two previous cases of influenza B-associated PB are known to us, one in the setting of congenital heart disease and the other with concurrent influenza A infection [4,10]. Influenza B is generally considered to be a less common and virulent viral strain than influenza A, and many of the cases of influenza A-associated PB were due to the novel H1N1 strain in 2009 in countries such as Mexico, Japan, and China. PB is also more commonly found in males more so than females. In our case series, both patients were males. However, both patients were atypical as they possessed no history of allergy, atopy, or asthma. Postoperatively, neither were ever found upon surveillance clinical examination to have developed asthma or reactive airway disease. We suspect that in the winter of 2012–2013, a more virulent strain of influenza B existed within our region of the country (Midwest) and state (Ohio) that contributed to the development of PB in our two patients. Unfortunately, the exact strain of influenza B for either child was never further characterized/typed at our institution. However, a review of Ohio Department of Health (DOH) Surveillance data was performed of state records of reportable cases of Influenza B infection for the dates when both our patients were diagnosed with their Influenza B-mediated PB (December 2012, February 2013). During the winter of 2012–2013, 60 reported cases of Influenza B were reported through February 28, 2013 to our state’s DOH. Ten cases of these Influenza-B cases were antigenically characterized and found to represent Influenza B/Brisbane/60/2008-like virus in 7 patients and Influenza B/ Wisconsin/1/2010-like virus in 3 patients. For both of our patients,
however, it is uncertain if either patient was reported to our state’s DOH and which exact Influenza B strain was responsible for their infection and associated PB. Thus, we were unable to discern the antigenic strain of Influenza B that was associated with PB in our 2 patients. To our knowledge, both patients had no prior physical contact, familial relation, or geographic proximity. Both cases were also chronologically separated by more than 2 months. We also suspect that their pneumothoraces and pneumomediastinum developed preoperatively from their associated vigorous cough as they attempted to mobilize and unknowingly expectorate their endobronchial casts. While aspirated foreign bodies are frequently noted in younger children and responsible for acute obstructive symptoms in this age cohort, in the wintertime months, other etiologies must be considered. In children with existing viral prodromal symptoms and any combination of pneumothorax, pneumomediastinum, mediastinal shift, asymmetric pulmonary hyperinflation, respiratory distress, and/or radiographic lung white-out, one should be suspicious for etiologies other than an airway foreign body. In these patients, an underlying infectious etiology as the source of a child’s acute airway obstruction with these findings should be entertained. Therefore, preoperative management for both patients included broad-spectrum IV antibiotics and anti-viral therapy until diagnostic/therapeutic rigid bronchoscopy was performed. Regardless of whether the etiology of the upper airway obstruction for both patients involved a bacterial tracheitis or virally mediated inflammatory plastic bronchitis, the preoperative management was identical. It was not until rigid bronchoscopy was performed that we were able to confirm the putative diagnosis of plastic bronchitis as the airway casts were identified and subsequently removed. In our two reported children with no witnessed or self-reported airway FB ingestion/aspiration, an airway FB was never discovered intra-operatively. The preoperative history suggested an acute infectious/inflammatory etiology for both patients’ airway obstruction which was confirmed intra-operatively as PB for both children. While uncommon, solitary influenza-B or any viral, inflammatory PB should be considered in the differential diagnosis of any child presenting during the winter months with acute respiratory distress and a preceding viral prodromal history.
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Conflicts of interest We have no personal or financial conflicts of interest to report as related to this publication.
References [1] P. Madsen, S.A. Shah, B.K. Rubin, Plastic bronchitis: new insights and a classification scheme, Paediatr. Respir. Rev. 6 (4) (2005) 292–300. [2] P. Do, I. Randhawa, T. Chin, K. Parsapour, E. Nussbaum, Successful management of plastic bronchitis in a child post Fontan: case report and literature review, Lung 190 (4) (2012) 463–468. [3] D. Preciado, S. Verghese, S. Choi, Aggressive bronchoscopic management of plastic bronchitis, Int. J. Pediatr. Otorhinolaryngol. 74 (7) (2010) 820–822. [4] R. Kunder, C. Kunder, H.Y. Sun, G. Berry, A. Messner, J. Frankovich, et al., Pediatric plastic bronchitis: case report and retrospective comparative analysis of epidemiology and pathology, Case Rep. Pulmonol. 2013 (2) (2013) 1–8. [5] D.J. Sun, Y.S. Yang, B.C. Wang, Plastic bronchitis associated with severe influenza A (H1N1) in children: a case report and review of the literature, Chin. J. Tuberc. Respir. Dis. 33 (November (11)) (2010) 837–839. [6] Y.J. Zheng, J.K. Deng, Z.W. Lu, H.L. Ma, J. Li, L. Wang, Clinical analysis of 8 children with plastic bronchitis associated with influenza A virus (H1N1) infection, Chin. J. Pediatr. 50 (July (7)) (2012) 521–524.
[7] C. Terano, M. Miura, R. Fukuzawa, Y. Saito, H. Arai, M. Sasaki, et al., Three children with plastic bronchitis associated with 2009 H1N1 influenza virus infection, Pediatr. Infect. Dis. J. 30 (January (1)) (2011) 80–82. [8] S. Hasegawa, R. Hirano, K. Hashimoto, Characteristics of atopic children with pandemic H1N1 influenza viral infection: pandemic H1N1 influenza reveals ‘occult’ asthma of childhood, Pediatr. Allergy Immunol. 22 (February (1)) (2011) e119–e123. [9] T. Uchimura, M. Mori, A. Nariai, S. Yokota, Analysis of cases of severe respiratory failure in children with influenza (H1N1) 2009 infection in Japan, J. Infect. Chemother. 18 (1) (2012) 59–65. [10] S. Kim, H.J. Cho, D.K. Han, Y.D. Choi, E.S. Yang, Y.K. Cho, et al., Recurrent plastic bronchitis in a child with 2009 influenza A (H1N1) and influenza B virus infection, J. Korean Med. Sci. 27 (September (9)) (2012) 1114–1119. [11] J. Deng, Y. Zheng, C. Li, Z. Ma, H. Wang, B.K. Rubin, Plastic bronchitis in three children associated with 2009 influenza A(H1N1) virus infection, Chest 138 (December (6)) (2010) 1486–1488. [12] J.E. Laubisch, D.M. Green, P.J. Mogayzel, W.R. Thompson, Treatment of plastic bronchitis by orthotopic heart transplantation, Pediatr. Cardiol. 32 (8) (2011) 1193–1195. [13] M. Tonan, S. Hashimoto, A. Kimura, H. Matsuyama, H. Kinose, M. Sawada, et al., Successful treatment of severe asthma-associated plastic bronchitis with extracorporeal membrane oxygenation, J. Anesth. 26 (2) (2012) 265–268. [14] J. Wilson, J. Russell, W. Williams, L. Benson, Fenestration of the Fontan circuit as treatment for plastic bronchitis, Pediatr. Cardiol. 26 (5) (2005) 717–719. [15] J. Zhang, X. Kang, Plastic bronchitis associated with influenza virus infection in children: a report of 14 cases, Int. J. Pediatr. Otorhinolaryngol. 79 (April) (2015) 481–486.
