Archives of Academic Emergency Medicine. 2021; 9(1): e16 https://doi.org/10.22037/aaem.v9i1.1014 CA S E RE P O RT Plastic Bronchitis in a Five-Year-Old Boy Treated Using Ex- tracorporeal Membrane Oxygenation; a Case Report Tsuyoshi Nojima1, Hiromichi Naito1∗, Takafumi Obara1, Kohei Tsukahara1, Atsunori Nakao1 1. Department of Emergency, Critical Care and Disaster Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama, Japan. Received: December 2020; Accepted: December 2020; Published online: 27 January 2021 Abstract: Plastic bronchitis is an uncommon disorder marked by the production of bronchial casts and acute respiratory failure development. In pediatric cases, influenza infection sometimes results in the obstruction of bronchi and leads to this potentially life-threatening condition. We report the case of a five-year-old boy with plastic bronchitis related to influenza A infection, which could only be recovered by the use of extracorporeal mem- brane oxygenation (ECMO). ECMO could effectively provide sufficient oxygenation for patients suffering from severe reversible acute respiratory failure. If patients infected with the influenza virus present acute respira- tory distress with total lung atelectasis, clinicians should consider the diagnosis of plastic bronchitis and the subsequent treatment interventions with ECMO in a severe cases. Keywords: Bronchitis; Influenza A virus; extracorporeal membrane oxygenation; respiratory distress syndrome Cite this article as: Nojima T, Naito H, Obara T, Tsukahara K, Nakao A. Plastic Bronchitis in a Five-Year-Old Boy Treated Using Extracorporeal Membrane Oxygenation; a Case Report. Arch Acad Emerg Med. 2021; 9(1): e16. 1. Introduction Plastic bronchitis is a life-threatening complication defined by the production of large, branching bronchial casts that are often expectorated but may be only recognized during bronchoscopy. Clinical manifestations may include fast and incremental respiratory distress with lung consolidation or atelectasis showing on chest radiograph. In most patients, bronchial casts are secondary to underlying heart or lung diseases known to be associated with allergy, asthma, or in- fluenza infection (1-4). Here, we report the case of a five-year-old boy with plastic bronchitis associated with influenza A successfully treated with extracorporeal membrane oxygenation (ECMO). Our case indicates that in pediatric patients suffering from severe plastic bronchitis with acute respiratory failure, venovenous (VV ) ECMO may be the only effective lifesaving procedure for providing sufficient oxygenation. If patients infected with the influenza virus present the combination of total lung atelec- tasis and acute respiratory distress, clinicians should con- ∗Corresponding Author: Hiromichi Naito; Okayama University Gradu- ate School of Medicine, Dentistry and Pharmaceutical Sciences, Depart- ment of Emergency, Critical Care, and Disaster Medicine. 2-5-1 Shikata, Okayama, Japan 700-8558. Tel: +81-86-235-7426, Fax: +81-86-235-7427, e- mail: naito.hiromichi@gmail.com sider the diagnosis of plastic bronchitis and intervene with early bronchoscopy and consider application of ECMO. 2. Cases presentation A five-year-old boy, 15 kilograms in weight and 107 cm in height, was admitted to a local hospital after three days of fever, coughing, and aggravating dyspnea. A rapid influenza test conducted using a nasopharyngeal swab was positive for the influenza A virus. He was transported to our tertiary emergency medical center due to respiratory failure. Upon physical examination after emergent transfer to our emer- gency department, his blood pressure was 174/101 mmHg, pulse rate was 179 bpm, respiratory rate was 30/minute, and body temperature was 37.8°C. His percutaneous oxygen sat- uration was 86% with a respirator delivering 100% oxygen, and breathing sounds were decreased in both lungs. His ve- nous blood gas analysis and blood chemical analysis results are shown in Table1. His chest X-ray and computed tomogra- phy showed pneumothorax in both lungs and consolidation in the right lung (Figure 1). Due to deoxygenation and deterioration as a result of respira- tory distress, the patient had to be intubated and placed un- der mechanical ventilation. Flexible bronchoscopy revealed thick secretions and right lower lobe bronchus lumen ob- struction with a rubbery, whitish substance consistent with This open-access article distributed under the terms of the Creative Commons Attribution NonCommercial 3.0 License (CC BY-NC 3.0). Downloaded from: http://journals.sbmu.ac.ir/aaem T. Nojima et al. 2 Table 1: Blood chemical analysis on arrival Variable Value Variable Value Venous blood gas Clinical chemistry test pH 7.201 CRP (mg/dL) 10.21 PaCO2 (mmHg) 73.7 TP (g/dL) 6.4 HCO3 (mmol/L) 27.4 ALB (g/dL) 3.7 BE (mmol/L) -8.1 AST (IU/L) 28 AG (mmol/L) 18.7 ALT (IU/L) 15 Lactate (mmol/L) 0.8 LDH (IU/L) 428 Complete blood count ALP (IU/L) 700 WBC (/µL) 34,010 G-GTP (IU/L) 10 Neut (%) 94 CK (IU/L) 56 Lymp (%) 3 T-Bil (mg/dL) 0.7 Mono (%) 2.5 S-AMY (IU/L) 26 Hb (g/dL) 13.1 BUN (mg/dL) 16.7 Plt (/µL) 40.5×104 Crea (mg/dL) 0.28 Blood coagulation test UA (mg/dL) 4.9 INR 1.22 Na ( mEq/L) 146 aPTT (sec) 29.2 K ( mEq/L) 4.6 Fibrinogen (mg/dL) 454 Cl ( mEq/L) 106 FDP (µg/mL) 34 Ca ( mg/dL) 9.4 D-dimer (µg/mL) 18.1 Figure 1: The chest X-ray and computed tomography on arrival showed pneumothorax and pleural effusion in both lungs. an endobronchial cast (Figure 2). The clinical diagnosis of acute respiratory failure, pneumothorax, and plastic bron- chitis associated with influenza A infection was made. Con- sidering persistent respiratory distress in 41 of peak inspi- ratory pressure and subsequent circulatory collapse, the pa- tient was placed on venous-arterial (VA) ECMO through the right cervical artery and vein with chest drainage tubes on both chest cavities. Initial VA-ECMO conditions were as fol- lows: oxygenator; Biocube (NIPRO, Japan), venous line; right internal jugular vein using BioMedicusT M NextGen catheter 17Fr (Medtronic, Mexico), arterial line; right internal jugular artery using BioMedicus cateter 12Fr (Medtronic, Mexico), blood flow 1.5L/min, speed 2,780 rpm, oxygen sweep gas 3.0L/min, and the fraction of oxygen 0.8 using heparin. An- timicrobial treatment included 2,250 mg/day of cefotaxime, 900 mg/day of vancomycin, and 150 mg/day of peramivir. Figure 3 shows the progress after hospitalization. On the fourth day, since initiation of VA-ECMO had sta- bilized the patient’s circulatory condition, VA-ECMO was converted to VV-ECMO through the right cervical vein and femoral vein. Initial VV-ECMO conditions were as follows: oxygenator; Biocube (NIPRO, Japan), venous line; right in- ternal jugular vein using BioMedicusT M NextGen catheter 17Fr (Medtronic, Mexico), arterial line; right femoral vein us- This open-access article distributed under the terms of the Creative Commons Attribution NonCommercial 3.0 License (CC BY-NC 3.0). Downloaded from: http://journals.sbmu.ac.ir/aaem 3 Archives of Academic Emergency Medicine. 2021; 9(1): e16 Figure 2: Photograph of a representative cast spontaneously expec- torated from a patient with plastic bronchitis during bronchoscopy. Figure 3: Progress of respiratory parameters of the patient during hospitalization. PIP: peak inspiratory pressure, VA-ECMO: Venous- Arterial extracorporeal membrane oxygenation, VV-ECMO: Veno- Venous extracorporeal membrane oxygenation, P/F: PaO2 /FiO2 . ing BioMedicusT M NextGen cateter 15Fr (Medtronic, Mex- ico), blood-flow 1.52 L/min, speed 2010 rpm, oxygen sweep gas 2.0 L/min, and the fraction of oxygen 1.0 using heparin. On the eighth day, his breathing improved, so we switched from VV-ECMO to ventilation management only. Mechan- ical ventilator conditions were as follows: assist mode and control mode by pressure control ventilation; positive end- expiratory pressure 5 cmH2O; the fraction of oxygen 0.6; pressure control 15 cmH2O; and respiratory rate 15. Fre- quent removal of purulent mucous plugging of the airway was required. ECMO support was discontinued after nine days. The patient was gradually weaned off respiratory sup- port, extubated on day 20, and transferred to a rehabilitation hospital on day 21. He has remained well over a 2-month follow-up period. 3. Discussion Plastic bronchitis is an uncommon and underrecognized but life-threatening condition marked by notable blockage of the large airways by mucous plugs (5). Prompt diagnosis and early intervention, including removing the firm, cohesive, branching casts using bronchoscopy, are critical. Mechan- ical ventilation is not always successful in critically ill plas- tic bronchitis patients. High airway pressure to achieve ad- equate oxygenation may result in barotrauma, as seen in our patient. The fast clinical deterioration that does not re- spond to standard therapies makes ECMO a critical adjunct therapy for plastic bronchitis treatment. The rubbery casts in the airway seen in plastic bronchitis are known to com- prise fibrin, mucin, or a mixture of both and can obstruct the airways completely or partially. According to a recent well-defined classification of casts based on their composi- tion and underlying pathology (6), casts are divided into two types: inflammatory casts comprising fibrin and dense in- flammatory infiltrate associated with respiratory infection or asthma (type 1), and acellular casts comprising mostly mucin related to congenital heart diseases (type 2). Histological ex- amination of the casts can point to underlying diseases and may be helpful in therapeutic decision making. Although acute mortality can occur in patients with both types of casts, standard therapeutic strategies have not been established. Type 1 cast survivors seem to be well-controlled with inhaled steroids. Optimal therapy for type 2 cast patients has not been fully determined; prognosis probably depends on un- derlying cardiac status. Various treatment options have been advocated (7). In addition to repeated bronchoscopic extrac- tion of casts, clinicians should consider inhalation heparin when treating the underlying disorders fail, and the casts are largely composed of fibrin (1, 8, 9). Heparin is ineffective for fibrin-containing casts but has anti-inflammatory prop- erties that can reduce the secretion of mucin. Chest physio- therapy, acetylcysteine, DNAse, and systemic corticosteroids have all been used (10). Since tissue factor activation of the fibrin pathway can attenuate vascular leak, case reports have shown that inhalation of recombinant tissue plasminogen activator can improve plastic bronchitis through fibrin de- polymerization (8). Of note, asthma medication administra- tion, like inhaled corticosteroids or beta-agonists, provides only a marginal benefit in most plastic bronchitis cases. 4. Conclusion In pediatric patients with fast and progressive respiratory distress and lung consolidation or atelectasis showing on chest radiograph associated with influenza infection, clini- cians should consider the possibility of plastic bronchitis and should intervene with early bronchoscopy treatment. For pa- tients with life-threatening progressive respiratory failure, in This open-access article distributed under the terms of the Creative Commons Attribution NonCommercial 3.0 License (CC BY-NC 3.0). Downloaded from: http://journals.sbmu.ac.ir/aaem T. Nojima et al. 4 whom significant cast obstruction may advance to cardiores- piratory collapse, prompt therapy with ECMO may be effec- tive and lifesaving, andit may facilitate full recovery of nor- mal pulmonary function. 5. Ethical issues Written informed consent was obtained from the patient for publication of this case report and accompanying images. This case report was approved by the Ethics Committee of Okayama University (ethics code: K2103-032 ). 6. Declarations 6.1. Acknowledgements No funding supported this study. 6.2. Authors’ relationships All authors met the criteria for authorship contribution based on the international committee of medical journal editors’ recommendations. 6.3. Conflict of interest The authors declare no conflicts of interest. 6.4. Funding None. References 1. Deng J, Zheng Y, Li C, Ma Z, Wang H, Rubin BK. Plastic bronchitis in three children associated with 2009 influenza A(H1N1) virus infection. Chest. 2010;138(6):1486-8. 2. Zhang X, Vinturache A, Ding G. Plastic bronchitis in a 3- year-old boy. CMAJ. 2019;191(48):E1336. 3. Okada Y, Okada A, Narumiya H, Iiduka R, Katsura K. Bloody Bronchial Cast Formation Due to Alveolar Hem- orrhage Associated with H1N1 Influenza Infection. In- tern Med. 2017;56(20):2747-51. 4. Kim S, Cho HJ, Han DK, Choi YD, Yang ES, Cho YK, et al. 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J Pediatr Surg. 2016;51(10):1640-3. 10. Madsen P, Shah SA, Rubin BK. Plastic bronchitis: new in- sights and a classification scheme. Paediatr Respir Rev. 2005;6(4):292-300. This open-access article distributed under the terms of the Creative Commons Attribution NonCommercial 3.0 License (CC BY-NC 3.0). Downloaded from: http://journals.sbmu.ac.ir/aaem Introduction Cases presentation Discussion Conclusion Ethical issues Declarations References