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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 13  |  Issue : 1  |  Page : 46-49

Plastic bronchitis in a 6-year-old boy: An unusual indication for rigid bronchoscopy


1 Department of Otorhinolaryngology, IMS and SUM Hospital, Siksha “O” Anusandhan (Deemed to be University), Bhubaneswar, Odisha, India
2 Department of Pediatrics, IMS and SUM Hospital, Siksha “O” Anusandhan (Deemed to be University), Bhubaneswar, Odisha, India

Date of Submission05-Aug-2019
Date of Acceptance17-Dec-2019
Date of Web Publication23-Jan-2020

Correspondence Address:
Dr. Santosh Kumar Swain
Department of Otorhinolaryngology, IMS and SUM Hospital, Siksha “O” Anusandhan (Deemed to be University), K8, Kalinganagar, Bhubaneswar - 751 003, Odisha
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/kleuhsj.kleuhsj_167_19

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  Abstract 


Plastic bronchitis (PB) is an extremely rare clinical entity where inspissated cast is found in endobronchial airway leading to respiratory distress. It is a large gelatinous or rigid branching airway cast. Removal of this endobronchial cast rapidly improves this condition. The underlying causes for this PB are pulmonary diseases, congenital heart disease, and few other conditions. The treatment of this condition includes rigid bronchoscopy and removal of this cast. Here, we present a case of PB in a previously healthy 6-year-old boy presenting with respiratory distress. This child was managed by rigid bronchoscopy and this cast was removed from the right bronchus.

Keywords: Mucous plug, pediatric patient, plastic bronchitis, respiratory distress


How to cite this article:
Swain SK, Choudhury J. Plastic bronchitis in a 6-year-old boy: An unusual indication for rigid bronchoscopy. Indian J Health Sci Biomed Res 2020;13:46-9

How to cite this URL:
Swain SK, Choudhury J. Plastic bronchitis in a 6-year-old boy: An unusual indication for rigid bronchoscopy. Indian J Health Sci Biomed Res [serial online] 2020 [cited 2020 Jun 1];13:46-9. Available from: http://www.ijournalhs.org/text.asp?2020/13/1/46/276418




  Introduction Top


Plastic bronchitis (PB) is a rare clinical entity of unknown etiology. It is characterized by the accumulation of fibrinous mucous plugs as a three-dimensional architecture in the bronchial tree. It often partially or completely blocks the airway and leads to respiratory distress.[1] Although the etiopathology of PB is not well understood, pediatric patients with the underlying cardiac diseases are prone to develop bronchial cases, particularly after surgical intervention, which direct systemic blood flow into the pulmonary circulation.[2] These endobronchial casts are more adhesive than mucous plugs and attend the shape of tracheobronchial tree.[3] The clinical presentations of the patients are cough, dyspnea, fever, and wheezing, which often confuse with inhaled foreign body in the bronchus. Medical treatment options for PB include inhaled or systemic corticosteroids, aerosolized acetylcysteine, aerosolized heparin and tissue plasminogen, which may be used to liquefy the PB and for its subsequent removal from the tracheobronchial airway. If this medical treatment fails, bronchoscopic intervention is required for better clearance of the airway.


  Case Report Top


A 6-year-old boy attended the emergency department with severe asthmatic attack but not responded to proper asthma medications. The child was shifted to the pediatric intensive care unit and on physical examination, he was in moderate respiratory distress with tachypnea, sternal retraction, and decreased breath sounds in the right upper and middle lobes. He was immediately treated with antibiotics, bronchodilators, steroids, and oxygen. He had also persistent dry cough and wheezing. Chest X-ray was done which showed no radiopaque foreign body in the tracheobronchial airway but showed haziness in the right bronchus, suggesting blockage at the area of the bronchus intermedius [Figure 1]. Then, the child was planned for rigid bronchoscopy and removal of foreign body from the right bronchus under general anesthesia. The rigid ventilating type of bronchoscope with venture connection was utilized for this procedure. During bronchoscopy, a tenacious black cast was seen in the right bronchus. A black-colored, soft branched-pattern, foreign body was removed [Figure 2]. This cast was removed by rigid bronchoscopy forceps along with suction. Postoperatively, the child symptomatically improved and showed no sign of respiratory distress; air entry in both sides was equal and normal in the chest. The child was discharged postoperatively on the 5th day. At the follow-up visit after 3 months of treatment, spirometry was done which showed normal respiratory function.
Figure 1: Chest X-ray (posteroanterior view) showing radiopaque cast in the right endobronchial airway

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Figure 2: Endobronchial cast after bronchoscopic removal

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  Discussion Top


PB is an uncommon, potential fatal clinical condition where extensive inspissated mucofibrinous bronchial casts mimick three-dimensional architecture of the tracheobronchial airway.[4] Its consistency is sticky and often difficult to remove by bronchoscopic procedure that differentiates this condition from ordinary mucus plug in the tracheobronchial airway.[5] PB can affect any age groups but it is more common among children.[4] Males and females are equally affected by PB, but one report shows female predominance.[6] The true prevalence of PB is still unknown. Certain conditions associated with PB are cyanotic congenital heart disease, bronchial asthma, lymphatic disorders, pulmonary infections, sickle cell anemia, and lymphoma. One study of pediatric PB revealed that 40% of patients had underlying cardiac defects, 31% had asthma or allergic disorders, and 29% had other disorders. They also documented that the overall mortality rate of PB is 16% and it is up to 29% in patients with cardiac defects. Similarly, life-threatening situations are much more in PB with cardiac defects.[7] The Seear classification of PB is divided into two categories, which is useful for diagnosis and appropriate adjunctive treatment. Type-I PB is described as hypercellular and usually associated with inflammatory mediators such as fibrin, neutrophils, and eosinophils. Inflammatory conditions of lungs such as asthma, acute chest syndrome, and allergic bronchopulmonary aspergillosis are included in Type-I PB. Type-II PB is described as a transudative, mucoid precipitate found with process, which increases central venous pressure as in congenital heart diseases and lymphatic leakage.[8] Type-II or acellular casts primarily consist of mucin with no acute inflammatory cells and sometimes a few mononuclear cellular components. These casts are usually seen in children with underlying cardiac problems. Recently, there is another classification of PB on the etiological basis. There are four etiological groups associated with this classification. Congenital heart disease is associated with acellular mucinous casts. Asthma and atopic disorders are associated with abundant eosinophils, Charcot–Leyden crystals, neutrophils, and fibrin. Lymphatic diseases have chylous casts and sometimes consist of fibrin. Sickle cell anemia is associated with casts which consist of fibrinous material and pigmented histiocytes in the surrounding fluid.[9] Once endobronchial casts are expectorated, it is more like cohesive and rubbery in consistency than ordinary mucus plug. The cast possesses varying proportions of mucin, fibrin, and cellular materials. The expectorate casts are often mistaken with different materials such as food materials such as noodles, chicken, and meat.

Patients usually present with mild symptoms of breathing difficulty, cough, to life-threatening condition such as death. The typical clinical presentations of the patient are dyspnea, wheezing, coughing, respiratory distress, and occasionally chest pain or fever.[10] Wheezing is the most common clinical finding in PB. The hallmark for PB is finding casts in bronchial tree during bronchoscopy. Imaging shows atelectasis and infiltrates on the affected side of the lungs and often hyperinflation in the contralateral side.[11] The expectorated bronchial cast varies in duration and frequency and sometimes may be complicated with hemoptysis. Some patients are unable to expectorate the bronchial cast spontaneously, which may delay or hide the diagnosis. Clinicians should intervene early if clinical presentations of airway obstruction are noticed. In severe cases of PB, substantial cast obstruction may lead to fatal outcome such as cardiorespiratory collapse.

Radiological studies reveal the site of the impactation of bronchial cast and atelectasis or infiltrates. Hyperinflations of lungs are often evident in the contralateral side of lungs. Computed tomography (CT) scan usually shows impacted casts in the bronchial airways. CT scan with axial view of the chest demonstrates the presence of atelectasis with blockage of endobronchial airway in the affected side. Diagnosis is confirmed by previously expectorated casts or bronchoscopy. Endobronchial casts often look whitish yellow and are often too thick in consistency to be suctioned out from the bronchus. In our case, the cast appeared blackish and soft in consistency. The lung biopsy specimen usually demonstrates mucin cell hyperplasia without any other pathological significance.

Treatment of PB includes removal of casts and addressing the underlying hypersecretory process. Immediate treatment of PB ranges from medical treatment with corticosteroids, different inhaled lytic agents, to bronchoscopy and potentially other surgical procedures. PB is usually removed by bronchial lavage by bronchoscopic approach, which is considered the standard treatment.[12] A mucolytic agent such as dornase alfa may be used to liquefy the dense and viscous PB.[13] Presently, 7% hypertonic saline solution which attracts water into thick and viscous mucous secretions has good outcome.[14] Rigid bronchoscopy shows tenacious secretions in the endobronchial airway. This cast can be removed by the bronchoscopy forceps. Premedication with injection atropine helps reduce airway secretions and prevent from bradycardia in response to airway manipulation by rigid bronchoscope.[15] Muscle relaxants are used for controlled ventilation which helps in easier removal of foreign body. Careful intermittent ventilation helps in proper oxygenation and prevents dislodgment of foreign body or cast further down in the bronchus.[16] In this case, rigid bronchoscopy procedure completely cleared the endobronchial airway and prevented from fatal airway complications. Every pediatrician or otolaryngologist must keep this disorder in mind.


  Conclusion Top


PB is an uncommon disease seen in the pediatric age group. If a pediatric patient presents with persistent cough even after adequate and prolonged treatment, a high suspect for this rare disease is mandatory. All pediatricians and otolaryngologists must keep this disorder in mind. Endoscopic evidence of PB in tracheobronchial airway is the gold standard diagnostic procedure. Bronchoscopy along with bronchial lavage is often considered the treatment of choice.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest



 
  References Top

1.
Walker PA, Shah SK, Letourneau PA, Allison ND, Cox CS. Treatment of plastic bronchitis using serial flexible bronchoscopy and aerosolized heparin therapy. Eur J Pediatr Surg 2013;23:157-60.  Back to cited text no. 1
    
2.
Ishman S, Book DT, Conley SF, Kerschner JE. Plastic bronchitis: an unusual bronchoscopic challenge associated with congenital heart disease repair. Int J Pediatr Otorhinolaryngol 2003;67:543-8.  Back to cited text no. 2
    
3.
Hasan RA, Black C, Reddy R. Plastic bronchitis in children. Fetal Pediatr Pathol 2012;31:87-93.  Back to cited text no. 3
    
4.
Brogan TV, Finn LS, Pyskaty Jr DJ, Redding GJ, Ricker D, Inglis A, et al. Plastic bronchitis in children: a case series and review of the medical literature. Pediatric pulmonology 2002 ;34:482-7.  Back to cited text no. 4
    
5.
Cajaiba MM, Borralho P, Reyes-Múgica M. The potentially lethal nature of bronchial casts: plastic bronchitis. Int J Surg Pathol 2008;16:230-2.  Back to cited text no. 5
    
6.
Eberlein MH, Drummond MB, Haponik EF. Plastic bronchitis: a management challenge. Am J Med Sci 2008;335:163-9.  Back to cited text no. 6
    
7.
Angelos PC, MacArthur CJ. Pediatric plastic bronchitis: A case report and literature review. Int J Pediatr Otorhinolaryngol Extra 2010;5:66-9.  Back to cited text no. 7
    
8.
Somers E, Day W, Derkay CS. Plastic bronchitis resulting in complete bronchial obstruction in an otherwise healthy child. Int J Pediatric Otorhinolaryngol Extra 2017;18:1-4.  Back to cited text no. 8
    
9.
Madsen P, Shah SA, Rubin BK. Plastic bronchitis: New insights and a classification scheme. Paediatr Respir Rev 2005;6:292-300.  Back to cited text no. 9
    
10.
Ugaki S, Lord DJ, Sherwood MC, Winlaw DS. Lymphangiography is a diagnostic and therapeutic intervention for patients with plastic bronchitis after the Fontan operation. J Thorac Cardiovasc Surg 2016;152:e47-9.  Back to cited text no. 10
    
11.
Barber BJ, Burch GH, Tripple D, Balaji S. Resolution of plastic bronchitis with atrial pacing in a patient with Fontan physiology. Pediatr Cardiol 2004;25:73-6.  Back to cited text no. 11
    
12.
Grizales CL, González LM, Fernández-Trujillo L. Plastic bronchitis: A case report. Resp Med Case Rep 2019;28:1-4..  Back to cited text no. 12
    
13.
Jones AP, Wallis C. Dornase alfa for cystic fibrosis. Cochrane Database of Systematic Reviews 2010(3):CD001127.  Back to cited text no. 13
    
14.
Donaldson SH, Bennett WD, Zeman KL, Knowles MR, Tarran R, Boucher RC. Mucus clearance and lung function in cystic fibrosis with hypertonic saline. N Engl J Med 2006;354:241-50.  Back to cited text no. 14
    
15.
Swain SK, Mohanty S, Sahu MC, Behera IC. Safe and effective anaesthesia during paediatric rigid bronchoscopy: An experience at a tertiary care centre of Eastern India. Pediatria Polska 2015;90:470-3.  Back to cited text no. 15
    
16.
Swain SK, Mishra S, Dash M. An unusual asymptomatic foreign body at tracheobronchial tree. Int J Phonosurg Laryngol 2014;4:40-2.  Back to cited text no. 16
    


    Figures

  [Figure 1], [Figure 2]



 

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