Efficacy of Fiberoptic Bronchoscopy In Non-Cardiac Pediatric Plastic Bronchitis: A Case Series

Efficacy of Fiberoptic Bronchoscopy In Non-Cardiac Pediatric Plastic Bronchitis: A Case Series | Authorea try { document.documentElement.classList.add('js'); } catch (e) { } var _gaq = _gaq || []; _gaq.push(['_setAccount', 'G-8VDV14Y67G']); _gaq.push(['_trackPageview']); (function() { var ga = document.createElement('script'); ga.type = 'text/javascript'; ga.async = true; ga.src = ('https:' == document.location.protocol ? 'https://ssl' : 'http://www') + '.google-analytics.com/ga.js'; var s = document.getElementsByTagName('script')[0]; s.parentNode.insertBefore(ga, s); })(); Skip to main content Preprints Collections Wiley Open Research IET Open Research Ecological Society of Japan All Collections About About Authorea FAQs Contact Us Quick Search anywhere Search for preprint articles, keywords, etc. Search Search ADVANCED SEARCH SCROLL This is a preprint and has not been peer reviewed. Data may be preliminary. 22 January 2025 V1 Latest version Share on Efficacy of Fiberoptic Bronchoscopy In Non-Cardiac Pediatric Plastic Bronchitis: A Case Series Authors : NNagarjun 0000-0003-3494-4870 [email protected] , K.R.Bharath Kumar Reddy , and Kavya C Authors Info & Affiliations https://doi.org/10.22541/au.173751998.82375188/v1 211 views 131 downloads Contents Abstract Information & Authors Metrics & Citations View Options References Figures Tables Media Share Abstract Plastic bronchitis (PB) is a rare condition characterized by the formation and expectoration of cohesive endobronchial casts that mimic the shape of local airways. It is often associated with cardiothoracic surgeries and various respiratory diseases, but pediatric cases without congenital heart disease remain poorly understood. This case series reports ten pediatric patients with non-cardiac PB, successfully managed through flexible bronchoscopy. The cohort included children aged 2-12 years, with a gender distribution of 50% boys and 50% girls. Underlying conditions included asthma, allergic histories, and primary ciliary dyskinesia. Infectious causes, including Influenza A and adenovirus, were identified in three cases. Bronchial casts were extracted with flexible bronchoscopy, using a suction channel, and one patient required a repeat bronchoscopy after the installation of dornase alfa for adherent casts. Histopathological examination revealed eosinophilic casts in the majority of patients. Treatment involved inhaled corticosteroids, with some patients receiving systemic steroids. No recurrences of PB were observed during follow-up, and all patients showed resolution of symptoms. This case series highlights the successful management of non-cardiac PB in children, emphasizing the role of bronchoscopy and tailored therapies. Further research is needed to explore optimal treatment strategies and the underlying pathophysiology of PB. ORIGINAL ARTICLE Efficacy of Fiberoptic Bronchoscopy In Non-Cardiac Pediatric Plastic Bronchitis: A Case Series Authors: Dr Nagarjun.N, Dr K.R. Bharath Kumar Reddy, Dr Kavya C Affiliations: Department of Pediatric Pulmonology and Sleep, Shishuka Childrens Hospital Informed Consent was obtained from all participants. Institutional Ethical Committee clearance was waived off for the study as it was only a descriptive analysis of findings, hence the exemption was provided. Abstract: Plastic bronchitis (PB) is a rare condition characterized by the formation and expectoration of cohesive endobronchial casts that mimic the shape of local airways. It is often associated with cardiothoracic surgeries and various respiratory diseases, but pediatric cases without congenital heart disease remain poorly understood. This case series reports ten pediatric patients with non-cardiac PB, successfully managed through flexible bronchoscopy. The cohort included children aged 2-12 years, with a gender distribution of 50% boys and 50% girls. Underlying conditions included asthma, allergic histories, and primary ciliary dyskinesia. Infectious causes, including Influenza A and adenovirus, were identified in three cases. Bronchial casts were extracted with flexible bronchoscopy, using a suction channel, and one patient required a repeat bronchoscopy after the installation of dornase alfa for adherent casts. Histopathological examination revealed eosinophilic casts in the majority of patients. Treatment involved inhaled corticosteroids, with some patients receiving systemic steroids. No recurrences of PB were observed during follow-up, and all patients showed resolution of symptoms. This case series highlights the successful management of non-cardiac PB in children, emphasizing the role of bronchoscopy and tailored therapies. Further research is needed to explore optimal treatment strategies and the underlying pathophysiology of PB. Introduction: Plastic bronchitis (PB), also referred to as cast bronchitis, fibrinous bronchitis, or Hoffman’s bronchitis, is a rare but intriguing condition first recognized several centuries ago. It is characterized by the formation and expectoration of cohesive endobronchial casts that replicate the shape of the local airways. The size of these casts can range from small, segmental pieces to large, tree-like structures capable of obstructing an entire lung. (1) In children, PB has been associated with a variety of underlying conditions, including cardiothoracic surgeries such as the Fontan procedure, infectious or inflammatory diseases, acute chest syndrome, and iatrogenic causes. (2) Despite its diverse etiologies, the condition remains challenging to manage due to its variable presentation and potential for significant airway obstruction. The cornerstone of PB management involves prompt removal of bronchial casts, with treatment strategies tailored to the underlying etiology. Therapeutic bronchoscopy, both flexible and rigid, plays a pivotal role in this process, complemented by adjunct therapies like corticosteroids, mucolytics, and immunomodulators based on the cast composition. In this report, we present a case series of pediatric plastic bronchitis not associated with congenital heart disease, successfully managed exclusively by flexible bronchoscopy. Our findings provide insights into the diagnostic approach, pathological characteristics, and tailored treatment strategies for non-cardiac PB in children. Case series: Ten children (n=10) with plastic bronchitis were included in our study. Demographic data, underlying diseases, and clinical characteristics are listed in Table 1. The cohort consisted of an equal number of boys and girls. The average age was 5.33 ± 1.73 years, with an age range of 2–12 years. Six patients (60%) had clearly defined asthma or allergic histories, two children (20%) had primary ciliary dyskinesia, and three patients (20%) developed bronchial casts without any underlying illness. None of the patients in the study had underlying cardiac disease or a history of cardiac surgery. The majority of children presented with symptoms of cough (100%), wheeze (80%), and respiratory distress (40%). An infectious cause was identified in three children (30%); two tested positive for Influenza A, while one tested positive for adenovirus. CT Thorax and 2D Echocardiography were performed on all children. Findings are detailed in Table 2. The right middle lobe was the most commonly affected (50%), followed by the left lower lobe (20%) and the right upper lobe (20%). 2D Echocardiography results were normal for all subjects (100%). All children (100%) underwent flexible bronchoscopy performed by a trained pediatric pulmonologist under general anesthesia. Two flexible bronchoscopes were used: 2.8 mm (80%) and 3.4 mm (20%), both equipped with a 1.2 mm suction channel. No additional instruments (e.g., forceps, basket, cryoprobe) were utilized for cast extraction. Casts were successfully extracted in a single attempt in all cases except one, where adherent casts required instillation of dornase alfa and a repeat bronchoscopy 24 hours later. (Table 2) Histopathological examination of casts was performed for nine children (90%), as shown in Table 3. Most of the casts were eosinophil-predominant (60%). Post-bronchoscopy, all children (100%) were initiated on inhaled steroids, and three (30%) received systemic steroids. Of the latter, two (66.6%) received pulse methylprednisolone (10 mg/kg/day), while one received oral steroids over six weeks. One patient, who underwent two bronchoscopies and dornase alfa instillation, was lost to follow-up after three months of pulse therapy. (Table 3) Notably, none of the patients experienced a recurrence of cast formation. At follow-up, all patients showed resolution of symptoms, underscoring the efficacy of the management protocol adopted in this study. Table 1: Patient demographics and presentation \tightlist 6.5 M Moderate Persistent Asthma Expectoration of Casts \tightlist 2.2 F Asthma with RML syndrome Persistent wheeze \tightlist 11 M Primary Ciliary Dyskinesia and Asthma Poorly controlled asthma \tightlist 2.8 F None Suspected Foreign body aspiration \tightlist 7 M Moderate persistent asthma Persistent cough \tightlist 3 F Under 5 wheeze- high probability of Asthma Acute Exacerbation of Asthma (Influenza A) \tightlist 3.3 M Primary Ciliary Dyskinesia and Asthma Persistent wheeze \tightlist 9 F Post-operative TEF and Asthma Persistent pneumonia \tightlist 4 M None Respiratory distress (Influenza A) \tightlist 4.5 F None Respiratory distress (Adenovirus) M: Male, F: Female, RML: Right middle lobe, TEF: Tracheo-esophageal fistula Table 2: Investigations and Management \tightlist Left Lower Lobe Collapse Normal, No PAH Flexible 2.8 Single No \tightlist Right Middle Lobe Collapse Normal, No PAH Flexible 2.8 Single No \tightlist Right Middle Lobe Collapse Normal, No PAH Flexible 3.4 Single No \tightlist Complete obstruction of right upper lobar bronchus Normal, No PAH Flexible 2.8 Single No \tightlist Right Middle Lobe Collapse Normal, No PAH Flexible 2.8 Single No \tightlist Left Lower Lobe Collapse Normal, No PAH Flexible 2.8 Single No \tightlist Right Middle Lobe Collapse Normal, No PAH Flexible 2.8 Single No \tightlist Right Lower Lobe Collapse Normal, No PAH Flexible 2.8 Single No \tightlist Right Upper Lobe Collapse Normal, No PAH Flexible 3.4 Double No \tightlist Right Middle Lobe Collapse Normal, No PAH Flexible 3.4 Single No PAH: Pulmonary artery hypertension Table 3: Cast analysis and Post Bronchoscopy management \tightlist Eosinophilic, CL crystals Nil Yes 6 months No - - \tightlist Lymphocytic Nil Yes 6 months No - - \tightlist Neutrophilic Nil Yes 6 months No - Azithromycin \tightlist Not available Nil Yes 12 months Yes 12 months \tightlist \tightlist Eosinophilic, CL crystals Nil Yes 6 months Yes 6 weeks - \tightlist Neutrophilic Nil Yes 6 months No \tightlist - \tightlist Eosinophilic Nil Yes 6 months No - - \tightlist Eosinophilic Nil Yes 6 months No - - \tightlist Eosinophilic Nil Yes 3 months Yes 3 months Dornase Alfa \tightlist Eosinophilic Nil Yes 3 months No \tightlist \tightlist CL crystals: Charcot-Leyden crystals Figure 1 showing eosinophilic cells and Charcot-leyden crystals on H&E staining Discussion: Plastic bronchitis (PB) is primarily diagnosed by identifying bronchial casts, either through bronchoscopy or in expectorated material. Seear’s classification, which divides bronchial casts into two types, remains widely used. (3) Type 1, the inflammatory type, is predominantly associated with respiratory diseases, while Type 2, the acellular type, is primarily linked to congenital heart disease and abnormal lymphatic reflux. With advancements in lymphatic imaging, PB can now be more specifically categorized as either lymphatic or eosinophilic. (4) Notably, eosinophilic PB differs from the lymphatic PB typically seen in congenital heart disease and lymphatic disorders. The etiology of PB is multifactorial. It is most commonly associated with congenital heart disease, particularly in patients with Fontan physiology, as well as pulmonary lymphatic abnormalities. Other associated conditions include sickle cell acute chest syndrome, respiratory infections, poorly controlled asthma, and allergic bronchopulmonary aspergillosis. (2) However, our case series exclusively included casts secondary to respiratory diseases. Infections and asthma are reported as the most common underlying causes in case reports and series.(5,6,7) Infectious agents linked to PB include Mycoplasma, Influenza, Adenovirus, and Boca virus.(5,8) In our series, Influenza and Adenovirus were identified as causative pathogens. PB can occur across all age groups but is more common in children aged 2–12 years, consistent with the demographic of our series.(9) The clinical presentation is variable, with cough being the most common symptom, followed by dyspnea, wheezing, chest pain, and fever. (10) Radiologically, atelectasis due to major airway obstruction is the most frequently observed finding on chest X-rays or computed tomography, with no specific lobe predilection in our series.(7) The primary mode of treatment for PB is the removal of bronchial casts through bronchoscopy, either flexible or rigid.(10,11,12) In our series, all casts were successfully extracted using flexible bronchoscopy. Although studies have described the use of tools such as baskets, forceps, and cryoprobes for cast removal, none were required in our cases.(6,13) In one child who tested positive for Influenza A, the casts were adherent to the bronchial walls, necessitating the instillation of mucolytic,dornase alfa and a repeat bronchoscopy 24 hours later, which was successful. Inhaled mucolytics including acetylcysteine and dornase alpha are commonly used in patients with plastic bronchitis. (14) Our findings are consistent with existing literature on plastic bronchitis. The pathological analysis of bronchial casts in our series revealed that the majority were eosinophilic, with Charcot-Leyden crystals (Figure 1) serving as a diagnostic hallmark.(1) This aligns with reports that eosinophilic casts are commonly associated with underlying respiratory conditions such as asthma. Other components identified in the casts, including neutrophils, lymphocytes, and mucin, further support the inflammatory nature of the disease. Treatment in our series was tailored to the underlying diagnosis and cast composition, a practice consistent with previous case reports and series.(1,2,7,10,11) Patients with eosinophilic casts were managed with inhaled corticosteroids (ICS) and systemic steroids, either intravenous or oral, similar to standard approaches described in the literature. For patients with neutrophilic casts, azithromycin was chosen in combination with ICS. Azithromycin was specifically selected for its immunomodulatory properties, which are known to help reduce neutrophilic inflammation, particularly in conditions such as asthma.(15) Follow-up data also align with previous observations. (16) All patients in our series were followed for up to six months, with no recurrence of cast formation, except for one patient lost to follow-up. This outcome is consistent with studies suggesting that addressing underlying conditions and implementing appropriate treatment strategies reduce the risk of recurrence.(11) The absence of significant risk factors for recurrence in our cohort further explains the favorable outcomes observed. (17) Our case series highlights the diverse presentations, underlying etiologies, and successful treatment strategies associated with PB in pediatric patients. Consistent with existing literature, our findings underscore the predominance of eosinophilic bronchial casts in respiratory-related PB and the efficacy of tailored treatments, including corticosteroids and azithromycin for their respective immunomodulatory properties. The importance of early bronchoscopy for both diagnostic and therapeutic purposes cannot be overstated, as evidenced by the successful cast removal in all our cases. Moreover, the use of mucolytics such as dornase alfa in cases of adherent casts demonstrates its utility as an adjunct in managing challenging cases. The absence of cast recurrence and the resolution of symptoms in our series further emphasize the effectiveness of individualized treatment and follow-up care. Conclusion: This case series reinforces the critical role of comprehensive clinical evaluation, bronchoscopy, and pathology in diagnosing and managing PB. By addressing underlying etiologies and utilizing evidence-based therapies, the risk of recurrence can be minimized, leading to favorable outcomes. Future studies with larger cohorts and longer follow-up periods are needed to further elucidate the pathophysiology, optimal treatments, and prognostic factors associated with PB. References: 1. Gipsman AI, Feld L, Johnson B, Needleman JP, Boas H, Lin N, DePasquale B, Pogoriler J, McDowell KM, Piccione JC. Eosinophilic plastic bronchitis: Case series and review of the literature. Pediatr Pulmonol. 2023 Nov;58(11):3023-3031. doi: 10.1002/ppul.26650. Epub 2023 Aug 22. PMID: 37606213; PMCID: PMC10928548. 2. Patel N, Patel M, Inja R, Krvavac A, Lechner AJ. Plastic Bronchitis in Adult and Pediatric Patients: A Review of its Presentation, Diagnosis, and Treatment. Mo Med. 2021 Jul-Aug;118(4):363-373. PMID: 34373673; PMCID: PMC8343636.) 3. Seear M, Hui H, Magee F, Bohn D, Cutz E. Bronchial casts in children: a proposed classification based on nine cases and a review of the literature. Am J Respir Crit Care Med. 1997;155(1):364–370 4. Madsen P, Shah S, Rubin B. Plastic bronchitis: new insights and a classification scheme. Paediatr Respir Rev. 2005;6(4):292–300. 5. Huang F, Gu W, Diwu J, Zhang X, He Y, Zhang Y, Chen Z, Huang L, Wang M, Dong H, Wang S, Wang Y, Zhu C, Hao C. Etiology and clinical features of infection-associated plastic bronchitis in children. BMC Infect Dis. 2023 Sep 7;23(1):588. doi: 10.1186/s12879-023-08529-w. PMID: 37679703; PMCID: PMC10486060. 6. Kallam EF, Kasi AS, Patki R, Silva GL, Simon DM, Caltharp S, Guglani L. Bronchoscopic interventions for plastic bronchitis in children without structural heart disease. Eur J Pediatr. 2021 Dec;180(12):3547-3554. doi: 10.1007/s00431-021-04161-5. Epub 2021 Jun 23. PMID: 34159443. 7. Nayır Büyükşahin H, Emiralioglu N, Sekerel BE, Soyer T, Oguz B, Güzelkaş I, Sunman B, Alboğa D, Akgül Erdal M, Yalcın E, Doğru D, Ozcelik U, Kiper N. Plastic bronchitis during childhood: Diversity of presentation, etiology, treatment, and outcomes. Pediatr Pulmonol. 2023 Sep;58(9):2559-2567. doi: 10.1002/ppul.26548. Epub 2023 Jun 6. PMID: 37278540. 8. Zhang FZ, Qin L, Yuan JX, Tang LF. Plastic bronchitis due to adenoviral infection: a case report. BMC Pediatr. 2020 Feb 10;20(1):61. doi: 10.1186/s12887-020-1954-0. PMID: 32039717; PMCID: PMC7008568. 9. Zhang Guangli, Luo Zhengxiu. Clinical features and differential diagnosis of plastic bronchitis in children[J]. Chinese Journal of Practical Pediatrics, 2021, 36(4): 248-250. DOI: 10.3760/cma.j.cn101070-20210113-00053 10. Li Y, Williams RJ, Dombrowski ND, Watters K, Daly KP, Irace AL, Visner GA, Rahbar R, Fynn-Thompson F. Current evaluation and management of plastic bronchitis in the pediatric population. Int J Pediatr Otorhinolaryngol. 2020 Mar;130:109799. doi: 10.1016/j.ijporl.2019.109799. Epub 2019 Nov 29. PMID: 31812839; PMCID: PMC9187852. 11. Wang L, Wang W, Sun JM, Ni SW, Ding JL, Zhu YL, Ding SG. Efficacy of fiberoptic bronchoscopy and bronchoalveolar lavage in childhood-onset, complicated plastic bronchitis. Pediatr Pulmonol. 2020 Nov;55(11):3088-3095. doi: 10.1002/ppul.25016. Epub 2020 Sep 24. PMID: 32770770. 12. Soyer T, Yalcin Ş, Emiralioğlu N, Yilmaz EA, Soyer O, Orhan D, Doğru D, Sekerel BE, Tanyel FC. Use of serial rigid bronchoscopy in the treatment of plastic bronchitis in children. J Pediatr Surg. 2016 Oct;51(10):1640-3. doi: 10.1016/j.jpedsurg.2016.03.017. Epub 2016 Apr 12. PMID: 27129763. 13. Moslehi MA. Foreign body retrieval by using flexible cryoprobe in children. J Bronchol Interventional Pulmonol. 2021;28(2):103–106 14. Manna SS, Shaw J, Tibby SM, et alTreatment of plastic bronchitis in acute chest syndrome of sickle cell disease with intratracheal rhDNaseArchives of Disease in Childhood 2003;88:626-627. 15. Schultz, K.D. and Oermann, C.M. (2003), Treatment of cast bronchitis with low-dose oral azithromycin. Pediatr. Pulmonol., 35: 139-143. https://doi.org/10.1002/ppul.10196 16. Yazan H, Girit S, Kut A, Calim M, Çakır FB, Nursoy MA, Çollak A, Çakır E. Clinical and Radiological Evaluation and Follow-Up of Patients with Noncardiac Plastic Bronchitis. Turk Arch Pediatr. 2023 Sep;58(5):515-518. doi: 10.5152/TurkArchPediatr.2023.23052. PMID: 37670550; PMCID: PMC10544299. 17. Clinical characteristics of plastic bronchitis and risk factors for recurrence in children. Zhongguo Dang Dai Er Ke Za Zhi. 2023 Jun 15;25(6):626-632. Chinese. doi: 10.7499/j.issn.1008-8830.2211122. PMID: 37382133; PMCID: PMC10321426. Informed Consent was obtained from all participants. Institutional Ethical Committee clearance was waived off for the study as it was only a descriptive analysis of findings, hence the exemption was provided. Information & Authors Information Version history V1 Version 1 22 January 2025 Copyright This work is licensed under a Non Exclusive No Reuse License. Keywords asthma eosinophilic flexible bronchoscopy non-cardiac plastic bronchitis Authors Affiliations NNagarjun 0000-0003-3494-4870 [email protected] Bangalore Hospital View all articles by this author K.R.Bharath Kumar Reddy Bangalore Hospital View all articles by this author Kavya C Bangalore Hospital View all articles by this author Metrics & Citations Metrics Article Usage 211 views 131 downloads .FvxKWukQNSOunydq8rnd { width: 100px; } Citations Download citation NNagarjun, K.R.Bharath Kumar Reddy, Kavya C. Efficacy of Fiberoptic Bronchoscopy In Non-Cardiac Pediatric Plastic Bronchitis: A Case Series. Authorea . 22 January 2025. DOI: https://doi.org/10.22541/au.173751998.82375188/v1 If you have the appropriate software installed, you can download article citation data to the citation manager of your choice. Simply select your manager software from the list below and click Download. For more information or tips please see 'Downloading to a citation manager' in the Help menu . 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