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      A Rare Case of Idiopathic Plastic Bronchitis

      case-report

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          Abstract

          Plastic bronchitis is a rare disorder characterized by formation of large, branching bronchial casts, which are often expectorated. We present an interesting case of a 35-year-old woman who presented for evaluation of a chronic cough productive of voluminous secretions. Clinical and radiological examination confirmed a total left lung atelectasis without any pathological mediastinal node. Flexible bronchoscopy demonstrated tenacious, thick, and sticky whitish secretions blocking the left stem bronchus. This material was extracted, and inspection demonstrated a bronchial cast, whose pathological analysis revealed necrotic epithelial cells, some eosinophils, and Charcot-Leyden crystals. Two days after bronchoscopy, the patient rejected more bronchial casts, and dyspnea improved. Control of chest x-ray revealed complete left lung aeration and the diagnosis of idiopathic plastic bronchitis was obtained. This article shows the interest in clinical practice to evoke the diagnosis of plastic bronchitis in front of a productive chronic cough. Our case illustrates a rare clinical presentation represented by an atelectasis of an entire lung.

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          Most cited references6

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          Plastic bronchitis: a management challenge.

          Plastic bronchitis is an uncommon and underdiagnosed entity, characterized by recurrent expectoration of large, branching bronchial casts. We describe a 39-year-woman with no prior lung disease who had episodic wheezing, severe dyspnea with expectoration of large and thick secretions, branching in appearance, which she described as resembling squid. A comprehensive evaluation revealed no specific cause and a diagnosis of idiopathic plastic bronchitis was made. In plastic bronchitis the bronchial casts may vary in size from small segmental casts of a bronchus to casts filling the airways of an entire lung. Plastic bronchitis can therefore present as an acute life-threatening emergency if mechanical obstruction of major airways occurs. The casts are differentiated into type I, inflammatory casts, or type II, acellular casts. The type I inflammatory casts are often associated with bronchial disease and often have an acute presentation. The acellular type of cast production is often chronic or recurrent. Numerous systemic illnesses are associated with plastic bronchitis, but often, as in our patient, no underlying cause can be identified. The treatment of plastic bronchitis includes acute therapy to aid the removal and expectoration of casts, and specific short- or long-term treatments attempting to address the underlying hypersecretory process. The therapeutic options are supported only by anecdotal evidence based on case reports as the rarity and heterogeneity of plastic bronchitis confounds systematic investigations of its treatment. Improved understanding of the regulation of mucus production may allow for new treatment options in plastic bronchitis and other chronic lung diseases characterized by hypersecretion of mucus.
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            Peripheral mucociliary clearance with high-frequency chest wall compression.

            We investigated the effects of high-frequency chest wall compression (HFCWC) on peripheral and tracheal mucus clearance in anesthetized spontaneously breathing dogs. HFCWC was achieved by oscillating the pressure in a thoracic cuff with a piston pump. Regional lung retention of a technetium-99m sulfur colloid aerosol was monitored with a gamma camera. A peripheral mucus clearance index (PMCI) was defined for each region of interest. The tracheal mucus clearance rate (TMCR) was determined by bronchoscopic visualization of marker particle transport. Phase I: In seven dogs, 30 min of HFCWC at 13 Hz with peak cuff pressure (Pcuff) 100-120 cmH2O was found to significantly enhance PMCI in regions immediately under the cuff. (delta PMCI = 24.4 +/- 4.6 in the basal peripheral region.) Phase II: Because of subpleural hemorrhage in phase I, the effect of HFCWC on TMCR at various Pcuff levels was studied in five dogs. The enhancement of TMCR by HFCWC reached a plateau level at Pcuff = 50 cmH2O. Phase III: HFCWC at 13 Hz with Pcuff = 50-60 cmH2O was found to significantly enhance PMCI in five dogs without the consequence of hemorrhage. Correlations were found between the enhancement of PMCI and TMCR by HFCWC. These results demonstrate that HFCWC is effective in enhancing both peripheral and central mucus clearance in dogs and safe when moderate pressures are applied.
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              Plastic Bronchitis in an Adult with Asthma

              Plastic bronchitis is a rare disease characterized by marked airway obstruction, via the formation of large gelatinous or rigid airway cast. In Korea, there were a few case reports with plastic bronchitis not in adults, but in children. So we report a case of an adult who was diagnosed as plastic bronchitis with eosinophilic casts, with no history of atopic and cardiac disease.
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                Author and article information

                Contributors
                Journal
                Case Rep Pulmonol
                Case Rep Pulmonol
                CRIPU
                Case Reports in Pulmonology
                Hindawi
                2090-6846
                2090-6854
                2017
                2 October 2017
                : 2017
                : 4789751
                Affiliations
                1Avicenne University Hospital, Pulmonary Unit, University of Rabat, Rabat, Morocco
                2Avicenne University Hospital, Anatomopathology Unit, University of Rabat, Rabat, Morocco
                3Avicenne University Hospital, Radiology Unit, University of Rabat, Rabat, Morocco
                Author notes

                Academic Editor: Akif Turna

                Author information
                http://orcid.org/0000-0003-0803-307X
                Article
                10.1155/2017/4789751
                5643106
                29098106
                c2e6c58d-b098-4263-874f-50fbc60ddacc
                Copyright © 2017 Mohammed Raoufi et al.

                This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                : 13 June 2017
                : 16 August 2017
                : 24 August 2017
                Categories
                Case Report

                Respiratory medicine
                Respiratory medicine

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