Unusual Combination of Tracheobronchopathia Osteochondroplastica and AA Amyloidosis

Department of Chest Diseases, Faculty of Medicine, University of Dicle, Diyarbakir, Turkey.
Yonsei medical journal (Impact Factor: 1.29). 10/2009; 50(5):721-4. DOI: 10.3349/ymj.2009.50.5.721
Source: PubMed

ABSTRACT Tracheobronchopathia osteochondroplastica (TO) is a rare disorder of unknown cause characterized by the presence of multiple submucosal osseous and/or cartilaginous nodules that protrude into the lumen of the trachea and large bronchi. A simultaneous diagnosis of TO and amyloidosis is rarely reported. In this report, a case initially suspected to be asthma bronchiole that could not be treated, was radiologically diagnosed as TO, and also secondary amyloidosis is presented. A 53 years, man patient reported a 3 years history of dyspnea. Pulmonary function tests (PFTs) showed an obstructive pattern. Chest X-rays revealed right middle lobe atelectasis. FOB and CT detected nodular lesions in the trachea and in the anterior and lateral walls of the main bronchi. AA amyloidosis was confirmed by endobronchial biopsy. In the abdominal fat pad biopsy, amyloidosis was not detected. Asthma bronchiole was excluded by PFTs. This case illustrates that it is possible for TO and amyloidosis to masquerade as asthma. TO and amyloidosis should be suspected in patients of older ages with asthma and especially with poorly treated asthmatic patients. Although nodular lesions in the anterior and lateral tracheobronchial walls are typical for TO, a biopsy should be obtained to exclude amyloidosis.

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    • "Biopsy of TO lesions sometimes can be a challenge due to the hard consistency of the bone nodules [26]. It was reported that histopathologic examination of TO nodules were usually unnecessary [40]. Nevertheless, because of unfamiliarity for many bronchoscopists with this condition and the need to exclude differential diagnoses, the histopathologic confirmation should be essential and reasonable in most cases [37]. "
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    ABSTRACT: Background and Study Aims Tracheobronchopathia osteochondroplastica (TO) is an uncommon disease of the tracheobronchial system that leads to narrowing of the airway lumen from cartilaginous and/or osseous submucosal nodules. The aim of this study is to perform a detailed review of this rare disease in a large cohort of patients with TO proven by fiberoptic bronchoscopy from China. Patients and Methods Retrospective chart review was performed on 41,600 patients who underwent bronchoscopy in the Department of Respiratory Medicine of Changhai Hospital between January 2005 and December 2012. Cases of TO were identified based on characteristic features during bronchoscopic examination. Results 22 cases of bronchoscopic TO were identified. Among whom one-half were male and the mean age was 47.45±10.91 years old. The most frequent symptoms at presentation were chronic cough (n = 14) and increased sputum production (n = 10). Radiographic abnormalities were observed in 3/18 patients and findings on computed tomography consistent with TO such as beaded intraluminal calcifications and/or increased luminal thickenings were observed in 18/22 patients. Patients were classified into the following categories based on the severity of bronchoscopic findings: Stage I (n = 2), Stage II (n = 6) and Stage III (n = 14). The result that bronchoscopic improvement was observed in 2 patients administered with inhaled corticosteroids suggested that resolution of this disease is possible. Conclusions TO is a benign disease with slow progression, which could be roughly divided into 3 stages on the basis of the characteristic endoscopic features and histopathologic findings. Chronic inflammation was thought to be more important than the other existing plausible hypotheses in the course of TO. Inhaled corticosteroids might have some impact on patients at Stage I/II.
    PLoS ONE 07/2014; 9(7):e102068. DOI:10.1371/journal.pone.0102068 · 3.23 Impact Factor
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    ABSTRACT: Objective: To report an atypical presentation of tracheobronchopathia osteochondroplastica (TO). Clinical presentation and intervention: A 59-year-old man was investigated for productive cough of 1 month. An antimycobacterial combination regime was initiated with a misdiagnosis of endobronchial tuberculosis. At follow-up, the patient reported worsening of his symptoms. CT revealed an increased intensity of the cartilage ring surrounding the trachea, and bronchoscopy showed tracheal stenosis with white, hard nodules on the airway submucosa. Histopathology confirmed the diagnosis of TO. Conclusion: This case showed that TO should be considered in patients with cough not explained by noninvasive testing and not responsive to empiric medications.
    Medical Principles and Practice 02/2013; 22(5). DOI:10.1159/000346662 · 1.34 Impact Factor
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    ABSTRACT: Tracheobronchopathia osteochondroplastica (TBPOCP) is an uncommon benign condition affecting the lumen of tracheobronchial tree and characterized by abnormal chondrification and ossification. TBPOCP is more frequent than it has been reported, as it can be asymptomatic or present with nonspecific respiratory symptoms. In this article we provide a review of the English literature on the condition and discuss its clinical features, general principles, diagnostic approaches, and current treatment recommendations for TBPOCP.
    The Clinical Respiratory Journal 05/2014; DOI:10.1111/crj.12166 · 1.51 Impact Factor

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