Yonsei Med J.  2009 Oct;50(5):721-724. 10.3349/ymj.2009.50.5.721.

Unusual Combination of Tracheobronchopathia Osteochondroplastica and AA Amyloidosis

• Klrbas G
• Dagll CE
• Tanrlkulu
• Ylldlz F
• Bukte Y
• Senyigit A
• Klyan E

• KMID: 1103830
• DOI: http://doi.org/10.3349/ymj.2009.50.5.721

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.

Keyword

Tracheobronchopathia osteochondroplastica; AA amyloidosis

MeSH Terms

Amyloidosis/complications/*diagnosis/pathology
Asthma/diagnosis
Bronchial Diseases/complications/*diagnosis/radiography
Diagnosis, Differential
Humans
Male
Middle Aged
Tracheal Diseases/complications/*diagnosis/radiography

Figure

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  Fig. 1 Computed tomography reveals irregular thickening of the tracheal wall with sparing of the membranous posterior wall and deformed tracheal cartilage rings without the evidence of external compression or submucosal calcification.

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  Fig. 2 Flexible bronchoscopic results show nodular lesions that arise from the anterior and lateral aspects of tracheal wall narrowing its lumen. The posterior tracheal wall was unaffected.

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  Fig. 3 Homogenous proteinous material with calcification under bronchus epithelium (hematoxylin-eosin ×100).

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  Fig. 4 Proteinous material was found to be positive with congo red stain (histochemistry ×200).

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  Fig. 5 Amyloid deposition shows green birefringence under polarized microscopy (histochemistry ×200).

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