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01.12.2016 | Case report | Ausgabe 1/2016 Open Access

Multidisciplinary Respiratory Medicine 1/2016

Tracheobronchopathia Osteochondroplastica: a rare case report of a non-smoker and non-atopic patient, with a long history of wheezing since childhood

Zeitschrift:
Multidisciplinary Respiratory Medicine > Ausgabe 1/2016
Autoren:
Alessandro G. Fois, Antonella Arcadu, Luigi Santoru, Rocco Trisolini, Vincenzo Marras, Giorgio C. Ginesu, Sara Canu, Lorenzo Cordero, Gabriella Diana, Pietro Pirina
Wichtige Hinweise

Competing interests

All authors declare that they have no competing interests.

Authors’ contributions

AGF, AA, and PP contributed to draft the study, write the manuscript and revise editing. AA, SC and GD contributed to the acquisition of patient data and figures. AGF, LS and RT performed fibrobroncoscopy and bronchial biopsies, and participated in the patient follow up. VM carried out the histological analysis, achieved the histological figures, discussed the patient biopsies and the histological aspect of TBPO. All authors gave a substantial contribution in the draft of this manuscript, in the acquisition of data and/or in the data interpretation, in the literature search and discussion, and in revising it critically. The final version of the manuscript was read and approved by all the authors.

Abstract

Background

Tracheobronchopathia Osteochondroplastica (TBPO) is an uncommon and benign condition characterized by osseous or metaplastic cartilaginous 1–3 mm nodules in the submucosa of the tracheo-bronchial tree. Posterior membranous wall of trachea is typically spared. Ecchondrosis and exostosis nodules can cause chronic inflammation and mucosal metaplasia, stiffness and airway obstruction. The prevalence of this disease, often asymptomatic or associated with nonspecific symptoms, is underestimated, and the mean age at diagnosis is 50 years.

Case presentation

We report a case of a 49 year old male, non-smoker. He was a smith, homeless, born in Romania and reported a diagnosis of asthma since childhood. He was admitted to our Respiratory Unit presenting low-grade fever with profuse sweating, cough, purulent sputum, and ground-glass opacity with irregularity in main bronchi detected by High-Resolution Computed Tomography (HRCT) scan.
Fibrobronchoscopy revealed the presence of mucosal irregularities up to the segmental bronchi entrance. Histological examination showed nodules of osseouscartilaginous nature, consistent with TBPO. Microbiological tests of Bronchoalveolar Lavage fluid also revealed an infection by Pseudomonas Aeruginosa.

Conclusion

TBPO is a rare disease characterized by wheezing, cough, hemoptysis, and recurrent pulmonary infections, with typical onset during adulthood. In the case reported, the symptoms began in childhood, although they had been misinterpreted as asthma. Even if childhood-onset is not reported in literature, it is likely that small changes occur in the first few years of life and become more evident in adulthood. The involvement of segmental and sub-segmental bronchi, usually spared in TBPO, could explain the presence of wheezing and non-productive cough reported by our patient since childhood.
Literatur
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