Display options
Cite
Fois AG, Arcadu A, Santoru L, et al. Tracheobronchopathia Osteochondroplastica: a rare case report of a non-smoker and non-atopic patient, with a long history of wheezing since childhood. Multidiscip Respir Med. 2016;11:16doi: 10.1186/s40248-016-0050-7.
Fois, A. G., Arcadu, A., Santoru, L., Trisolini, R., Marras, V., Ginesu, G. C., Canu, S., Cordero, L., Diana, G., & Pirina, P. (2016). Tracheobronchopathia Osteochondroplastica: a rare case report of a non-smoker and non-atopic patient, with a long history of wheezing since childhood. Multidisciplinary respiratory medicine, 1116. https://doi.org/10.1186/s40248-016-0050-7
Fois, Alessandro G, et al. "Tracheobronchopathia Osteochondroplastica: a rare case report of a non-smoker and non-atopic patient, with a long history of wheezing since childhood." Multidisciplinary respiratory medicine vol. 11 (2016): 16. doi: https://doi.org/10.1186/s40248-016-0050-7
Fois AG, Arcadu A, Santoru L, Trisolini R, Marras V, Ginesu GC, Canu S, Cordero L, Diana G, Pirina P. Tracheobronchopathia Osteochondroplastica: a rare case report of a non-smoker and non-atopic patient, with a long history of wheezing since childhood. Multidiscip Respir Med. 2016 Apr 19;11:16. doi: 10.1186/s40248-016-0050-7. eCollection 2016. PMID: 27096087; PMCID: PMC4836196.
Copy Download .nbib Format: NLM
Share it on

Multidiscip Respir Med. 2016 Apr 19;11:16. doi: 10.1186/s40248-016-0050-7. eCollection 2016.

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

Multidisciplinary respiratory medicine

Alessandro G Fois, Antonella Arcadu, Luigi Santoru, Rocco Trisolini, Vincenzo Marras, Giorgio C Ginesu, Sara Canu, Lorenzo Cordero, Gabriella Diana, Pietro Pirina

Affiliations

  1. Department of Respiratory Disease, University of Sassari, viale san Pietro 46/b, 07100 Sassari, Italy.
  2. Thoracic Endoscopy and Pulmonology Unit, Maggiore Hospital, Bologna, Italy.
  3. Pathology Unit, University of Sassari, Sassari, Italy.
  4. Department of Clinical and Experimental Medicine, Surgical Clinic, University of Sassari, Sassari, Italy.

PMID: 27096087 PMCID: PMC4836196 DOI: 10.1186/s40248-016-0050-7

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.

Keywords: Rare diseases; Respiratory sounds; Tracheal disease; Tracheobronchopathia Osteochondroplastica

References

  1. Mayo Clin Proc. 1989 Sep;64(9):1091-6 - PubMed
  2. PLoS One. 2014 Jul 11;9(7):e102068 - PubMed
  3. Hum Pathol. 1993 Sep;24(9):970-6 - PubMed
  4. Yonsei Med J. 2009 Oct 31;50(5):721-4 - PubMed
  5. J Laryngol Otol. 1985 Mar;99(3):305-10 - PubMed
  6. J Bronchology Interv Pulmonol. 2013 Jan;20(1):84-6 - PubMed
  7. Lung. 2008 Sep-Oct;186(5):293-7 - PubMed
  8. Ann Otol Rhinol Laryngol. 1990 Sep;99(9 Pt 1):689-94 - PubMed
  9. Respiration. 1995;62(3):151-4 - PubMed
  10. Clin Respir J. 2015 Oct;9(4):386-91 - PubMed
  11. Chest. 2000 Feb;117(2):385-92 - PubMed
  12. Chest. 1992 Oct;102(4):990-4 - PubMed
  13. J Clin Pathol. 1967 Nov;20(6):814-20 - PubMed
  14. Chest. 1981 Dec;80(6):706-9 - PubMed
  15. Virchows Arch. 1997 Nov;431(5):359-63 - PubMed
  16. Medicine (Baltimore). 2001 Nov;80(6):378-90 - PubMed
  17. Ann Thorac Surg. 2006 Apr;81(4):1480-2 - PubMed

Publication Types