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

Fois AG, Arcadu A, Santoru L, Trisolini R, Marras V, Ginesu GC, Canu S, Cordero L, Diana G, Pirina P - Multidiscip Respir Med (2016)

Bottom Line: The prevalence of this disease, often asymptomatic or associated with nonspecific symptoms, is underestimated, and the mean age at diagnosis is 50 years.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.

View Article: PubMed Central - PubMed

Affiliation: Department of Respiratory Disease, University of Sassari, viale san Pietro 46/b, 07100 Sassari, Italy.

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.

No MeSH data available.


Related in: MedlinePlus

HRCT. Ground glass consolidation in anterior segment of right upper lobe
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Fig1: HRCT. Ground glass consolidation in anterior segment of right upper lobe

Mentions: He had a clinical history of fatty liver disease (FLD) and renal lithiasis. He reported a history of wheezing since childhood, with non-productive cough, diagnosed at different stages as asthma. He had no family history of respiratory diseases, including tuberculosis (TB), or asthma and atopy. He was admitted to our Respiratory Unit with suspected pulmonary tuberculosis and with symptoms characterized by a low-grade fever with profuse sweating combined with productive cough of purulent sputum, dyspnea, wheezing, and chest pain. At diagnosis the patient showed a severe obstructive ventilatory deficit, not reversible after inhalation of short acting beta 2 agonists. A Chest X-Ray prescribed by his general practitioner showed diffuse interstitial thickening without parenchymal consolidation. He was treated with a broad-spectrum antibiotic but the symptoms persisted for a few weeks. Chest X-ray didn’t show any parenchymal consolidation, whilst a High Resolution CT scan (HRCT) showed the presence of ground-glass opacity in the anterior segment of the right upper lobe, of suspected tubercular origin (Fig. 1).Fig. 1


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

Fois AG, Arcadu A, Santoru L, Trisolini R, Marras V, Ginesu GC, Canu S, Cordero L, Diana G, Pirina P - Multidiscip Respir Med (2016)

HRCT. Ground glass consolidation in anterior segment of right upper lobe
© Copyright Policy - OpenAccess
Related In: Results  -  Collection

License 1 - License 2
Show All Figures
getmorefigures.php?uid=PMC4836196&req=5

Fig1: HRCT. Ground glass consolidation in anterior segment of right upper lobe
Mentions: He had a clinical history of fatty liver disease (FLD) and renal lithiasis. He reported a history of wheezing since childhood, with non-productive cough, diagnosed at different stages as asthma. He had no family history of respiratory diseases, including tuberculosis (TB), or asthma and atopy. He was admitted to our Respiratory Unit with suspected pulmonary tuberculosis and with symptoms characterized by a low-grade fever with profuse sweating combined with productive cough of purulent sputum, dyspnea, wheezing, and chest pain. At diagnosis the patient showed a severe obstructive ventilatory deficit, not reversible after inhalation of short acting beta 2 agonists. A Chest X-Ray prescribed by his general practitioner showed diffuse interstitial thickening without parenchymal consolidation. He was treated with a broad-spectrum antibiotic but the symptoms persisted for a few weeks. Chest X-ray didn’t show any parenchymal consolidation, whilst a High Resolution CT scan (HRCT) showed the presence of ground-glass opacity in the anterior segment of the right upper lobe, of suspected tubercular origin (Fig. 1).Fig. 1

Bottom Line: The prevalence of this disease, often asymptomatic or associated with nonspecific symptoms, is underestimated, and the mean age at diagnosis is 50 years.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.

View Article: PubMed Central - PubMed

Affiliation: Department of Respiratory Disease, University of Sassari, viale san Pietro 46/b, 07100 Sassari, Italy.

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.

No MeSH data available.


Related in: MedlinePlus