Tracheobronchopathia osteoplastica: Its relationship to primary tracheobronchial amyloidosis

Sakula, Alex

doi:10.1136/thx.23.1.105

Cited by 83 publications

(25 citation statements)

References 18 publications

(15 reference statements)

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“…It has also been considered as the terminal stage of the bronchial primary amyloidosis [5,6], but more recent studies have shown that they are distinct pathological processes [7,8]. Tracheobronchopathia osteochondroplastica is often asymptomatic, but can cause dry or productive cough, dyspnoea, inspiratory stridor, dry throat, dysphonia, bronchial obstruction, haemoptysis, recurrent pneumonia, and, occasionally, difficult tracheal intubation before surgery.…”

Section: Discussionmentioning

confidence: 99%

Mycobacterium avium-intracellulare associated with tracheobronchopathia osteochondroplastica

Baugnée¹,

Delaunois²

1995

Eur Respir J

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Despite its low incidence, tracheobronchopathia osteochondroplastica can be associated with atypical mycobacterial disease. Eur Respir J., 1995, 8, 180- Infection with Mycobacterium avium-intracellulare complex (MAC) has become one of the most frequent complications of the acquired immunodeficiency syndrome (AIDS), but primary pulmonary colonizations and infections have been described in non-AIDS patients with and without pre-existing bronchopulmonary disease. We report one patient with MAC pneumonia associated with tracheobronchopathia osteochondroplastica. Case reportA previously healthy and nonsmoking woman aged 53 yrs presented with symptoms of dry cough, night sweats and weakness of 2 yrs duration. On admission, physical examination was remarkable only for clubbing of the fingers: Her physical status was fair (weight 72 kg, height 1.72m); pulse 76 beats·min -1 ; blood pressure 130/60 mmHg; temperature 37.2°C; and respiratory rate 12 breaths·min -1 , with normal breath sounds over both lung fields. The erythrocyte sedimentation rate was 22 mm·h -1 , but routine blood, coagulation and serum biochemistry studies were within normal limits. A test for antibodies to human immunodeficiency virus (HIV) was also negative.The chest roentgenogram showed a reticulonodular infiltration of the right upper lobe, with a small atelectasis in the ventral segment, but without any cavitation or pleural effusion ( fig. 1). This infiltration was already obvious on a chest X-ray performed 2 yrs previously. The computed tomography (CT) scan of the thorax ( fig. 2) revealed localized bronchiectasis of the right upper lobe, surrounded by soft infiltrates and small rounded well-circumscribed nodules.A tuberculin skin test with 5 tuberculin units (TU) purified protein derivative (PPD) induced a very small wheal. Acid-fast bacilli from the sputum were observed on Ziehl-Neelsen staining.The patient was treated with isoniazid, rifampicin and pyrazinamide for 4 months. Sputum cultures remained repeatedly positive for Mycobacterium avium-intracellulare. No other potential pathogens were identified.Eur Respir J, 1995, 8, 180-182 DOI: 10.1183 Printed in UK -all rights reserved

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Section: Discussionmentioning

confidence: 99%

Mycobacterium avium-intracellulare associated with tracheobronchopathia osteochondroplastica

Baugnée¹,

Delaunois²

1995

Eur Respir J

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“…An association with amyloidosis [7,8] was suggested, but not confirmed in a large group of patients. Furthermore, its relationship with malignancies is unclear [9,10].…”

Section: Discussionmentioning

confidence: 99%

“…The possibility that a disturbance in calcium phosphate metabolism plays a role in the aetiology has not been proved [11]. Very few cases have been described in children, the youngest patient being 12 yrs of age; therefore, there is little evidence of a congenital process [7].…”

Section: Discussionmentioning

confidence: 99%

An unusual cause of recurrent fever

Los¹,

Schramel²,

Harten³

et al. 1997

Eur Respir J

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A 49 year old man was referred to our hospital in 1994, because of intermittent fever and production of mucopurulent sputum. His complaints started at 43 yrs of age. The symptoms were mostly self-limiting, and occurred five times a year. Over the years, periods of bronchitis had increased to 12 times a year. In addition, a sinusitis with Klebsiella ozaenae became apparent, which was treated with antibiotics and intranasal beclomethasone dipropionate. Coughing was most severe during the night, with production of mucopurulent sputum. The patient's exercise tolerance was normal; he had worked for more than 10 years as a shoemaker. He has no smoking history.Physical examination and routine laboratory investigations were normal. Posteroanterior and lateral chest radiography and lung function tests were normal. A fibreoptic bronchoscopy was performed and showed many abnormalities of the trachea and bronchi (figs. 1 and 2). Specimens were taken for microbiological analysis, and biopsies were sampled for histological examination ( fig. 3). Because of the extent of the abnormalities seen during bronchoscopy, computed tomography (CT) of the chest was performed (figs. 4 and 5).

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“…Although the cause of the condition is unknown, several theories have been proposed. Sakula [9] advocated the theory that bronchiectasis is a form of primary localized amyloidosis of the lower respiratory tract that has undergone ossification.…”

Section: Discussionmentioning

confidence: 99%

Tracheobronchopathia osteochondroplastica discovered during tracheal intubation for general anesthesia

et al. 2018

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Tracheobonchopathia osteochondroplastica (TO) is a rare disorder, where there is benign dysplasia of the trachea and occasionally large bronchi. We report the case of a 55-year-old woman who was discovered to have TO at the time of a difficult intubation. This case was also unusual because the patient had presented no previous symptoms despite the presence of extensive endotracheal lesions. The final diagnosis of TO was made after subsequent bronchoscopy in the post-operative period.

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Tracheobronchopathia osteoplastica: Its relationship to primary tracheobronchial amyloidosis

Cited by 83 publications

References 18 publications

Mycobacterium avium-intracellulare associated with tracheobronchopathia osteochondroplastica

Mycobacterium avium-intracellulare associated with tracheobronchopathia osteochondroplastica

An unusual cause of recurrent fever

Tracheobronchopathia osteochondroplastica discovered during tracheal intubation for general anesthesia

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