|Year : 2017 | Volume
| Issue : 2 | Page : 89-91
Tracheopathia and tracheobronchopathia osteochondroplastica: Computed tomographic findings of an uncommon entity – a series of two case reports
Mohd Ilyas, Ghanshyam Dev
Department of Radiodiagnosis and Imaging, Government Medical College, Jammu, Jammu and Kashmir, India
|Date of Web Publication||28-Apr-2017|
Department of Radiodiagnosis and Imaging, Government Medical College, Jammu - 180 001, Jammu and Kashmir
Source of Support: None, Conflict of Interest: None
Tracheopathia and tracheobronchopathia osteochondroplastica refer to idiopathic, non-malignant disease of large airways featured by sub-mucosal cartilaginous to osseous nodules overlying the cartilaginous rings, which may be focal or diffuse. We report two cases, one having tracheopathia osteochondroplastica and other having tracheobronchopathia osteochondroplastica. It is generally an overlooked diagnosis due to lack of familiarity among the clinicians. Our aim to present these cases is to avoid under-diagnosis/misdiagnosis whenever such a condition is encountered in daily practice.
Keywords: Computed radiography, tracheobronchopathia osteochondroplastica, tracheopathia
|How to cite this article:|
Ilyas M, Dev G. Tracheopathia and tracheobronchopathia osteochondroplastica: Computed tomographic findings of an uncommon entity – a series of two case reports. Adv Hum Biol 2017;7:89-91
|How to cite this URL:|
Ilyas M, Dev G. Tracheopathia and tracheobronchopathia osteochondroplastica: Computed tomographic findings of an uncommon entity – a series of two case reports. Adv Hum Biol [serial online] 2017 [cited 2020 Apr 4];7:89-91. Available from: http://www.aihbonline.com/text.asp?2017/7/2/89/205390
| Introduction|| |
Tracheopathia (involving trachea only) and tracheobronchopathia (involving trachea as well as main bronchi) osteochondroplastica are characterised by diffuse or focal nodularities consisting of cartilaginous or osseous tissue involving the anterior and lateral walls of tracheobronchial tree with sparing of posterior membranous (pars membranacea) wall of tracheobronchial tree. On computed tomography (CT), these appear as calcified nodules 1–3 mm in diameter lining the anterolateral tracheobronchial walls. These conditions may be cause of persistent cough, dyspnoea, recurrent infections, hoarseness or recurrent haemoptysis unexplained by the common aetiologies. It is important to be aware of this condition as a differential diagnosis of neoplasms as well as other mimicking benign conditions such as tracheobronchial amyloidosis or Wegener's granulomatosis, wherein posterior wall is not spared, to avoid unnecessary chemotherapy or surgery. Chest radiograph may be unremarkable in these cases unless complicated by some infection. CT with multiplanar reformations remains the most appropriate modality for diagnosis. We used 128-slice dual-source dual-energy Somatom Definition Flash (Siemens Healthcare, Germany) for CT of these two patients with multiplanar reformations.
| Case Reports|| |
A 66-year-old female patient was referred to our department for non-contrast CT of the chest. She had a history of recurrent cough, fever and intermittent chest pain for the past 4–6 months. She had no history of smoking or past anti-tubercular drug therapy. Physical examination revealed a respiratory rate of 16 breaths/min with no evidence of wheeze or crackles. Her temperature was 98.8°F. A posteroanterior chest radiograph from 15 days back was unremarkable. Plain CT scan of the chest was performed and multiplanar reformations also reproduced. The study revealed an air space consolidation involving right upper lobe with air bronchograms appearing as a soft tissue density on soft tissue window [Figure 1]a and [Figure 1]b. The attractive findings on soft tissue window were multiple calcific nodularities involving whole of the tracheal anterior wall (spared posterior membranous wall) with involvement of main as well as lobar bronchi on both sides, protruding into the lumen with anterior wall thickening [Figure 1]a,[Figure 1]b,[Figure 1]c,[Figure 1]d. There was no evidence of tracheal stenosis or narrowing. The diagnosis of right upper lobe air space consolidation with tracheobronchopathia osteochondroplastica was made and the patient referred to a pulmonary physician for further management.
|Figure 1: (a) Axial soft tissue window computed tomography picture of the chest showing soft tissue attenuation consolidation in right upper lobe and calcific nodularities of both main bronchi. (b) Coronal soft tissue window reformatted computed tomography picture showing right upper lobe consolidation and calcific irregular nodularities involving the trachea, main bronchi and lobar bronchi. (c) Sagittal soft tissue window computed tomography image showing the calcific nodularities along the anterior wall of trachea sparing the posterior membranous wall (characteristic of tracheobronchopathia osteochondroplastica). (d) Axial computed tomography image at the level of inferior part of thyroid showing the calcific nodules along the anterolateral walls of trachea with sparing of posterior wall.|
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A 56-year-old male patient was referred to our department for CT angiography of the neck and brain vessels. The patient had a history of multiple episodes of syncope and mild right-sided weakness with a history of intermittent episodes of cough and irritation in the throat. His physical examination was unremarkable. Non-contrast CT scan of the chest and neck was obtained for pre-planning of CT angiography which revealed small 1–3 mm calcific nodularities along the anterolateral tracheal wall with sparing of posterior tracheal wall; however, these nodularities did not extend into the main bronchi [Figure 2]a,[Figure 2]b,[Figure 2]c. CT angiography was later performed which revealed 70% narrowing of the left internal carotid artery. The diagnosis of left internal carotid artery stenosis with incidental finding of tracheopathia osteochondroplastica was made and the patient referred to a cardiologist for further management.
|Figure 2: (a) Sagittal reformatted computed tomography image showing the calcific nodularities along the anterior wall of trachea sparing the posterior membranous wall. (b) Coronal reformatted computed tomography image showing the calcific nodular densities along the lateral walls of trachea (these densities did not extend up to bronchi) - tracheopathia osteochondroplastica early stage. (c) Axial soft tissue window computed tomography image of the neck showing calcific densities along lateral walls of the trachea.|
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| Discussion|| |
Being an uncommon disease, tracheopathia and tracheobronchopathia osteochondroplastica are usually underdiagnosed due to lack of familiarity of the imaging findings. It is a slowly progressing, benign disease of the large airways. The first description was given by Wilks in 1857. It mostly affects the persons in the fifth to seventh decades of life (as in our cases).
The aetiology of the nodules remains uncertain with no apparent association with smoking. No definitive trigger has yet been identified. Whatever the trigger, there is histopathological evidence that bone morphogenetic protein-2 synergistically with transforming growth factor-beta-1 plays a role.
The course of the disease is usually asymptomatic and symptoms arise in the later stages of disease, which involve recurrent cough, hoarseness of voice, recurrent infections, haemoptysis and dyspnoea due to tracheal or bronchial stenosis. These symptoms are not explainable by the common aetiologies in these patients.
Tracheopathia and tracheobronchopathia osteochondroplastica are three times more common in males than females.
Diagnosis is based on CT, bronchoscopy and histopathological findings. CT study reveals (in our cases too) irregular thickening and calcified nodularities involving anterior and lateral tracheobronchial walls sparing the posterior membranous wall of trachea. In our series, one having findings limited to trachea was labelled as tracheopathia and other with extensive involvement of trachea, main bronchi and lobar bronchi was labelled as having tracheobronchopathia osteochondroplastica.
The conditions to be differentiated from these entities include tracheobronchial amyloidosis and Wegener's granulomatosis, wherein posterior wall is not spared.
There is no definitive management and treatment is limited to symptomatic cases. The palliative management includes airway humidification, control of infections by antibiotics, and in severe cases where significant tracheal or bronchial stenosis is there, bronchoscopic interventions can be performed.
| Conclusion|| |
Tracheopathia and tracheobronchopathia osteochondroplastica are an uncommon benign disorder of large airways usually presenting in the fifth to seventh decades of life. It is usually asymptomatic and overlooked diagnosis. This condition needs to be kept in mind while evaluating the patients having chronic cough, recurrent infections, dyspnoea or haemoptysis where no other cause could be attributed. CT is the most important and effective modality for the diagnosis of this entity which reveals calcific nodular densities with irregular wall thickening along the anterior and lateral tracheobronchial walls. It is to be differentiated from other mimicking conditions such as certain neoplasms, tracheobronchial amyloidosis and Wegener's granulomatosis with the characteristic feature of sparing the posterior membranous tracheal wall. If advanced disease is noted, prompt intervention should be performed.
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Conflicts of Interest
There are no conflicts of interest.
| References|| |
Williams SM, Jones ET. General case of the day. Tracheobronchopathia osteochondroplastica. Radiographics 1997;17:797-9.
Meyer CN, Døssing M, Broholm H. Tracheobronchopathia osteochondroplastica. Respir Med 1997;91:499-502.
Wilks S. Ossific deposit on the larynx, trachea and bronchi. Trans Pathol Soc Lond 1857;8:88.
Tajima K, Yamakawa M, Katagiri T, Sasaki H. Immunohistochemical detection of bone morphogenetic protein-2 and transforming growth factor-beta 1 in tracheobronchopathia osteochondroplastica. Virchows Arch 1997;431:359-63.
Raess PW, Cowan SW, Haas AR, Zhang PJ, Litzky LA, Miller WT Jr., et al.
Tracheobronchopathia osteochondroplastica presenting as a single dominant tracheal mass. Ann Diagn Pathol 2011;15:431-5.
Jabbardarjani HR, Radpey B, Kharabian S, Masjedi MR. Tracheobronchopathia osteochondroplastica: Presentation of 10 cases and review of the literature. Lung 2008;186:293-7.
Prakash UB. Tracheobronchopathia osteochondroplastica. Semin Respir Crit Care Med 2002;23:167-75.
[Figure 1], [Figure 2]