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Abstract: Case Reports |

TRACHEOBRONCHOPATHIA OSTEOCHONDROPLASTICA: A CASE SERIES OF FIVE PATIENTS FREE TO VIEW

Scott I. Hagedorn, MD*; Shannon McCrann, MD; Janet N. Myers, MD
Author and Funding Information

National Naval Hosptal, Bethesda, MD


Chest


Chest. 2009;136(4_MeetingAbstracts):28S. doi:10.1378/chest.136.4_MeetingAbstracts.28S-c
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Published online

INTRODUCTION:  Tracheobronchopathia Osteochondroplastica (TPO) is a rare disorder characterized by submucosal calcified nodules. Several case reports have described its presentation. However, long term prognosis in TPO is poorly understood. We present five cases of TPO with follow up evaluations at varying intervals.

CASE PRESENTATION:  Our cases included four women and one man. Mean age was 68.2 years (59–72) at time of diagnosis. All presented with cough, post-nasal drainage, or gastric reflux symptoms. One patient recently started an angiotensin enzyme inhibitor and presented with mild hemoptysis. Pulmonary function tests were normal in three patients, obstructive in one, and restrictive in one. All patients underwent bronchoscopic evaluation revealing round, hard, sumbucosal nodules with normal overlying mucosa. Follow-up examinations and CAT scans were performed on all patients. None of the patients had evidence of advancing disease. Two of the patients were followed for greater than six years. Neither patient had progression of symptoms or nodules as seen on virtual bronchoscopy.

DISCUSSIONS:  TPO is a rare disorder of unclear etiology; however, abnormal bone morphogenetic protein 2 may play a role in initiating calcium deposition. Case series suggest 88 percent of patients have upper respiratory symptoms and cough at presentation. Diagnosis is made by direct visualization of characteristic lesions under bronchoscopy. In many cases, symptoms resolve but reported complications include airway obstruction, post-ostructive pneumonia, recurrent pneumonia, atelectasis, and difficult intubation. Rare cases of progressive disease have been reported. These patients were treated with laser ablation or tracheostomy. Treatment is usually directed at underlying disorders such as chronic obstructive pulmonary disease, asthma, bronchiectais, gastric reflux disease or upper respiratory cough syndrome. Minimal literature exists describing the long term behavior of TPO lesions or optimal follow-up. In our patients, the lesions did not change on follow up virtual bronchoscopy, suggesting TPO may be a reactive process to a single event leading to permanent changes.

CONCLUSION:  TPO is a rare usually benign disorder; however, progressive disease has been reported. The etiology and long term behavior of TPO lesions are poorly understood. Treatment is directed at comorbid respiratory disorders that cause chronic cough. As our images show, virtual bronchoscopy offers a potential non-invasive method of diagnosis and follow-up imaging.

DISCLOSURE:  Scott Hagedorn, No Financial Disclosure Information; No Product/Research Disclosure Information

Tuesday, November 3, 2009

4:30 PM - 6:00 PM

References

Leske V, Lazor R, Coetmeur, B, et al. Tracheobrochpathia Osteochondroplastica: A Study of 41 Patients.Medicine.80, 6:378–390. [CrossRef]
 
Thomas, D., Stonell and Hasan. Tracheobronchopathia osteoplastica: incidental finding at tracheal intubation. British J. of Anaesthesia.87 (3).:515. [CrossRef]
 

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References

Leske V, Lazor R, Coetmeur, B, et al. Tracheobrochpathia Osteochondroplastica: A Study of 41 Patients.Medicine.80, 6:378–390. [CrossRef]
 
Thomas, D., Stonell and Hasan. Tracheobronchopathia osteoplastica: incidental finding at tracheal intubation. British J. of Anaesthesia.87 (3).:515. [CrossRef]
 
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