Tracheobronchopathia osteochondroplastica: A rare cause of chronic cough with haemoptysis

Abstract

A case of tracheobronchopathia osteochondroplastic (TPO) was diagnosed in a 69-year old male with prolonged cough. TPO is a rare condition of unknown cause and only sporadic cases have been reported. The condition is benign, characterized by submucosal nodules growing from the submucosal layer of the airways, protruding into the bronchial lumen. The bronchscopic view together with bronchial cartilage with abnormal distributed mineralization of the histologic examination of theses nodules leads to the correct diagnosis. Mild cases are treated symptomatically, whereas we tried an inhaled corticosteroid. Prominent protrusions in the trachea or the bronchi must be removed. In most cases the disease is stable over years but progressive forms have been reported. TPO may cause chronic refractory cough, which eventually is the only prominent symptom of this disease.

Dyspnea, Dysphonia, and Cough

“…11 Some feel that this may support the theory that TPO is congenital, with nodules progressing so slowly over the course of the patient's life and most patients remaining asymptomatic. 2,6,13 Due to the wide array of symptoms, most patients are initially diagnosed with more common conditions such as asthma or chronic bronchitis. Although the etiology is not clear, associations with chronic inflammation, atrophic rhinitis, silicosis, amyloidosis, tracheal botryomycosis, and mycobacterioses are reported.…”

“…4 There is no gender predilection, with the majority of patients presenting during the sixth decade of life, although children as young as 5 years of age have been diagnosed with TPO. 2,6 Treatment for TPO is typically supportive but also includes diagnosing and treating other underlying conditions that may exacerbate TPO, such as asthma, allergic rhinitis, and chronic obstructive pulmonary disease. However, patients with moderate to severe disease may present with chronic cough, hoarseness, dyspnea, hemoptysis, or recurrent respiratory infections.…”

Traqueobroncopatia osteocondroplástica: relato de 2 casos e revisão bibliográfica

“…According to Secrest, it is estimated that only 5% of the cases are diagnosed during the person's life 9 . There is no correlation with smoking, however, some studies point to an association with chronic tracheal inflammatory processes, arguing that it can be a possible factor associated with disease progression 9,10 . Its etiology and pathogenesis are unknown; however, it is believed that chronic infection, congenital anomalies, chemical or mechanical irritation, degenerative and metabolic disorders, chronic inflammation and the final stage of primary amyloidosis.…”

“…Histologically, the mucosal bed may look normal, with areas of inflammation and necrosis, abnormal proliferative cartilaginous or bony formations on the submucosa; there may be squamous metaplasia of the columnar epithelium, calcium deposits, fragments of adipocytes, with active hematopoietic medullar bone tissue 10,11 . In 2001, Leske et al reported that in 40 biopsies they found the following changes: ossification (58%), squamous metaplasia of the respiratory epithelium (48%), cartilage (38%), calcification (20%) and amyloidosis 13% 6,8,14 .…”

“…Occasionally, tracheostomy is necessary. Prognosis is favorable and depends on the extension and location of the nodular lesions [8][9][10]13 . In the cases hereby reported we decided to follow up the patients annually in the clinic.…”