Tracheobronchopathia osteochondroplastica associated with skin cancer: a case report and review of the literature

Abstract

Background: Tracheobronchopathia osteochondroplastica (TO) is a rare disorder of unknown cause affecting the large airways. It is characterized by the accumulation of bony and cartilaginous nodules in the tracheal and bronchial mucosa. Approximately 300 cases of tracheobronchopathia osteochondroplastica have been reported since Wilks first identified it in 1857. Tomography and computed tomography scanning (CT) can be suggestive but final verification requires biopsy. Neoplastic disorders are, among others, blamed in etiology. We describe here, for the first time, a case of TO associated with skin cancer.

Case presentation: A 40-year-old man with a scalp cancer was admitted for further evaluation of an occasional dry cough. Her medical history was otherwise unremarkable, and physical examination showed no abnormalities.The chest CT scan demonstrated multiple nodular densities in the trachea and proximal bronchi. The fiberoptic bronchoscopy showed multiple nodules in the trachea suggesting a malignant infiltration.Microscopic examination of the biopsy material revealed fragments of normal cartilage and bone formation with normal mucosa which confirmed the diagnosis of TO.Patient underwent surgery for scalp cancer. For TO, case has followed up. At twelve-month follow up, scalp tumor did not recur and cough ceased.

Conclusion: TO is a rare, benign disease that should be kept in mind especially in patients with tracheal irregularities in their chest imaging. Association with malignant tumors is reported.In patients with malignancy, TO can easily be misdiagnosed if it is not known as a diagnosis possibility of malignant invasion of the trachea. Therefore, it is important to be aware of this possibility, in order to prevent unnecessary treatments to patients.

Figure 1

Skin carcinoma: Macroscopic view (A), and cranial computed tomography scan (B). Exophytic lesion of the scalp.

Figure 2

Chest computed tomography scan: Axial sections (A) and coronal reconstruction (B) showing thickening and calcified lesions of the trachea.

Figure 3

Bronchoscopic view showed multiple, irregular nodules in the trachea (A) and the main bronchi (B), sparing the posterior wall.

Figure 4

Pathological examination of specimen’s biopsy showed squamous metaplasia with ectopic bony tissue under the respiratory epithelium. (Haematoxylin, eosin, and safran × 400).