The common histologic types of endobronchial hamartoma are chondromatous and lipoid hamartoma.1 Osteochondromatous hamartoma with a bone marrow component has been very infrequently reported in English publications.2 We describe an extremely rare case of endobronchial osteochondromatous hamartoma that was centrally located and extracted satisfactorily by flexible bronchoscopy.

A 67-year-old woman came to our hospital complaining of intermittent cough that had been evolving over the previous 2 months. The patient was a non-smoker. No anomalies were found on laboratory testing, including hemogram as well as microbiological sputum tests. Simple chest radiograph revealed no anomalies. For a more detailed evaluation of the cough, thoracic computed tomography was carried out. This demonstrated a 1cm endobronchial lesion with no specific parenchymatous anomalies (Fig. 1a). The patient underwent bronchoscopy in order to assess the endobronchial lesion. Bronchoscopy demonstrated a polypoid mucosal lesion located in the left lower lobe bronchus (Fig. 1b). During bronchoscopy, its consistency was observed to be as hard as a bone fragment, and we were then able to completely extract the polypoid lesion with the biopsy forceps (Fig. 1c). Although the lesion had a tendency to hemorrhage, the hemorrhage was controlled with cold saline solution and an adrenalin solution at a ratio of 1:20000. The anatomic pathology findings demonstrated cartilaginous cells accompanied by bone tissue with myeloid elements, compatible with endobronchial osteochondromatous hamartoma (Fig. 1d and e). After the exeresis of the endobronchial lesion, the cough was resolved. On follow-up, the patient showed no evidence of relapse (Fig. 1f).

Fig. 1.

(a) Computed tomography revealed an endobronchial lesion measuring 1cm in diameter (arrow). (b) Polypoid mucous lesion located in the left lower lobar bronchus as seen on flexible bronchoscopy. (c) After the total extraction of the polypoid mucous lesion with biopsy forceps. (d) The endobronchial polypoid mass was mainly made up of bone, cartilage and fatty tissue and covered with respiratory epithelium (hematoxylin–eosin stain, ×40). (e) The bone contained myeloid elements, which would suggest true bone marrow (hematoxylin–eosin stain, ×200). (f) Computed tomography showing the elimination of the polypoid lesion 12 months after the bronchoscopic procedure.

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The lesion of this patient is an exceptional case of endobronchial osteochondromatous hamartoma. Few cases have been published of these lesions with a bone marrow component that have been surgically resected.2 Their treatment should be individualized according to the clinical manifestations of each patient and each hamartoma.3,4 Oishi et al.2 surgically resected a relatively large osteochondromatous hamartoma that could not be extracted by broncoscopy. In contrast, our group was able to extract the lesion during flexible bronchoscopy. This case was characterized by a polypoid lesion that was relatively small in diameter. This could be the reason why the lesion was able to be completely extracted by bronchoscopy without significant complications. Endoscopic extraction presents advantages for symptom control and can avoid thoracotomy due to endobronchial lesion.5,6

In the present case, the lesion had a consistency that was as hard as a bone fragment. On biopsy, the hard consistency was different from the spongiform consistency that has generally been described.1 This seems to be associated with the bone marrow component. In addition, during the bronchoscopy extraction there was a tendency to hemorrhage. Said hemorrhagic tendency is due to the bone marrow content of the tumor. We speculate that a relatively small endobronchial osteochondromatous hamartoma could be extracted without any risk with biopsy forceps during flexible bronchoscopy.