INTRODUCTION: Primary tracheal tumors are rare, and composed of various benign and malignant pathologies. We present a case of an incidentally discovered tracheal tumor in a male patient that was initially diagnosed as a papilloma. On complete resection of the tumor and subsequent analysis it was identified as a tracheal hamartoma
CASE PRESENTATION: A 65-year old male, former smoker, presented to the out-patient pulmonary office for an evaluation of a pulmonary nodule. He denied any complaints of shortness of breath, wheezing, chronic cough, pedal edema, anorexia or weight loss. He also denied a history of prior lung disease, TB or TB exposure. On a recent CT scan of his chest he was noted to have sub-cm pulmonary nodule and an 8mm polypoidal lesion in his trachea His past medical history was significant for congestive heart failure, diabetes mellitus and benign hypertension. His vital signs and physical examination was unremarkable. After reviewing all the relevant data and images he was scheduled for an out-patient bronchoscopy On inspection bronchoscopy a verrucous lesion was noted in the right lateral wall of the lower mid trachea. Multiple biopsies of the lesion were sent for histopathological analysis The histopathology revealed submucosal fibrosis and chronic inflammation with an inflamed squamous papilloma; however the HPV stains were subsequently negative The patient was rescheduled for a repeat bronchoscopy and complete resection of the lesion. Polyp snares with cautery to 40W was used for cutting the polyp, subsequently APC at 40W and 0.4s pulses was used for further destruction of the tumor. The histopathology revealed abundant hypocellular stroma with myxochondroid appearance and a few mature adipocytes. The overlying respiratory epithelium showed regional squamous metaplasia but no squamous hyperplasia, koilocytosis or squamous atypia to warrant a diagnosis of squamous papilloma. It was diagnosed as a Tracheal Hamartoma.
DISCUSSION: Primary tracheal tumors are rare, with most of them arising in the lower one-half to two-thirds of the trachea. The name hamartoma, first introduced by Albrecht in 1904 is derived from the Greek words for ‘error’ and ‘tumor’. On histological examination all the elements of a normal bronchus are seen with cartilage, muscle and epithelial tissue, together with ciliated epithelium lining the clefts in the tumor. Only 3% of hamartomas are endobronchial and there are only 12 cases reported of tracheal hamartomas. In a review of 128 benign tumors of the bronchus and trachea over a 30 year period, Raymond Hurst included 38 cases of hamartoma, but no cases of tracheal hamartoma. Most cases have been reported in men with only three present in women. Bronchoscopic resection is a safe and effective therapy in the management of airway hamartomas. Cosio reports that in 45 cases of endobronchial hamartoma, 42 had a complete resection through endoscopic laser.
CONCLUSIONS: The tracheal origin of the hamartoma in our patient is an unusual and relatively rare entity. Resection of the tumor is safe and should be considered if the patient has symptoms or if the diagnosis is uncertain.
Reference #1 Tracheal hamartoma. IJ Hurst and KG Nelson; Chest 1977;72;661-662
Reference #2 Endobronchial hamartomas. Cosio BG, Villena V, Echave-Sustaeta J, de Miguel E, Alfaro J, Hernandez L, Sotelo T. Respiratory Department, Hospital 12 de Octubre, Madrid, Spain. Chest 2002;122:202-5.
Reference #3 Tracheal hamartoma detected by abnormal breath sounds. Akira Kunisawa Et al; Journal of Bronchology 2000;7;160-163
DISCLOSURE: The following authors have nothing to disclose: Shailesh Pinto, Maria Del Mar Cirino-Marcano, Jay Dobkin, Changcheng Zhu
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