INTRAPULMONARY TERATOMA: A RARE PRESENTATION OF BRONCHIECTASIS

Abstract

TOPIC: Lung Pathology TYPE: Medical Student/Resident Case Reports INTRODUCTION: Intrapulmonary teratomas (IPT) are a rare extra-gonadal manifestation of teratomas and are a challenging diagnosis to make. We present a patient with IPT presenting with hemoptysis. CASE PRESENTATION: A 49-year-old man presented with intermittent non-massive hemoptysis throughout the previous year. Labs were normal with the exception of normocytic anemia with hemoglobin of 12.1 g/dL. CT chest with contrast revealed a necrotic mass in the central left upper lobe invading the left upper lobe bronchus and obliterating the left pulmonary artery with severe bronchiectatic changes. Multiple mediastinal lymph nodes were enlarged. Flexible bronchoscopy was performed and demonstrated occlusion of the left upper lobe with a dense, white mass, and hair-like material within the bronchus. Attempts at biopsy with the cryoprobe demonstrated friable mucosa that easily bled. No intervention was necessary as spontaneous hemostasis occurred. Attempts at forceps biopsy were unsuccessful because of dense, calcified material. As a result, the cryoprobe was used. Cryoprobe biopsies revealed inflammatory cells without evidence of malignancy. Cultures including bacterial, fungal, and AFB were negative. Lack of diagnosis, severe bronchiectasis, and recurrent hemoptysis prompted the recommendation for resection, and he underwent left thoracotomy with interpleural pneumolysis. Due to involvement of the left pulmonary artery, lung sparing surgery could not be safely achieved and ultimately required a left pneumonectomy. Final pathology was consistent with a lung teratoma. DISCUSSION: Teratomas are germ cell tumors commonly located in the sacrococcygeal region, anterior mediastinum, retroperitoneum, cranium, ovary, or gonads. Rarely they are intrapulmonary in location (1). Less than 100 IPT cases have been reported to date, and usually reported in the left upper lobe. (2,7). Patients present with chest pain, cough, fever, dyspnea, bronchiectasis, or recurrent pneumonia (5). Patients may expectorate hair (trichoptysis), which is considered pathognomonic (3). Untreated, patients are at risk of malignant transformation, tumor rupture, hemoptysis, and airway obstruction from compression. (5). CT chest shows well-defined round or lobulated masses that are smooth in contour with areas of peripheral translucency and may contain fat, calcium, or fluid (5,6). Irregular or ill-defined margins can suggest malignant transformation (4).The only definitive treatment is complete surgical resection and diagnosis is often made concomitantly from the gross pathology. Early resection is crucial in order to prevent complications (6). CONCLUSIONS: IPT is a rare cause of endobronchial obstruction and highlights the need for early bronchoscopic evaluation of hemoptysis. Early diagnosis and resection of IPT is critical to prevent complications and provide definitive treatment. REFERENCE #1: Khan J, Aslam F, Fatimi S, Ahmed R. Cough, fever and a cavitary lung lesion-an intrapulmonary teratoma. J Postgrad Med. 2005;51(4):330-1. REFERENCE #2: Faria RA, Bizon JA, Junior RS, Neto VD, Botter M, Saieg MA. Intrapulmonary teratoma. J Bras Pneumol 2007; 33: 612– 615. REFERENCE #3: Siddiqui, F.A., Jain, A., Maheshwari, V. and Beg, M.H. (2010), FNA diagnosis of teratoma lung: A case report. Diagn. Cytopathol., 38: 758-760. https://doi.org/10.1002/dc.21318Webb, W., Brant, W. E., & Helms, C. A. (1991). Fundamentals of Body CT (1st ed.). Philadelphia, PA: W. B. Saunders Company.Sasaka, K., Kurihara, Y., Nakajima, Y., Seto, Y., Endo, I., Ishikawa, T., & Takagi, M. (1998). Spontaneous rupture: A complication of benign mature teratomas of the mediastinum. American Journal of Roentgenology, 170(2), 323-328. doi:10.2214/ajr.170.2.9456938 Mardani P, Naseri R, Amirian A, et al. Intrapulmonary mature cystic teratoma of the lung: Case report of a rare entity. BMC Surgery. 2020;20:1-6. Ditah C, Templin T, Mandal R, Pinchot JW, Macke RA. Isolated intrapulmonary teratoma. J Thorac Cardiovasc Surg. 2016 Dec;152(6):e129-e131. doi: 10.1016/j.jtcvs.2016.07.056. Epub 2016 Aug 5. PMID: 27576374. DISCLOSURES: No relevant relationships by Beren Chandler, source=Web Response No relevant relationships by Garret Duron, source=Web Response No relevant relationships by Lindsey Goldberg, source=Web Response No relevant relationships by Joanna Kosko, source=Web Response No relevant relationships by Jason McClune, source=Web Response No relevant relationships by Lourens Willekes, source=Web Response