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Ann Thorac Surg 2007;83:1194-1196
© 2007 The Society of Thoracic Surgeons

Case Reports

Intrapulmonary Teratoma: An Exceptional Disease

Sandeep S. Rana, MS, MCh, Naveen Swami, MS*, Sudhir Mehta, MS, MCh, Jaswinder Singh, MS, MCh, Suvkanta Biswal, MS

Department of Cardio-thoracic & Vascular Surgery, Post Graduate Institute of Medical Education & Research, Chandigarh, India

Accepted for publication July 31, 2006.

* Address correspondence to Dr Swami, SR, Dept of CTVS, PGIMER, Chandigarh, India (Email: nav_swami_5275{at}yahoo.com).


   Abstract
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 Abstract
Introduction
 Case Reports
 Comment
 References
 
Intrathoracic teratomas almost always occur in the mediastinum, but occasionally, they may be found in the lung as intrapulmonary teratomas. Intrapulmonary teratomas have histologic findings that are similar to those of teratoma from other sites. Two successive patients with intrapulmonary teratomas presented to us in a variable manner. The clinical and radiologic features and the histopathologic findings are presented, and the relevant literature is discussed.


   Introduction
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 Abstract
 Introduction
Case Reports
 Comment
 References
 
Intrathoracic teratomas almost always occur in the mediastinum, but occasionally, they may be found in the lung as intrapulmonary teratomas. In clinical practice, an intrapulmonary teratoma is a rare entity: Until 1996, only 65 cases had been reported in the English and Japanese literature [1]. Typically, cystic teratomas are located in the midline. A patient with an intrapulmonary teratoma presents with fever, cough, hemoptysis, chest pain, and hair expectoration (trichoptysis) [2]. We are report 2 patients with intrapulmonary teratomas that presented in different ways.


   Case Reports
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 Abstract
 Introduction
 Case Reports
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 References
 
Patient 1
A 28-year-old woman presented with a 3-month history of nonproductive cough. A chest roentgenogram revealed inhomogeneous opacity in the upper zone of left lung with linear radiopaque shadows suggestive of calcification. A computed tomography (CT) scan of the chest similarly revealed a cystic lesion (5 x 4 inch) with a fatty component and calcification. A cytologic examination of a CT scan-guided aspiration showed a complete range of myeloid cells, including myeloblasts, myelocytes, and metamyelocyte, along with mature adipose tissue, suggestive of myelolipoma.

The patient underwent a posterolateral thoracotomy, during which a large cystic mass (15 x 10 cm) was found extending from the pericardium to the thoracic wall. It was adherent to pericardium and had multiple bronchial communications. The cystic mass was completely resected, and bronchial openings were closed.

On the cut section, areas of variegated appearance with yellowish homogenous and a few hard (teeth or bone) areas were seen. Histologic examination revealed fragments of stratified squamous epithelium with pilosebaceous units, extensive areas of adipose tissue, cartilage, and cysts lined by respiratory epithelium, and focal lesions of bony lamellae, suggestive of a mature teratoma.

Patient 2
A 30-year-old woman presented with frequent exacerbation of cough and copious expectoration. After the baseline investigations, sputum examination revealed pulmonary tuberculosis and a chest CT scan demonstrated features of bronchiectasis of the right upper and middle lobe, with miliary nodules with right paratracheal lymphadenopathy.

The patient was treated with antitubercular chemotherapy, but after a short remission, experienced a relapse of illness. Lung abscess and empyema thoracis subsequently developed. A CT scan demonstrated multiple collections of varying sizes (7 to 9 Hounsfield units) with air-fluid levels and air-bronchogram (Fig 1). The patient was treated conservatively with CT-guided pigtail drainage in view of her poor general condition.


Figure 1
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  		Fig 1. A contrast-enhanced computed tomography scan of the chest demonstrates multiple collections of varying sizes (7 to 9 Hounsfield units) with air-fluid levels.

 
After 6 months, the patient’s general condition had improved, and she was admitted for surgical intervention. The patient had a significant right mediastinal shift and moderate restriction with spirometry examination. Intraoperatively, we found a cystic mass in the zone of the right upper and middle lobes, with bronchial communications.

On the cut section, the mass displayed white cheesy material with a quantity of hair. Histopathologic analysis revealed stratified squamous lined fragments along with multiple pilosebaceous units, and the adjoining areas showed appendiceal submucosa with prominent lymphoid follicles and gastric mucosal epithelium. In addition, there was a large area of thymic tissue with normal morphology. The surrounding lung parenchyma showed dilated bronchioles with ulcerated mucosal epithelium. The overall features were consistent with a mature teratoma.

Postoperatively, both patients had a good recovery and were discharged to their homes.


   Comment
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 Abstract
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 Case Reports
 Comment
References
 
Teratomas are tumors originating from totipotential cells. They may arise from extragonadal sites, such as from the intracranial, cervicopharyngeal, intrathoracic, intraabdominal, and sacrococcygeal region. Intrapulmonary teratomas are extremely rare. According to Asano and colleagues [1], 65 cases have been reported in the literature from 1839 until 1996, including 35 from Japan, seven from Korea, and rest are in the English literature [3].

An analysis of various studies by Joo and colleagues [3] suggests that intrathoracic teratomas, including mediastinal and intrapulmonary teratoma, have a common genesis and are thought to originate from the displaced thymic tissue of the third pharyngeal pouch. They reference another study that indicated that intrapulmonary teratomas arise from aberrant thymic tissue, and a further report that proposed that the primordial teratomatous focus in the potential mediastinum is caught up by the respiratory outgrowth and hence locates within the lung [3]. Another theory suggested that primary pulmonary germ cell tumors represent an unusual differentiation of somatic cell line [4].

Because intrapulmonary teratomas are extremely rare, it is necessary to exclude the possibility of a mediastinal teratoma or metastasis from an extrapulmonary germ cell tumor. Morgan and colleagues [5] reported 7 patients with metastatic intrapulmonary teratomas arising from testicular embryonal and teratocarcinoma, and those were indistinguishable from primary tumor.

Direct bronchial communication is a distinguishing feature of intrapulmonary teratomas and has been reported in half of the English cases [5] and in two thirds of the cases in the Korean literature [3]. Joo and colleagues [3] also stressed the stressed the need to identify the exact anatomic location by endobronchial approach. Intrapulmonary teratomas predominantly locate in the upper lobe (65%), mostly in the anterior segment, as was found in both of our patients.

Although about two third of teratomas present in females, intrapulmonary teratomas do not show any predilection for women. Most intrapulmonary teratomas have occurred in first or second decade of life, with ages ranging from 10 to 68 years [6].

A preoperative clinical diagnosis is difficult because of some nonspecific symptoms, such as chest pain, fever, cough, hemoptysis, loss of weight, and features of pneumonia or bronchiectasis [6]. Trichoptysis is a strong evidence of intrapulmonary teratoma, but is an uncommon feature [6]. Hemoptysis is three times more common in patients with intraoperative evidence of bronchial communication or in tumors with aberrant pancreatic tissue [5].

Radiologically, intrapulmonary teratomas have most often presented as a lobulated mass, but they may be seen as a cavitary lesion, as areas of consolidation, or as a peripheral translucency. A tumor with intramural calcification is a characteristic feature, as was illustrated in one of our patients. A cavity with peripheral translucency is a distinguishing feature of intrapulmonary teratomas from mediastinal teratomas [6]. This feature indicates air within the cavity arising from bronchial communication.

CT scans demonstrate discrete areas of soft tissue, high local fat content, or punctate calcification, or a combination of these, and is extremely valuable to detect a ruptured teratoma. In a ruptured teratoma, the internal density becomes heterogeneous, the tumor margin becomes irregular, and the fat component shows a bursting configuration [6]. Magnetic resonance imaging is another emerging method that can be used to preoperatively diagnose a pulmonary teratoma [7].

Histologically, intrapulmonary teratomas may contain any tissue originating from one of the three germinal layers. A high percentage (approximately 30%) of teratomas are of the immature type and therefore have malignant potential [6]. Tumors with pancreatic tissue are prone to rupture owing to enzymatic reactions. Because of its malignant potential and possibility for rupture, surgical resection of an intrapulmonary teratoma is advocated [6]; options may range from segmentectomy to pneumonectomy.

In conclusion, an intrapulmonary teratoma is an exceptionally rare tumor. The diagnosis has to rely on the radiologic imaging, which demonstrates calcification, cavitations, and peripheral translucent areas. Trichoptysis is the only clinical feature that can be diagnostic. Because of its potential for rupture or malignancy, surgical removal is the curative treatment.


   References
Top
 Abstract
 Introduction
 Case Reports
 Comment
 References
 

  1. Asano S, Hoshikawa Y, Yamane Y, Ikeda M, Wakasa H. An intrapulmonary teratoma associated with bronchiectasia containing various kinds of primordium: a case report and review of the literature Virchows Arch 2000;436:384-388.[Medline]
  2. Üstün MÖ, Demircan A, Paksoy N, Özkaynak C, Tüzüner S. A case of intrapulmonary teratoma presenting with hair expectoration Thorac Cardiovasc Surg 1996;44:271-273.[Medline]
  3. Joo M, Kang YK, Lee HK, et al. Intrapulmonary and gastric teratoma: report of two cases J Korean Med Sci 1999;14:330-334.[Medline]
  4. Miller RR, Champagne K, Nevin Murrey RC. Primary pulmonary germ cell tumor with blastomatous differentiation Chest 1994;106:1595-1596.
  5. Morgan DE, Sanders C, McElvein RB, Nath H, Alexander CB. Intrapulmonary teratoma: a case report and review of the literature J Thorac Imag 1992;7:70-77.[Medline]
  6. Zenker D, Aleksic I. Intrapulmonary cystic benign teratoma: a case report and review of the literature Ann Thorac Cardiovasc Surg 2004;10:290-292.[Medline]
  7. Iwasaki T, Iuchi K, Matsumura A, Sueki H, Yamamoto S, Mori T. Intrapulmonary mature teratoma Jpn J Thorac Cardiovasc Surg 2000;48:468-472.[Medline]



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