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• W2023285220 abstract "A double-located mediastinal and intrapulmonary cystic teratoma is a rare condition to be considered by thoracic surgeons. Clinical or radiologic diagnosis of a ruptured mediastinal teratoma into adjacent structures may be highly suggestive. An atypical presentation may indicate cautiousness for complete surgical excision. We report the case of a 14-year-old girl presenting with chronic chest pain. The radiologic work-up showed a large cystic mediastinal tumor and a heterogeneous intrapulmonary left upper-lobe lesion. We discuss the radiologic differential diagnosis of this atypical double-located thoracic tumor and the surgical strategy for complete excision. A double-located mediastinal and intrapulmonary cystic teratoma is a rare condition to be considered by thoracic surgeons. Clinical or radiologic diagnosis of a ruptured mediastinal teratoma into adjacent structures may be highly suggestive. An atypical presentation may indicate cautiousness for complete surgical excision. We report the case of a 14-year-old girl presenting with chronic chest pain. The radiologic work-up showed a large cystic mediastinal tumor and a heterogeneous intrapulmonary left upper-lobe lesion. We discuss the radiologic differential diagnosis of this atypical double-located thoracic tumor and the surgical strategy for complete excision. Cystic teratomas are usually unique germ cell tumors that are located in the anterior mediastinum [1Duwe B. Sterman D. Musani A. Tumors of the mediastinum.Chest. 2005; 128: 2893-2909Crossref PubMed Scopus (325) Google Scholar]. They typically appear as uncomplicated encapsulated lesions that consist of fluid, calcifications, or fat tissue, or both [1Duwe B. Sterman D. Musani A. Tumors of the mediastinum.Chest. 2005; 128: 2893-2909Crossref PubMed Scopus (325) Google Scholar]. Acute rupture is rare and mostly occurs into adjacent structures such as the pleural space, pericardium, and lung [2Hiraiwa T. Hayashi T. Kaneda M. et al.Rupture of a benign mediastinal teratoma into the right pleural cavity.Ann Thorac Surg. 1991; 51: 110-112Abstract Full Text PDF PubMed Scopus (31) Google Scholar, 3Yu C.W. Hsieh M.J. Hwang K.P. Huang C.C. Ng S.H. Ko S.F. Mediastinal mature teratoma with complex rupture into the pleura, lung, and bronchus complicated with mycoplasma pneumonia.J Thorac Cardiovasc Surg. 2007; 133: 1114-1115Abstract Full Text Full Text PDF PubMed Scopus (10) Google Scholar]. Clinical presentation combined with specific computed tomography (CT) features usually leads to an easy diagnosis. We discuss here the main differential diagnosis and surgical strategy in the case of a double-localized mediastinal and intrapulmonary cystic tumor with unspecific chronic symptoms. A 14-year-old girl from Algeria was referred to our center for a voluminous anterior mediastinal tumor discovered at chest roentgenogram in the context of chronic chest pain. A chest CT scan showed a multiloculated, rather homogenous mediastinal cystic lesion measuring 90 × 55 mm (Fig 1). A second bullous cystic lesion was visualized in the left upper lobe (Fig 2), with pleural thickening and suspicion of a communication cord between the 2 lesions. There was no mediastinal adenomegaly. Results of echinococcosis serologic analysis were negative. On the basis of the patient's origin and the radiologic findings, we suspected a diagnosis of mediastinal hydatid cyst ruptured into the lung.Fig 2A chest computed tomography scan in the parenchymal window shows a bullous cystic lesion in the upper left lobe associated with pleural thickening.View Large Image Figure ViewerDownload (PPT) Surgical en bloc excision was planned, with particular care to avoid any tumor fragmentation, and hypertonic saline solution was used in the operative field. A median sternotomy revealed a 10-cm, solid, nonhomogenous mediastinal mass in contact with the 2 phrenic nerves. Both pleurae were opened, revealing dense bilateral adhesions. The lesion was first dissected, with both phrenic nerves preserved throughout their mediastinal course. A frozen section of the surrounding tissue showed no signs of malignancy, allowing us to preserve both nerves. The innominate vein and both thymic horns were dissected free of the tumor. The en bloc resection required partial pericardial excision. No tumor fragmentation occurred during this procedure. This was followed by an atypical pulmonary resection of the left upper lobe (wedge resection). The pulmonary lesion was indeed well defined and located in the anteroapical segment. Postoperative histopathologic examination showed a unilocular cyst, filled by a yellowish fluid and a grumous material mixed with hair. Although adhesions were present between the cyst and the surrounding thymic, pericardial, and pleural structures, no sign of macroscopic infiltration was observed. The tumor was microscopically composed of mature, adult-type tissues, including squamous and respiratory-type epithelium, sweat glands, sebaceous glands, mature cartilage, and hair follicles (Fig 3). There was neither necrosis nor hemorrhage. No immature (embryonic or fetal) or germinal- or somatic-type malignant features were observed in the tumor. The tumor was surrounded by a fibroinflammatory tissue rich in lymphocytes, histiocytes, and foreign-body giant cells. The diagnosis of mature cystic teratoma was confirmed. A subpleural cystic lesion, filled with a yellowish material, was found in the parenchymal resection. The lung cyst microscopically corresponded to a foreign-body reaction against the hair and fat (Fig 4, Fig 5).Fig 4A photomicrograph shows lung parenchyma supplanted by fibroinflammatory tissue (hematoxylin and eosin stain, original magnification ×25).View Large Image Figure ViewerDownload (PPT)Fig 5A higher magnification illustrates the important foreign-body giant cell reaction (hematoxylin and eosin stain, original magnification ×100).View Large Image Figure ViewerDownload (PPT) The patient's postoperative course was uncomplicated, with transient elevation of the left diaphragm that resolved spontaneously by postoperative week 2. We report the case of a patient with a mature mediastinal teratoma with secondary intrapulmonary localization. Teratomas are the most common mediastinal germ cell tumors [1Duwe B. Sterman D. Musani A. Tumors of the mediastinum.Chest. 2005; 128: 2893-2909Crossref PubMed Scopus (325) Google Scholar]. Symptoms of uncomplicated mediastinal teratomas include chronic cough and chest pain. Rupture is the major complication. Acute pain is a sign of such complication that can occur in the pleura [2Hiraiwa T. Hayashi T. Kaneda M. et al.Rupture of a benign mediastinal teratoma into the right pleural cavity.Ann Thorac Surg. 1991; 51: 110-112Abstract Full Text PDF PubMed Scopus (31) Google Scholar], bronchial tree [3Yu C.W. Hsieh M.J. Hwang K.P. Huang C.C. Ng S.H. Ko S.F. Mediastinal mature teratoma with complex rupture into the pleura, lung, and bronchus complicated with mycoplasma pneumonia.J Thorac Cardiovasc Surg. 2007; 133: 1114-1115Abstract Full Text Full Text PDF PubMed Scopus (10) Google Scholar], or even the pericardial space [4Sasaka K. Kurihara Y. Nakajima Y. et al.Spontaneous rupture: a complication of benign mature teratomas of the mediastinum.AJR Am J Roentgenol. 1998; 170: 323-328Crossref PubMed Scopus (69) Google Scholar]. Mediastinal teratoma rupture into the lung has been very rarely described [5Pikin O. Kolbanov K. Kazakevich V. Korolev A. Mediastinal mature cystic teratoma perforating into the lung.Interact Cardiovasc Thorac Surg. 2010; 11: 827-829Crossref PubMed Scopus (9) Google Scholar]. Trichoptysis (hair expectoration) is rare but specific of this complication [2Hiraiwa T. Hayashi T. Kaneda M. et al.Rupture of a benign mediastinal teratoma into the right pleural cavity.Ann Thorac Surg. 1991; 51: 110-112Abstract Full Text PDF PubMed Scopus (31) Google Scholar]. Finally, teratomas can also be hemorrhagic [6Cheung Y.C. Ng S.H. Wan Y.L. Pan K.T. Ruptured mediastinal cystic teratoma with intrapulmonary bronchial invasion: CT demonstration.Br J Radiol. 2001; 74: 1148-1149PubMed Google Scholar]. In our case report, despite the presence of a secondary pulmonary tumor, the patient's chronic chest pain was not clearly suggestive of acute rupture of a mediastinal primitive lesion. Therefore, clinical diagnosis for cystic tumors should also include double-located hydatid cysts, because these tumors essentially affect adolescents in endemic areas and are poorly symptomatic until growth or rupture [7Shameem M. Bhargava R. Ahmad Z. Fatima N. Nazir Shah N. Mediastinal hydatid cyst rupturing into the pleural cavity associated with pneumothorax: case report and review of the literature.Can Respir J. 2006; 13: 211-213PubMed Google Scholar]. Main symptoms also include chest pain and cough. The rupture of a mediastinal lesion into the pleural cavity is a rare but previously described pathology [7Shameem M. Bhargava R. Ahmad Z. Fatima N. Nazir Shah N. Mediastinal hydatid cyst rupturing into the pleural cavity associated with pneumothorax: case report and review of the literature.Can Respir J. 2006; 13: 211-213PubMed Google Scholar]. Because the serologic test is not specific for a diagnosis, chest CT is considered the most efficient diagnostic method [8Allen M.S. Presentation and management of benign mediastinal teratomas.Chest Surg Clin N Am. 2002; 12: 659-664Abstract Full Text Full Text PDF PubMed Scopus (25) Google Scholar]. Hydatid disease rather typically presents as homogeneous air-filled fluid cavity masses with air-fluid levels [8Allen M.S. Presentation and management of benign mediastinal teratomas.Chest Surg Clin N Am. 2002; 12: 659-664Abstract Full Text Full Text PDF PubMed Scopus (25) Google Scholar]. A preoperative diagnosis is mandatory because of the perioperative measures to prevent the surrounding tissues from being flooded by cystic material and the recommendations for a conservative operation. The doubled-located cystic lesion in this patient, rather homogenous at CT and clinically poorly suggestive of a complicated lesion, had to be managed with caution. This presentation was first suggestive of double-located hydatid disease, not excluding a diagnosis of complicated teratoma. In such an atypical presentation, surgical management should consider systematic use of intraoperative hypertonic saline solution and a complete tumor excision to avoid tumor fragmentation and spillage." @default.
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• W2023285220 date "2013-09-01" @default.
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• W2023285220 title "Mediastinal Teratoma With Coexisting Parenchymal Pulmonary Cystic Lesion" @default.
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