Case Study
A large neglected pleomorphic adenoma of the lung: Report of a rare case
Asian Cardiovascular & Thoracic Annals 2014, Vol. 22(5) 620–622 ß The Author(s) 2013 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav DOI: 10.1177/0218492313479959 aan.sagepub.com
Ioannis Gakidis1, Petros T Mihos1, Christos Chatziantoniou1, Athanasios Stamatelopoulos1, Christos Eftychiadis2 and Constantinos Mantas1
Abstract Primary pleomorphic adenomas of the lung are very rare tumors that have peculiar clinical and oncologic features. We report here on the diagnostic and therapeutic approach for a patient with a large neglected pulmonary pleomorphic adenoma that presented initially as pneumonia.
Keywords Adenoma, pleomorphic, lung neoplasms
Introduction Although pleomorphic adenomas are the most common tumors of salivary glands, they rarely appear in the lungs.1 Due to the rarity and unpredictable oncologic behavior, there is no clear consensus regarding management, prognosis, and survival.2 We describe the diagnostic and therapeutic approach in a young patient with a large neglected pleomorphic adenoma of the lung.
Case report A 24-year-old woman was admitted to our department with a pleomorphic adenoma of the right upper lobe of the lung. The tumor had been identified as pneumonia at another hospital during assessment and diagnosis of a lower pulmonary tract infection 3 months prior to admission. At that time, chest radiography identified an ill-defined lesion of the right upper lobe that was confused with the pulmonary solidification of pneumonia (Figure 1). After resolution of pneumonia, a residual opacity remained, and the patient underwent a computed tomography (CT) scan of the chest (Figure 2). The CT scan revealed an irregularly margined nodular tumor with dimensions of 3.5 3.4 cm, in the right upper lobe and in close contact with the pulmonary hilum without enlargement of the hilar and mediastinal lymph nodes. A subsequent bronchoscopy identified an exophytic lesion obstructing the orifice of
the posterior segment of the right upper lobe. A biopsy taken during the bronchoscopy revealed a pleomorphic adenoma of the lung. The patient was discharged with the advice of further surgical treatment. The delay in admission to our department was due to her personal reasons. Due to the time elapsed from the initial diagnosis, chest radiography and CT were repeated to identify local or distant spread of the lesion. The chest radiography revealed no significant changes, but the CT scan identified growth of the tumor to new dimensions of 4.6 3.5 cm (Figure 3). CT scans of the brain and abdomen revealed no pathology. The patient underwent a right upper lobectomy. Macroscopic examination showed a well-demarcated whitish solid mass that was 5 3 3 cm in size and had a nodular appearance on the cut surface. Light microscopy revealed sheets, trabeculae, or islands of epithelial or myoepithelial cells embedded in a myxoid matrix. Ducts were present with an outer layer of myoepithelial cells and an inner area of epithelial cells, containing material that was positive for periodic acid Schiff 1 Department of Thoracic Surgery, General Hospital of Attica ‘‘KAT’’, Kifisia, Athens, Greece 2 Department of Pathology, General Hospital of Attica ‘‘KAT’’, Kifisia, Athens, Greece
Corresponding author: Ioannis Gakidis, MSc, 29 Praxitelous, Kamatero, Athens 13451, Greece. Email: [email protected]
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Figure 1. An ill-defined lesion of the right upper lobe that was confused with the pulmonary solidification of pneumonia.
Figure 3. The preoperative chest computed tomography showing a large irregularly margined tumor.
Figure 2. A residual opacity in the right upper lung field after the resolution of pneumonia.
Figure 4. Ducts with epithelial and myoepithelial cells as well as periodic acid Schiff-positive material. Hematoxylin and eosin staining, original magnification 10.
staining. No mitotic activity, pleomorphism, or necrosis was found (Figure 4). Immunohistochemical analysis using antibodies against S-100, AE1/AE3, Cam 5.2, CK5, CK7, CK14, CK17, CK18, and CK19, highlighted both epithelial and myoepithelial cells, while antibodies against vimentin and smooth muscle actin highlighted the myoepithelial cells. The final diagnosis was pleomorphic adenoma of the lung. Ultrasound examination of the salivary glands ruled out a metastatic nature of the tumor. Six months after surgery, the patient was doing well without tumor recurrence.
Discussion Pleomorphic adenoma of the lung is very rare. Due to the fact that only case reports have appeared sporadically, no definite incidence has been recorded.2 Patients
described in the literature have been between the ages of 8 and 74 years, and no sex predominance has been identified.2 It seems that this tumor comprises approximately 1% of all primary lung adenomas.3 The symptoms that occur due to pleomorphic adenomas depend on tumor location.4 Endoluminal tumors may present with dyspnea and hemoptysis. Our patient had no dyspnea or hemoptysis despite the fact that a portion of the tumor protruded into the bronchus of the right upper lobe. The lesion was asymptomatic and discovered only by an evaluation and diagnosis of pneumonia. It is important to note that the lesion remained asymptomatic during the 3 months prior to definitive surgical treatment, despite a substantial growth in size. The oncologic behavior of pleomorphic adenomas depends on the location, size, and cytological evidence
Downloaded from aan.sagepub.com at University of Victoria on June 4, 2015
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Asian Cardiovascular & Thoracic Annals 22(5)
of malignancy at the time of diagnosis and treatment. Malignancy in association with primary lung pleomorphic adenoma has seldom been reported.5 However, larger less-circumscribed tumors with infiltrative features can recur or metastasize.3 Surgery is the treatment of choice for pleomorphic adenomas of the lung. All patients must have long-term follow-up due to the possibility of malignant behavior.6 Treatment of patients with advanced or recurrent disease has not been clearly established, despite the partial response that has been reported after radio- or chemotherapy.7 Funding This research received no specific grant from any funding agency in the public, commerical, or not-for-profit sectors.
Conflict of interest statement None declared.
2. Fitchett J, Luckraz H, Gibbs A and O’Keefe P. A rare case of primary pleomorphic adenoma in main bronchus. Ann Thorac Surg 2008; 86: 1025–1026. 3. Payne WS, Schier J and Woolner LB. Mixed tumors of the bronchus (salivary gland type). J Thorac Cardiovasc Surg 1965; 49: 663–668. 4. Carretta A, Libretti L, Taccagni G and Zannini P. Salivary gland-type mixed tumor (pleomorphic adenoma) of the lung. Interact Cardiovasc Thorac Surg 2004; 3: 663–665. 5. Weissferdt A and Moran C. Pulmonary salivary glandtype tumors with features of malignant tumor (carcinoma ex pleomorphic adenoma). A clinicopathologic study of five cases. Am J Clin Pathol 2011; 136: 793–798. 6. Ang KL, Dhannapuneni VR, Morgan WE and Soomro IN. Primary pulmonary pleomorphic adenoma. An immunohistochemical study and review of the literature. Arch Pathol Lab Med 2003; 127: 621–622. 7. Sakamoto H, Uda H, Tanaka T, Oda T, Morino H and Kikui M. Pleomorphic adenoma in the periphery of the lung. Arch Pathol Lab Med 1991; 115: 393–396.
References 1. Tanigaki T, Shoyama Y, Iwasaki M, Abe Y, Nakamura M and Inoue H. Pleomorphic adenoma in the lung. Monaldi Arch Chest Dis 2002; 57: 30–32.
Downloaded from aan.sagepub.com at University of Victoria on June 4, 2015
A large neglected pleomorphic adenoma of the lung: Report of a rare case
Asian Cardiovascular & Thoracic Annals 2014, Vol. 22(5) 620–622 ß The Author(s) 2013 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav DOI: 10.1177/0218492313479959 aan.sagepub.com
Ioannis Gakidis1, Petros T Mihos1, Christos Chatziantoniou1, Athanasios Stamatelopoulos1, Christos Eftychiadis2 and Constantinos Mantas1
Abstract Primary pleomorphic adenomas of the lung are very rare tumors that have peculiar clinical and oncologic features. We report here on the diagnostic and therapeutic approach for a patient with a large neglected pulmonary pleomorphic adenoma that presented initially as pneumonia.
Keywords Adenoma, pleomorphic, lung neoplasms
Introduction Although pleomorphic adenomas are the most common tumors of salivary glands, they rarely appear in the lungs.1 Due to the rarity and unpredictable oncologic behavior, there is no clear consensus regarding management, prognosis, and survival.2 We describe the diagnostic and therapeutic approach in a young patient with a large neglected pleomorphic adenoma of the lung.
Case report A 24-year-old woman was admitted to our department with a pleomorphic adenoma of the right upper lobe of the lung. The tumor had been identified as pneumonia at another hospital during assessment and diagnosis of a lower pulmonary tract infection 3 months prior to admission. At that time, chest radiography identified an ill-defined lesion of the right upper lobe that was confused with the pulmonary solidification of pneumonia (Figure 1). After resolution of pneumonia, a residual opacity remained, and the patient underwent a computed tomography (CT) scan of the chest (Figure 2). The CT scan revealed an irregularly margined nodular tumor with dimensions of 3.5 3.4 cm, in the right upper lobe and in close contact with the pulmonary hilum without enlargement of the hilar and mediastinal lymph nodes. A subsequent bronchoscopy identified an exophytic lesion obstructing the orifice of
the posterior segment of the right upper lobe. A biopsy taken during the bronchoscopy revealed a pleomorphic adenoma of the lung. The patient was discharged with the advice of further surgical treatment. The delay in admission to our department was due to her personal reasons. Due to the time elapsed from the initial diagnosis, chest radiography and CT were repeated to identify local or distant spread of the lesion. The chest radiography revealed no significant changes, but the CT scan identified growth of the tumor to new dimensions of 4.6 3.5 cm (Figure 3). CT scans of the brain and abdomen revealed no pathology. The patient underwent a right upper lobectomy. Macroscopic examination showed a well-demarcated whitish solid mass that was 5 3 3 cm in size and had a nodular appearance on the cut surface. Light microscopy revealed sheets, trabeculae, or islands of epithelial or myoepithelial cells embedded in a myxoid matrix. Ducts were present with an outer layer of myoepithelial cells and an inner area of epithelial cells, containing material that was positive for periodic acid Schiff 1 Department of Thoracic Surgery, General Hospital of Attica ‘‘KAT’’, Kifisia, Athens, Greece 2 Department of Pathology, General Hospital of Attica ‘‘KAT’’, Kifisia, Athens, Greece
Corresponding author: Ioannis Gakidis, MSc, 29 Praxitelous, Kamatero, Athens 13451, Greece. Email: [email protected]
Downloaded from aan.sagepub.com at University of Victoria on June 4, 2015
Gakidis et al.
621
Figure 1. An ill-defined lesion of the right upper lobe that was confused with the pulmonary solidification of pneumonia.
Figure 3. The preoperative chest computed tomography showing a large irregularly margined tumor.
Figure 2. A residual opacity in the right upper lung field after the resolution of pneumonia.
Figure 4. Ducts with epithelial and myoepithelial cells as well as periodic acid Schiff-positive material. Hematoxylin and eosin staining, original magnification 10.
staining. No mitotic activity, pleomorphism, or necrosis was found (Figure 4). Immunohistochemical analysis using antibodies against S-100, AE1/AE3, Cam 5.2, CK5, CK7, CK14, CK17, CK18, and CK19, highlighted both epithelial and myoepithelial cells, while antibodies against vimentin and smooth muscle actin highlighted the myoepithelial cells. The final diagnosis was pleomorphic adenoma of the lung. Ultrasound examination of the salivary glands ruled out a metastatic nature of the tumor. Six months after surgery, the patient was doing well without tumor recurrence.
Discussion Pleomorphic adenoma of the lung is very rare. Due to the fact that only case reports have appeared sporadically, no definite incidence has been recorded.2 Patients
described in the literature have been between the ages of 8 and 74 years, and no sex predominance has been identified.2 It seems that this tumor comprises approximately 1% of all primary lung adenomas.3 The symptoms that occur due to pleomorphic adenomas depend on tumor location.4 Endoluminal tumors may present with dyspnea and hemoptysis. Our patient had no dyspnea or hemoptysis despite the fact that a portion of the tumor protruded into the bronchus of the right upper lobe. The lesion was asymptomatic and discovered only by an evaluation and diagnosis of pneumonia. It is important to note that the lesion remained asymptomatic during the 3 months prior to definitive surgical treatment, despite a substantial growth in size. The oncologic behavior of pleomorphic adenomas depends on the location, size, and cytological evidence
Downloaded from aan.sagepub.com at University of Victoria on June 4, 2015
622
Asian Cardiovascular & Thoracic Annals 22(5)
of malignancy at the time of diagnosis and treatment. Malignancy in association with primary lung pleomorphic adenoma has seldom been reported.5 However, larger less-circumscribed tumors with infiltrative features can recur or metastasize.3 Surgery is the treatment of choice for pleomorphic adenomas of the lung. All patients must have long-term follow-up due to the possibility of malignant behavior.6 Treatment of patients with advanced or recurrent disease has not been clearly established, despite the partial response that has been reported after radio- or chemotherapy.7 Funding This research received no specific grant from any funding agency in the public, commerical, or not-for-profit sectors.
Conflict of interest statement None declared.
2. Fitchett J, Luckraz H, Gibbs A and O’Keefe P. A rare case of primary pleomorphic adenoma in main bronchus. Ann Thorac Surg 2008; 86: 1025–1026. 3. Payne WS, Schier J and Woolner LB. Mixed tumors of the bronchus (salivary gland type). J Thorac Cardiovasc Surg 1965; 49: 663–668. 4. Carretta A, Libretti L, Taccagni G and Zannini P. Salivary gland-type mixed tumor (pleomorphic adenoma) of the lung. Interact Cardiovasc Thorac Surg 2004; 3: 663–665. 5. Weissferdt A and Moran C. Pulmonary salivary glandtype tumors with features of malignant tumor (carcinoma ex pleomorphic adenoma). A clinicopathologic study of five cases. Am J Clin Pathol 2011; 136: 793–798. 6. Ang KL, Dhannapuneni VR, Morgan WE and Soomro IN. Primary pulmonary pleomorphic adenoma. An immunohistochemical study and review of the literature. Arch Pathol Lab Med 2003; 127: 621–622. 7. Sakamoto H, Uda H, Tanaka T, Oda T, Morino H and Kikui M. Pleomorphic adenoma in the periphery of the lung. Arch Pathol Lab Med 1991; 115: 393–396.
References 1. Tanigaki T, Shoyama Y, Iwasaki M, Abe Y, Nakamura M and Inoue H. Pleomorphic adenoma in the lung. Monaldi Arch Chest Dis 2002; 57: 30–32.
Downloaded from aan.sagepub.com at University of Victoria on June 4, 2015
