Mixed Lung Mucoepidermoid Carcinoma and Adenocarcinoma With Identical Mutations in an Epidermal Growth Factor Receptor Gene Jie-Yang Jhuang, MD, Yueh-Hung Chou, MD, Syue-Fong Hua, and Min-Shu Hsieh, MD Department of Pathology, Far East Memorial Hospital, New Taipei City, and Department of Pathology, National Taiwan University Hospital and National Taiwan University College of Medicine, Taipei, Taiwan
Lung cancers presenting two different histologic types are relatively rare. This paper presents a case report of mixed lung cancer comprising mucoepidermoid carcinoma and conventional adenocarcinoma, a combination that has not been reported previously. These two carcinomas showed distinct morphologic and immunohistochemical features. However, gene analysis revealed identical mutations in each component, which indicates they possess a monoclonal origin. Specifically, we identified the same mutation in exon 19 of the epidermal growth factor receptor gene. Molecular analysis further substantiated a monoclonal origin with divergent differentiation, as opposed to the collision of discrete tumors. (Ann Thorac Surg 2014;98:695–7) Ó 2014 by The Society of Thoracic Surgeons
L
ung cancers with two different histologic variants that have previously been identified include adenosquamous carcinomas presenting both squamous cell carcinoma and adenocarcinoma, small cell carcinomas with either squamous cell carcinoma or adenocarcinoma, and carcinosarcomas presenting a mixture of carcinomatous and sarcomatous components [1]. Molecular analysis has revealed identical genetic mutations within the different histologic variants of these combined cancers. Evidence from those previous studies indicates a monoclonal origin with divergent differentiation, as opposed to the collision of discrete tumors [2]. This current study reports a rare case of lung cancer presenting combined mucoepidermoid carcinoma and adenocarcinoma. Identical mutations in the epidermal growth factor receptor were identified in each of the components. This is the first case report of a combined salivary gland-type tumor with conventional adenocarcinoma in the lung. This patient in this case study is a 58-year-old woman with a history of Hashimoto disease with multinodular goiter. The patient denied smoking or consuming alcohol. Chest roentgenography and computed tomography (CT) revealed a single 4-cm mass lesion over the right upper lobe of the Accepted for publication Oct 11, 2013. Address correspondence to Dr Hsieh, Department of Pathology, National Taiwan University Hospital, 7 Chung San South Rd, Taipei 10002, Taiwan; e-mail: [email protected].
Ó 2014 by The Society of Thoracic Surgeons Published by Elsevier Inc
lung (Fig 1A). Transbronchial biopsy identified this as an adenocarcinoma. No metastasis was observed in a brain CT scan, bone scan, or positron emission tomography scan. After this diagnosis, lobectomy of the right upper lobe and dissection of the lymph nodes were performed by thoracotomy with thoracoscopic assist. A single firm, gray tumor measuring 4 3 cm was found located in the right upper lobe. Microscopic examination revealed that the tumor was located in the peribronchial area of the lung (Fig 1B) and comprised two different histologic components: a relatively solid central portion, surrounded by typical lung adenocarcinoma (Fig 1C). The adenocarcinoma presented mixed lepidic, acinar, and papillary patterns lined by cuboidal or low columnar cells with apical snouting and immunoreactivity to thyroid transcription factor-1 (TTF-1) (Fig 2A). The central portion of the tumor presented characteristics typically observed in mucoepidermoid carcinoma (MEC). Specifically, we observed mixed mucusproducing cells, intermediate cells, and squamoid cells arranged within irregular solid nests and some with cystic dilation (Fig 2B). The intermediate and squamoid cells were immunoreactive to p63, and the mucus-producing cells were highlighted by mucicarmine staining. The TTF-1 immunostaining was negative in the MEC component. Furthermore, one of the group 12 (lobar) lymph nodes presented metastatic carcinoma composed entirely of MEC (Fig 3A). Neither component was immunoreactive for anaplastic lymphoma kinase (ALK [5A4 clone]). Epidermal growth factor receptor (EGFR) gene mutation analysis of exons 18, 19, 20, and 21 was performed by polymerase chain reaction and gene sequencing in both tumor components. This investigation revealed identical mutations in exon 19 in both the adenocarcinoma and MEC. Specifically, we found a deletion of nucleotides 2237–2255 and a thymidine insertion (Del 2237–2255 ins T, Del E746–S752 ins V; Fig 3B). Based on these molecular characteristics, we believe that the different components within this mixed lung cancer possess a monoclonal origin and subsequently underwent divergent differentiation. According to the seventh edition of TNM staging for lung and pleural tumors, the clinical stage of this patient was T2aN1M0, stage IIA. The patient has been disease free, without local recurrence or renewed metastasis, for 1 year since the operation.
Comment Salivary gland-type lung cancers are relatively rare neoplasms of the lung, compared with conventional small cell and non-small cell carcinomas. Among these, adenoid cystic carcinoma and MEC are the most common [1, 3]. Nonetheless, the incidence of MEC is less than 1% of all lung cancers, most of which arise from bronchial secretory glands in the large airways [1]. Similar to other salivary gland-type lung cancers, MEC does not stain for TTF-1, a marker for lung adenocarcinoma with terminal respiratory unit differentiation. Owing to its endobronchial location, MEC usually presents symptoms associated with airway obstruction, such as wheezing, 0003-4975/$36.00 http://dx.doi.org/10.1016/j.athoracsur.2013.10.035
FEATURE ARTICLES
CASE REPORTS
696
CASE REPORT JHUANG ET AL MIXED LUNG CARCINOMA
Ann Thorac Surg 2014;98:695–7
Fig 1. (A) Chest computed tomography scan revealed a speculated mass lesion over the right upper lobe. (B) The tumor was located in the peribronchial region (hematoxylin and eosin stain, 20). (C) The tumor contained two distinct histologic components, a mucoepidermoid carcinoma (left side of this figure) occupying the central portion of the tumor, surrounded by conventional adenocarcinoma (right side of this figure) (hematoxylin and eosin stain, 20).
Fig 2. (A) Adenocarcinoma with typical acinar and papillary patterns (hematoxylin and eosin stain, 200) as well as TTF-1(þ) tumor cells (200) (inset). (B) The mucoepidermoid carcinoma presented characteristic mucin-producing cells, squamoid cells, and intermediate cells (hematoxylin and eosin stain, 200). Squamoid and intermediate cells were highlighted using p63 immunostain (200) (inset). FEATURE ARTICLES
hemoptysis, or obstructive pneumonia [1]. Surgical excision is the treatment of choice because MEC is unresponsive to chemotherapy and radiotherapy [1, 3]. The tumor described in this case report presented two distinct areas of MEC and adenocarcinoma. The MEC was centrally located, surrounded by adenocarcinoma. Each component comprised more than 10% of the tumor and displayed different immunophenotypes. The histogenesis of this combined lung cancer may have been the collision of separate tumors or a single tumor originating from common progenitor cells capable of divergent differentiation. Molecular testing can be used to confirm whether combined lung cancers are monoclonal. In this study, EGFR gene mutation analysis revealed identical deletions in exon 19 of both tumor components. These results strongly support a monoclonal origin. Monoclonal origins have been observed in lung adenosquamous Fig 3. (A) One of the group 12 (lobar) lymph nodes was metastasized by mucoepidermoid carcinoma (MEC) (hematoxylin and eosin stain, 100). (B) Both adenocarcinoma (Ade) and mucoepidermoid carcinoma presented the same EGFR mutation in exon 19 with a deletion of nucleotides 2237–2255 and a thymidine insertion.
carcinomas as well as cases of combined small cell carcinoma and non-small cell carcinoma [1, 2]. A comprehensive search of relevant literature indicated that this is the first case report of a combined salivary gland-type lung cancer and adenocarcinoma with identical EGFR gene mutations in each component. Clinical trials have proven that target therapies, such as those involving tyrosine kinase inhibitors (TKI), can effectively treat advanced lung adenocarcinomas harboring sensitive EGFR mutations. However, previous research investigating EGFR mutations in MEC yielded diverse results. For example, two studies did not find an EGFR mutation in exons 18–21 [3, 4], whereas two other studies reported the presence of missense mutations in exon 21 (L858R and L861Q) [5, 6]. At present, the use of TKI among patients with MEC has been limited; however, some research has reported a positive response to TKI, even without sensitive
Ann Thorac Surg 2014;98:697–9
References 1. Yousem SA, Nicholson AG. Mucoepidermoid carcinoma. In: Travis WD, Brambilla E, Muller-Hermelink HK, Harris CC, eds. World Health Organization classification of tumours. Tumours of the lung, pleura, thymus and heart. Lyon, France: IARC press; 2004:63–4. 2. Tochigi N, Dacic S, Nikiforova M, Cieply KM, Yousem SA. Adenosquamous carcinoma of the lung: a microdissection study of KRAS and EGFR mutational and amplification status in a western patient population. Am J Clin Pathol 2011;135:783–9. 3. Macarenco RS, Uphoff TS, Gilmer HF, et al. Salivary glandtype lung carcinomas: an EGFR immunohistochemical, molecular genetic, and mutational analysis study. Mod Pathol 2008;21:1168–75. 4. Xi JJ, Jiang W, Lu SH, Zhang CY, Fan H, Wang Q. Primary pulmonary mucoepidermoid carcinoma: an analysis of 21 cases. World J Surg Oncol 2012;10:232. 5. Han SW, Kim HP, Jeon YK, et al. Mucoepidermoid carcinoma of lung: potential target of EGFR-directed treatment. Lung Cancer 2008;61:30–4. 6. Yu Y, Song Z, Gao H, et al. EGFR L861Q mutation is a frequent feature of pulmonary mucoepidermoid carcinoma. J Cancer Res Clin Oncol 2012;138:1421–5. 7. Rossi G, Sartori G, Cavazza A, Tamberi S. Mucoepidermoid carcinoma of the lung, response to EGFR inhibitors, EGFR and K-RAS mutations, and differential diagnosis. Lung Cancer 2009;63:159–60. 8. Lindeman NI, Cagle PT, Beasley MB, et al. Molecular testing guideline for selection of lung cancer patients for EGFR and ALK tyrosine kinase inhibitors: guideline from the College of American Pathologists, International Association for the Study of Lung Cancer, and Association for Molecular Pathology. J Thorac Oncol 2013;8:823–59.
A Novel Approach to Endobronchial Closure of a Bronchial Pleural Fistula Jason Akulian, MD, Vikas Pathak, MD, Mark Lessne, MD, Kelvin Hong, MD, David Feller-Kopman, MD, Hans Lee, MD, and Lonny Yarmus, DO Section of Interventional Pulmonology, Division of Pulmonary and Critical Care, University of North Carolina in Chapel Hill, Chapel Hill, North Carolina; and Section of Interventional Radiology, Division of Radiology, and Section of Interventional Pulmonology, Division of Pulmonary and Critical Care, Johns Hopkins University, Baltimore, Maryland Accepted for publication Sept 23, 2013. Address correspondence to Dr Yarmus, Interventional Pulmonology, Department of Medicine, Johns Hopkins Hospital, 1800 Orleans St, Ste 7125, Baltimore, MD 21287; e-mail: [email protected].
Ó 2014 by The Society of Thoracic Surgeons Published by Elsevier Inc
697
Bronchopleural fistula presents an important and challenging management problem after lung parenchymal resection. The mainstay of treatment has been surgical revision of the bronchial stump, however increasingly endobronchial therapies are being employed. We report the novel use of a liquid embolic agent with an Amplatzer vascular plug to seal a chronic bronchopleural fistula. Using rigid bronchoscopy, fluoroscopy, radio opaque liquid embolic agent, and the Amplatzer vascular plug, we were able to demonstrate not only feasibility but also safety and a marked reduction in symptoms consistent with successful closure of the bronchopleural fistula. (Ann Thorac Surg 2014;98:697–9) Ó 2014 by The Society of Thoracic Surgeons ronchopleural fistula (BPF) is a communication between the pleural space and the bronchial tree. The majority of cases occur after pneumonectomy, with subsequent infection, chemotherapy, radiotherapy, and persistent spontaneous pneumothorax [1]. The reported incidence of BPF ranges from 0.5% to 20% and has been shown to be associated with the underlying etiology, surgical technique, and experience of the surgeon [1]. Complications associated with BPF are due to a loss of pleural space sterility after the establishment of direct communication with the airway. These complications include but are not limited to empyema, aspiration pneumonia, adult respiratory distress syndrome, and death [2]. The diagnosis and treatment of BPF represents a challenging problem and an important source of postoperative morbidity and mortality. We report the first case in the literature utilizing a fluoroscopically visualizable liquid embolic agent in conjunction with the Amplatzer Vascular Plug II (AVP [St. Jude Medical, St. Paul, MN]) to endobronchially seal a BPF and cement the AVP in place using rigid bronchoscopy and fluoroscopy.
B
A 66-year-old man with a history of adenocarcinoma of the lung who had undergone right pneumonectomy presented with stump breakdown and recurrent empyema. He was treated with tube thoracostomy and antibiotics, followed by several failed attempts at endobronchial closures. Finally, an Eloesser flap with window was created. After several years with an improvement in symptoms and an exhaustive assessment for stump healing and closure including V/Q scan, bronchoscopies with direct visual inspection, Omnipaque (GE Healthcare, Waukesha, WI) fluoroscopic evaluation, contrast chest computed tomography, and negative cultures of both bronchial stump and pleural space, the flap was surgically reversed. Three months after Eloesser flap closure, the patient began to have recurrent episodes of pleural sepsis due to recurrence of the BPF. Because of his poor surgical candidacy and based on prior reports of success [3, 4], an endobronchial closure using an AVP was attempted. Under rigid bronchoscopic guidance, the AVP was placed through a guide sheath into the right main bronchus, intentionally obstructing the stump. After this, 0.9 mL cyanoacrylate glue was injected into the space between the 0003-4975/$36.00 http://dx.doi.org/10.1016/j.athoracsur.2013.09.105
FEATURE ARTICLES
EGFR mutations [5, 7]. According to guidelines outlined by the College of American Pathologists, the International Association for the Study of Lung Cancer, and the Association for Molecular Pathology, EGFR and ALK tests are recommended for adenocarcinomas and mixed lung cancers presenting an adenocarcinoma component [8]. In this case of mixed lung cancer, we identified sensitive EGFR mutations, which is in line with the criteria proposed by the College of American Pathologists and supports the use of TKI for this patient in the event of distant metastasis or tumor recurrence.
CASE REPORT AKULIAN ET AL NOVEL CLOSURE OF BRONCHIAL PLEURAL FISTULA
Lung cancers presenting two different histologic types are relatively rare. This paper presents a case report of mixed lung cancer comprising mucoepidermoid carcinoma and conventional adenocarcinoma, a combination that has not been reported previously. These two carcinomas showed distinct morphologic and immunohistochemical features. However, gene analysis revealed identical mutations in each component, which indicates they possess a monoclonal origin. Specifically, we identified the same mutation in exon 19 of the epidermal growth factor receptor gene. Molecular analysis further substantiated a monoclonal origin with divergent differentiation, as opposed to the collision of discrete tumors. (Ann Thorac Surg 2014;98:695–7) Ó 2014 by The Society of Thoracic Surgeons
L
ung cancers with two different histologic variants that have previously been identified include adenosquamous carcinomas presenting both squamous cell carcinoma and adenocarcinoma, small cell carcinomas with either squamous cell carcinoma or adenocarcinoma, and carcinosarcomas presenting a mixture of carcinomatous and sarcomatous components [1]. Molecular analysis has revealed identical genetic mutations within the different histologic variants of these combined cancers. Evidence from those previous studies indicates a monoclonal origin with divergent differentiation, as opposed to the collision of discrete tumors [2]. This current study reports a rare case of lung cancer presenting combined mucoepidermoid carcinoma and adenocarcinoma. Identical mutations in the epidermal growth factor receptor were identified in each of the components. This is the first case report of a combined salivary gland-type tumor with conventional adenocarcinoma in the lung. This patient in this case study is a 58-year-old woman with a history of Hashimoto disease with multinodular goiter. The patient denied smoking or consuming alcohol. Chest roentgenography and computed tomography (CT) revealed a single 4-cm mass lesion over the right upper lobe of the Accepted for publication Oct 11, 2013. Address correspondence to Dr Hsieh, Department of Pathology, National Taiwan University Hospital, 7 Chung San South Rd, Taipei 10002, Taiwan; e-mail: [email protected].
Ó 2014 by The Society of Thoracic Surgeons Published by Elsevier Inc
lung (Fig 1A). Transbronchial biopsy identified this as an adenocarcinoma. No metastasis was observed in a brain CT scan, bone scan, or positron emission tomography scan. After this diagnosis, lobectomy of the right upper lobe and dissection of the lymph nodes were performed by thoracotomy with thoracoscopic assist. A single firm, gray tumor measuring 4 3 cm was found located in the right upper lobe. Microscopic examination revealed that the tumor was located in the peribronchial area of the lung (Fig 1B) and comprised two different histologic components: a relatively solid central portion, surrounded by typical lung adenocarcinoma (Fig 1C). The adenocarcinoma presented mixed lepidic, acinar, and papillary patterns lined by cuboidal or low columnar cells with apical snouting and immunoreactivity to thyroid transcription factor-1 (TTF-1) (Fig 2A). The central portion of the tumor presented characteristics typically observed in mucoepidermoid carcinoma (MEC). Specifically, we observed mixed mucusproducing cells, intermediate cells, and squamoid cells arranged within irregular solid nests and some with cystic dilation (Fig 2B). The intermediate and squamoid cells were immunoreactive to p63, and the mucus-producing cells were highlighted by mucicarmine staining. The TTF-1 immunostaining was negative in the MEC component. Furthermore, one of the group 12 (lobar) lymph nodes presented metastatic carcinoma composed entirely of MEC (Fig 3A). Neither component was immunoreactive for anaplastic lymphoma kinase (ALK [5A4 clone]). Epidermal growth factor receptor (EGFR) gene mutation analysis of exons 18, 19, 20, and 21 was performed by polymerase chain reaction and gene sequencing in both tumor components. This investigation revealed identical mutations in exon 19 in both the adenocarcinoma and MEC. Specifically, we found a deletion of nucleotides 2237–2255 and a thymidine insertion (Del 2237–2255 ins T, Del E746–S752 ins V; Fig 3B). Based on these molecular characteristics, we believe that the different components within this mixed lung cancer possess a monoclonal origin and subsequently underwent divergent differentiation. According to the seventh edition of TNM staging for lung and pleural tumors, the clinical stage of this patient was T2aN1M0, stage IIA. The patient has been disease free, without local recurrence or renewed metastasis, for 1 year since the operation.
Comment Salivary gland-type lung cancers are relatively rare neoplasms of the lung, compared with conventional small cell and non-small cell carcinomas. Among these, adenoid cystic carcinoma and MEC are the most common [1, 3]. Nonetheless, the incidence of MEC is less than 1% of all lung cancers, most of which arise from bronchial secretory glands in the large airways [1]. Similar to other salivary gland-type lung cancers, MEC does not stain for TTF-1, a marker for lung adenocarcinoma with terminal respiratory unit differentiation. Owing to its endobronchial location, MEC usually presents symptoms associated with airway obstruction, such as wheezing, 0003-4975/$36.00 http://dx.doi.org/10.1016/j.athoracsur.2013.10.035
FEATURE ARTICLES
CASE REPORTS
696
CASE REPORT JHUANG ET AL MIXED LUNG CARCINOMA
Ann Thorac Surg 2014;98:695–7
Fig 1. (A) Chest computed tomography scan revealed a speculated mass lesion over the right upper lobe. (B) The tumor was located in the peribronchial region (hematoxylin and eosin stain, 20). (C) The tumor contained two distinct histologic components, a mucoepidermoid carcinoma (left side of this figure) occupying the central portion of the tumor, surrounded by conventional adenocarcinoma (right side of this figure) (hematoxylin and eosin stain, 20).
Fig 2. (A) Adenocarcinoma with typical acinar and papillary patterns (hematoxylin and eosin stain, 200) as well as TTF-1(þ) tumor cells (200) (inset). (B) The mucoepidermoid carcinoma presented characteristic mucin-producing cells, squamoid cells, and intermediate cells (hematoxylin and eosin stain, 200). Squamoid and intermediate cells were highlighted using p63 immunostain (200) (inset). FEATURE ARTICLES
hemoptysis, or obstructive pneumonia [1]. Surgical excision is the treatment of choice because MEC is unresponsive to chemotherapy and radiotherapy [1, 3]. The tumor described in this case report presented two distinct areas of MEC and adenocarcinoma. The MEC was centrally located, surrounded by adenocarcinoma. Each component comprised more than 10% of the tumor and displayed different immunophenotypes. The histogenesis of this combined lung cancer may have been the collision of separate tumors or a single tumor originating from common progenitor cells capable of divergent differentiation. Molecular testing can be used to confirm whether combined lung cancers are monoclonal. In this study, EGFR gene mutation analysis revealed identical deletions in exon 19 of both tumor components. These results strongly support a monoclonal origin. Monoclonal origins have been observed in lung adenosquamous Fig 3. (A) One of the group 12 (lobar) lymph nodes was metastasized by mucoepidermoid carcinoma (MEC) (hematoxylin and eosin stain, 100). (B) Both adenocarcinoma (Ade) and mucoepidermoid carcinoma presented the same EGFR mutation in exon 19 with a deletion of nucleotides 2237–2255 and a thymidine insertion.
carcinomas as well as cases of combined small cell carcinoma and non-small cell carcinoma [1, 2]. A comprehensive search of relevant literature indicated that this is the first case report of a combined salivary gland-type lung cancer and adenocarcinoma with identical EGFR gene mutations in each component. Clinical trials have proven that target therapies, such as those involving tyrosine kinase inhibitors (TKI), can effectively treat advanced lung adenocarcinomas harboring sensitive EGFR mutations. However, previous research investigating EGFR mutations in MEC yielded diverse results. For example, two studies did not find an EGFR mutation in exons 18–21 [3, 4], whereas two other studies reported the presence of missense mutations in exon 21 (L858R and L861Q) [5, 6]. At present, the use of TKI among patients with MEC has been limited; however, some research has reported a positive response to TKI, even without sensitive
Ann Thorac Surg 2014;98:697–9
References 1. Yousem SA, Nicholson AG. Mucoepidermoid carcinoma. In: Travis WD, Brambilla E, Muller-Hermelink HK, Harris CC, eds. World Health Organization classification of tumours. Tumours of the lung, pleura, thymus and heart. Lyon, France: IARC press; 2004:63–4. 2. Tochigi N, Dacic S, Nikiforova M, Cieply KM, Yousem SA. Adenosquamous carcinoma of the lung: a microdissection study of KRAS and EGFR mutational and amplification status in a western patient population. Am J Clin Pathol 2011;135:783–9. 3. Macarenco RS, Uphoff TS, Gilmer HF, et al. Salivary glandtype lung carcinomas: an EGFR immunohistochemical, molecular genetic, and mutational analysis study. Mod Pathol 2008;21:1168–75. 4. Xi JJ, Jiang W, Lu SH, Zhang CY, Fan H, Wang Q. Primary pulmonary mucoepidermoid carcinoma: an analysis of 21 cases. World J Surg Oncol 2012;10:232. 5. Han SW, Kim HP, Jeon YK, et al. Mucoepidermoid carcinoma of lung: potential target of EGFR-directed treatment. Lung Cancer 2008;61:30–4. 6. Yu Y, Song Z, Gao H, et al. EGFR L861Q mutation is a frequent feature of pulmonary mucoepidermoid carcinoma. J Cancer Res Clin Oncol 2012;138:1421–5. 7. Rossi G, Sartori G, Cavazza A, Tamberi S. Mucoepidermoid carcinoma of the lung, response to EGFR inhibitors, EGFR and K-RAS mutations, and differential diagnosis. Lung Cancer 2009;63:159–60. 8. Lindeman NI, Cagle PT, Beasley MB, et al. Molecular testing guideline for selection of lung cancer patients for EGFR and ALK tyrosine kinase inhibitors: guideline from the College of American Pathologists, International Association for the Study of Lung Cancer, and Association for Molecular Pathology. J Thorac Oncol 2013;8:823–59.
A Novel Approach to Endobronchial Closure of a Bronchial Pleural Fistula Jason Akulian, MD, Vikas Pathak, MD, Mark Lessne, MD, Kelvin Hong, MD, David Feller-Kopman, MD, Hans Lee, MD, and Lonny Yarmus, DO Section of Interventional Pulmonology, Division of Pulmonary and Critical Care, University of North Carolina in Chapel Hill, Chapel Hill, North Carolina; and Section of Interventional Radiology, Division of Radiology, and Section of Interventional Pulmonology, Division of Pulmonary and Critical Care, Johns Hopkins University, Baltimore, Maryland Accepted for publication Sept 23, 2013. Address correspondence to Dr Yarmus, Interventional Pulmonology, Department of Medicine, Johns Hopkins Hospital, 1800 Orleans St, Ste 7125, Baltimore, MD 21287; e-mail: [email protected].
Ó 2014 by The Society of Thoracic Surgeons Published by Elsevier Inc
697
Bronchopleural fistula presents an important and challenging management problem after lung parenchymal resection. The mainstay of treatment has been surgical revision of the bronchial stump, however increasingly endobronchial therapies are being employed. We report the novel use of a liquid embolic agent with an Amplatzer vascular plug to seal a chronic bronchopleural fistula. Using rigid bronchoscopy, fluoroscopy, radio opaque liquid embolic agent, and the Amplatzer vascular plug, we were able to demonstrate not only feasibility but also safety and a marked reduction in symptoms consistent with successful closure of the bronchopleural fistula. (Ann Thorac Surg 2014;98:697–9) Ó 2014 by The Society of Thoracic Surgeons ronchopleural fistula (BPF) is a communication between the pleural space and the bronchial tree. The majority of cases occur after pneumonectomy, with subsequent infection, chemotherapy, radiotherapy, and persistent spontaneous pneumothorax [1]. The reported incidence of BPF ranges from 0.5% to 20% and has been shown to be associated with the underlying etiology, surgical technique, and experience of the surgeon [1]. Complications associated with BPF are due to a loss of pleural space sterility after the establishment of direct communication with the airway. These complications include but are not limited to empyema, aspiration pneumonia, adult respiratory distress syndrome, and death [2]. The diagnosis and treatment of BPF represents a challenging problem and an important source of postoperative morbidity and mortality. We report the first case in the literature utilizing a fluoroscopically visualizable liquid embolic agent in conjunction with the Amplatzer Vascular Plug II (AVP [St. Jude Medical, St. Paul, MN]) to endobronchially seal a BPF and cement the AVP in place using rigid bronchoscopy and fluoroscopy.
B
A 66-year-old man with a history of adenocarcinoma of the lung who had undergone right pneumonectomy presented with stump breakdown and recurrent empyema. He was treated with tube thoracostomy and antibiotics, followed by several failed attempts at endobronchial closures. Finally, an Eloesser flap with window was created. After several years with an improvement in symptoms and an exhaustive assessment for stump healing and closure including V/Q scan, bronchoscopies with direct visual inspection, Omnipaque (GE Healthcare, Waukesha, WI) fluoroscopic evaluation, contrast chest computed tomography, and negative cultures of both bronchial stump and pleural space, the flap was surgically reversed. Three months after Eloesser flap closure, the patient began to have recurrent episodes of pleural sepsis due to recurrence of the BPF. Because of his poor surgical candidacy and based on prior reports of success [3, 4], an endobronchial closure using an AVP was attempted. Under rigid bronchoscopic guidance, the AVP was placed through a guide sheath into the right main bronchus, intentionally obstructing the stump. After this, 0.9 mL cyanoacrylate glue was injected into the space between the 0003-4975/$36.00 http://dx.doi.org/10.1016/j.athoracsur.2013.09.105
FEATURE ARTICLES
EGFR mutations [5, 7]. According to guidelines outlined by the College of American Pathologists, the International Association for the Study of Lung Cancer, and the Association for Molecular Pathology, EGFR and ALK tests are recommended for adenocarcinomas and mixed lung cancers presenting an adenocarcinoma component [8]. In this case of mixed lung cancer, we identified sensitive EGFR mutations, which is in line with the criteria proposed by the College of American Pathologists and supports the use of TKI for this patient in the event of distant metastasis or tumor recurrence.
CASE REPORT AKULIAN ET AL NOVEL CLOSURE OF BRONCHIAL PLEURAL FISTULA
