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References

1. Shanmugan G, MacArthur K, Pollock JCS. Congenital lungmalformations-antenatal and postnatal evaluation and man-agement. Eur J Cardiothorac Surg 2005;27:45–52.

2. Papagiannopoulos K, Hughes S, Nicholson AG, Goldstraw P.Cystic lung lesions in the pediatric and adult population:surgical experience at the Brompton Hospital. Ann ThoracSurg 2002;73:1594– 8.

3. MacSweeney F, Papagiannopoulos K, Goldstraw P, SheppardMN, Corrin B, Nicholson AG. An assessment of the expandedclassification of congenital cystic adenomatoid malformationsand their relationship to malignant transformation. Am J SurgPath 2003;27:1139– 46.

4. Travis WD, Colby TV, Corrin B, Shimosato Y, Brambilla E.Histologic and graphical text slides for the histologic typing of lung and pleural tumors. In: Travis WD, ed; World HealthOrganization Pathology Panel. WHO international histologi-cal classification of tumors. 3rd ed. Berlin: Springer Verlag,Berlin; 1999:5.

5. Benjamin DR, Cahill JL. Bronchioloalveolar carcinoma of thelung and congenital cystic adenomatoid malformation. Am JClin Pathol 1991;95:889–892.

6. Lantuejoul S, Ferretti GR, Goldstraw P, Hansell DM, Bram-

billa E, Nicholson AG. Metastases from bronchioloalveolarcarcinomas associated with long-standing type 1 congenitalcystic adenomatoid malformations. A report of two cases.Histopathology 2006;48:204–6.

Bronchial Epithelial-MyoepithelialCarcinomaTung-Ying Chao, MD, An-Shen Lin, MD,

Chien-Hao Lie, MD, Yu-Hsiu Chung, MD, Jui-Wei Lin, MD, and Meng-Chih Lin, MD

Division of Pulmonary and Critical Care Medicine,Department of Internal Medicine, and Department of Pathology, Chang Gung Memorial Hospital-Kaohsiung

Medical Center, Chang Gung University College of Medicine,Kaohsiung; and Department of Respiratory Care, Chang GungInstitute of Technology, Chiayi, Taiwan

An epithelial-myoepithelial tumor is an extremely rarepulmonary neoplasm. Only 21 cases have been reported todate. This report presents a case of left main bronchialepithelial-myoepithelial carcinoma in a 43-year-old woman.No mediastinal lymph nodes were enlarged in computedtomographic scan, and no tumor growth was noted beyondthe bronchial cartilage layer by endobronchial ultrasoundimaging. This report highlights the usefulness of endo-bronchial ultrasound imaging for determining the depth oftumor invasion and choosing an alternative approach to

surgical resection.(Ann Thorac Surg 2007;83:689–91)

© 2007 by The Society of Thoracic Surgeons

Although epithelial-myoepithelial tumors are rela-tively common in salivary glands, they are rarelyfound in the respiratory tract  [1].  To date, 21 cases with

various biologic features have been reported in the lung[2, 3].  This report describes an epithelial-myoepithelial

carcinoma obstructing the left main bronchus in a 43-year-old woman. Endobronchial ultrasound (EBUS) im-aging demonstrated that tumor growth was limited to thebronchial cartilage layer. We used EBUS imaging toassist in the staging of this tumor and treated it withelectrocautery.

A 43-year-old woman (nonsmoker) presented with pro-gressive exertional dyspnea after right thyroid goitersurgery at a local hospital 1 year earlier. The patientdenied having weight loss, bloody sputum, and anycough. The physical examination showed expiratorywheezing over the left chest. The chest roentgenogram

finding was normal. Spirometry indicated a forced expi-ratory volume of first second of 1.24 liters, forced vitalcapacity 1.70 liters, and the percentage of forced expira-tory volume in 1 second was 57.4%.

A computed tomographic (CT) chest scan revealed a2-cm endobronchial tumor mass over the distal left mainbronchus without mediastinal lymph node enlargement.Bronchoscopy revealed a 2-cm well-circumscribed hy-pervascular tumor located at the anterolateral wall of theleft main bronchus, 2.5 cm distal to the carina ( Fig 1A).The movable tumor obstructed nearly 90% of the lumen,but the scope could pass through the narrowed routewithout causing bleeding from the tumor.

A forceps biopsy was used to obtain a specimen forhistologic diagnosis, and electrocauterization was used tocontrol bleeding and for temporary relief of dyspnea.Microscopically, the tumor showed ductular and a solidarchitecture. The solid section consisted of plasmacytoidcells. The glands were composed of an inner layer of epithelial cells and an outer layer of myoepithelial cells(Figs 2 and 3). No cell atypia or mitosis was noted. Ductalepithelial cells were positive for cytokeratin 7 and epi-thelial membrane antigen. Myoepithelial cells were pos-itive for smooth muscle actin and S100. No cytokeratin 20,neuron-specific enolase, chromogranin A, or synapto-physin was found in the tumor cells. Most of the tumorcells were also positive for P53 and CD117. About 2.8% of 

Accepted for publication July 13, 2006.

Address correspondence to Dr Chao, Pulmonary and Critical CareDivision, Chang Gung Memorial Hospital-Kaohsiung Medical Center,

Chang Gung University College of Medicine; No 123, Ta-Pei Rd, Niao-Sung Hsiang, Kaohsiung County, Taiwan; e-mail: tychao@adm.cgmh.org.tw.

 Fig 1. (A) Bronchoscopic picture shows a 2-cm well-circumscribed,hypervascular tumor that nearly totally obstructs the left main bron-chus. (B) No residual tumor was identified over left distal mainbronchus 3 months after electrocauterization.

689Ann Thorac Surg CASE REPORT CHAO ET AL2007;83:689 –91 BRONCHIAL EPITHELIAL-MYOEPITHELIAL CARCINOMA

© 2007 by The Society of Thoracic Surgeons 0003-4975/07/$32.00Published by Elsevier Inc doi:10.1016/j.athoracsur.2006.07.025

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the tumor cells stained positive for Ki-67, indicatinglow-grade malignancy.

Surgical resection was advised, but the patient refusedbecause of the possibility of pneumectomy. EBUS imag-ing later showed that tumor growth was limited to thebronchial cartilage layer; therefore, curative electrosur-

gery was performed. Three months later, a chest CT scanand a bronchoscopic examination demonstrated no re-sidual lesions over the distal left main bronchus (Fig 1B).The specimen from a repeated bronchoscopic biopsy at 3and 6 months revealed only chronic inflammation.

Comment

Epithelial-myoepithelial carcinomas arising in the lungare very rare. Only 21 cases have been reported   [2, 3].Diagnosis is based on the identification of myoepithelialcells, with spindle cells, clear cells, or plasmacytoiddifferentiation or a mixture of phenotypes, along with a

variable abundance of duct-forming epithelium. Immu-nohistochemically, the basal layer of the glandular ele-ments is shown to be myoepithelium by staining forS-100 and smooth muscle actin. The luminal layer stainedpositive for cytokeratin and epithelial membrane anti-gen. Tumors described as predominantly myoepithelio-matous are aggressive and have a high mitotic rate. Thepresence of tumor necrosis and nuclear pleomorphismare typically adverse prognostic factors. Immunochemi-cal staining with the lower Ki-67 index generally meanslow-grade malignancy [2, 3].

In all reported cases, the tumor site was predominantlyendobronchial, with one exception. Clinical symptoms

presented as endobronchial lesions with dyspnea andcough. All patients underwent complete resection (lobec-tomy or pneumonectomy)   [2].   Distant metastases areextremely rare [4, 5].

Clinically, this patient had no obstructive pneumonitisor distant metastases by chest CT scan and low-grade

malignancy on the pathologic view. Surgical resectionwas still advised to ensure tumor eradication; however,because of the possibility of pneumectomy, this younghealthy patient rejected surgical treatment. Curativeelectrocauterization was done under the assurance of limited bronchial airway invasion by EBUS imaging.

EBUS has been available for several years and is anexceptional instrument in differentiating between endo-bronchial tumors that have infiltrated the airway andthose that are just compressing it [6, 7]. In this case, EBUSimaging did not show tumor growth beyond the cartilagelayer.

Electrocauterization is useful for controlling bleeding,debulking, and removal of endobronchial tumors [8]. Theoutcome has been acceptable thus far, and regular,long-term follow-up of the patient is mandatory. Cura-tive electrosurgery is an option for management of thisrare low-grade malignancy. In our opinion, EBUS imag-ing is useful for preoperative diagnosis and treatment of an endobronchial tumor.

References

1. Fonseca I, Soares J. Epithelial-myoepithelial carcinoma of thesalivary glands: a study of 22 cases. Virchous Arch A PatholAnat Histopathol 1993;422:389–96.

2. Fulford LG, Kamata Y, Okudera K, et al. Epithelial-myoepithelial carcinomas of the bronchus. Am J Surg Pathol2001;25:1508–14.

3. Ru Kun, Srivastava A, Tischler, AS. Bronchial epithelial-myoepithelial carcinoma. Arch Pathol Lab Med 2004;128:92–4.

4. Higashiyama M, Kodama K, Yokouchi H, et al. Myoepitheli-oma of lung: report of two cases and review of the literature.Lung Cancer 1998;20:47–56.

5. Miura K, Harada H, Aiba S, Tsutsui Y. Myoepithelial carci-noma of the lung arising from bronchial submucosa. Am JSurg Pathol 2000;24:1300–4.

6. Herth F, Ernst A, Schulz M, Becker H. Endobronchial ultra-sound reliably differentiates between airway infiltration andcompression by tumor. Chest 2003;123:458–62.

7. Miyazu Y, Miyazawa T, Kurimoto N, Iwamoto Y, Kanoh K,Kohno N. Endobronchial ultrasonography in the assessment

 Fig 2. Neoplastic plump cells with focal ductular formation (hema-toxylin and eosin stain, original magnification 100).

 Fig 3. Florid neoplastic cells in sheets with plasmacytoid appearance(hematoxylin and eosin stain, original magnification  200).

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of centrally located early-stage lung cancer beforephotodynamic therapy. Am J Respir Crit Care Med 2002;165:832–7.

8. Hooper RG, Jackson FN. Endobronchial electrocautery. Chest1988;94;595–98.

Multiple Central EndobronchialChondroid HamartomaMin-Woong Kang, MD, Jong Hee Han, MD,

 Jeong Hwan Yu, MD, Yong Ho Kim, MD,Myung Hoon Na, MD, Jae Hyeon Yu, MD,Seung Pyung Lim, MD, Young Lee, MD,

 Jin-Hwan Kim, MD, Dae Young Kang, MD, and Ju-Ock Kim, MD

Departments of Thoracic and Cardiovascular Surgery,Radiology, Pathology, and Pulmonology, College of Medicine,Chungnam National University, Daejeon, Korea

Benign neoplasm of the lung is rare, and pulmonaryhamartoma is the most common form of benign neo-plasm of the lung. Most pulmonary hamartomas areparenchymal hamartomas, and endobronchial hamarto-mas are very rare and usually occur as a single mass. Wereport a case of a 55-year-old man presenting with mul-tiple endobronchial chondroid hamartomas that had notbeen confirmed preoperatively. The patient received bi-lobectomy, and the postoperative course was uneventful.There was no evidence of recurrence or complicationsduring the 6-month follow-up period. Reports of multi-ple endobronchial chondroid hamartomas are rare in theliterature, and the awareness of this form of benigndisease is important in the differential diagnosis of

pulmonary neoplasms.(Ann Thorac Surg 2007;83:691–3)

© 2007 by The Society of Thoracic Surgeons

Pulmonary hamartoma is a very rare form of benignneoplasm of the lung [1, 2].   In most cases, pulmo-nary hamartomas occur as peripheral and solitary le-sions. In this report, we present a patient with multipleendobronchial chondroid hamartomas, which have beenrarely reported in the literature.

A 55-year-old man was referred to our hospital with a6-month history of cough and sputum and a recent

episode of hemoptysis. Physical examination on admis-sion was not remarkable except for the decreased breath-ing sound in the right lung field. No abnormalities werefound in the blood tests. Chest roentgenogram showed a4-mm-sized calcified nodule in the right middle lungfield. Endoscopic examination showed separated bron-chial epithelial thickening between the right middle andlower lobe bronchi, and the anterior segmental bronchusof the right lower lobe was near-totally occluded by a

tumor-like mass. Biopsy specimens were taken from theseparated bronchial epithelial thickening site and theendobronchial mass; however, there were no specificfindings other than severe dysplasia.

The axial computed tomography (CT) images (Sensa-tion 16, Siemens Medical, Erlangen, Germany) showed

multiple separated endobronchial masses within thebronchus intermedius, lobar, and segmental bronchi of 

Accepted for publication June 6, 2006.

Address correspondence to Dr Lim, Department of Thoracic and Cardio-vascular Surgery, College of Medicine, Chungnam National University,Daejeon, Korea 301-721; e-mail: splim@cnu.ac.kr.

 Fig 1. (A) Axial computed tomography scan demonstrates a low-density mass (arrow) in the bronchus intermedius. (B) Coronal mul-tiplanar reformation image clearly shows the mass in the bronchusintermedius (arrow). Note the masses within the right middle lobeand lower lobe bronchi (arrowheads). Multiplanar reformationhelped confirm the multiplicity and endobronchial location of lesionsin a shorter time than it took to review the more numerous axialimages. (C) Virtual bronchoscopy demonstrates the mass (arrows).Cartilaginous rings are shown in the normal bronchial wall(arrowhead).

691Ann Thorac Surg CASE REPORT KANG ET AL2007;83:691–3 ENDOBRONCHIAL CHONDROID HAMARTOMA

© 2007 by The Society of Thoracic Surgeons 0003-4975/07/$32.00Published by Elsevier Inc doi:10.1016/j.athoracsur.2006.06.014

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