{"paper_id":"2177f622-e35a-4add-bf1f-21d4a30fc8aa","body_text":"C A S E R E P O R T Open Access\nEndobronchial endometriosis presenting as\ncentral-type lung cancer: a case report\nJuan-Han Yu 1,2, Xu-Yong Lin 1,2, Liang Wang 1,2, Yang Liu 1,2, Chui-Feng Fan 1,2, Yong Zhang 1,2 and En-Hua Wang 1,2*\nAbstract\nA 45-year-old female patient was referred to our hospital for complaining of dyspnea and coughing in the past four\nmonths. The computed tomography scanning demonstrated a central lesion in the upper lobe of the left lung\nclose to the hilar, and the subsequent bronchoscopy revealed a polypoid lesion of the distal of the left main\nbronchus. This patient was diagnosed clinically as “possibly central-type lung cancer ”. However, the pathologic\nresult of the surgically excised polypoid lesion was endobronchial endometriosis.\nVirtual Slides: The virtual slide(s) for this article can be found here: http://www.diagnosticpathology.diagnomx.eu/\nvs/1077439085928525\nKeywords: Endometriosis, Bronchus, Pathology, Immunohistochemistry\nBackground\nE n d o m e t r i o s i si sd e f i n e da st h ep r e s e n c eo fe n d o m e t r i a l\ntissue, including endometr ial glands and stroma, in the\nbody areas out of the uterus. Endometriosis foci are usually\nlocated in the pelvis and abdomen, and rarely in the\nthorax. Moreover, the thoracic endometriosis commonly\naffects the lung parenchyma, pleura, and diaphragm, and\nthe prominent clinical manifestations are recurrent\nhemoptysis, pneumothorax, hemothorax, and asymptom-\natic pulmonary nodules [1,2]. We herein report a rare case\nof endobronchial endometriosis presenting as central-type\nlung cancer.\nCase presentation\nA 45-year-old female without a history of smoking was\nreferred to our hospital for complaining of dyspnea and\ncoughing in the past four months. The patient had no\nf e v e ro rh a e m o p t y s i s .N op r i o rh i s t o r yo fd y s m e n o r r h e a ,\ndyspareunia, or pelvic pain was found as well. The physical\nexamination showed the left lung breath sounds weakened\ncompared with contralateral. On computed tomography\n(CT) scanning, a round-like lesion of 1.5 × 1.7 cm in size\nwas observed in the upper lobe of the left lung close to\nthe hilar, and the diagnosis was “possible of central-type\nlung cancer ”. The bronchoscopy (BF-1 T260, Olympus,\nTokyo, Japan) revealed a polypoid lesion in the distal of\nthe left main bronchus, and the lumen was blocked\n(Figure 1). The bronchoscopic diagnosis was “possible of\ncentral-type lung cancer ” as well. Pathologic result of\nbronchoscopic biopsy showed that there was only some\nsquamous epithelium. Thus, the patient underwent the\nlobectomy in our hospital. No adjuvant treatment\nwas performed and the patient was found well without\nrecurrence at 2 years after surgery.\nMaterials and methods\nThe resected specimens were fixed with 10% neutral-\nbuffered formalin and embedded in paraffin blocks. Tissue\nblocks were cut into 4- μm slides, deparaffinized in xylene,\nrehydrated with graded alcohols, and immunostained with\nthe following antibodies: cytokeratin (CK, AE1/AE3),\nthyroid transcription factor 1 (TTF-1, 8G7G3/1), ER\n(SP1), CD10 (56C6), and Ki67 (MIB-1) (MaiXin, China).\nSections were then stained with a streptavidin-peroxidase\nsystem (KIT-9720, Ultrasensitive TM S-P , MaiXin, China).\nThe chromogen used was diaminobenzidine tetrahydro-\nchloride substrate (DAB kit, MaiXin, China). All the\nsamples were slightly counterstained with hematoxylin,\ndehydrated, and mounted. For the negative controls, each\nsample was incubated with PBS instead of the primary\nantibody as above described.\n* Correspondence: wangeh@hotmail.com\n1Department of Pathology, the First Affiliated Hospital and College of Basic\nMedical Sciences, China Medical University, Shenyang 110001, China\n2Institute of pathology and pathophysiology, China Medical University,\nShenyang 110001, China\n© 2013 Yu et al.; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative\nCommons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and\nreproduction in any medium, provided the original work is properly cited.\nYu et al. Diagnostic Pathology 2013, 8:53\nhttp://www.diagnosticpathology.org/content/8/1/53\n\nResults\nGrossly, the resected lung tissue was about 14.0×4.5×3.0 cm\nin size. The polypoid lesion (1.5×1.3×1.0 cm in size) was\nlocated in the bronchus and the cut surface showed a\npink-red color. Histologically, the normal bronchial\nepithelium, submucosal glands, cartilage, and alveolar\nepithelium were found in the section. Of note, there\nwere some ectopic glandular structures surrounded by\ndensely distributed endometrial-like stromal cells under\nthe bronchial mucosa. The ectopic glands predominantly\nshowed a single layer of columnar cells similar to the\nendometrial epithelium lined with the basal nuclei, and no\nmarked cytological atypia was observed. The mitosis was\nrare (Figure 2).\nImmunohistochemical staining showed that all the\nepithelium and the glands were positive for CK, while\nthe remaining alveolar epithelial cells were positive for\nTTF-1. The ectopic glands showed positive staining for\nER, and the densely distributed stromal cells were\npositive for both ER and CD10 (Figure 3). Ki67 index\nwas less than 5%. According to the morphological and\nimmunohistochemical findings, the final diagnosis was\nendobronchial endometriosis.\nDiscussion\nThe occurrence of endometriosis in areas other than the\nabdomen and pelvis is uncommon, even rare in the thorax.\nThoracic endometriosis is usually located in the lung\nparenchyma, pleura, and diaphragm. The endometriosis\nof the bronchus is extremely rare. To our knowledge,\nthere were only five cases of thoracic endometriosis\nreported to occur in the bronchus previously [2-6]. The\ntypical clinical manifestations of thoracic endometriosis\ninclude pneumothorax (72%), hemoptysis (14%), or\nhemothorax (12%) coincident with menstrual bleeding,\nand only 2% cases were presented as pulmonary nodules\n[7,8]. Moreover, catamenial hemoptysis was documented\nin all the five patients with endobronchial endometriosis\n[2-6], suggesting that patients with tracheal endometriosis\nare prone to hemoptysis. However, in our case, the patient\nonly showed dyspnea and coughing without hemoptysis,\nand the case history gave no other valuable information.\nThe CT scanning and bronchoscopy, which have\nbeen considered to be valuable in diagnosing thorax\nendometriosis, both have a yield in this extremely rare\ncondition. It was reported that asymptomatic pulmonary\nnodules was documented in only 2% patients with\nthoracic endometriosis [7]. While of the 5 cases with\ntracheal endometriosis, no patient presented as pulmonary\nnodules [2-6]. In our case, CT scanning detected a\nround-like lesion in the upper lobe of the left lung close\nto the hilar. The bronchoscopy revealed that there was a\nFigure 1 Bronchoscopic examination showing a pink-red\npolypoid lesion blocked the left main bronchus.\nFigure 2 The endometrial-like glands and stromal cells were found under the bronchial epithelium (A). The ectopic glandular structures\nsurrounded by a densely distributed endometrial-like stroma, and the glands predominantly showed a single layer of columnar cells similar to the\nendometrial epithelial lining with basal nuclei, and no marked cytological atypia could be observed ( B). A, HE×100; B, HE×200.\nYu et al. Diagnostic Pathology 2013, 8:53 Page 2 of 4\nhttp://www.diagnosticpathology.org/content/8/1/53\n\npolypoid lesion in the distal of the left main bronchus.\nAccordingly, the clinical diagnosis was considered as\n“possibly central-type lung cancer ”. Though the broncho-\nscopic biopsy was performed, the result was of little value\nin precluding neoplasm of the lung. Therefore, surgical\ntreatment was imperative.\nOn pathology sections, endometrial glands and stromal\ncells were found under the bronchial epithelium. Further-\nmore, the immunohistochemistry was performed to\nconfirm the diagnosis. The immunohistochemical results\nrevealed that all the epithelium and the glands were\npositive for CK, while the stromal cells were negative.\nAll the epithelium and glandular structures which were\npositive for CK showed no atypia. The alveolar epithelial\ncells were positive for TTF-1. The ectopic glands showed\npositive staining for ER, and the stromal cells were\npositive for both ER and CD10. Taken together, the\npossibility of pulmonary tumors exhibiting atypical\nmicroscopic changes could be ruled out [9,10], and the\ndiagnosis of this case is endobronchial endometriosis.\nIn this case, the patient underwent lobectomy, which\ncould not prevent recurrence. Cancer antigen CA-125 may\nbe useful to monitor the progress of endometriosis [11,12].\nConclusion\nReports of endobronchial endometriosis are extremely\nrare. Diagnosis of etiology remains challenging due to the\nabsence of specific clinical characteristics, especially when\nthe features of CT scanning and bronchoscopy could not\nexclude the possibility of central-type lung cancer. In this\ncondition, to make a right diagnosis can only depend on\npathological examination. Endobronchial endometriosis\nhad to be considered in the differential diagnosis of\n“central-type lung cancer ”, particularly in patients with\nknown endometriosis.\nConsent\nWritten informed consent was obtained from the patient\nfor publication of this case report and accompanying\nimages. A copy of the written consent is available for\nreview by the Editor-in Chief of this Journal.\nCompeting interests\nThe authors declare that they have no competing interests.\nAuthors’ contributions\nYJH and LXY participated in the histopathological evaluation, performed the\nliterature review, acquired photomicrographs and drafted the manuscript.\nFCF and LY carried out the immunohistochemical stains evaluation. ZY\nconceived and designed the study. WEH gave the final histopathological\ndiagnosis and revised the manuscript. WL edited the manuscript. All the\nauthors read and approved the final manuscript.\nReceived: 25 February 2013 Accepted: 26 March 2013\nPublished: 3 April 2013\nFigure 3 The bronchial epithelium and the glands were positive for CK, while the stromal cells were negative (A). The alveolar epithelial\ncells were positive for TTF-1 ( B). The ectopic glands and the endometrial-like stromal cells were positive for ER ( C). The endometrial-like stromal\ncells were positive for CD10 ( D). A-D, SP×200.\nYu et al. Diagnostic Pathology 2013, 8:53 Page 3 of 4\nhttp://www.diagnosticpathology.org/content/8/1/53\n\nReferences\n1. Celik A, Ayd ın E, Yaz ıcı U, Agack ıran Y, Karaoglanoglu N: A rare case of\nhemoptysis: intrapulmonary cavitary lesion appearing as a thoracic\nendometriosis. Case Rep Pulmonol 2012, 201(2):351305.\n2. Hope-Gill B, Prathibha BV: Catamenial haemoptysis and clomiphene\ncitrate therapy. Thorax 2003, 58(1):89–90.\n3. Kuo PH, Wang HC, Liaw YS, Kuo SH: Bronchoscopic and angiographic\nfindings in tracheobronchial endometriosis.Thorax 1996, 51(10):1060–1061.\n4. Butler H, Lake KB, Van Dyke JJ: Bronchial endometriosis and\nbronchiectasis. A possible relationship. Arch Intern Med 1978, 138(6):991–992.\n5. Bateman ED, Morrison SC: Catamenial haemoptysis from endobronchial\nendometriosis–a case report and review of previously reported cases.\nRespir Med 1990, 84(2):157–161.\n6. Rosenberg SM, Riddick DH: Successful treatment of catamenial\nhaemoptysis with danazol. Obstet Gynecol 1981, 57:130–131.\n7. Channabasavaiah AD, Joseph JV: Thoracic endometriosis: revisiting the\nassociation between clinical presentation and thoracic pathology based\non thoracoscopic findings in 110 patients. Medicine (Baltimore) 2010,\n89:183–188.\n8. Visouli AN, Darwiche K, Mpakas A, Zarogoulidis P, Papagiannis A, Tsakiridis K,\nMachairiotis N, Stylianaki A, Katsikogiannis N, Courcoutsakis N, Zarogoulidis\nK: Catamenial pneumothorax: a rare entity? Report of 5 cases and review\nof the literature. J Thorac Dis 2012, 4(Suppl 1):17–31.\n9. Lin XY, Wang Y, Fan CF, Liu Y, Yu JH, Dai SD, Wang L, Wang EH: Pulmonary\nsclerosing hemangioma presenting with dense spindle stroma cells: a\npotential diagnostic pitfall. Diagn Pathol 2012, 7:174.\n10. Xu HT, Lin XY, Li QC, Wang EH: The alveolar epithelial differentiation of\nglandular inner lining cells in a mucoepidermoid carcinoma of the lung:\na case report. Diagn Pathol 2012, 7:137.\n11. Emmanuel R, Léa M, Claude P, Antonio V, Marianne Z, Christophe P,\nChristophe B: Ileocolic intussusception due to a cecal endometriosis: case\nreport and review of literature. Diagn Pathol 2012, 7:62.\n12. Bedaiwy MA, Falcone T: Laboratory testing for endometriosis. Clin Chim\nActa 2004, 340(1–2):41–56.\ndoi:10.1186/1746-1596-8-53\nCite this article as: Yu et al. : Endobronchial endometriosis presenting as\ncentral-type lung cancer: a case report. Diagnostic Pathology 2013 8:53.\nSubmit your next manuscript to BioMed Central\nand take full advantage of: \n• Convenient online submission\n• Thorough peer review\n• No space constraints or color figure charges\n• Immediate publication on acceptance\n• Inclusion in PubMed, CAS, Scopus and Google Scholar\n• Research which is freely available for redistribution\nSubmit your manuscript at \nwww.biomedcentral.com/submit\nYu et al. Diagnostic Pathology 2013, 8:53 Page 4 of 4\nhttp://www.diagnosticpathology.org/content/8/1/53","source_license":"CC0","license_restricted":false}