{"paper_id":"fb2cbd22-8cef-41fd-863f-b673907c568b","body_text":"A Case of Catamenial Hemoptysis Treated by \nBronchial Artery Embolization\nSuk Pyo Shin, M.D.\n1\n, Chi Young Park, M.D.\n1\n, Ji Hyun Song, M.D.\n1\n, Hong Min Kim, M.D.\n1\n, Daniel \nMin, M.D.\n1\n, Sang Hwan Lee, M.D.\n1\n, San Ha Kang, M.D.\n1\n, Gyeong Sik Jeon, M.D.\n2\n and Ji-Hyun Lee, \nM.D.\n1\nDepartments of  \n1\nInternal Medicine and \n2\nRadiology, CHA Bundang Medical Center, CHA University, Seongnam, Korea\nCatamenial hemoptysis is a rare condition, characterized by recurrent hemoptysis associated with the presence \nof intrapulmonary or endobronchial endometrial tissue. Therapeutic strategies proposed for intrapulmonary \nendometriosis with catamenial hemoptysis consist of medical treatments and surgery. Bronchial artery embolization \nis a well-established modality in the management of massive or recurrent hemoptysis, but has seldom been used \nfor the treatment of catamenial hemoptysis. We report a case of catamenial hemoptysis associated with pulmonary \nparenchymal endometriosis, which was successfully treated by a bronchial artery embolization.\nKeywords: Endometriosis; Hemoptysis; Embolization, Therapeutic\nnary parenchyma or in the airway\n3\n.\nVarious treatment modalities such as hormonal therapy, \nsurgery or medical conservative treatment have been at -\ntempted, but controversies exist about optimal management \nof catamenial hemoptysis. Bronchial artery embolization \n(BAE) is a well established minimally invasive treatment \nmodality for hemoptysis and few have been reported for the \nmanagement of catamenial hemoptysis. Here, we describe a \ncase of catamenial hemoptysis caused by pulmonary paren -\nchymal endometriosis successfully treated with BAE.\nCase Report\nA 34-year-old married woman was admitted to pulmonary \ndepartment with a 4-day history of hemoptysis. Hemoptysis \noccurred from the 3rd day of menstruation till 6th day and the \ntotal amount of hemoptysis was about 150 mL. She had no \nhistory of previous hemoptysis event. She had medical history \nof an appendectomy 20 years ago and pelvic inflammatory \ndisease 2 years ago. She gave birth by normal spontaneous \nvaginal delivery (gravida 1, para 1) 10 years ago and she had \nnot had a past history of obstetric or gynecological procedures \nbefore developing hemoptysis. Her medical history was oth -\nerwise unremarkable and she did not have a significant family \nhistory. She denied using smoking, excessive alcohol and illicit \nCopyright © 2014\nThe Korean Academy of Tuberculosis and Respiratory Diseases.\nAll rights reserved.\nIntroduction\nThoracic endometriosis is a rare disorder characterized by \na presence of functional endometrial tissue within the pleura, \nthe lung parenchyma or the airway\n1\n. The tissue is responsive \nto circulating sex hormones and clinical manifestations are re-\nlated to the menstrual cycle. Clinically, thoracic endometriosis \nincludes four well-recognized entities, namely, catamenial \npneumothorax, catamenial hemothorax, catamenial hemop-\ntysis, and lung nodules\n2\n. In catamenial hemoptysis, the source \nof bleeding is an endometrial implant located in the pulmo -\nCASE REPORT\nhttp://dx.doi.org/10.4046/trd.2014.76.5.233\nISSN: 1738-3536(Print)/2005-6184(Online) • Tuberc Respir Dis 2014;76:233-236\n233\nAddress for correspondence: Ji-Hyun Lee, M.D.\nDivision of Respiratory and Critical Care Medicine, Department of \nInternal Medicine, CHA Bundang Medical Center, CHA University, 59 \nYatap-ro, Bundang-gu, Seongnam 463-712, Korea\nPhone: 82-31-780-6140, Fax: 82-31-780-6143\nE-mail: plmjhlee@cha.ac.kr\nReceived: Oct. 14, 2013\nRevised: Nov. 5, 2013\nAccepted: Nov. 21, 2013\ncc It is identical to the Creative Commons Attribution Non-Commercial \nLicense (http://creativecommons.org/licenses/by-nc/3.0/).\n\nSP Shin et al.\n234 Tuberc Respir Dis 2014;76:233-236 www.e-trd.org\ndrugs. She had no complaints of weight loss, fever, dyspnea, \npalpitations, gastrointestinal symptoms or a history of bleed-\ning. Her physical examination was within normal limits. Chest \nX-ray (Figure 1) had no abnormal findings. Chest computed \ntomography (CT) scan was performed on the 2nd hospital \nday, which was fourth day of menstruation and the CT scan \nshowed a focal consolidation with adjacent ground glass \nopacity (GGO) in the posterior basal segment of the left lower \nlobe (Figure 2A), but there’ s no evidence of enlarged bronchial \nartery or vascular abnormality. Fiberoptic bronchoscopy \nshowed a small amount of blood clot in the bronchi of left \nlower lobe. No endobronchial lesion was detected during the \ninvestigation. Bronchial washing fluid showed no acid fast \nbacilli, bacteria or abnormal cells. Hemoptysis was spontane-\nously resolved with the cessation of menstruation and did not \nrecur during the rest of admission period. We assumed the \nillness as catamenial hemoptysis due to pulmonary endome-\ntriosis, and she was discharged. After discharge from the first \nadmission, follow-up chest CT scan was performed for follow-\ning up the lung lesion at 26th day of menstrual cycle. Previ -\nously noted focal consolidation with adjacent GGO lesion was \nalmost disappeared and only a noncalcified 5-mm-sized nod-\nule was left (Figure 2B). Four days passed and at the first day \nof the next menstrual cycle, hemoptysis recurred. The amount \nof hemoptysis ranged 100 to 150 mL and she was admitted \nagain. Chest CT scan was performed and reappeared larger \nnodule with focal GGO was seen at the same location (Figure \n2C). T o control the bleeding, we decided to perform bronchial \narteriography embolization. Digital subtraction technique \nwith a digital subtraction angiography unit was used. Femoral \nartery was punctured, then thoracic aortography and followed \nselective left bronchial arteriography were done with a 5-Fr \ncatheter. Diagnostic angiography showed a small nodular \nstaining on left lower lung zone (Figure 3A) that corresponds \nwith the lesion of previous chest CT scan. After determination \nof the pathologic vascularity, embolization was performed \nwith 355 to 500 μm Contour polyvinyl alcohol particles. After \nembolization, control angiography showed occlusion of the \nartery feeding the lesion (Figure 3B). There was no more he -\nmoptysis after the embolization. After discharge, she has been \nfollowed up for 5 months without hemoptysis. We diagnosed \nthis case as catamenial hemoptysis because the hemoptysis \nevents and size change of the lesion on chest CT scans were \nsynchronized with the menstruation cycle, and both the CT \nscan and the bronchoscopy excluded other possible causes of \nhemoptysis. \nDiscussion\nCatamenial hemoptysis is a cyclic pulmonary hemorrhage \nFigure 1. Initial posteroanterior chest radiograph shows no specific \nabnormality on the lung field. \nFigure 2. (A) Chest computed tomography (CT) scan performed on 4th day of menstruation shows focal consolidation with surrounding ground \nglass opacity in the posterior basal segment of the left lower lobe. (B) In the CT scan on the 26th day of menstruation, previously noted focal con-\nsolidation with surrounding ground glass opacity in the left lower lobe has been resolved. Residual small noncalcified nodule is noted. (C) In the \nCT scan on the 1st day of the next menstruation, focal ground glass opacity and nodule is noted at the same location as previous one. \n\n\nBronchial artery embolization in catamenial hemoptysis\nhttp://dx.doi.org/10.4046/trd.2014.76.5.233\n 235www.e-trd.org\nthat is synchronized with female menstruation, which is asso-\nciated with the presence of intrapulmonary or endobronchial \nectopic endometrial tissue. The mechanism regarding tho -\nracic endometriosis is not fully understood and three theories \nhave been proposed to explain the presence of intrathoracic \nendometrial implants: coelomic metaplasia, retrograde men-\nstruation with subsequent transperitoneal-transdiaphragmat-\nic migration of endometrial tissue and lymphatic or hematog-\nenous embolization from the uterus or pelvis\n4\n. None of these \ntheories can explain all the clinical manifestations, and the \ndisease probably has a multifocal etiology. The theory of coe-\nlomic metaplasia is based on the concept that both endome-\ntrium and pleural mesothelium share the same embryologic \norigin. Pathologic stimuli induce precursor cells of the pleura \ninto endometrial cells. The second theory is based on the con-\ncept that movement of fluids in the peritoneal cavity follows \npredictable patterns: namely “peritoneal fluid circulation.” It \nimplies a preferable flow direction from the pelvis to the right \nsubdiaphragmatic area through the right paracolic gutter\n5\n. \nDuring the process of the flow, endometrial tissue could enter \ninto the thorax through either congenital or acquired dia -\nphragmatic defects\n5,6\n. However, these theories cannot explain \nthe occurrence of intrapulmonary endometriosis. The last \ntheory of transplantation of endometrium through lymphatic \nor vascular embolization can explain intrapulmonary endo -\nmetriosis. Trauma or manipulation of uterine tissue would \nbe a factor predisposing to microembolization. For example, \nthe nationwide Korean report of 19 patients with catamenial \nhemoptysis showed that 16 (84%) patients had a history of \nobstetric or gynecological procedures before development \nof hemoptysis\n7\n. Also, in a study that followed 4 patients with \ncatamenial hemoptysis, all patients had history of one or two \ndilatations and curettages before diagnosis of catamenial \nhemoptysis\n8\n. However, this patient had not had a past history \nof obstetric or gynecological procedures before developing \nhemoptysis.\nThe diagnosis of pulmonary parenchymal endometriosis \nis usually assumed on the basis of the clinical history and \nthe exclusion of other causes of recurrent hemoptysis. Chest \nroentgenogram may reveal pulmonary opacities or nodular \ninfiltrates, but findings could be normal even in a patient with \ncurrent bleeding\n4\n. The diagnostic use of bronchoscopy is lim-\nited, because most cases of pulmonary endometriosis involve \nthe distal pulmonary parenchyma rather than the mucosa of \nlarge bronchi and also the bleeding site may only be apparent \nduring menstruation. Chest CT scan is useful for detection of \nthe lesion and exclusion of other causes for hemoptysis. CT \nsigns of pulmonary endometriosis include ill-defined or well-\ndefined nodules, thin-walled cavities, bullous formations and \nground glass opacities\n2\n. These lesions, which are expressions \nof the endometrial implants and/or secondary hemorrhage, \nmay change in size during the menstrual cycle\n4,8\n. The lesion \non CT scans in this patient also showed the characteristic \nchange according to her menstrual cycle.\nThere is no guideline for the treatment of catamenial he -\nmoptysis. Hormonal therapy has been considered as the first \nchoice in patients with thoracic endometriosis. It includes oral \ncontraceptives, progestational drugs, danazol, and gonado -\ntropin-releasing hormone agonists which suppress the endo-\nmetrial tissue. It has been proved to be effective in controlling \nsymptoms, but controlled trials on the efficacy of these drugs \nare lacking. Also, heavy side effects of the hormonal therapy \nhave been observed and symptoms often recur after discon -\ntinuation\n7\n. Moreover patients who consider pregnancy cannot \ntake these drugs. \nMedical conservative management could be another op -\nFigure 3.  (A) Left bronchial angiography \nshows a small nodular staining in left \nlower lung field (arrow). (B) Left bron -\nchial angiography after embolization \nshows no distal flow and disappearance \nof nodular staining. \n\n\nSP Shin et al.\n236 Tuberc Respir Dis 2014;76:233-236 www.e-trd.org\ntion, because most of the hemoptysis events associated with \npulmonary emdometriosis are not lethal and most patients \nfrom case series are almost women of childbearing ages. In \na study reported in Korea, 4 patients taken only conservative \nmanagement had been followed approximately for five years \nand hemoptysis spontaneously disappeared after several epi-\nsodes of minor bleeding\n8\n.\nSurgical treatment has been advocated if medical treatment \nfails, intolerable drug-related side effects occur, or symptoms \nrecur after the cessation of hormonal therapy\n3\n. Wedge resec-\ntion or lobectomy can be applied to these cases according to \nthe extent and location of the lesion. Video-assisted thoraco -\nscopic surgery, endoscopic laser treatment\n9\n or open surgery \ncan be done\n7\n.\nIn our case we had observed the patient after the first epi -\nsode, but the hemoptysis recurred in the next menstruation \ncycle. Since the amount of bleeding was more than that of \nthe previous episode, we decided to try BAE prior to surgery. \nIn general, BAE is a well-known alternative to surgery in the \nmanagement of hemoptysis. Clinically, BAE has been widely \napplied for the treatment of hemoptysis caused by bron -\nchiectasis, tuberculosis, aspergillosis, lung cancer or chest \ntrauma. Possible rare complications of BAE are spinal cord \ninjury, esophageal ulceration, stroke, bronchial infarction and \ntransient chest pain. BAE may be more lifesaving and provide \nbetter long term control of recurrent bleeding and give better \nquality of life than medical conservative management alone \ndoes in massive or even if not massive but socially or physi -\ncally recurrent troublesome hemoptysis\n10,11\n.\nBlood supply of lung parenchymal endometriosis has not \nbeen well described. In some case reports or case series, \npathologic findings of the removed pulmonary endometrial \ntissues revealed the presence of expanded bronchovascular \nbundles\n12\n or thin-walled large capillaries or bronchial arter -\nies\n13\n.\nDespite these findings, BAE has not been frequently used \nto control catamenial hemoptysis. We found only one report \nfrom Kervancioglu et al.\n14\n, in which hemoptysis with multiple \npulmonary endometriosis was successfully treated by BAE \nwithout recurrence for 3 months of follow-up. On the other \nhand, Katoh et al.\n15\n found no abnormalities on bronchial and \npulmonary angiograms in their clinically suspected pulmo -\nnary endometriosis patients. In this case, we found a small \nnodular staining in bronchial angiogram matched to the \nlesion on the chest CT scan. After BAE, the patient has not \nshown hemoptysis during the follow up period of 5 months \nwith regular menses. In conclusion, we reported a patient of \ncatamenial hemoptysis treated with BAE. This case suggests \nthe possibility that BAE might be an alternative therapeutic \nstrategy for the patient with catamenial hemoptysis of intra -\npulmonary endometriosis. \nConflicts of Interest\nNo potential conflict of interest relevant to this article was \nreported.\nReferences\n1. Honore GM. 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Thorax \n1968;23:434-7.\n14. Kervancioglu S, Andic C, Bayram N, Telli C, Sarica A, Sirikci A. \nBronchial artery embolization in the management of pulmo -\nnary parenchymal endometriosis with hemoptysis. Cardio -\nvasc Intervent Radiol 2008;31:824-7.\n15. Katoh O, Yamada H, Aoki Y, Matsumoto S, Kudo S. Utility of \nangiograms in patients with catamenial hemoptysis. Chest \n1990;98:1296-7.","source_license":"CC0","license_restricted":false}