{"paper_id":"7a85e329-3411-4e78-91f1-bb0d4d18b988","body_text":"American Journal of Medical Case Reports, 2015, Vol. 3, No. 11, 362-366 \nAvailable online at http://pubs.sciepub.com/ajmcr/3/11/4 \n© Science and Education Publishing \nDOI:10.12691/ajmcr-3-11-4 \n \nCatamenial Haemothorax Masquerading Pulmonary \nTuberculosis? A Case Report of a 28 Year Old Woman \nwho Presented with Recurrent Haemothorax and \nEmpyema Thoracis  \nSolomon Gumanga*, Godfred Takyi, Hakeem Ofosu, Sunday Imogie, Bill Koomson \nTamale Teaching Hospital, Ghana \n*Corresponding author: gumangask@yahoo.co.uk \nReceived June 04, 2015; Revised September 25, 2015; Accepted October 09, 2015 \nAbstract  Catamenial haemothorax is a clinical manifestation of endometriosis of the pleural cavity. This is a case \nreport of a 28 year old woman who presented with recurrent catamenial haemothorax who had histological \nconfirmation of pelvic endometriosis and uterine leiomyoma. A presumptive diagnosis of catamenial haemothorax \nwas made as it was not possible to get a histological confirmation of endometriosis of the pleural cavity. She was \ntreated effectively with gonadotropin-releasing hormone GnRH agonist-leuprorelin. Antituberculosis treatment was \nhowever added when she developed empyema thoracis instead of haemothorax at a time she was experiencing \namenorrhoea. Pulmonary tuberculosis being an endemic disease remained the main differential diagnosis but was \nnever confirmed at any stage by investigations conducted. She has since not experienced any recurrence in eighteen \nmonths of follow-up after treatment was completed. Conclusion: Pulmonary or thoracic endometriosis though rare; \nshould be considered seriously in women of reproductive age who present with recurrent catamenial haemothorax. \nDue to obscured locations of the endometriosis lesions in the thoracic cavity; the diagnosis may be difficult to \nconfirm since access to investigative tools is limited in developing countries. \nKeywords: catamenial haemothorax, pulmonary endometriosis, thoracic endometriosis syndrome, extra pelvic \nendometriosis \nCite This Article:  Solomon Gumanga, Godfred Takyi, Hakeem Ofosu, Sunday Imogie, and Bill Koomson, \n“Catamenial Haemothorax Masquerading Pulmonary Tuberculosis? A Case Report of a 28 Year Old Woman who \nPresented with Recurrent Haemothorax and Empyema Thoracis.” American Journal of Medical Case Reports, vol. \n3, no. 11 (2015): 362-366. doi: 10.12691/ajmcr-3-11-4. \n1. Introduction \nEndometriosis is the presence of functional endometrial \ntissue[endometrial glands and stroma  outside the uterine \ncavity [1,2,3], most common locations are dependant parts \nof the female pelvis such as the ovaries,  fallopian tubes, \nuterosacral ligaments, sigmoid colon and pelvic \nperitoneum [1,2,3]. Some e xtra pelvic locations of \nendometriosis include  the umbilicus, abdominal scars, \nkidneys, pleural cavity, lungs,  transverse colon , breasts, \nlymph nodes, nasal mucosa, the arms and legs [1,2,3]. The \ntrue incidence of endometriosis in unknown, however it is \nestimated to be between 5% to 15% of women in their \nreproductive years [1,2,3]. There are several theories to \nexplain the histogenesis of endometriosis but the most \npopular theory is that endometriosis results from \nretrograde menstruation [2,3]. In contrast to the theory of \nretrograde menstruation, is the theory that endometriosis \narises from metaplasia of coelomic epithelium or \nproliferation of embryonic rest and the theory of the \nendometrium being transplanted via the vascular and \nlymphatic systems in rare and remote location [2,4,5]. \nIn thoracic endometriosis, there is functioning endometrial \ntissue in the pleural cavity, lung parenchyma, airways and \ndiaphragm [1,2]. It may manifest as catamenial \npneumothorax, catamenial h aemoptysis, cat amenial \nhaemothorax, catamenial ha emopnuemothorax and lung \nnodules [2,3,5] depending on the location on the \nendometriosis lesions within the thoracic cavity . The true \nincidence of thoracic endometriosis is not known however \n75% of  thoracic endometriosis cases reported present as \ncatamenial pneumothorax whereas catamenial haemoptysis \nand catamenial haemothorax represent 8.3% of cases \n[3,6]. \nIt is estimated that about a third of women hospitalized for \nspontaneous pneumothorax are actually experiencing \ncatamenial pneumothorax [3,7], endometriosis of the lungs \nor pulmonary endometriosis. Catamenial haemothorax is the \nusual clinical manifestation of endometriosis affecting the \npleural cavity [3,8,9].  \nCatamenial h aemothorax presents as haemorrhagic \npleural effusion together with non specific symptoms such \nas pleuric chest pains, cough and shortness of breath \n\n American Journal of Medical Case Reports 363 \n \nwhich may be occurring periodically  with the \nmenstruation[2]. Most of the cases previously reported \nwere diagnosed based on the clinical history of the patient \nas histological confirmation of ectopic endometriosis is \nnot always done [1]. Investigations such as chest x -rays, \nCT, MRT, bronchoscopy may  be useful in diagnosis of  \nconditions in the chest but have limitations  and \ninconsistent findings [1,2]. \nThe treatment of endometriosis can be medical, surgical \nor both [2,3]. Medical treatment is usually the first line of \nmanagement of thoracic endometriosis usuing GnRH \nantagonist such as d anazol, hormonal agents such as  \nprogestatins, oral contraceptive pills, and GnRH analogues \n[2,3]. The use of these drugs for medical  treatment is \naimed at induction of amenorrhea or suppression of ovarian \nfunction. Recurrent bleeding in the ectopic implants is one \nof the most important pathophysiological proce sses to \ninterrupt [3] for an improvement to be noticed . Patients \npresenting with catamenial pneumothorax, catamenial \nhaemopnuemothorax or catamenial haemothorax as in the \ncase reported would initially require surgical intervention \nsuch as thoracocentesis and chest tube placement as first -\nstep in the emergency room until further action is taken  \n[3]. After performing the emergency lifesaving \nthoracocentesis and chest tube placement , when the \npatient’s condition becomes stable, the option of medical \ntreatment can then be initiated. Hysterectomy and bilateral \nsalpingo-oophorectomy is the treatment of last resort \nwhen other options have failed [3], this should be reversed \nfor those in the later ages of the reproductive years and do \nnot want to have any more  children. \n Our experience in the management of this unusual case \nof recurrent catamenial haemothorax is being reported so \nthat attention is drawn to thoracic endometriosis when it \npresents as a clinical entity. \n2. Case Report \nA 28 year old nulliparous woman  was referred for \ngynaecological consultation with a diagnosis of uterine \nleiomyomata and suspected  thoracic endometriosis in \nearly 2013 due to recurrent h aemothorax. She presented \nfive months earlier to the hospital  with fever, chill,  \nanorexia and a productive cough which lasted ab out one \nmouth. She was treated  with broad spectrum antibiotics \nfor right  lobar pneumonia  after investigations that \nincluded sputum negative smear and chest X -rays. \nSubsequently she developed episodes of  haemothorax \noccurring periodically with  her menstruations. She  was \ntreated with broad  spectrum oral antibiotics on three \noccasions after varying amounts  of 1-1.5 litres  of \nhaemorrhagic pleural effusion was drained;  but sputum \nsmears an d laboratory studies of the effusion  remained \nnegative for malignancy, acid fast bacilli [AFB] and other \norganisms. Chest x-rays however confirmed right sided \npleural effusion on all three occasions  before \nthoracocentesis and placement of chest tube was \nperformed. She experience monthly recurre nce of \nhaemothorax during her mense s and also lost up to 12kg \nof weight during 5-6 months  duration of illness but she \ndid not have recurrent fever , persistent cough or \nhaemoptysis. A s the diagnosis of pulmonary tubercu losis \ncould not be confirmed , she was referred  for \ngynaecologist and card iothoracic specialist consultation s \nand investigation on suspicion of thoracic endometriosis \nor malignancy.  \nShe did not have any family history of significance and \nthere was no p ositive history of contact with tuberculosis. \nShe was singled, lived alone in  an apartment and did not \nsmoke tobacco or drink alcohol . Her menarche was at 11 \nyears, she had a menstrual cycle length of 24-26 days and \nbleeds for 7 days . She  had positive history of \ndysmenorrhea and chronic pelvic pain  of three years \nduration, and had episodes right sided chest pain  which \nradiated to her shoul ders during her menses which was  \nmanaged on analgesics a few months before  the monthly \nrecurrences of haemothorax. She did not experience  \nvaginal discharge, intermenstrual bleeding, or dyspareunia.  \nOn examination, she was calm and  well looking, body \nmass index of 21.1kg/m2 with no pallor, jaundiced or \nfever. The thyroid gland and both breasts were normal \nwith no peripheral lymphadenopathy. Her respiratory was \n20 cycles per minute with reduced  air entry on the right \nmiddle and lower lobes  with dull percussion note. Air \nentry was adequate on the left lung with vesicular  breath \nsounds and no crepitation. Other findings including the \ncardiovascular and gastrointestinal  systems, liver, both \nkidneys and  the spleen were all normal. There was a \nmulti-nodular mass in the lower abdomen arising from the \npelvis about 26 week’s size gestational uterus. The mass \nwas not tender and there was sign of free fluid in the \nabdomen. The vulva and vagina were normal and t he \ncervix was closed and posterior.  The adnexae and pouch \nof Douglas were normal and there was no blood or vaginal \ndischarge noticed. \nSummary of results of some investigations done during \nthe period before her referral included normal \nhaematological investigations, negative sputum for Acid \nFast Bacilli[AFB ], routine examination and culture of \npleural aspirate  were all negative for malignancies and \ninfections. She however had ultrasound reports confirming \nleimyoma of the uterus.  M ultiple chest x -rays and CT -\nscan showing  previous episodes of right sided \nhaemothorax \nFigure 1a, Figure 1b, Figure 2a and Figure \n3a and repeat chest x -rays showing normal findings after \nthoracosentesis and chest tube drainage  as in Figure  2b \nand Figure 3b below.  \n \nFigure 1a. CT scan of the chest and abdomen s howing massive right \npleural effusion \n\n364 American Journal of Medical Case Reports  \n \n \nFigure 1b. Section of CT scan of the chests showing massive right \npleural effusion and normal left lungs  \n \nFigure 2a. Chest  X-ray  showing massive right pleural effusion . March \n2013 \n \nFigure 2b. Chest  X-ray  showing drainage of  right effusion  4 days after \nchest tube placement \nAfter the gynaecological and cardioth oracic surgical \nspecialist consultation, the need for additional \ninvestigations including a repeat of CT-Scan or MRI of \nthe chest, and possibly  tissue biopsies of any lesions \nwithin the pleural cavity  for histological studies  were \ndiscussed with her. She was not prepared financially for \nthese additional investigations when  she experienced a \nrecurrent episode of haemothorax with severe resp iratory \nembarrassment on the last day of her menses in May 2013. \nShe was admitted  to the intensive care u nit where \nthoracostomy was performed and  chest tube was retained \nfor five days after chest x -rays confirmed the recurrence \nof right haemothorax with left side remaining normal . An \nultrasound scan done showed multiple uterine myomas  \nwith some significant amount of free fluid in the abdomen \nwhich was not obvious from clinical examination. \n \nFigure 3a. Chest  X-ray  showing massive right pleural effusion. May \n2013 \n \nFigure 3b. Chest  X-ray  showing drainage of  right effusion after chest \ntube placement \nShe was given prophylactic parenteral broad spectrum \nantibiotics and analgesics for 72hrs and was transfuse d \nthree pints of whole blood when haemoglobin was noticed \nto be 8.0g/dl  and was symptomatic for anaemia . Other \nlaboratory investigations done included serological test for \nhuman immunodeficiency virus , repeat routine \nexaminations and cultures of pleural effusion were \nnegative for malignancies, AFB and other bacterial \ninfections. The pleural effusion was com pletely drained  \nand exploratory laparotomy and drainage of the free fluid \nin the ab domen was performed.  Finding at laparotomy \nwas some 18 0-200mls of chocolate brown colored fluid \nwhich was drained. Other significant finding include d the \nmulti nodular uterine myomas and severe  pelvic \nendometriosis confirmed by histopathology of biopsies of \ntissues taken from different sites within the pelvis.  \nThe multi-displinary team of specialist upon discussion \nagreed on a presumptive diagnosis of thoracic \nendometriosis with catamenial pneumothorax and the need \nto commerce an effective treatment using GnRH \nanalogues [leuprorelin]. \n\n American Journal of Medical Case Reports 365 \n \nThe treatment of GnRH analogues injections  \n[leuprorelin] were given monthly for three months. Three \nweeks after comme nt of the GnRH analogues treatment \nshe developed empyema pleurae instead of haem othorax. \nShe had thoracostomy  and insertion of draina ge tube \nwhich drained about 1200mls of purulent effusion, \nlaboratory investigations conducted on the purulent pleural \naspirate still did not confirm pulmonary tuberculosis ; \nantituberculosis treatment was however also initiated as a \nprecautionary measure. She had myomectomy successfully \nperformed five months  after commencing treatment with \nGnRH analogues when the size of the uterine leiomyomas \nregressed to about 16 weeks gestation. The adhesions in \nthe pelvis had also regressed with disappearance of much \nof endometriosis lesion s seen during the earlier \nlaparotomy. Her m enses resumed about six weeks after \nthe last GnRH analogue injection; it was heavy and lasted \nfor five days and  was treated with tranexa mic acid. She \nhas since had regular menstruation established without \ndysmenorrhea or recurrence of haemothorax and she is \nalso not experiencing chronic pelvic pains either during \nmore than eighteen months of follow-up.  \n3. Discussion \nThis is a case of recurrent catamenial haemothorax due \nto thoracic endometriosis in a 28 year old nulliparous \nwoman was managed successfully with GnRH agonist \n[leuprorelin]. The  presumptive diagnosis of thoracic \nendometriosis was based on recurrent  catamenial \nhaemothorax; a condition which was  not confirmed by \nhistology since tissue could not be taken from the pleural \ncavity. Results of cytological studies of the effusion were \nnot diagnostic of endometriosis, malignancy or pulmonary \ntuberculosis.  \nA history of recurrent catamenial heamoptysis or \ncatamenial haemothorax when pulmonary tuberculosis \ncannot be confirmed  should draw attention for  the \ndiagnosis of pulmonary or thoracic endometriosis to be \nconsidered. The diagnosis of extra -pelvic endometriosis \nmay be difficult or delayed[2] as in this case reported due \nto the rare nature of the condition  or the patient’s \nsymptoms in relation  to the menstrual cycle may not be \napparent [1]. Pulmonary endometriosis o r thoracic \nendometriosis usually presents in women of the \nreproductive age group with a mean age of 34 years with \nover 90% of the lesions  located within the right \nhemithorax and 14% presenting as  catamenial \nhaemothorax [2,3,7,12]. Some 50-84% of women with  \npulmonary or thoracic endometriosis have a history of \npelvic endometriosis with the symptoms of pelvic \nendometriosis preceding some 5 -7 years [3,7,12]. Though \nhistological confirmation of thoracic endometriosis was \nnot obtained, there was however histological confirmation \nof pelvic endometriosis  in this same case. The clinical \nsymptoms of catamenial haemothorax and effectiveness of \ntreatment with GnRH analogue  provides basis for the \ncorrectness of the presumptive diagnosis.  Treatment with \nGnRH analogue provided dual advantage of shrinking the \nlarge uterine myomas and suppressing symptoms of pelvic \nendometriosis making th e myomectomy procedure easier \nfor the large myomas which presented as an additional \npathology.  \nImaging studies or bronchoscopy during men ses and \nrepeated during the midcycle may assist in the diagnosis \nof pleural or bronchopulmonary disease. The disappearance \nof the previous findings strengthens the clinical suspicion  \n[13]. The C T findings for pulmonary endometriosis may \ninclude well -defined opacities, nodular lesions, thin -wall \ncavities, or bullous formations, transient radiologic \ndensities in the affected part of  the lung [1,14]. Chest X-\nrays were done during the management of this case and it \nonly provided value in diagnosis of the pleur al effusion \nand its drainage but not its cause.  \nAlthough antituberculosis treatment was added wh en \nshe developed empyema thoracis  instead of haemothorax \nat a time she was experiencing amenorrhoea, the cause of \nthe empyema may be from bacteria infection du e to the \nthoracocentesis and insertion of drainage tube on several \noccasions as the diagnosis of pulmonary tuberculosis was \nstill not confirmed by any of the investigations that were \nconducted on the purulent pleural aspirate.  Since thoracic \nendometriosis is not common, the diagnosis may be \ndelayed or missed as the symptoms are not specific. \nPulmonary tuberculosis may be the primary focus of \ninvestigations and pose a differential diagnostic challenge \nin tuberculosis endemic parts of the world when thoracic  \nendometriosis presents as catamenial hemoptysis as in a \ncase reported by Yuz et al [11] or catamenial haemoptysis \nas in this case report It is important a presumptive \ndiagnosis be made based on symptoms and recurrence of \nperiodic haemothorax at the time of  menstruation so that \ntreatment options are considered early.  \nPatients with thoracic endometriosis usually un dergo \neither surgery or medical  treatments with GnRH agonists \nconsidered more effective in controlling recurrences of \ncatamenial pneumothorax, par ticularly when used for \nprolonged periods of up to 1 year  \n[3,15,16] though no \nlarge-scale randomized trial has been conducted and the \noptimal treatment regimen still remains c ontroversial \n[1,4,16]. \n4. Conclusion \nPulmonary or thoracic endometriosis though rare;  \nshould be considered seriously in women of reproductive \nage who present with recurrent catamenial haemothorax. \nDue to obscured locations of the endometriosis lesions in \nthe thoracic cavity; the diagnosis may be difficult to \nconfirm since access to investi gative tools is limited in \ndeveloping countries where other diseases such as \npulmonary tuberculosis  could masquerade the condition \nareas where it is endemic. \nReferences \n[1] Huang et al. Endometriosis of the lung: report of a case and \nliterature review. Eu ropean Journal of Medical Research.  2013, \n18:13. http://www.eurjmedres.com/content/18/1/13 . \n[2] Parisa Azizad -Pinto, David Clarke, Thoracic endometriosis \nSyndrome: Case Report and Review of the literatur e: Perm J 2014 \n18[3]: 61-65. http:/dx.doi.org/10.7812/TPP/13 -154. \n[3] Camran Nezhat, Babak Hajhosseini, Elizabeth Buescher, Asrafjah \nHussein, Georgios E. Hilaris, Michal Sellin. Thoracic \nEndometriosis Syndrome.  \nhttp://laparoscopy.blogs.com/prevention_management_3/2011/01/\nthoracic-endometriosis.html. \n\n366 American Journal of Medical Case Reports  \n \n[4] Droegenmueller W. Endometriosis and Adenomyosis. In: \nStenchever MA, Droegenmueller W, Herbst AL, Mishell DR, \neditors. Comprehensive Gynecology. St. Louis: Mosby; 2001;  \n531-584. \n[5] Okeke TC, Ikeako LC, Ezenyeaku CC: Endometriosis. Niger J \nMed 2011, 20[2]: 191-199. \n[6] Roberts LM, Redan J, Reich H; extraperitoneal endometriosis with \ncatamenial pneumothoraces, a review of t he literature . JSLS 2003 \n7[4]: 371-375. \n[7] Joseph J, Sahn SA. Thoracic endometriosis syndrome: new \nobservations from an analysis of 110 cases. Am J Med. Feb 1996; \n100[2]:164-170. \n[8] Jubanyik KJ, Comite F. Extrapelvic endometriosis.  Obstet \nGynecol Clin North Am. Jun 1997;  24[2]:411-440. \n[9] Nezhat CR BG, Nezhat F, Buttram VC Jr., Nezhat CH. \nEndometriosis: Advanced Management and Surgical Techniques . \nNew York: Springer-Verlag. \n[10] Alifano M, Trisolini R, Cancellieri A, Regnard JF. Thoracic \nendometriosis: current knowledge. Ann Thorac Surg. 2006;  81[2]: \n761-769. \n[11] Yu Z , Fleischman JK , Rahman HM , Mesia AF , Rosner F .  \nCatamenial he moptysis and pulmonary endometriosis: a case \nreport. Mt Sinai J Med. 2002; 69[4]: 261-3. \n[12] Korom S, Canyurt H, Missbach A, et al. Catamenial \npneumothorax revisited: clinical approach and systematic review \nof the literature. J Thorac Cardiovasc Surg. Oct 2004;  128[4]: 502-\n508. \n[13] Hope-Gill B, Prathibha BV. Catamenial haemoptysis and \nclomiphene citrate therapy. Thorax. Jan 2003; 58[1]: 89-90. \n[14] Orriols R, Muna z X, Alvarez A, Sampol G: Chest CT scanning: \nutility in lung endometriosis. Respir Med 1998, 92[6]:  876-877. \n[15] Hilaris GE NC. Endometriosis and pelvic pain. In: Wetter PA KM, \nLevinson CJ, Kelley WE Jr, McDougall EM, Nezhat C, ed. \nPrevention and Management of Laparoendoscopic Surgical \nComplications. Miami: The Society of Laparoendoscopic \nSurgeons; 2005. \n[16] Augoulea A, Lambrinoudaki I, Christodoulakos G: Thoracic \nendometriosis syndrome. Respiratory 2008; 75[1]: 113-119.","source_license":"CC0","license_restricted":false}