{"paper_id":"47f7c34e-6600-42d1-afef-c338659c2074","body_text":"10 \nInternational Journal of Reproduction, Gynaecology and Obstetrics \nOnline ISSN: 2706-5464, Print ISSN: 2706-5456 \nReceived: 28-11-2020; Accepted: 11-12-2020; Published: 26-12-2020 \nwww.gynecologyjournal.in \nVolume 2; Issue 1; 2020; Page No. 10-13 \nMedical management of bronchial endometriosis: Case report and literature review \nMohamed Abdelrahman*, Amy Worrall, Feras J Elkharouf, Conor Harrity \nGynecology Registrar, Department of Gynecology, Royal College of Physicians of Ireland, Ireland \n \n \nAbstract \nWe are presenting 29 year old female complaining of catamina l haemoptysis 3 months post -delivery of her second child. She \nhad no previous history of endometriosis or chronic  pelvic pain. She was investigated thoroughly by re spiratory and \nimmunology teams for all possible diagnosis and clinically the final diagnosis  was bronchial endometriosis. This case showed \nthat combined oral contraceptive pills has successful role of management of lung endometriosis. \n \nKeywords: bronchial endometriosis, post-delivery, respiratory, immunology \n \nIntroduction \nEndometriosis is the presence of functional endometrial-like \ntissue outside of the uterus  It is most commonly in the \novaries, uterosacral ligaments, and pelvic peritone um the \ncondition was first reported in 1860  [1]. Endometr iosis is \nreported in 5% to 15% of females worldwide during t heir \nreproductive life [2]. In some rare cases ectopic endometrial \ntissue has been found in distant sites, such as the  umbilicus, \nabdominal scars, breasts, the extremities, pleural cavity and \nlung parenchyma [3, 4] . The symptoms of extra -pelvic \nendometriosis are not always synchronous with the \nmenstrual cycle, and diagnosis can be particularly difficult.  \nBronchial endometriosis is an uncommon condition, a nd \nusually associated with cyclical haemoptysis and chest pain. \nDiagnosis mainly depends on clinical suspicion base d on \npresenting symptoms and history, with confirmation by \nhistopathological assessment [5]. Clinical examination often \nreveals only occult signs and symptoms. Reported cases that \nhad cyclical pain and haemoptysis often have severe \ndecidual adhesions and distortion of tissue around the \ndecidua [6]. Additionally, the pathologist’s role is made \ndifficult by the atypical histopathological feature s, from \ntypical endometrial glands to an abundance of fibrous tissue, \non a background of lunch parenchyma and bronchial tissue. \nThe major modality of treatment for bronchial \nendometriosis is surgical excision through bronchos copy, \nwhich usually provides a positive outcome  [7]. Medical \nmanagement such as use of contraceptive progestogen s, \ngonadotropin-releasing hormone agonists, androgens, and \nnon-steroidal anti-inflammatory drugs have been used with \nmixed effect, and often only have benefits for limi ted time \nperiods, and the long term side effects of chronic use [8]. \n \nCase Report \nA 27 year old female, Para 2+0 presented to a terti ary \nuniversity hospital complaining of several episodes  of \ncyclical cataminal haemoptysis (coughing blood duri ng \nmenstruation). The patient had a prior uncomplicate d \nspontaneous normal vaginal delivery of her second child 3 \nmonths previously, with a routine postnatal course.  Her \nmenses resumed eight weeks post -delivery. Despite having \nno pre-existing respiratory problems, she developed severe  \nnew onset pulmonary bleeding post-partum, coinciding with \nmenstruation. Each episode of haemoptysis was estimated at \n15ml volume with each period, and she presented to acutely \nto the hospital emergency department and required i npatient \nadmission each time. The patient never reported any cyclical \npelvic pain, or abnormal gynaecological symptoms. She was \ninitially assessed by the respiratory physicians, a s each \nepisode accompanied with significant dyspnoea. On \nphysical examination, right -sided coarse crepitations with \ndecreased air entry was noted on auscultation. PA Chest \nradiography demonstrated an opacity and regular mar kings \nin the right lower lung lobe. All haematological an d \nbiochemical investigations, including full blood co unt, c -\nreactive protein, liver function tests and renal fu nction tests, \nwere normal. To further assess, a CT pulmonary angi ogram \n(CTPA) was arranged. This identified a focal area o f \n“ground glass” changes within the superior segment of the \nright lower lobe, and was deemed to likely be eithe r \ninfectious or inflammatory  by the reporting radiologist. No \npulmonary embolus, plural effusion, thoracic \nlymphadenopathy or osseous abnormality were seen. S he \nwas started on broad spectrum antibiotics (pending \nmicrobiological analysis of sputum culture) with th e \nrecommendation to r epeat the CT thorax in 4 -6 weeks, and \ndischarged home for outpatient follow up with the report. \nA month after the initial presentation she re -attended the \nemergency room with heavy respiratory bleeding and \nshortness of breath, again with menstruation. A re peat \nCTPA was performed, and interestingly this showed a n \nincrease in the previously identified “ground -glass” changes \nwith new extension into the anterior aspect of the apical \nsegment of the right lower lobe. No evidence of pul monary \nhaemorrhage or nodule s were seen. During this repeat \nCTPA the patient had active haemoptysis. Following review \nand discussion at the regional respiratory multi -disciplinary \nmeeting a bronchoscopy was recommend, and subsequen tly \ncompleted during this emergency admission. \nBronchoalviolar lavage (BAL) (it is a diagnostic method of \nthe lower respiratory system in which a bronchoscop e is \npassed through the mouth or nose into an appropriate airway \nin the lungs, with a measured amount of fluid intro duced \nand then collected for examination) was performed, and \nhistology revealed an abundant alveolar presence. \n\nInternational Journal of Reproduction, Gynaecology and Obstetrics  www.gynecologyjournal.in \n11 \nEosinophils were not identified on May  Grünewald-Giemsa \n(MGG) Stain (MGG is stain used for staining of blood, bone \nmarrow smears and clinical cytological specimens), and no \nmalignant cells were seen. A full immunology screen was  \nthen completed including: Anti -neutrophil cytoplasm \nantibodies (ANCA) and DNA by crithidia luciliae (it  is a \nflagellate parasite that uses the housefly,  Musca domestica, \nas a host) [20]. As part of the fa mily of Trypanosomatidae, it \nis characterised by the presence of a  kinetoplast, a complex \nnetwork of interlocking circular  double-stranded DNA  (ds \nDNA) molecules, which were negative. Complement C3 \nand C4 levels were within normal range. \nThe respiratory team at this stage noted that the haemoptysis \nwas cataminal – as it occurred recurrently during the days \nwhen the patient was actively menstruating. A gynae cology \nconsultation was then sought. She had no previous \ngynaecological history of note. A recommendation to  \ncommence the patient on a course of triptorelin pam oate \n(Decapeptyl) 11.25mg injection for 3 months to achi eve \novarian and menstrual suppression was made by the \nattending consultant. Due to the possibility of \nhypoestrogenic vasomotor symptoms, and po tential \nosteopenia if longer term use was needed, the abili ty to \nincorporate add -back hormone replacement therapy (HRT) \nfor symptom control and bone-protection was discussed. An \nultrasound pelvis was performed to assess for struc tural \nabnormalities, but wa s unremarkable. A provisional \nworking diagnosis of pulmonary endometriosis was no w \nmade, and an MRI Thorax was arranged to further \ninvestigate. Unusually this showed no T1 hyper -intensities \nwithin the lung parenchyma or plural or diaphragm t o \nsuggest endometriosis deposits (figure 3, 4).  \nNo episodes of cataminal haemoptysis occurred in th e 3 \nmonths followed the triptorelin pamoate injection. A \nsubsequent CT thorax showed complete resolution of \n“ground-glass” opacification, with appearance suspicious \nfor prior pulmonary haemorrhage in the anterior basal low er \nlobe, but with no acute findings suspicious for cur rent \npulmonary haemorrhage (figure 1,  2). She had minimal \nhormonal side effects from the ovarian suppression,  with \nonly mild but tolerable episodes of warm flushing and minor \nmood change. \nFor long term management a patient centred discussi on was \nhad outlining risks and benefits of the treatment o ptions. \nWith no personal contraindications for combined ora l \ncontraceptive pill use, the patient was commenced  on \nYasmin (0.03mg Ethinylestradiol/3mg Drospirenone) f or \nthree months continuously, avoiding the pill-free weeks. She \nagain suffered no episodes of cataminal haemoptysis  \nthroughout, however did reported ongoing light head aches. \nShe was switched Microlite (100mcg Levonorgestrel/20mcg \nEthinylestradiol) for 6 months, again “back -to-back”, with \nno break. She reported no adverse side effects for three \nmonths, but at this point had a small break -through bleed, \ntogether with some mild chest pain, but no haemopty sis. \nSince then for the last she has been maintained on the \nLevonorgestrel/Ethinylestradiol combination, taking  pill \nfree period every six months for one week, without \ncomplaints or further episodes. \n \n \nFig 1 \n \n \n \nFig 2 \n \n \n \nFig 3 \n \n \n \nFig 4 \n\n\nInternational Journal of Reproduction, Gynaecology and Obstetrics  www.gynecologyjournal.in \n12 \nDiscussion \nBronchial endometriosis is a rare condition of non -pelvic \nendometriosis [9] . Approximately 60% of cases also have \ntrue co-existing pelvic endometriosis [9, 10, 11] . Simultaneous \nextra-pelvis endometriosis of the diaphragm and the visceral \npleura are found in 38.8% and 29.6% of cases respec tively \n[10]. Endometriosis of the lung parenchyma is much less  \ncommon [9, 11]. The pathophysiology of cyclical haemoptysis \nremains unclear, though three existing predominant theories \ndominate the literature  [12]. The leading hypothesis, \nproposed by Schron and Ruysh  [13], is a variant of \nSampson’s theory of retrograde menstruation. This m odel \nsuggests that endometrial -like cells enter the peritoneal \ncavity then pass to pleural space by lymphatic chan nels. An \nalternative second hypothesis considers that high l evels of \nprostaglandin F2 at the time of ovulation may resul t in \nvasospasm and associated ischemia in the lung paren chyma. \nA combination of prostaglandin -induced bronchospasm, \nmay cause the alveoli to rupture resulting in haemo ptysis, \nand also explain the associated risk of pneumothora x with \nthoracic endometriosis [13]. Thirdly, an anatomical theory is \nthat the loss of t he cervical mucus plug during menses \nresults in communication between the environment, \nperitoneal cavity, and subsequently the pleural spa ce [14]. As \nmany women will have retrograde menstruation with t heir \nmonthly periods, yet thoracic endometriosis is rar e, none of \nthese proposed models can completely explain this \nphenomenon. More recent theories in molecular and cellular \npathophysiology have looked at a spread of endometr ial \nmicro RNAs through exosomal trafficking [15], though this is \na novel theory for e xtra-pelvic endometriosis spread and as \nof yet has not been demonstrated in bronchial \nendometriosis. \nDiagnosis of bronchial endometriosis is almost alwa ys \ngrounded initially on clinical suspicion  [11]. The majority of \npatients present to hospital with a combination of cataminal \nhaemoptysis, shortness of breath, cough, and pleuri sy. \nInvestigations including chest radiography, CT thor ax, MRI \nthorax and bronchoscopy can also give help to suppo rt the \nclinical suspicion for a diagnosis of bronchial \nendometriosis. However, in the majority of cases reported, \ndiagnosis is usually made, and treatment performed,  with \nthe use of video -assisted thoracoscopic surgery (VATS)  [14]. \nThis allows direct visualisation of any pathology, and \ntargeted biopsy of any suspected endo metriotic lesion  [14]. \nThe initial management is often symptom control thr ough \nmedical treatment; which brings side -effect profiles, \nrecurrence risks and long -term high cost to the patients \nwithout definite treatment of the lesion.  Surgical \ninterventions s uch as chemical pleurodesis, pleurectomy, \nresection and lobectomy have proven successful \nmanagement options [14]. \nWhile in this case no evidence of pelvic endometrio sis or \ngynaecological disease was found on radiological im ages; \nthe CT findings for pulmona ry endometriosis may include \nwell-defined opacities, nodular lesions, thin-wall cavities, or \nbullous formations, but in cases with haemoptysis t hey have \ntransient radiologic densities in the part of the l ung [16]. In \nthis case, the CT revealed an opacity a t the anterior part of \nthe lower right lobe, histopathology of bronchial l avage \nsamples did not indicate ectopic endometrial pathol ogy. \nGenerally, diagnostic requirements for bronchial \nendometriosis is the presentation of periodic haemo ptysis \nthat is synch ronous with menstruation. Most previously \nreported cases were diagnosed based on the clinical  history \nof the patient; and a histological confirmation of ectopic \nendometriosis is not always completed and reported.  \nUnfortunately, though bronchoscopic attempt s at tissue \nsampling were made, we were unable to get positive \nhistological samples. However, the presence of cata minal \nhaemoptysis, synchronous to menses combined with a \nresponse to ovulation suppression confirmed the diagnosis. \nRecently, VATS for surgica l resection to treat cyclical \nhaemoptysis was reported to be safer and less invas ive than \nlobectomy [17, 18, 19] . Most of the time management of cases \nof endometriosis treated with hormonal therapy, typ ically \ngonadotrophic-releasing hormone analogs, or pro stogenic \ndrugs, but these remain controversial. We report he re that a \nlight dose of combined oral contraceptive pill cont inuous \nuse for one year gave excellent symptomatic relief for our \npatient with this benign thoracic lesion. \n \nConclusion \nWe present the use of the combined oral contraceptive pill \nfor symptomatic treatment of bronchial endometriosis, as an \neffective option for medical management.  \n \nAcknowledgements \nThanks to Dr. Conor Harrity for his contribution as  the lead \nof the gynaecology service in B eaumont Hospital and Dr. \nElkharouf and Dr. Amy Worrall for their contributio n to the \nmanuscript. \n \nReferences \n1. Fleishman SJ, Davidson JF. Vicarious menstruation, a \nlikely case of pulmonary endometriosis. Lancet. 195 9; \n2(7093):88-89. \n2. Schuld J, Justinger C, Wa gner M, Bo hle RM, Kollmar \nO, Schilling MK  et al. Bronchobiliary fistula: a rare \ncomplication of hepatic endometriosis. Fertil Steri l. \n2011; 95(2):804-808. \n3. Hilaris GE, Payne CK, Osias J, Cannon W, Nezhat CR.  \nSynchronous rectovaginal, urinary bladder, and \npulmonary endometriosis. JSLS. 2005; 9(1):78-82. \n4. Simoglou C, Zarogoulidis P, Machairiotis N, Por podis \nK, Simoglou L, Mitrakas A. et al . Abdominal wall \nendometrioma mimicking an incarcerated hernia: a case \nreport. Int J Gen Med. 2012; 5:569-571. \n5. Giudice LC, Kao LC. Endometriosis. Lancet. 2004 ; \n364(9447):1789-1799. Doi: 10.1016 /S0140-6736(04) \n17403-5. \n6. Suginami H, Hamada K, Yano K. A case of \nendometriosis of the lung treated with danazol. Obs tet \nGynecol. 1985; 66(3 Suppl):68S-71S. \n7. Park YB, Heo GM, M oon HK, Cho SJ, Shin YC, Eom \nKS. Et al. Pulmonary endometriosis resected by video -\nassisted thoracoscopic surgery. Respirology. 2006 ; \n11(2):221-223. Doi:10.1111/j.1440-1843.2006.00829.x. \n8. Shiota Y, Umemura S, Arikita H, Ho rita N, Hiyama J, \nTetsuya Ono T et al. A case of parenchymal pulmonary \nendometriosis, diagnosed by cytologic examination o f \nbronchial washing. Respiration. 2001 ; 68 (4):439. Doi: \n10.1159/000050545. \n9. Schwarz O. Endometriosis of the Lung. Am J Obstet \nGynecol. 1938; 36:887-889. \n10. Veeraswamy A, Lewis M, Mann A , Koti kela S, \nHajhosseini B, Nezhat C  et al . Extragenital \nendometriosis. Clin Obstet Gynecol.  2010; 53 (2):449-\n\nInternational Journal of Reproduction, Gynaecology and Obstetrics  www.gynecologyjournal.in \n13 \n466.  \n11. Rousset-Jablonski C, Alifano M, Plu -Bureau G  et al . \nCata- menial pneumothorax and endometriosis -related \npneumothorax: clinical features an d risk factors. Hum \nReprod, 2011.  \n12. Peikert T, Gillespie DJ, Cassivi SD. Catamenial \npneumothorax. Mayo Clin Proc. 2005; 80(5):677-680.  \n13. Nezhat C. Pelvic pain, Infertility, Pregnancy and \nEndometriosis: Ancient Conditions, Ancient \nCorrelations and their Ancient Treatments.  Fertil Steril \naccepted for publication, 2012.  \n14. Cowl CT, Dunn WF, Deschamps C. Visualization of \ndiaphragmatic fenestration associated with catameni al \npneumothorax. Ann Thorac Surg . 1999; 68 (4):1413-\n1414.  \n15. Klemmt PA, Starzinski -Powitz A. Molec ular and \ncellular pathogenesis of endometriosis. Current \nwomen's health reviews. 2018; 14(2):106-16. \n16. Orriols R, Munoz X, Alvarez A, Sampol G: Chest CT \nscanning: utility in lung endometriosis. Respir Med . \n1998; 92(6):876-877. \n17. Park YB, Heo GM, M oon HK, Cho S J, Shin YC, Eom \nKS. Et al. Pulmonary endometriosis resected by video -\nassisted thoracoscopic surgery. Respirology. 2006;  \n11(2):221-223.  \n18. De Z iegler D, Borghese B, Chapron C.  Endometriosis \nand infertility: pathophysiology and managem ent. \nLancet. 2010; 376(9742):730-738.  \n19. Lee CH, Huang YC, Huang SF, Wu YK, Kuo KT.  \nThoracic endometriosis: rare presentation as a soli tary \npulmonary nodule with ecc entric cavitations. Thorax. \n2009; 64(10):919-920.  \n20. Jenkins DW. \"Pathogens, Parasites and Predators of \nMedically Import ant Arthropods. Annotated List and \nBibliography\". Bull. World Health Organ . 1964; \n(30suppl):1-150.","source_license":"CC0","license_restricted":false}