{"paper_id":"6157e616-1846-4a4a-b147-a8a57980fab5","body_text":"International Journal of Case Reports and Images, Volume 15, Issue 2, 2024; Pages 47–52. ISSN: 0976-3198\nInt J Case Rep Images 2024;15(2):47–52.   \nwww.ijcasereportsandimages.com\nAdelekan et al. 47\nCASE REPORT  PEER REVIEWED | OPEN ACCESS\nCatamenial pneumothorax and endometriosis: Report of a \nrare case\nOluwaseun Adelekan, Moaz Alowami, Pooja Vasudev\nABSTRACT\nIntroduction: Catamenial pneumothorax is a rare \ncondition which is characterized by the recurrent \naccumulation of air within the pleural cavity without \nrelated respiratory diseases. It happens in women of \nreproductive age, usually within 72 hours before or after \nonset of menstruation. It occurs in association with \ncharacteristic pleural lesions, right-sided location of the \npneumothorax, and concomitant thoracic endometriosis. \nThe objective is to report a rare case of thoracic \nendometriosis in a 35-year-old woman with recurrent \ncatamenial pneumothorax.\nCase Report:  A 35-year-old female presented with \npersistent and recurrent right pneumothorax usually \noccurring immediately after her menstruation. She had \nno personal history of lung problems, asthma, bronchitis, \nor any family history of any chronic lung disease. \nComputed tomography (CT) of the chest showed a large \nhydropneumothorax on the right side resulting in near-\ncomplete collapse of the right lung and mediastinal shift \nfrom right to left. The gross specimen comprised the \nright upper lobe resection of the lung, which weighed 4.9 \ngrams and measured 7.7 cm × 1.5 cm × 1.2 cm. The pleura \nwas mottled brown-gray with adhesions. Histopathologic \nfindings showed pleural endometriosis (pleural \nendometrial glands and stroma present), consistent \nwith catamenial pneumothorax. Associated pleural \nOluwaseun Adelekan1, Moaz Alowami2, Pooja Vasudev1\nAffiliations: 1Department of Pathology and Molecular Medi -\ncine, McMaster University, St Joseph’s Healthcare L222-5, St \nLuke Wing, 50 Charlton Avenue East, Hamilton, Ontario L8N \n4A6, Canada; 2Department of Radiology, St Joseph’s Health-\ncare, 50 Charlton Avenue East, Hamilton, Ontario L8N 4A6, \nCanada.\nCorresponding Author:  Oluwaseun Adelekan, Department \nof Pathology and Molecular Medicine, McMaster University, \nSt Joseph’s Healthcare L222-5, St Luke Wing, 50 Charlton \nAvenue East, Hamilton, Ontario L8N 4A6, Canada; Email: \nOluwaseun.adelekan@medportal.ca\nReceived: 11 June 2024\nAccepted: 13 July 2024\nPublished: 04 September 2024\nadhesions with acute and chronic inflammation and mild \nemphysematous changes in the lung parenchyma were \nalso noted. Immunohistochemistry with immunostains \nestrogen receptor (ER) and CD10 highlighted the \nendometrial glands and stroma which confirmed the \ndiagnosis.\nConclusion: There should be a high index of suspicion \nof catamenial pneumothorax in women of reproductive \nage who present with pneumothorax during the \nperimenstrual period. Adequate sampling of lung wedge \nresections and careful microscopic examination, followed \nby confirmation with immunohistochemistry, play \nimportant roles in the diagnosis of this rare condition.\nKeywords: Catamenial pneumothorax, Endometriosis, \nImmunohistochemistry\nHow to cite this article\nAdelekan O, Alowami M, Vasudev P. Catamenial \npneumothorax and endometriosis: Report of a rare \ncase. Int J Case Rep Images 2024;15(2):47–52.\nArticle ID: 101469Z01OA2024\n*********\ndoi: 10.5348/101469Z01OA2024CR\nINTRODUCTION\nCatamenial pneumothorax (CP) was first described \nby Lillington et al. in 1972 [1]. It is considered as a rare \ncondition and subtype of pneumothorax which is related \nto menstruation and endometriosis. This condition is \nusually misdiagnosed and characterized by the recurrent \naccumulation of air within the pleural cavity without \nconcomitant respiratory diseases. The typical clinical \nmanifestation is spontaneous pneumothorax, or this \ncondition may also be asymptomatic. It happens in \nreproductive age women, usually within 72 hours of onset \nof menstruation [2]. Thoracic endometriosis, right side \ninvolvement, and distinctive pleural lesions (multiple \n\nInternational Journal of Case Reports and Images, Volume 15, Issue 2, 2024; Pages 47–52. ISSN: 0976-3198\nInt J Case Rep Images 2024;15(2):47–52.   \nwww.ijcasereportsandimages.com\nAdelekan et al. 48\ndiaphragmatic fenestrations, brown nodules on the \ndiaphragm) are characteristic of this condition [2].\nCASE REPORT\nA 35-year-old woman, premenopausal, para 1 \npresented to the emergency department with sudden \nonset of shortness of breath and chest pain. This episode \ncoincided with the first three days of her menstruation. She \nalluded to prior episodes of left shoulder pain and chest \npain occurring during her menstruation. She also had a \npast history of a spontaneous right hydropneumothorax \nfour weeks prior to presentation for which she had a chest \ntube insertion drainage in the emergency room as well as \ncare in the intensive care unit. Additionally, there was also \na history of a small left apical pneumothorax two years \nprior to presentation which required no intervention. A \nhistory of pelvic and peritoneal endometriosis, infertility \nand 2 laparoscopies of the pelvis was also noted. However, \nthere was no history of chronic lung disease, recent \nsurgical procedures, or trauma. There was no additional \npertinent personal or family medical history.\nClinical examination revealed tachypnea, mediastinal \nshift from right to left and reduced breath sounds. \nThe laboratory results were within reference range. \nChest X-ray showed right-sided moderate to large \nhydropneumothorax (Figure 1). A chest CT performed, \nrevealed bilateral pleural nodularity in keeping with \npleural-based endometriosis deposits and right basal \natelectatic changes also noted (Figure 2). A transvaginal \nultrasound (US) showed severe endometriosis including \nextra-pelvic endometriosis of the abdominal wall. \nSpecifically, rectal and severe posterior compartment \nendometriosis was noted. There was also severe \nobliteration of the rectouterine pouch and benign cystic \nlesion in the left adnexa. There was free blood in the right \nadnexa likely due to a retrograde menstruation. In spite \nof several chest drains, her lung did not expand and she \ncontinued to have persistent air leaks. She underwent a \nflexible video-assisted bronchoscopy, right video-assisted \nthoracoscopic surgery (VATS). Intra-operatively, there \nwere adhesions seen between the right lung and the \nchest wall and an area of abnormality on the lateral wall \nof the right upper lobe between the apical and posterior \nsegments. The pleura was also abnormal in this area. A \nwedge resection of the right upper lobe of the lung was \nperformed and the specimen was sent for pathology \nanalysis.\nGrossly, the specimen comprised a wedge resection \nof the upper lobe of the right lung, which measured \n7.7 cm × 1.5 cm × 1.2 cm and weighed 4.9 grams. \nThe pleura of the lung was mottled brown-gray with \nadhesions. The lung parenchyma was tan and appeared \nrelatively unremarkable. Microscopic examination \nshowed endometrial glands and stroma surrounded by \nhemorrhage as well as fibrosis of the pleura (Figure 3). \nThere was also acute and chronic inflammation in addition \nto subpleural chronic inflammation (Figure 4). Mild \nemphysematous change of lung parenchyma was also \nnoted. Immunohistochemical stains performed confirmed \nthe endometrial glands and stroma to be estrogen receptor \n(ER) positive (Figure 5) and the endometrial stroma to be \nCD10 positive (Figure 6).\nThe postoperative course was uneventful, and the \npatient was discharged four days after surgery. She was \nsubsequently placed on Leuprolide injections every \nthree months and Norlutate as an add -back hormone \nfor a period of one year. She has not had any recurrence \nof shortness of breath or chest pain in the last two \nyears following her surgery. She remains quite stable \nfrom a thoracic standpoint. However, she continues to \nexperience pelvic and abdominal pain while on medical \nmanagement and is scheduled to have a laparoscopic \nresection of bowel endometriosis.\nFigure 1: Chest X-ray demonstrating a right-sided moderate to \nlarge hydropneumothorax.\nFigure 2: Chest CT scan showing bilateral pleural nodularity in \nkeeping with pleural-based endometriosis deposits and right \nbasal atelectatic changes also noted.\n\nInternational Journal of Case Reports and Images, Volume 15, Issue 2, 2024; Pages 47–52. ISSN: 0976-3198\nInt J Case Rep Images 2024;15(2):47–52.   \nwww.ijcasereportsandimages.com\nAdelekan et al. 49\nDISCUSSION\nRisk factors associated with catamenial pneumothorax \ninclude previous pelvic surgery, uterine scraping, \ninfertility, mean age of 32–35 years, and symptoms of \npelvic endometriosis [2]. Screening with these factors \nmay enhance the diagnostic yield [3]. Our index case \nsimilarly was 34 years and had a history of infertility and \nhad undergone 2 laparoscopic pelvic procedures prior to \nthe presentation of catamenial pneumothorax. The typical \nclinical manifestation of catamenial pneumothorax \ninvolves spontaneous pneumothorax preceding or in \nsynchrony with menses, usually presenting with pain, \nshortness of breath and cough. Scapular and or thoracic \npain preceding or in synchrony with menses, history \nof previous episode(s) of spontaneous pneumothorax \nwith or without previous surgical interventions is \nhighly suspicious [4, 5]. Most of the cases of catamenial \npneumothorax are right-sided, similar to this case but \nsome can be left or bilateral. The pathogenesis of this \ndisease entity is likely multifactorial. Several theories \nhypothesize the causation of this condition including the \nretrograde menstruation theory, coelomic metaplasia \ntheory, lymphatic and hematogenous dissemination \ntheory, and the prostaglandin theory [6].\nA detailed history and physical examination are \nimportant prerequisites to the diagnostic work-up. The \nwork-up is composed of imaging modalities such as chest \nX-ray (CXR), computed tomography (CT), and magnetic \nresonance imaging (MRI) [7]. Chest X-ray and CT chest \nare the most sensitive in detecting pneumothorax. \nRadiological findings include pneumomediastinum, \npneumoperitoneum, ground-glass opacities, bronchial-\nwall thickening, thin-walled cavities within the lung \nparenchyma, or bullous formation [8, 9]. Chest X-ray \nin our case revealed a right-sided moderate to large \nhydropneumothorax and a chest CT revealed bilateral \npleural nodularity in keeping with pleural-based \nFigure 3: Micrograph at ×100 showing endometrial glands and \nstroma surrounded by hemorrhage and fibrosis. H&E stain.\nFigure 4: Micrograph at ×100 showing endometrial glands with \nstroma and hemorrhage. H&E stain.\nFigure 5: Micrograph at ×100 showing endometrial glands and \nstroma staining with estrogen receptor.\nFigure 6: Micrograph at ×100 showing endometrial stroma \nstaining with CD10.\n\nInternational Journal of Case Reports and Images, Volume 15, Issue 2, 2024; Pages 47–52. ISSN: 0976-3198\nInt J Case Rep Images 2024;15(2):47–52.   \nwww.ijcasereportsandimages.com\nAdelekan et al. 50\nendometriosis deposits and right basal atelectatic \nchanges. Interventional procedures like bronchoscopy, \nvideo laparoscopy, and video-assisted thoracic surgery \nare also included in the work-up regimen.\nThe gold standard of surgical intervention is video-\nassisted thoracic surgery (VATS) [10]. Depending on the \nvariability of the case presentation, alternative procedures \nmay also be performed including thoracotomy, and \npleurodesis (chemically or surgically). Most commonly \nreported intraoperative findings included diaphragmatic \nlesions (38.8%), endometriosis of the visceral pleura \n(29.6%), discrete lesions such as bullae, blebs, or scarring \n(23.1%) and no findings (8.5%) [3]. In this case, there \nwere adhesions seen between the right lung and lateral \nchest wall with some pleural abnormality; however, no \novert diaphragmatic lesions or endometriosis of the lung \npleura were observed intraoperatively.\nThoracic endometriosis has been considered as \nhistologically “proven” after identification of endometrial \nstroma and glands in the thoracic lesions and as \n“probable” after identification of stroma only [4, 5, 11–13]. \nIn our case, histologic examination showed endometrial \nglands and stroma surrounded by hemorrhage as \nwell as fibrosis of the pleura; which was confirmed by \nimmunohistochemical stains.\nAs far as treatment is concerned, VATS in conjunction \nwith hormonal therapy for at least six months has been \nfound to show an improvement in prognosis and a \ndecrease in the possibility of a recurrence of catamenial \npneumothorax [14]. Medical management includes \nhormonal therapy in the form of gonadotropin releasing \nhormones and aromatase inhibitors preferably for \na period of 6–12 months. Similarly, our index case \nunderwent VATS and was treated with Leuprolide for \none-year post-surgery with no recurrence of symptoms of \nthoracic endometriosis.\nCONCLUSION\nThere should be a high index of suspicion of \ncatamenial pneumothorax in women of reproductive age \nwho present with pneumothorax during or around the \nperimenstrual period. Adequate sampling of lung wedge \nresections and careful microscopic examination, followed \nby confirmation with immunohistochemistry play \nimportant roles in the diagnosis of this rare condition.\nREFERENCES\n1. Lillington GA, Mitchell SP, Wood GA. Catamenial \npneumothorax. JAMA 1972;219(10):1328–32.\n2.\t Marjański\t T,\tSowa\tK,\tCzapla\tA,\tRzyman\tW.\tCatamenial\t\npneumothorax\t–\tA\treview\tof\tthe\tliterature.\tKardiochir\t\nTorakochirurgia Pol 2016;13(2):117–21.\n3.\t Korom\t S,\t Canyurt\t H,\t Missbach\t A,\t et\t al.\t Catamenial\t\npneumothorax revisited: Clinical approach and \nsystematic review of the literature. J Thorac \nCardiovasc Surg 2004;128(4):502–8.\n4. Rousset-Jablonski C, Alifano M, Plu-Bureau G, et al. \nCatamenial pneumothorax and endometriosis-related \npneumothorax: Clinical features and risk factors. \nHum Reprod 2011;26(9):2322–9.\n5.\t Visouli\t AN,\tDarwiche\tK,\tMpakas\tA,\tet\tal.\tCatamenial\t\npneumothorax: A rare entity? Report of 5 cases and \nreview of the literature. J Thorac Dis 2012;4(Suppl \n1):17–31. \n6. Hope-Gill B, Prathibha BV. Catamenial haemoptysis \nand clomiphene citrate therapy. Thorax \n2003;58(1):89–90.\n7. Nezhat C. A multidisciplinary approach to \ndiaphragmatic endometriosis. MDedge ObGyn News \n2017;1–7.\n8. Ciudad MJ, Santamaría N, Bustos A, Ferreirós J, \nCabeza B, Gómez A. Imaging findings in catamenial \npneumothorax. [Article in Spanish]. Radiologia \n2007;49(4):263–7.  \n9. Rousset P, Rousset-Jablonski C, Alifano M, Mansuet-\nLupo A, Buy JN, Revel MP. Thoracic endometriosis \nsyndrome: CT and MRI features. Clin Radiol \n2014;69(3):323–30.\n10.\t Kolos\t A,\t Dzhieshev\t Z,\t Dikolaev\t V,\t Amangaliev\t A.\t\nCatamenial pneumothorax. Exp Clin Transplant \n2015;13 Suppl 3:144–5.\n11. Legras A, Mansuet-Lupo A, Rousset-Jablonski C, et \nal. Pneumothorax in women of child-bearing age: An \nupdate classification based on clinical and pathologic \nfindings. Chest 2014;145(2):354–60. \n12. Ciriaco P, Negri G, Libretti L, et al. Surgical \ntreatment of catamenial pneumothorax: A single \ncentre experience. Interact Cardiovasc Thorac Surg \n2009;8(3):349–52.   \n13.\t Alifano\t M,\t Jablonski\t C,\t Kadiri\t H,\t et\t al.\t Catamenial\t\nand noncatamenial, endometriosis-related \nor nonendometriosis-related pneumothorax \nreferred for surgery. Am J Respir Crit Care Med \n2007;176(10):1048–53.\n14. Toffolo Pasquini M, Auvieux R, Tchercansky A, \nBuero A, Chimondeguy D, Mendez J. Catamenial \npneumothorax. [Article in Spanish]. Medicina (B \nAires) 2022;82(1):147–50.\n*********\nAcknowledgments\nImage credits. Dr. Abdullah Alabousi, MD, FRCPC, \nDepartment of Radiology, McMaster University, \nHamilton, Ontario.\nAuthor Contributions\nOluwaseun Adelekan – Conception of the work, Design \nof the work, Acquisition of data, Analysis of data, \nInterpretation of data, Drafting the work, Revising the \nwork critically for important intellectual content, Final \napproval of the version to be published, Agree to be \naccountable for all aspects of the work in ensuring that \nquestions related to the accuracy or integrity of any part \nof the work are appropriately investigated and resolved\n\nInternational Journal of Case Reports and Images, Volume 15, Issue 2, 2024; Pages 47–52. ISSN: 0976-3198\nInt J Case Rep Images 2024;15(2):47–52.   \nwww.ijcasereportsandimages.com\nAdelekan et al. 51\nMoaz Alowami – Conception of the work, Design of the \nwork, Acquisition of data, Analysis of data, Interpretation \nof data, Drafting the work, Revising the work critically \nfor important intellectual content, Final approval of the \nversion to be published, Agree to be accountable for all \naspects of the work in ensuring that questions related \nto the accuracy or integrity of any part of the work are \nappropriately investigated and resolved\nPooja Vasudev – Conception of the work, Design of the \nwork, Acquisition of data, Analysis of data, Interpretation \nof data, Drafting the work, Revising the work critically \nfor important intellectual content, Final approval of the \nversion to be published, Agree to be accountable for all \naspects of the work in ensuring that questions related \nto the accuracy or integrity of any part of the work are \nappropriately investigated and resolved\nGuarantor of Submission\nThe corresponding author is the guarantor of submission.\nSource of Support\nNone.\nConsent Statement\nWritten informed consent was obtained from the patient \nfor publication of this article.\nConflict of Interest\nAuthors declare no conflict of interest.\nData Availability\nAll relevant data are within the paper and its Supporting \nInformation files.\nCopyright\n© 2024 Oluwaseun Adelekan et al. This article is \ndistributed under the terms of Creative Commons \nAttribution License which permits unrestricted use, \ndistribution and reproduction in any medium provided \nthe original author(s) and original publisher are properly \ncredited. Please see the copyright policy on the journal \nwebsite for more information.\nABOUT THE AUTHORS\nArticle citation:  Adelekan O, Alowami M, Vasudev P. Catamenial pneumothorax and endometriosis: Report of a \nrare case. Int J Case Rep Images 2024;15(2):47–52.\nOluwaseun Adelekan  is a resident doctor in the Department of Diagnostic and Clinical Pathology \nat McMaster University in Hamilton, Ontario, Canada. She earned the undergraduate degree \n(M.B.B.S) from the College of Health Sciences, University of Ilorin, Ilorin, Nigeria and postgraduate \ndegree (FMCPath) from the National Postgraduate Medical College of Nigeria. She has published \nfive\tresearch\t papers\tin\tnational\t and\tinternational\t academic\t journals.\tHer\tresearch\t interests\t include\t\nhematopathology, blood transfusion and gynecological pathology.\nEmail: Oluwaseun.adelekan@medportal.ca\nMoaz Alowami is currently a research assistant at McMaster University as well as a clinical assistant \nat a walk-in clinic at Hamilton, Ontario, Canada. He earned his undergraduate degree (MBBS/MD) \nfrom Libyan International Medical University in Benghazi, Libya. He has published a total of 3 research \npapers so far in national and international academic journals. His research interests include pathology, \ninternal medicine, and primary care medicine. He intends to pursue residency training in Canada. \nEmail: Drmoazalowami95@gmail.com\nPooja Vasudev is assistant professor at Department of Pathology and Molecular Medicine, McMaster \nUniversity, Hamilton, Ontario, Cananda. She earned the undergraduate degree (M.B.B.S.) from \nUniversity of Delhi, Delhi, India and postgraduate degree (M.D. Pathology) from Department of \nPathology/Punjab University, Chandigarh, India, followed by residency in Anatomical Pathology at \nMcMaster University, Hamilton, Ontario, Canada. Currently, she is the Fellowship Director for the \nBreast Pathology Fellowship program, McMaster University. She has published numerous research \npapers in national and international academic journals. Her research interests include breast carcinoma \nspecial types, breast biomarkers, and unusual case reports related to skin and lung pathology. She \nintends to pursue more extensive research in breast biomarkers reporting in future.\n\n\nInternational Journal of Case Reports and Images, Volume 15, Issue 2, 2024; Pages 47–52. ISSN: 0976-3198\nInt J Case Rep Images 2024;15(2):47–52.   \nwww.ijcasereportsandimages.com\nAdelekan et al. 52\nAccess full text article on\nother devices\nAccess PDF of article on\nother devices\n\nSubmit your manuscripts at\nwww.edoriumjournals.com","source_license":"CC0","license_restricted":false}