{"paper_id":"97a7c96d-de0e-41d8-a779-1c09ea78ebd8","body_text":"International Journal of\nRespiratory and Pulmonary Medicine\nAlves. Int J Respir Pulm Med 2023, 10:190\nVolume 10 | Issue 2\nDOI: 10.23937/2378-3516/1410190\n• Page 1 of 3 •\nISSN: 2378-3516\nOpen Access\nCitation: Alves MG (2023) Catamenial Pneumothorax. Int J Respir Pulm Med 10:190. doi.\norg/10.23937/2378-3516/1410190\nAccepted: December 04, 2023; Published: December 06, 2023\nCopyright: © 2023 Alves MG. This is an open-access article distributed under the terms of the Creative \nCommons Attribution License, which permits unrestricted use, distribution, and reproduction in any \nmedium, provided the original author and source are credited\nAlves. Int J Respir Pulm Med 2023, 10:190\nCatamenial Pneumothorax\nMarina Gabriela Alves*\na Serviço de Pneumologia, Centro Hospital de Lisboa Ocidental, Lisbon, Portugal\n*Corresponding author: Marina Gabriela Alves, a Serviço de Pneumologia, Centro Hospital de Lisboa Ocidental, Lisbon, \nPortugal\nAbstract\nCatamenial pneumothorax is the most common form \nof thoracic endometriosis syndrome. It is a form of \nspontaneous recurrent pneumothorax, occurring mostly in \nwomen of reproductive age, typically within 72h from the \nonset of menstruation. Although aetiology is unknown, \nseveral hypothesis have been raised trying to explain the \npathogenesis behind it. We present two cases of women \nin reproductive age, with episodes of recurrent right \npneumothorax. In surgery were visualized diaphragmatic \ndefects compatible with the diagnosis of catamenial \npneumothorax. After surgical correction and hormonal \ntreatment,they are asymptomatic and without new episodes \nof pneumothorax.\nCAse RepoRt\nCheck for\nupdates\nAs recurrences are common, the best recommended \ntreatment is a combination of surgery and hormone \ntherapy [1].\nCatamenial pneumothorax aetiology is still unknown, \nalthough several theories try to explain it.\nWe report in this article 2 cases of young women \nwith recurrent spontaneous pneumothorax, surgically \ndiagnosed with catamenial pneumothorax.\nCase 1\nA 37-year-old patient with a history of bronchial \nasthma and allergic rhinitis, non-smoker. She went \nto the emergency department due to progressively \nworsening dyspnoea, cough and productive cough and \nfever. An X-ray showed a large right pneumothorax. A \nchest tube was placed in the 5\nth intercostal space, in \nthe midaxillary line. After documenting the resolution \non chest CT, the chest tube was removed. CT showed \nno blebs or bullae. However, there was recurrence \nof the pneumothorax during hospitalization. For this \nreason, she was transferred to Thoracic Surgery, and \nunderwent wedge resection of the right upper lobe \nand mechanical pleurodesis by video-assisted thoracic \nsurgery (VATS). Subsequently, due to a new recurrence \nof pneumothorax, she needed a new surgery, and were \nvisualized diaphragmatic fenestrations suggestive of \ncatamenial secondary spontaneous pneumothorax. \nShe underwent right pleurectomy and diaphragmatic \ntalcage. After the surgical intervention, she initiated \nhormone suppression with monthly goserelin for 6 \nmonths, without recurrence.\nCase 2\nA 41-year-old patient with only known history \nIntrodution\nThoracic endometriosis (TE) is a clinical condition \nmanifested clinically by catamenial pneumothorax, \nhemoptysis, catamenial hemothorax and pulmonary \nnodules [1-3].\nThe most common clinical presentation of TE is \ncatamenial pneumothorax [1,2,4-7]. However, it is also \nknown that absence of TE can occur, associated with \ndiaphragmatic defects [8]. Catamenial pneumothorax \nwas initially described in 1958 by Maurer, et al. [9]. It is \na rare form of secondary spontaneous pneumothorax, \nunder diagnosed. It occurs mostly in adult women, \nwith a peak of incidence between 30-35 years. It \nis characterized by recurrent pneumothoraces, in \nperimenstrual period, usually occurring within 72 hours \nbefore or after onset of menstruation [1, 8,10], mostly \nin the right hemithorax [1, 4,6,8,10]. It corresponds to \n3-6% of cases of recurrent spontaneous pneumothorax \nand a third of all cases of spontaneous pneumothorax in \nwomen at reprodutive age [1,2,4].\n\nISSN: 2378-3516\nDOI: 10.23937/2378-3516/1410190\nAlves. Int J Respir Pulm Med 2023, 10:190\n• Page 2 of 3 •\nrelapses and that this theory would not justify the \nprevalence of the right side [10]. Another theory based \non an anatomical explanation defends that dissolution \nof the cervical mucus plug during the menstrual period \nallows communication between the peritoneal cavity \nand the external environment. Air migrates through \nthe fallopian tubes into the abdominal cavity, and from \nthere through fenestrations or congenital diaphragmatic \ndefects into the pleural space. Since those defects are \nmore frequent on the right, this theory justifies the \nright hemithorax prevalence found in the literature \nand the low rate of pneumothorax recurrence after \nsurgical correction of the defects [1, 4-6,11]. There \nare reported cases of postpartum and postcoital \ncatamenial pneumothoraces that also help to support \nthis hypothesis [1,4].\nThe migration theory advocates the migration \nof endometrial tissue from the uterus through the \nperitoneal flow into the subdiaphragmatic space. \nThe cyclic necrosis of these diaphragmatic implants \nproduces defects that allow the passage of endometrial \ncells into the thoracic cavity. Due to the preferential \nflow of peritoneal fluids through the right paracolic \ngutter, most endometrial tissue migrates to the right \nhemidiaphragm [6,8]. Resulting from the cyclic necrosis \nof implants in the visceral pleura, alveolar rupture may \noccur [1,4,11].\nThe metastatic theory defends that occur \ntransdiaphragmatic migration of endometrial tissue \nthrough the peritoneal cavity into the pleural space \nvia transdiaphragmatic, hematogenous lymphatic or \ncongenital diaphragmatic fenestrations [1, 4], with \nsubsequent necrosis of those endometrial foci in the \nproximity of visceral pleural causing air leak [6]. Some \nof rhinitis. Previous admitted one month earlier for \nspontaneous right pneumothorax. She came back \nwith pain located in the right shoulder and irradiation \nto the scapula, worst with deep inspiration, with one \nday of evolution. She denied associated trauma. No \nclinical signs of infection. She wasn´t menstruated. \nChest radiography showed right pneumothorax without \ndeviation of mediastinal structures. After placement of \nthe chest tube, an apical pneumothorax chamber was \nmaintained. As it was a second episode, the thoracic \nsurgery was contacted. She underwent atypical right \nsuperior lobe resection with pleurectomy and talcage, \nby VATS. Multiple diaphragmatic fenestrations were \nseen, some pericentimetric with hepatic exposure. \nNo intrathoracic endometriosis, bleds, or bullae were \nvisualized. She was discharged with an indication to \ncontinue treatment with goserelin.\nDiscussion\nWe present two cases women with episodes \nof recurrent spontaneous right pneumothorax, \nidentificated as catamenial. Catamenial pneumothorax \nis usually unilateral, occurring mostly in the right \nhemithorax, although it can occur on the left or \nbe bilateral [4-6]. The aetiology of catamenial \npneumothorax is still unknown, but there are several \ntheories that try to explain the mechanisms that lead to \nit. Rossi and Goplerud in 1974, in their hormonal theory, \nclaimed that high levels of Prostaglandin F2 during \novulation lead to vasospasm, with consequent ischemia, \nand bronchospasm, responsible for alveolar rupture \n[1,4-6,11]. Blebs and/or bullae can be more susceptible \nto rupture during menstrual-related hormonal changes \n[6]. However, it was also found that continuous use of \nnon-steroidal anti-inflammatory drugs did not prevent \n \nFigure 1: Intraoperative findings of the 2 nd patient. Multiples diaphragmatic fenestrations. Liver can be seen underneath \nthe perforations.\n\nISSN: 2378-3516\nDOI: 10.23937/2378-3516/1410190\nAlves. Int J Respir Pulm Med 2023, 10:190\n• Page 3 of 3 •\nOne year after surgery and continuous hormonal \ntherapy our patients remain asymptomatic and without \nrecurrence of pneumothorax.\nReferences\n1. Elia S, Felice L, Varvaras D, Sorrenti G, Mauriello A, \net al. (2015) Catamenial pneumothorax due to solitary \nlocalization of diaphragmatic endometriosis. Int J Surg \nCase Rep 12: 19-22.\n2. Celik B, Gurz S, Pirzirenli MG, Kefeli M (2013) Recurrent \nCatamenial pneumothorax caused by diaphragmatic \nfenestration. Respir Care 58: 168-171.\n3. MacDuff A, Arnold A, Harvey J (2010) Management of \nspontaneous pneumothorax: British thoracic society pleural \ndisease guideline 2010. Thorax 65: 18-31.\n4. Visouli AN, Darwiche K, Mpakas A, Zarogoulidis P, \nPapagiannis A, et al. (2012) Catamenial pneumothorax: A \nrare entity? Report of 5 cases and review of the literature. J \nThorac Dis 4: 17-31.\n5. Korom S, Canyurt H, Missbach A, Schneiter D, Kurrer MO, \net al. (2004) Catamenial pneumothorax revisited: Clinical \napproach and systematic review of the literature. J Thorac \nCardiovasc Surg 128: 502-508.\n6. Visouli AN, Zarogoulidis K, Kougioumtzi I, Huang H, Li Q, \net al. (2014) Catamenial pneumothorax. J Thorac Dis 6: \nS448-S460.\n7. Alifano M, Trisolini R, Cancellieri A, Regnard JF (2006) \nThoracic endometriosis: Current knowledge. Ann Thorac \nSurg 81: 761-769.\n8. Alifaro M, Roth T, Broet SC, Schussler O, Magdeleinat P, et \nal. (2003) Catamenial pneumothorax. A prospective study. \nChest 124: 1004-1008.\n9. Maurer ER, Schaal JA, Mendez FL (1958) Chronic recurring \nspontaneous pneumothorax due to endometriosis of the \ndiaphragm. JAMA 168: 2013-2014.\n10. Peiket T, Gillespie DJ, Cassivi SD (2005) Catamenial \nPneumothorax. Case report. Mayo Clin Proc 80: 677-680.\n11. Kawaguchi Y, Fujita T, Hanaoka J (2015) Catamenial \npneumothorax with bullae. Ann Thorac Surg 99: 1075-\n1078.\n12. Subotic D, Mikovic Z, Atanasijadis N, Savic M, Moskovljevic \nD, et al. (2016) Hormonal therapy after the operation for \ncatamenial pneumothorax - is it always necessary? J \nCardiothorac Surg 11: 66.\n13. Garner M, Ahmed E, Gatiss S, West D (2018) Hormonal \nmanipulation after surgery for catamenial pneumothorax. \nInteract CardioVasc Thorac Surg 26: 319-322.\nstudies show a high prevalence of diaphragmatic defects \n(50-62.5%) [10].\nIn endometriosis occurs an increase in the levels of \nCA-125. Although not specific, it can guide the suspicion \ntowards an earlier diagnosis. CA-125 is a tumour marker \nused in the monitoring of ovarian cancer, although it \ncan be elevated in several benign conditions [1,4,6].\nThese two women were submitted to VATS, that are \ndescribed in the literature as the preferred approach \n[5]. In both cases, endometriosis was not seen, there \nwas only evidence of diaphragmatic fenestrations that \nsuggested the diagnosis.\nThe absence of findings frequently during \nvideothoracoscopy makes catamenial pneumothorax \nan underdiagnosed entity [1].\nTreatment involves surgery (pulmonary resection, \npleurectomy, chemical or mechanical pleurodesis and \ndiaphragmatic reconstruction) and hormonal therapy \n[1,4,7].\nA high recurrence rate has been documented with \nthe use of hormone therapy or surgery alone. Post-\nsurgical recurrence is reported to be, depending on the \nstudy, 8 to 40%. and may happen several years after the \nfirst episode [2,3,6].\nHormonal therapy with gonadotropin-releasing \nhormone is important to prevent recurrence, inducing \nhypogonadotropic hypogonadism and amenorrhea \nafter surgery, for at least 6 to 12 months [4,6,7,12,13].\nConclusion\nCatamenial pneumothorax is an entity described \nas rare, although it is responsible for a large number \nof spontaneous pneumothorax. For this reason, we \nshould suspect of it in women of reproductive age, with \nsuggestive history and recurrent pneumothorax, even in \nabsence of symptoms related to pelvic endometriosis.\nConservative treatment is insufficient. VATS allow \nbetter visualization of the pleural space and diaphragm. \nConsidering the high frequency of diaphragmatic \nabnormalities, early surgical exploration should be \npursued in these patients. Hormone therapy after \nsurgery is responsible for reducing the recurrence rate.","source_license":"CC0","license_restricted":false}