{"paper_id":"14418f66-7bcf-4b95-baa5-f3a11814d6da","body_text":"25\nYonago Acta medica 2003;46:25–28\nAbbreviation:  VATS, video-assisted thoracoscopic sur-\ngery\nA Case of Catamenial Pneumothorax Treated by Video-\nAssisted Thoracoscopic Surgery\nSakiko Yasui, Yuji Taniguchi, Yoshimasa Suzuki, Kazuhiko Makihara, Koichiro\nOkada, Norimasa Ito, Kiyosuke Ishiguro and Shigetsugu Ohgi\nDivision of Organ Regeneration Surgery, Department of Surgery, School of Medicine, Tottori Uni-\nversity Faculty of Medicine, Yonago 683-8504 Japan\nThis is a case of a 47-year-old female who had a medical history of right pneumothorax for\nthe second time.  The pneumothorax, accompanying the start of menstruation, recurred\nand the patient was hospitalized.  From the medical history, a catamenial pneumothorax\nwas suspected.  As for intraoperative findings, many small fenestrations of 1 mm or 3 mm\nwere present in the border region with the muscle bundle of the central tendon of the\ndiaphragm.  The lesion site of the diaphragm and the apex area as a biopsy were partially\nexcised under video-assisted thoracoscopic surgery.  Although a postoperative Gn-RH\nagonist was started for endometriosis, it was stopped because side effects appeared.  Because\nthe right pneumothorax recurred in accordance with the start of menstruation, the treat-\nment was changed to danazol.  To date, the pneumothorax has not recurred.\nKey words:  catamenial pneumothorax; diaphragmatic fenestration; hormonal therapy; video-assisted\nthoracoscopic surgery\nCatamenial pneumothorax is a disease in which a\npneumothorax repeats in accordance with the men-\nstruation cycle (Maurer et al., 1958).  While there\nare many unknown points regarding the mechanism\nof its occurrence, however, this disease is assumed\nto be caused by endometriosis.  Thus, because endo-\nmetriosis cases have increased recently, this disease\nis expected to increase in the future as well.  We\nexperienced a case of a patient in whom a perforat-\ned site of the diaphragm was excised under video-\nassisted thoracoscopic surgery (VATS) and the dis-\nease recurred when hormonal therapy was stopped\nafter surgery.  The case is reported here with a dis-\ncussion of the literature.\nPatient Report\nIn 1998, a 45-year-old female consulted a local phy-\nsician because right chest pain and dyspnea appear-\ned suddenly.  The symptoms were alleviated by\nconservative treatment, specifically an analgetic.  In\nthe middle of December 2000, right chest pain and\ndyspnea recurred and the patient again consulted\nthe physician.  The symptoms were not alleviated\nby the same treatment.  On December 27, she was\nreferred to the Clinic of Respiratory Internal Medi-\ncine of Tottori University Hospital and admitted as\na patient.  Diagnosis as right spontaneous pneumo-\nthorax was made and the symptoms were alleviated\nby continuous chest drainage.  Because the pain oc-\ncurred at the start of her menstruaction, catamenial\npneumothorax was suspected.  The patient was fol-\nlowed-up at the outpatient section after discharge.\n\n26\nS. Yasui et al.\nFig. 1.  Preoperative Chest roentgenogram showing a\nright pneumothorax after continuous chest drainage.\nThe arrows indicate visceral pleura.\nOn February 18, 2001, pain recurred 2 days prior to\nher menstruation.  On February 21, she consulted\nthe Clinic again.  Because a recurrence of the right\npneumothorax was found, she was immediately\nhospitalized in the Clinic of Thoracic Surgery, Tottori\nUniversity Hospital.\nThe patient had experienced 2 pregnancies but\nonly 1 delivery.  Her menstruation was regular, but\nwith dysmenorrhea.  There was no past illness that\nwas especially notable.\nOn admission, her height was 155 cm, body\nweight 45 kg, and nutrition was slightly poor.  Body\ntemperature was 36.6˚C, blood pressure 100/72\nmmHg and pulse rate 70/min.  Ausculation revealed\nfaint respiratory sounds in the right lung.  There\nwere no rale sounds or cardiac murmur.  There were\nno abnormal physical findings in the abdomen.\nChest X-rays showed a right pneumothorax\nwithout bulla or pleural effusion.  Although conti-\nnuous chest drainage was conducted,  complete pul-\nmonary expansion was not obtained (Fig. 1).  Com-\nputed tomography of the chest showed no bulla or\nother abnormal findings.  Electrocardiograms also\nshowed no abnormalities.  The bleeding time and\ncoagulation time were normal, and no abnormali-\nties were found in hematological examinations, uri-\nnalysis, biochemical examinations, etc.\nOn February 28, 2001, VATS was performed\nin a recumbent position on the inferior left side\nunder general anesthesia with separate ventilation.\nA thoracoport of 5.5 mm was inserted through the\n5th intercostal middle axillary line and the inside of\nthe thorax was observed with a hard scope.  Irregu-\nlar hypertrophic pleura were found at the apex area\nin the right thorax, but no bulla or adhesions were\npresent.  Subsequently, a thoracoport of 11.5 mm\nwas inserted through the 4th intercostal anterior\naxillary line, and another one of 5.5 mm through the\n6th intercostal scapular angle for further observa-\ntion.  A small fenestration of about 3 mm in dia-\nmeter was found in the tendon center of the dia-\nphragm and air bubbles were present around it.  De-\nposits of a blackish brown color and small pores of\nabout 1 mm in diameter were also observed around\nthe diaphragm.  From these findings, the disease\nwas diagnosed as catamenial pneumothorax.  The\ntendon center of the diaphragm, including the small\npores, was resected using an ENDO GIA Universal\n30-2.5 (United States Surgical Corp., Norwalk,\nCT).  The hypertrophic pleura in the apex part were\nalso resected, using an ENDO GIA Universal 45-\n2.5 (United States Surgical Corp.).\nThe sizes of the excised sections were 33 × 11\nmm from the diaphragm (Fig. 2) and 33 × 5 mm\nfrom the apex.  In the diaphragm, there was a slit-\nlike partial change of 7 mm, and mild chronic in-\nflammation was found around it, which is not gen-\nerally observable in disease as endometriosis.  In\nthe apex section, a severe pneumonic change and a\nnon-specific fibrous formation were found just un-\nder the pleura, but no tissue indicating endometrio-\nsis or pneumothorax.\nAlthough a postoperative atelectasis occurred\nin the right pulmonary inferior lobe, it improved on\nday 5.  Subsequently, its course was favorable.\nDuring gynecological examination, endometriosis\nof the intraperitoneal organs was not detected.\nHowever, since the serum CA125 level increased to\n\n27\n Catamenial pneumothorax\n76.3 and laparoscopy examination was not per-\nformed, the possibility of endometriosis was un-\ndeniable.  The patient was discharged 15 days after\nsurgery, and administration of leuprorelin acetate,\nan Gn-RH agonist, was started.  Because symptoms\nof carpal tunnel syndrome appeared, which might\nhave been caused by the growth of smooth muscle,\nits administration was stopped 4 months later.\nAfter 4 months, following the cessation of hormo-\nnal therapy, on November 10, 2001, 1 day prior to\nthe start of her menstruation, the pneumothorax\nwith right chest pain recurred.  However, the col-\nlapse was so mild that the symptoms cured natural-\nly.  On November 16, oral administration of dana-\nzol was started.  At present, the pneumothorax has\nnot recurred after 1 year.\nDiscussion\nAlthough there is no consistent view regarding a\nmechanism for the occurrence of catamenial pneu-\nmothorax, there are 3 hypotheses:  the intraperi-\ntoneal air theory (Maurer et al., 1958), the subpleu-\nral endometrial implant theory (Lillington et al.,\n1972) and the prostaglandin F2α theory (Rossi and\nGoplerud, 1974).  As for the intraperitoneal air the-\nory, it is assumed that air entering the abdominal\ncavity through the uterine and fallopian tubes at the\ntime of menstruation could enter the thorax through\na defective pore due to diaphragmatic endometrio-\nsis, and thus cause a pneumothorax (Maurer et al.,\n1958).  In our patient, because small fenestrations\nwere found in the right diaphragm and pneumotho-\nrax was not found in the lung, this hypothesis is\nconsidered highly likely.  Histologically, although\nno endometrial tissue was found in the diaphragm,\nthe tissue might have fallen out, causing formation\nof the small pore (Shiraishi, 1991).\nThis disease was diagnosed as catamenial\npneumothorax before surgery, because it was right\npneumothorax in a middle-aged female and the\nsymptoms repeated around the start of her men-\nstruation.  Presence of the diaphragmatic fenestra-\ntions, as an intraoperative finding, and her having a\nnormal lung, created a relatively certain diagnostic\nbasis.  After surgery, hormonal therapy was started\nand pneumothorax did not recur during that period.\nFig. 2.  The excised diaphragm showing the perforation penetrating the intraperi-\ntoneum.  The scale indicates 1 cm.\n\n\n28\nS. Yasui et al.\nFour months after the treatment was stopped, right\npneumothorax recurred in accordance with the start\nof her menstruation.  From the positive effects of\nthe hormonal therapy, it was again highly possible\nthat this case was a catamenial pneumothorax.\nTreatments for this patient focused on surgical\nand hormonal therapies.  Indication for therapy de-\npended on the severity and frequency of the pneu-\nmothorax, age and patient’s desire for pregnancy.\nSurgical therapy included closure of the diaphrag-\nmatic fenestration and pleural symphysis, with the\nfrequent use of VATS.  However, the recurrence\nrate of cases in which diaphragmatic lesions were\nexcised is high, 21% (Banba et al., 1983).  In our\npatient, because a large number of preliminary le-\nsions were observed in the diaphragm during sur-\ngery, the recurrence after surgery may have been\ndue to a new fistula occurring at another site on the\ndiaphragm.  However, this surgery is used normally\nwhen the pneumothorax is causing the problem, yet\nmay simultaneously be a very useful method for\ncreating a diagnostic standard for discounting natu-\nral pneumothorax due to bulla, observation of dia-\nphragmatic defective pores and allopatric endo-\nmetriosis in the pleura (Inagaki et al., 1999).\nHormonal therapy follows drug treatment for\nendometriosis, including testosterone derivatives,\nGn-RH agonists and oral contraceptives (Slabbynck\net al., 1991).  However, it is difficult to continue this\ntherapy because of the appearance of side effects\n(Banba et al., 1983), and because when it is stopped,\nthe symptoms recur; thus this therapy has not been\nestablished as a complete treatment method.  To es-\ntablish a more effective treatment method, it will be\nnecessary to elucidate endometriosis as a prognos-\ntic background as well as other related pathologic\nconditions.\nReferences\n 1 Bamba J, Masaki M, Kohda S, Matsushita H.  Treat-\nment of catamenial pneumothorax.  Nippon Kyobu\nShikkan Gakkai Zasshi 1983;42:571–577 (in Japa-\nnese with English abstract).\n 2 Inagaki M, Usui S, Okazaki H, Funakoshi N.  A case\nof catamenial pneumothorax resected of diaphragmat-\nic endometriosis thoracoscopicaly.  Nippon Kokyuki\nGakkai Zasshi 1999;13:779–783 (in Japanese with\nEnglish abstract).\n 3 Lillington GA, Mitchell SP, Wood GA.  Catamenial\npneumothorax.  JAMA1972;219:1328–1332.\n 4 Maurer ER, Schaal JA, Mendez FL.  Chronic recur-\nrent spontaneous pneumothorax due to endometrio-\nsis of the diaphragm.  JAMA 1958;168:2013–2014.\n 5 Rossi NP, Goplerud CP.  Recurrent catamenial pneu-\nmothorax.  Arch Surg 1974;109:173–176.\n 6 Shiraishi T.  Catamenial pneumothorax:  report of a\ncase and review of the Japanese and non-Japanese\nliterature.  Thorac Cardiovasc Surg 1991;39:304–\n307.\n 7 Slabbynck H, Laureys M, Impens N, De Vroey P,\nSchandevyl W.  Recurring catamenial pneumotho-\nrax treated with a Gn-RH analogue.  Chest 1991;100:\n851.\nReceived December 9, 2002; accepted December 18, 2002\nCorresponding author:  Sakiko Yasui","source_license":"CC0","license_restricted":false}