Yonago Acta medica 2003;46:25–28

A Case of Catamenial Pneumothorax Treated by VideoAssisted Thoracoscopic Surgery
Sakiko Yasui, Yuji Taniguchi, Yoshimasa Suzuki, Kazuhiko Makihara, KoichiroOkada, Norimasa Ito, Kiyosuke Ishiguro and Shigetsugu Ohgi
Division of Organ Regeneration Surgery, Department of Surgery, School of Medicine, Tottori University Faculty of Medicine, Yonago 683-8504 Japan
This is a case of a 47-year-old female who had a medical history of right pneumothorax forthe second time. The pneumothorax, accompanying the start of menstruation, recurredand the patient was hospitalized. From the medical history, a catamenial pneumothorax
was suspected. As for intraoperative findings, many small fenestrations of 1 mm or 3 mm
were present in the border region with the muscle bundle of the central tendon of thediaphragm. The lesion site of the diaphragm and the apex area as a biopsy were partially
excised under video-assisted thoracoscopic surgery. Although a postoperative Gn-RH
agonist was started for endometriosis, it was stopped because side effects appeared. Because
the right pneumothorax recurred in accordance with the start of menstruation, the treatment was changed to danazol. To date, the pneumothorax has not recurred.
Key words: catamenial pneumothorax; diaphragmatic fenestration; hormonal therapy; video-assisted
thoracoscopic surgery

Patient ReportCatamenial pneumothorax is a disease in which apneumothorax repeats in accordance with the menstruation cycle (Maurer et al., 1958). While there
are many unknown points regarding the mechanismof its occurrence, however, this disease is assumed
to be caused by endometriosis. Thus, because endometriosis cases have increased recently, this disease
is expected to increase in the future as well. We
experienced a case of a patient in whom a perforated site of the diaphragm was excised under videoassisted thoracoscopic surgery (VATS) and the disease recurred when hormonal therapy was stopped
after surgery. The case is reported here with a discussion of the literature.

In 1998, a 45-year-old female consulted a local physician because right chest pain and dyspnea appeared suddenly. The symptoms were alleviated byconservative treatment, specifically an analgetic. In
the middle of December 2000, right chest pain anddyspnea recurred and the patient again consulted
the physician. The symptoms were not alleviatedby the same treatment. On December 27, she wasreferred to the Clinic of Respiratory Internal Medicine of Tottori University Hospital and admitted asa patient. Diagnosis as right spontaneous pneumothorax was made and the symptoms were alleviatedby continuous chest drainage. Because the pain occurred at the start of her menstruaction, catamenial
pneumothorax was suspected. The patient was followed-up at the outpatient section after discharge.

Abbreviation: VATS, video-assisted thoracoscopic surgery

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On February 18, 2001, pain recurred 2 days prior toher menstruation. On February 21, she consulted
the Clinic again. Because a recurrence of the rightpneumothorax was found, she was immediatelyhospitalized in the Clinic of Thoracic Surgery, Tottori
University Hospital.
The patient had experienced 2 pregnancies but
only 1 delivery. Her menstruation was regular, butwith dysmenorrhea. There was no past illness thatwas especially notable.
On admission, her height was 155 cm, bodyweight 45 kg, and nutrition was slightly poor. Body
temperature was 36.6˚C, blood pressure 100/72
mmHg and pulse rate 70/min. Ausculation revealed
faint respiratory sounds in the right lung. There
were no rale sounds or cardiac murmur. There wereno abnormal physical findings in the abdomen.
Chest X-rays showed a right pneumothorax
without bulla or pleural effusion. Although continuous chest drainage was conducted, complete pulmonary expansion was not obtained (Fig. 1). Computed tomography of the chest showed no bulla orother abnormal findings. Electrocardiograms also
showed no abnormalities. The bleeding time andcoagulation time were normal, and no abnormalities were found in hematological examinations, urinalysis, biochemical examinations, etc.
On February 28, 2001, VATS was performed
in a recumbent position on the inferior left side
under general anesthesia with separate ventilation.
A thoracoport of 5.5 mm was inserted through the5th intercostal middle axillary line and the inside of
the thorax was observed with a hard scope. Irregular hypertrophic pleura were found at the apex areain the right thorax, but no bulla or adhesions werepresent. Subsequently, a thoracoport of 11.5 mm
was inserted through the 4th intercostal anterior
axillary line, and another one of 5.5 mm through the6th intercostal scapular angle for further observation. A small fenestration of about 3 mm in diameter was found in the tendon center of the diaphragm and air bubbles were present around it. Deposits of a blackish brown color and small pores ofabout 1 mm in diameter were also observed aroundthe diaphragm. From these findings, the disease
was diagnosed as catamenial pneumothorax. Thetendon center of the diaphragm, including the smallpores, was resected using an ENDO GIA Universal
30-2.5 (United States Surgical Corp., Norwalk,CT). The hypertrophic pleura in the apex part werealso resected, using an ENDO GIA Universal 452.5 (United States Surgical Corp.).
The sizes of the excised sections were 33 × 11mm from the diaphragm (Fig. 2) and 33 × 5 mmfrom the apex. In the diaphragm, there was a slitlike partial change of 7 mm, and mild chronic inflammation was found around it, which is not generally observable in disease as endometriosis. Inthe apex section, a severe pneumonic change and anon-specific fibrous formation were found just under the pleura, but no tissue indicating endometriosis or pneumothorax.
Although a postoperative atelectasis occurredin the right pulmonary inferior lobe, it improved on
day 5. Subsequently, its course was favorable.
During gynecological examination, endometriosis
of the intraperitoneal organs was not detected.
However, since the serum CA125 level increased toFig. 1. Preoperative Chest roentgenogram showing aright pneumothorax after continuous chest drainage.
The arrows indicate visceral pleura.

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Fig. 2. The excised diaphragm showing the perforation penetrating the intraperitoneum. The scale indicates 1 cm.

76.3 and laparoscopy examination was not performed, the possibility of endometriosis was undeniable. The patient was discharged 15 days aftersurgery, and administration of leuprorelin acetate,
an Gn-RH agonist, was started. Because symptoms
of carpal tunnel syndrome appeared, which might
have been caused by the growth of smooth muscle,
its administration was stopped 4 months later.
After 4 months, following the cessation of hormonal therapy, on November 10, 2001, 1 day prior to
the start of her menstruation, the pneumothorax
with right chest pain recurred. However, the collapse was so mild that the symptoms cured naturally. On November 16, oral administration of danazol was started. At present, the pneumothorax has
not recurred after 1 year.

ral endometrial implant theory (Lillington et al.,1972) and the prostaglandin F2α theory (Rossi andGoplerud, 1974). As for the intraperitoneal air theory, it is assumed that air entering the abdominal
cavity through the uterine and fallopian tubes at thetime of menstruation could enter the thorax througha defective pore due to diaphragmatic endometriosis, and thus cause a pneumothorax (Maurer et al.,1958). In our patient, because small fenestrations
were found in the right diaphragm and pneumothorax was not found in the lung, this hypothesis isconsidered highly likely. Histologically, althoughno endometrial tissue was found in the diaphragm,
the tissue might have fallen out, causing formationof the small pore (Shiraishi, 1991).
This disease was diagnosed as catamenial
pneumothorax before surgery, because it was rightpneumothorax in a middle-aged female and thesymptoms repeated around the start of her menstruation. Presence of the diaphragmatic fenestrations, as an intraoperative finding, and her having anormal lung, created a relatively certain diagnostic
basis. After surgery, hormonal therapy was startedand pneumothorax did not recur during that period.

Discussion
Although there is no consistent view regarding amechanism for the occurrence of catamenial pneumothorax, there are 3 hypotheses: the intraperitoneal air theory (Maurer et al., 1958), the subpleu27

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Four months after the treatment was stopped, rightpneumothorax recurred in accordance with the startof her menstruation. From the positive effects of
the hormonal therapy, it was again highly possiblethat this case was a catamenial pneumothorax.
Treatments for this patient focused on surgicaland hormonal therapies. Indication for therapy depended on the severity and frequency of the pneumothorax, age and patient’s desire for pregnancy.
Surgical therapy included closure of the diaphragmatic fenestration and pleural symphysis, with thefrequent use of VATS. However, the recurrence
rate of cases in which diaphragmatic lesions wereexcised is high, 21% (Banba et al., 1983). In ourpatient, because a large number of preliminary lesions were observed in the diaphragm during surgery, the recurrence after surgery may have been
due to a new fistula occurring at another site on thediaphragm. However, this surgery is used normallywhen the pneumothorax is causing the problem, yet
may simultaneously be a very useful method for
creating a diagnostic standard for discounting natural pneumothorax due to bulla, observation of diaphragmatic defective pores and allopatric endometriosis in the pleura (Inagaki et al., 1999).
Hormonal therapy follows drug treatment forendometriosis, including testosterone derivatives,
Gn-RH agonists and oral contraceptives (Slabbyncket al., 1991). However, it is difficult to continue thistherapy because of the appearance of side effects(Banba et al., 1983), and because when it is stopped,
the symptoms recur; thus this therapy has not been
established as a complete treatment method. To es-tablish a more effective treatment method, it will benecessary to elucidate endometriosis as a prognostic background as well as other related pathologicconditions.

References
1 Bamba J, Masaki M, Kohda S, Matsushita H. Treatment of catamenial pneumothorax. Nippon Kyobu
Shikkan Gakkai Zasshi 1983;42:571–577 (in Japanese with English abstract).
2 Inagaki M, Usui S, Okazaki H, Funakoshi N. A caseof catamenial pneumothorax resected of diaphragmatic endometriosis thoracoscopicaly. Nippon Kokyuki
Gakkai Zasshi 1999;13:779–783 (in Japanese withEnglish abstract).
3 Lillington GA, Mitchell SP, Wood GA. Catamenialpneumothorax. JAMA1972;219:1328–1332.
4 Maurer ER, Schaal JA, Mendez FL. Chronic recurrent spontaneous pneumothorax due to endometriosis of the diaphragm. JAMA 1958;168:2013–2014.
5 Rossi NP, Goplerud CP. Recurrent catamenial pneumothorax. Arch Surg 1974;109:173–176.
6 Shiraishi T. Catamenial pneumothorax: report of acase and review of the Japanese and non-Japanese
literature. Thorac Cardiovasc Surg 1991;39:304–307.
7 Slabbynck H, Laureys M, Impens N, De Vroey P,Schandevyl W. Recurring catamenial pneumothorax treated with a Gn-RH analogue. Chest 1991;100:851.

Received December 9, 2002; accepted December 18, 2002
Corresponding author: Sakiko Yasui28