Journal of the Formosan Medical Association (2014) 113, 385e387
Available online at www.sciencedirect.com
journal homepage: www.jfma-online.com
CASE REPORT
Catamenial pneumothorax associated with multiple diaphragmatic perforations and pneumoperitoneum in a reproductive woman Lin Baoquan a, Zou Liangjian b,*, Wang Qiang b, Jin Hai b, Chen Hezhong b, Xu Zhiyun b a b
Department of Cardiothoracic Surgery, Fuzhou General Hospital, Fuzhou, China Department of Cardiothoracic Surgery, Changhai Hospital, Shanghai, China
Received 8 January 2011; received in revised form 11 March 2011; accepted 25 April 2011
KEYWORDS catamenial pneumothorax; diaphragmatic perforations; pneumoperitoneum
Catamenial pneumothorax (CP) is considered to be an extremely rare entity, characterized by recurrent pneumothorax occurring between the day before and within 72 hours after the onset of menses, usually in the right-side thorax cavity in women of reproductive age.The etiology remains obscure. We report a rare case of CP complicated with multiple diaphragmatic perforations as the only thoracoscopy finding, and also with right-side isolated pneumoperitoneum confirmed by a chest X-ray. This case strongly supports the hypothesis that CP may be caused by the air through the perforations of the diaphragm. Copyright ª 2012, Elsevier Taiwan LLC & Formosan Medical Association. All rights reserved.
Introduction Catamenial pneumothorax (CP) is considered to be an extremely rare entity, characterized by recurrent pneumothorax occurring between the day before and within 72 hours after the onset of menses, usually in the right-side Conflicts of interest: The authors have no conflicts of interest relevant to this article. * Corresponding author. Department of Cardiothoracic Surgery, Changhai Hospital, 168 Changhai Road, Shanghai 200433, People’s Republic of China. E-mail address: [email protected] (Z. Liangjian).
thorax cavity in women of reproductive age.1,2 The etiology remains obscure. We report a rare case of CP complicated with multiple diaphragmatic perforations as the only thoracoscopy finding, and also with right-side isolated pneumoperitoneum confirmed by a chest X-ray. This case strongly supports the hypothesis that CP may be caused by the air through the perforations of the diaphragm.
Case report A 36-year-old nonsmoking woman was admitted to our hospital due to multiple recurrent pneumothoraces. During
0929-6646/$ - see front matter Copyright ª 2012, Elsevier Taiwan LLC & Formosan Medical Association. All rights reserved. doi:10.1016/j.jfma.2011.04.006
386 the past decade, she had 17 episodes of right-side chest discomfort and dyspnea, all occurring within 48 hours after the onset of menstruation. Chest symptoms commenced at the age of 29 years. The first documented pneumothorax was at the age of 30, which was drained by needle pleurocentesis in the right second intercostal space. In the next 8 years, the patients suffered from anther 14 episodes of right-side chest pain and mild dyspnea. The symptoms persisted for about 3e4 days each time and were alleviated by conservative treatment, specifically an analgesic. A chest X-ray and computed tomography documented rightside pneumothorax several times. In the middle of February 2008, the patient had severe dyspnea. A chest X-ray revealed right lung compression of 45% accompanied by minimal pleural effusion. She was treated by tube thoracostomy and continuous chest drainage and was discharged after 10 days of treatment. Four days before this hospitalization, she had her 17th chest discomfort. On admission, the chest roentgenograph showed no obvious pneumothorax, but revealed crescent pneumoperitoneum in the right subdiaphragmatic space (Fig. 1). The chest CT revealed small amounts of pneumothorax at the apex area in the right pleural cavity. The medical history was reviewed, and a chest radiograph taken from another hospital also showed pneumoperitoneum. Considering the frequent recurrence of the episodes, we scheduled the video-assisted thoracoscopic surgery (VATS). At exploration, the pleura and lung were found to be normal without any bulla or severe adhesions. The only finding of the operation was multiple 1e5 mm circular diaphragmatic perforations in the central tendon of the diaphragm. On the basis of these findings, a conversion to
Figure 1 The chest roentgenograph showed crescent subphrenic air in the right side (white arrow).
L. Baoquan et al. video-assisted mini-thoracotomy was made, and a partial diaphragmatic resection, including the morbid area, was carried out. The defect was then repaired with X-shaped stitches (Fig. 2). Pleural brushing and talc pleurodesis were performed subsequently. The diaphragm pathology showed no evidence of endometriosis.The patient had uneventful recoveries. During the 26-month follow-up, the patient was symptom-free and suffered no recurrence.
Discussion The rarity of CP makes understanding of its pathophysiology and verification of etiological mechanisms difficult.Several hypotheses have been proposed. Thoracic endometriosis seems to play the most important role in the development of CP. During the menstrual phase, the endometrial ectopic tissue in the visceral pleural ruptures, which leads to the air leakage.1,3 The second mechanism is the presence of diaphragmatic perforations (congenital or acquired) allowing the passage of air from the peritoneal into the pleural cavity, which is aspirated from the genital tract in the absence of the cervical mucous plug during menses.4,5 There exist other hypotheses, for example, Fonseca6 believed that the increased intrapulmonary concentration of prostaglandin PGF2, a potent bronchoconstrictor associated with menstruation , provoked alveolar rupture which led to CP. Our case was diagnosed as CP before surgery, because it was right pneumothorax in an ovulating woman with symptoms repeating at the beginning of her menstruation, despite the absence of pelvic endometriosis symptoms. The presence of the multiple diaphragmatic fenestrations as an intraoperative finding, and her having a normal lung, created a relatively certain diagnostic basis. The mean age at presentation of CP was 35 0.6 years, with a range from 15 to 54 years.7 Direct visualization of the multiple diaphragm perforations without pathological evidence of thoracic endometriosis, and no recurrence after repair of the morbid diaphragm, lends support to the proposed causes involving diaphragm perforations as avenues of thoracic air collection. However, the major problem with this hypothesis is that pneumoperitoneum has been exceptionally described in association with pneumothorax, thus questioning the reliability of this theory. Glauser and Bartlett8 reported five cases of pneumoperitoneum complicated with pneumothorax. However, these occurred during positive pressure ventilation or after trauma or surgery to the neck or thorax. The air often dissected through the mediastinum or retroperitoneum, and ruptured into the peritoneal cavity; a pneumoperitoneum was then formed. In addition to the multiple diaphragmatic perforations shown in Fig. 2, an interesting point about our case, is the radiological sign of pneumoperitoneum before VATS, which seems to argue for the transdiaphragmatic passage of air as the pathogenic mechanism of CP. In the absence of a mucus plug in the menstrual period, air is aspirated from outside, proceeds through the fallopian tube, and penetrates the chest cavity through the perforations of the right diaphragm. Lung bases probably constitute a transient barrier for such passage, and the blockage of the air on the
Transdiaphragmatic passage of air lead to catamenial pneumothorax
387
Figure 2 The perioperative finding of the multiple diaphragmatic perforations (white arrows). (A) Thoracoscopy finding; (B) resected specimen of the diaphragm; (C) video-assisted mini-thoracotomy; and (D) diaphragmatic resection and repair with X-shaped stitches
moment of passage being represented by crescent pneumoperitoneum in our radiologic sign, which implies the occurrence of a pneumoperitoneum as an intermediate step. In our case, the pneumoperitoneum is not an accidental sign; it can also be seen in more than one episode of CP. This theory of transfallopian ascent of air is further strengthened by the observation that CP has been cured by tubal ligation or hysterectomy.9 Although it cannot explain all cases, CP has been described as a recurrence in a few patients after hysterectomy.4 However, none of the other mechanisms mentioned above would be expected to produce a pneumoperitoneum. Our demonstration of CP, concomitant with pneumoperitoneum and multiple diaphragmatic perforations, strongly supports the theory that CP may result from air migrating at menstruation through the fallopian tubes, through the peritoneal cavity, and then through diaphragmatic defects into the pleural cavity. From our experience, a thorough and careful examination of the pleura and lung tissue, especially surfaces of the diaphragm, is of great importance to rule out perforations or possible endometrial implants of the diaphragm in all women (especially in the reproductive age) who undergo surgery for recurrent pneumothorax. VATS is the preferred procedure, providing minimally invasive treatment and enlarging the vision.
References 1. Alifano M, Trisolini R, Cancellieri A, Francois Regnard J. Thoracic endometriosis: current knowledge. Ann Thorac Surg 2006;81:761e9. 2. Alifano M, Roth T, Camilleri Broet S, Schussler O, Magdeleinat P, Regnard JF. Catamenial pneumothorax: a prospective study. Chest 2003;124:1004e8. 3. Korom S, Canyurt H, Missbach A, Schneiter D, Kurrer MO, Haller U, et al. Catamenial pneumothorax revisited: clinical approach and systematic review of the literature. J Thorac Cardiovasc Surg 2004;128:502e8. 4. Bagan P, Le Pimpec Barthes F, Assouad J, Souillamas R, Riquet M. Catamenial pneumothorax: retrospective study of surgical treatment. Ann Thorac Surg 2003;75:378e81. 5. Alifano M, Jablonski C, Kadiri H, Falcoz P, Gompel A, Sophie Camilleri-BS, et al. Catamenial and noncatamenial, endometriosis-related or nonendometriosis-related pneumothorax referred for surgery. Am J Resp Crit Care Med 2007;176:1048e53. 6. Fonseca P. Catamenial pneumothorax: a multifactorial etiology. J Thorac Cardiovasc Surg 1998;116:872e3. 7. Joseph J, Sahn SA. Thoracic endometriosis syndrome: new observations from an analysis of 110 cases. Am J Med 1996;100: 164e70. 8. Glauser FL, Bartlett RH. Pneumoperitoneum in association with pneumothorax. Chest 1974;66:536e40. 9. Slasky BS, Siewers RD, Lecky JW, Zajko A, Burkholder JA. Catamenial pneumothorax: the roles of diaphragmatic defects and endometriosis. Am J Roentgenol 1982;138:639e43.
Available online at www.sciencedirect.com
journal homepage: www.jfma-online.com
CASE REPORT
Catamenial pneumothorax associated with multiple diaphragmatic perforations and pneumoperitoneum in a reproductive woman Lin Baoquan a, Zou Liangjian b,*, Wang Qiang b, Jin Hai b, Chen Hezhong b, Xu Zhiyun b a b
Department of Cardiothoracic Surgery, Fuzhou General Hospital, Fuzhou, China Department of Cardiothoracic Surgery, Changhai Hospital, Shanghai, China
Received 8 January 2011; received in revised form 11 March 2011; accepted 25 April 2011
KEYWORDS catamenial pneumothorax; diaphragmatic perforations; pneumoperitoneum
Catamenial pneumothorax (CP) is considered to be an extremely rare entity, characterized by recurrent pneumothorax occurring between the day before and within 72 hours after the onset of menses, usually in the right-side thorax cavity in women of reproductive age.The etiology remains obscure. We report a rare case of CP complicated with multiple diaphragmatic perforations as the only thoracoscopy finding, and also with right-side isolated pneumoperitoneum confirmed by a chest X-ray. This case strongly supports the hypothesis that CP may be caused by the air through the perforations of the diaphragm. Copyright ª 2012, Elsevier Taiwan LLC & Formosan Medical Association. All rights reserved.
Introduction Catamenial pneumothorax (CP) is considered to be an extremely rare entity, characterized by recurrent pneumothorax occurring between the day before and within 72 hours after the onset of menses, usually in the right-side Conflicts of interest: The authors have no conflicts of interest relevant to this article. * Corresponding author. Department of Cardiothoracic Surgery, Changhai Hospital, 168 Changhai Road, Shanghai 200433, People’s Republic of China. E-mail address: [email protected] (Z. Liangjian).
thorax cavity in women of reproductive age.1,2 The etiology remains obscure. We report a rare case of CP complicated with multiple diaphragmatic perforations as the only thoracoscopy finding, and also with right-side isolated pneumoperitoneum confirmed by a chest X-ray. This case strongly supports the hypothesis that CP may be caused by the air through the perforations of the diaphragm.
Case report A 36-year-old nonsmoking woman was admitted to our hospital due to multiple recurrent pneumothoraces. During
0929-6646/$ - see front matter Copyright ª 2012, Elsevier Taiwan LLC & Formosan Medical Association. All rights reserved. doi:10.1016/j.jfma.2011.04.006
386 the past decade, she had 17 episodes of right-side chest discomfort and dyspnea, all occurring within 48 hours after the onset of menstruation. Chest symptoms commenced at the age of 29 years. The first documented pneumothorax was at the age of 30, which was drained by needle pleurocentesis in the right second intercostal space. In the next 8 years, the patients suffered from anther 14 episodes of right-side chest pain and mild dyspnea. The symptoms persisted for about 3e4 days each time and were alleviated by conservative treatment, specifically an analgesic. A chest X-ray and computed tomography documented rightside pneumothorax several times. In the middle of February 2008, the patient had severe dyspnea. A chest X-ray revealed right lung compression of 45% accompanied by minimal pleural effusion. She was treated by tube thoracostomy and continuous chest drainage and was discharged after 10 days of treatment. Four days before this hospitalization, she had her 17th chest discomfort. On admission, the chest roentgenograph showed no obvious pneumothorax, but revealed crescent pneumoperitoneum in the right subdiaphragmatic space (Fig. 1). The chest CT revealed small amounts of pneumothorax at the apex area in the right pleural cavity. The medical history was reviewed, and a chest radiograph taken from another hospital also showed pneumoperitoneum. Considering the frequent recurrence of the episodes, we scheduled the video-assisted thoracoscopic surgery (VATS). At exploration, the pleura and lung were found to be normal without any bulla or severe adhesions. The only finding of the operation was multiple 1e5 mm circular diaphragmatic perforations in the central tendon of the diaphragm. On the basis of these findings, a conversion to
Figure 1 The chest roentgenograph showed crescent subphrenic air in the right side (white arrow).
L. Baoquan et al. video-assisted mini-thoracotomy was made, and a partial diaphragmatic resection, including the morbid area, was carried out. The defect was then repaired with X-shaped stitches (Fig. 2). Pleural brushing and talc pleurodesis were performed subsequently. The diaphragm pathology showed no evidence of endometriosis.The patient had uneventful recoveries. During the 26-month follow-up, the patient was symptom-free and suffered no recurrence.
Discussion The rarity of CP makes understanding of its pathophysiology and verification of etiological mechanisms difficult.Several hypotheses have been proposed. Thoracic endometriosis seems to play the most important role in the development of CP. During the menstrual phase, the endometrial ectopic tissue in the visceral pleural ruptures, which leads to the air leakage.1,3 The second mechanism is the presence of diaphragmatic perforations (congenital or acquired) allowing the passage of air from the peritoneal into the pleural cavity, which is aspirated from the genital tract in the absence of the cervical mucous plug during menses.4,5 There exist other hypotheses, for example, Fonseca6 believed that the increased intrapulmonary concentration of prostaglandin PGF2, a potent bronchoconstrictor associated with menstruation , provoked alveolar rupture which led to CP. Our case was diagnosed as CP before surgery, because it was right pneumothorax in an ovulating woman with symptoms repeating at the beginning of her menstruation, despite the absence of pelvic endometriosis symptoms. The presence of the multiple diaphragmatic fenestrations as an intraoperative finding, and her having a normal lung, created a relatively certain diagnostic basis. The mean age at presentation of CP was 35 0.6 years, with a range from 15 to 54 years.7 Direct visualization of the multiple diaphragm perforations without pathological evidence of thoracic endometriosis, and no recurrence after repair of the morbid diaphragm, lends support to the proposed causes involving diaphragm perforations as avenues of thoracic air collection. However, the major problem with this hypothesis is that pneumoperitoneum has been exceptionally described in association with pneumothorax, thus questioning the reliability of this theory. Glauser and Bartlett8 reported five cases of pneumoperitoneum complicated with pneumothorax. However, these occurred during positive pressure ventilation or after trauma or surgery to the neck or thorax. The air often dissected through the mediastinum or retroperitoneum, and ruptured into the peritoneal cavity; a pneumoperitoneum was then formed. In addition to the multiple diaphragmatic perforations shown in Fig. 2, an interesting point about our case, is the radiological sign of pneumoperitoneum before VATS, which seems to argue for the transdiaphragmatic passage of air as the pathogenic mechanism of CP. In the absence of a mucus plug in the menstrual period, air is aspirated from outside, proceeds through the fallopian tube, and penetrates the chest cavity through the perforations of the right diaphragm. Lung bases probably constitute a transient barrier for such passage, and the blockage of the air on the
Transdiaphragmatic passage of air lead to catamenial pneumothorax
387
Figure 2 The perioperative finding of the multiple diaphragmatic perforations (white arrows). (A) Thoracoscopy finding; (B) resected specimen of the diaphragm; (C) video-assisted mini-thoracotomy; and (D) diaphragmatic resection and repair with X-shaped stitches
moment of passage being represented by crescent pneumoperitoneum in our radiologic sign, which implies the occurrence of a pneumoperitoneum as an intermediate step. In our case, the pneumoperitoneum is not an accidental sign; it can also be seen in more than one episode of CP. This theory of transfallopian ascent of air is further strengthened by the observation that CP has been cured by tubal ligation or hysterectomy.9 Although it cannot explain all cases, CP has been described as a recurrence in a few patients after hysterectomy.4 However, none of the other mechanisms mentioned above would be expected to produce a pneumoperitoneum. Our demonstration of CP, concomitant with pneumoperitoneum and multiple diaphragmatic perforations, strongly supports the theory that CP may result from air migrating at menstruation through the fallopian tubes, through the peritoneal cavity, and then through diaphragmatic defects into the pleural cavity. From our experience, a thorough and careful examination of the pleura and lung tissue, especially surfaces of the diaphragm, is of great importance to rule out perforations or possible endometrial implants of the diaphragm in all women (especially in the reproductive age) who undergo surgery for recurrent pneumothorax. VATS is the preferred procedure, providing minimally invasive treatment and enlarging the vision.
References 1. Alifano M, Trisolini R, Cancellieri A, Francois Regnard J. Thoracic endometriosis: current knowledge. Ann Thorac Surg 2006;81:761e9. 2. Alifano M, Roth T, Camilleri Broet S, Schussler O, Magdeleinat P, Regnard JF. Catamenial pneumothorax: a prospective study. Chest 2003;124:1004e8. 3. Korom S, Canyurt H, Missbach A, Schneiter D, Kurrer MO, Haller U, et al. Catamenial pneumothorax revisited: clinical approach and systematic review of the literature. J Thorac Cardiovasc Surg 2004;128:502e8. 4. Bagan P, Le Pimpec Barthes F, Assouad J, Souillamas R, Riquet M. Catamenial pneumothorax: retrospective study of surgical treatment. Ann Thorac Surg 2003;75:378e81. 5. Alifano M, Jablonski C, Kadiri H, Falcoz P, Gompel A, Sophie Camilleri-BS, et al. Catamenial and noncatamenial, endometriosis-related or nonendometriosis-related pneumothorax referred for surgery. Am J Resp Crit Care Med 2007;176:1048e53. 6. Fonseca P. Catamenial pneumothorax: a multifactorial etiology. J Thorac Cardiovasc Surg 1998;116:872e3. 7. Joseph J, Sahn SA. Thoracic endometriosis syndrome: new observations from an analysis of 110 cases. Am J Med 1996;100: 164e70. 8. Glauser FL, Bartlett RH. Pneumoperitoneum in association with pneumothorax. Chest 1974;66:536e40. 9. Slasky BS, Siewers RD, Lecky JW, Zajko A, Burkholder JA. Catamenial pneumothorax: the roles of diaphragmatic defects and endometriosis. Am J Roentgenol 1982;138:639e43.
