- Email: [email protected]
Pathologic finding of restenosis in stent-implanted right ventricle–pulmonary artery extracardiac conduit
Ann Thorac Surg 1999;68:1411–3
out any signs of infection or valvular dysfunction and is taking fluconazole daily.
Comment In our series of 200 homografts implanted from 1990 to 1998, two cases of PVE were identified; both were of fungal origin. Although these observations are made on a limited population of CryoLife (Kennesaw, GA) homograft valve recipients, they represent a study looking specifically at the relative susceptibility of the homograft to fungal PVE. Several factors may have contributed to the development of fungal endocarditis. In patient 1, the recipient was elderly, had been hospitalized many times in the past, had previously received chemotherapy and radiation for cancer, and had had a pacemaker inserted after the valve grafting procedure. Insertion of central venous catheters, prolonged use of antibiotics, total parenteral nutrition, and immunosuppression of the valve recipient may have all contributed to the enhanced risk of contamination with an opportunistic organism such as Candida albicans. Similar factors have been associated with a dramatic increase in the documented number of nosocomial bloodstream infections due to Candida albicans (487% increase from 1980 to 1989) in large teaching hospitals [3, 4]. Causes for PVE in patient 2 were not as obvious. The recipient was young with no apparent predisposing factors for infection. Although fungal seeding from the recipient remains a possible cause for PVE, the clinical profile and very early onset suggests that the source of infection originated from the implanted donor valve. Interestingly, the Morbidity and Mortality Weekly Report recently cited a case of Candida albicans PVE associated with a contaminated donor aortic valve homograft. The Candida albicans cultured from the donor valve trimmings during processing and at the time of its removal from the recipient were genetically similar as revealed by DNA finger-printing [5]. This report led us to consider the possibility of factory-acquired infection. CryoLife, with the largest series, reported more than 22,000 homografts implanted with 22 cases of fungal PVE. Information from CryoLife regarding processing of their homograft valves is proprietary. Nonetheless, the information they did provide indicates that all donor homograft valves are tested for sterility by culturing trimmed paravalvular tissue. These homografts, even if initially cultured positive for fungus, are sterilized in warm (37°C) solution for 24 hours with multiple antibiotics and antifungal agents to destroy common bacterial and fungal contaminants before cryopreservation. After cryopreservation and storage, these valves are recultured for organisms and then distributed for clinical use [5]. In summary, our experience emphasizes the need for meticulous prevention of fungal infection preoperatively in the recipient, but for maximum assurance of homograft sterility, such measures should include mandatory elimination of fungal-contaminated homografts from the donor pool. Once fungal PVE is diagnosed, removal and replacement of the infected valve along with systemic antifungal therapy is required [2]. Postoperative adjuvant medical management with 6 to 7 weeks of © 1999 by The Society of Thoracic Surgeons Published by Elsevier Science Inc
CASE REPORT NEMOTO ET AL RESTENOSIS OF STENTED EXTRACARDIAC CONDUIT
1411
intravenous amphotericin B followed by oral fluconazole is recommended. However, the appropriate duration of treatment with fluconazole in the absence of symptoms remains uncertain (6 months versus lifelong) [6]. This disparity of treatment length is based on the chance of a second reoccurrence versus the risk of developing PVE with a fluconazole-resistant fungus. Moreover, the rarity of fungal PVE and the scarcity of literature on the subject fail to provide treatment guidelines. Dr V. Paul Addonizio is a John A. Hartford Foundation Scholar, New York, New York.
References 1. O’Brien MF, Stafford EG, Gardner MA, et al. Allograft valve replacement: long-term follow-up. Ann Thorac Surg 1995;60: S65–70. 2. Cowgill LD, Addonizio VP, Hopeman AR, et al. A practical approach to prosthetic valve endocarditis. Ann Thorac Surg 1987;43:450–7. 3. Melgar GR, Nasser RM, Gordon SM, et al. Fungal prosthetic valve endocarditis in 16 patients: an 11 year experience in a tertiary care hospital. Medicine 1997;76:94 –103. 4. Nasser RM, Melgar GR, Longworth DL, et al. Incidence and risk of developing fungal prosthetic valve endocarditis after nosocomial candidemia. Am J Med 1997;103:25–32. 5. Clark E, Chia J, Waterman S, et al. Candida albicans endocarditis associated with a contaminated aortic valve allograft— California, 1996. MMWR 1997;46:261–3. 6. Muehrcke DD, Lytle BW, Cosgrove DM III. Surgical and long-term antifungal therapy for fungal prosthetic valve endocarditis. Ann Thorac Surg 1995;60:538– 43.
Pathologic Finding of Restenosis in Stent-Implanted Right Ventricle– Pulmonary Artery Extracardiac Conduit Shintaro Nemoto, MD, Akira Sakai, MD, Yutaka Miyoshi, MD, Kiyomitsu Yasuhara, MD, and Masashi Seguchi, MD Departments of Cardiovascular Surgery and Pediatric Cardiology, Seirei Hamamatsu General Hospital, Shizuoka, Japan
We describe an excised specimen of a stent-implanted valved equine pericardial extracardiac conduit in the right heart. It appears from careful pathologic examination that the stent acted as a nidus for thrombus formation followed by thick neo-intimal development over the stent, which caused restenosis. Restenosis occurred despite anticoagulation. (Ann Thorac Surg 1999;68:1411–3) © 1999 by The Society of Thoracic Surgeons Accepted for publication March 25, 1999. Address reprint requests to Dr Nemoto, Laboratories of Cardiac Molecular and Cellular Physiology, Baylor College of Medicine, Department of Medicine—Cardiology, Veterans Affairs Medical Center, Room 243, Bldg 110, 2002 Holcombe Blvd, Houston, TX 77030.
0003-4975/99/$20.00 PII S0003-4975(99)00736-5
1412
CASE REPORT NEMOTO ET AL RESTENOSIS OF STENTED EXTRACARDIAC CONDUIT
Ann Thorac Surg 1999;68:1411–3
A
lthough stent implantation has improved the outcome of the use of right ventricle–pulmonary artery conduits, a major problem with restenosis still exists. We present the case of a patient in whom a stent that had not fully expanded served as the nidus for severe restenosis.
A male baby was born with tetralogy of Fallot, pulmonary atresia, and aortopulmonary window. When he was 6 months old, total repair was performed using a handmade equine pericardial extracardiac conduit (14 mm in diameter) with a fabricated trileaflet valve. Three years after repair, the conduit was severely stenosed, and balloon dilation was attempted. The pressure gradient across the conduit was reduced from 90 mm Hg to 42 mm Hg. One year later, there was major restenosis (gradient, 72 mm Hg). A second balloon dilation was not successful. When the patient was 5 years of age, a balloon-expandable stent (Palmaz-Schatz, Johnson & Johnson Interventional Systems) was successfully implanted. The gradient decreased from 70 mm Hg to 30 mm Hg. Aspirin (2 mg 䡠 kg⫺1 䡠 day⫺1) was administered after this operation. One year after the implantation, the patient had a high fever and complete atrioventricular block. Methicillinresistant Staphylococcus aureus was detected in blood cultures. An echocardiogram showed severe restenosis of the conduit. After signs of infection had disappeared with antibiotic therapy, conduit replacement was performed. With the patient under cold cardioplegic arrest, the right ventricular outflow tract was reconstructed by the technique of Cerfolio and colleagues [1]. Briefly, both the side and posterior parts of the fibrous tissue surrounding the conduit were preserved. A handmade monocuspid polytetrafluoroethylene patch was anastomosed to the preserved fibrous tissue, distal pulmonary artery stump, and proximal right ventricular incision. The patient made a good recovery. A postoperative echocardiogram showed a substantial reduction in the right ventricular to left ventricular pressure ratio (0.45). The pathologic findings were as follows: Grossly, a continuous fibrous peel was present on the surface of the stent and in the space between the stent and the conduit wall (Fig 1). This peel was the major cause of conduit stenosis. The stent had not been uniformly deployed throughout the conduit, and this resulted in the space in which the peel developed. The stent along with thick granular fibrin seemed to be floating in the right ventricular cavity at the proximal end of the conduit (Fig 2). Histologic examination of the fibrous peel revealed similar fibrous connective tissue proliferation with hyalinosis.
Fig 1. Cross section of excised conduit at distal anastomosis level. A continuous fibrous peel was present on the surface of the stent and in the space between the stent and the conduit wall, and it constituted the major cause of conduit stenosis. (graduation ⫽ 1 mm.)
with full expansion of the stent, a fibrous peel developed in the Dacron conduit, which was excised [2]. Thus, even full stent deployment may not prevent this process. In contrast, Ando and coworkers [5] reported that degenerated valve leaflets and external compression by the sternum were the main causes of stenosis in a valved equine pericardial conduit. Intimal peel formation was not prominent in the absence of a stent. It appears from our findings and those of others that the stent acts as a nidus for thrombus formation, which is followed by development of a thick neo-intima over the stent. This possible mechanism of stenosis is different from that reported by Powell and colleagues [6] in which recurrent obstruction of stent-implanted homografts in the right ventricle–pulmonary artery position was caused by external compression and progressive stenosis outside the stented region. In conclusion, although the indications for stenting
Comment The pathologic finding in this case of stenosis may differ from the findings reported by Almagor [2], Hosking [3], and their associates in stented conduits. One characteristic of the stenosis in those reports was progressive thickening of the fibrous peel because of organization of thrombus between the peel and the Dacron conduit [4]. However, even
Fig 2. Cross section of excised conduit at proximal anastomosis level. The stent had not been uniformly deployed, and this created the space between it and the conduit wall. The stent along with thick granular fibrin seemed to be floating in the right ventricular cavity.
Ann Thorac Surg 1999;68:1413– 4
CASE REPORT COWL ET AL CATAMENIAL PNEUMOTHORAX
1413
stenosed right ventricle–pulmonary artery conduits are still evolving, it should be emphasized that an incompletely deployed stent can serve as a nidus for thrombus formation and progressive obstruction, even when anticoagulants are used. We thank Dr Hiroshi Kobayashi of Seirei Hamamatsu General Hospital and Dr Yasuko Tomizawa of Tokyo Women’s Medical College for histologic studies and discussion.
References 1. Cerfolio RJ, Danielson GK, Puga FJ, et al. Results of an autologous tissue reconstruction for replacement of obstructed extracardiac conduits. J Thorac Cardiovasc Surg 1995;110:1359– 66. 2. Almagor Y, Prevosti LG, Bartorelli AL, et al. Balloon expandable stent implantation in stenotic right heart valved conduits. J Am Coll Cardiol 1990;16:1310– 4. 3. Hosking MC, Williams WG, Freedom RM, et al. Intravascular stent prosthesis for right ventricular outflow obstruction. J Am Coll Cardiol 1992;20:373– 80. 4. Agarwal KC, Edwards WD, Feldt RH, Danielson GK, Puga FJ, McGoon DC. Clinicopathological correlates of obstructed right-sided porcine-valved extracardiac conduits. J Thorac Cardiovasc Surg 1981;81:591– 601. 5. Ando M, Imai Y, Takanashi Y, Hoshino S, Seo K, Terada M. Fate of trileaflet equine pericardial extracardiac conduit used for the correction of anomalies having pulmonic ventricle– pulmonary arterial discontinuity. Ann Thorac Surg 1997;64: 154– 8. 6. Powell AJ, Lock JE, Keane JF, Perry SB. Prolongation of RV-PA conduit life span by percutaneous stent implantation. Intermediate-term results. Circulation 1995;92:3282– 8.
Visualization of Diaphragmatic Fenestration Associated With Catamenial Pneumothorax Clayton T. Cowl, MD, MS, William F. Dunn, MD, and Claude Deschamps, MD Division of Pulmonary and Critical Care Medicine and Section of General Thoracic Surgery, Mayo Medical Center and Mayo Graduate School of Medicine, Rochester, Minnesota
Catamenial pneumothorax is a rare entity of unknown etiology characterized by recurrent accumulation of air in the thoracic space during or preceding menstruation. We documented the presence of a diaphragmatic fenestration during thoracoscopy, lending support for hypotheses involving diaphragmatic defects as possible avenues of air collection in the thorax. (Ann Thorac Surg 1999;68:1413– 4) © 1999 by The Society of Thoracic Surgeons Accepted for publication March 26, 1999. Address reprint requests to Dr Cowl, Division of Pulmonary and Critical Care Medicine, Mayo Medical Center, 200 First St SW, Rochester, MN 55905; e-mail: [email protected].
© 1999 by The Society of Thoracic Surgeons Published by Elsevier Science Inc
Fig 1. Chest radiograph revealed complete right-sided lung collapse in this otherwise healthy 39-year-old woman.
A
lthough cases of spontaneous pneumothorax are relatively common, recurrent pneumothoraces associated with menstruation are rare. Catamenial pneumothoraces, coined from the Greek root meaning monthly [1], were first described in the late 1950s by Maurer and colleagues [2], but its physiologic mechanism remains unclear. A 39-year-old woman was referred for evaluation of recurrent right-sided pneumothoraces. She was in excellent health 12 years before presentation with no history of pelvic endometriosis before she developed episodes of chest discomfort characterized by a “dull ache” in the right anterosuperior chest. At first, the pain episodes resolved spontaneously. Gradually, however, the pain, dyspnea, and a sensation of a “fluid motion” in her chest persisted. Right-sided apical pneumothoraces were confirmed radiographically several times each year, and, in retrospect, usually at the onset of menstruation. Although frequent, the pneumothoraces were never more than 15% of the hemithoracic volume and the patient had not previously undergone needle or chest tube decompression. Trial therapies with danazol, leuprolide, and medroxyprogesterone acetate were unsuccessful. Three weeks before presentation the patient noted increasing dyspnea and thoracic pain on exertion, then experienced right-sided neck pressure associated with right lung collapse (Fig 1). Video-assisted thoracoscopic exploration of the right pleural cavity was subsequently performed. A 1-cm circular diaphragmatic fenestration was identified (Fig 2) and sutured closed, followed by mechanical pleurodesis. No intrathoracic endometriosis, blebs, or bullae were visualized. Serum prostaglandin levels were normal. Postoperative recovery was uneventful and she remained asymptomatic 9 months after her procedure. 0003-4975/99/$20.00 PII S0003-4975(99)00735-3
out any signs of infection or valvular dysfunction and is taking fluconazole daily.
Comment In our series of 200 homografts implanted from 1990 to 1998, two cases of PVE were identified; both were of fungal origin. Although these observations are made on a limited population of CryoLife (Kennesaw, GA) homograft valve recipients, they represent a study looking specifically at the relative susceptibility of the homograft to fungal PVE. Several factors may have contributed to the development of fungal endocarditis. In patient 1, the recipient was elderly, had been hospitalized many times in the past, had previously received chemotherapy and radiation for cancer, and had had a pacemaker inserted after the valve grafting procedure. Insertion of central venous catheters, prolonged use of antibiotics, total parenteral nutrition, and immunosuppression of the valve recipient may have all contributed to the enhanced risk of contamination with an opportunistic organism such as Candida albicans. Similar factors have been associated with a dramatic increase in the documented number of nosocomial bloodstream infections due to Candida albicans (487% increase from 1980 to 1989) in large teaching hospitals [3, 4]. Causes for PVE in patient 2 were not as obvious. The recipient was young with no apparent predisposing factors for infection. Although fungal seeding from the recipient remains a possible cause for PVE, the clinical profile and very early onset suggests that the source of infection originated from the implanted donor valve. Interestingly, the Morbidity and Mortality Weekly Report recently cited a case of Candida albicans PVE associated with a contaminated donor aortic valve homograft. The Candida albicans cultured from the donor valve trimmings during processing and at the time of its removal from the recipient were genetically similar as revealed by DNA finger-printing [5]. This report led us to consider the possibility of factory-acquired infection. CryoLife, with the largest series, reported more than 22,000 homografts implanted with 22 cases of fungal PVE. Information from CryoLife regarding processing of their homograft valves is proprietary. Nonetheless, the information they did provide indicates that all donor homograft valves are tested for sterility by culturing trimmed paravalvular tissue. These homografts, even if initially cultured positive for fungus, are sterilized in warm (37°C) solution for 24 hours with multiple antibiotics and antifungal agents to destroy common bacterial and fungal contaminants before cryopreservation. After cryopreservation and storage, these valves are recultured for organisms and then distributed for clinical use [5]. In summary, our experience emphasizes the need for meticulous prevention of fungal infection preoperatively in the recipient, but for maximum assurance of homograft sterility, such measures should include mandatory elimination of fungal-contaminated homografts from the donor pool. Once fungal PVE is diagnosed, removal and replacement of the infected valve along with systemic antifungal therapy is required [2]. Postoperative adjuvant medical management with 6 to 7 weeks of © 1999 by The Society of Thoracic Surgeons Published by Elsevier Science Inc
CASE REPORT NEMOTO ET AL RESTENOSIS OF STENTED EXTRACARDIAC CONDUIT
1411
intravenous amphotericin B followed by oral fluconazole is recommended. However, the appropriate duration of treatment with fluconazole in the absence of symptoms remains uncertain (6 months versus lifelong) [6]. This disparity of treatment length is based on the chance of a second reoccurrence versus the risk of developing PVE with a fluconazole-resistant fungus. Moreover, the rarity of fungal PVE and the scarcity of literature on the subject fail to provide treatment guidelines. Dr V. Paul Addonizio is a John A. Hartford Foundation Scholar, New York, New York.
References 1. O’Brien MF, Stafford EG, Gardner MA, et al. Allograft valve replacement: long-term follow-up. Ann Thorac Surg 1995;60: S65–70. 2. Cowgill LD, Addonizio VP, Hopeman AR, et al. A practical approach to prosthetic valve endocarditis. Ann Thorac Surg 1987;43:450–7. 3. Melgar GR, Nasser RM, Gordon SM, et al. Fungal prosthetic valve endocarditis in 16 patients: an 11 year experience in a tertiary care hospital. Medicine 1997;76:94 –103. 4. Nasser RM, Melgar GR, Longworth DL, et al. Incidence and risk of developing fungal prosthetic valve endocarditis after nosocomial candidemia. Am J Med 1997;103:25–32. 5. Clark E, Chia J, Waterman S, et al. Candida albicans endocarditis associated with a contaminated aortic valve allograft— California, 1996. MMWR 1997;46:261–3. 6. Muehrcke DD, Lytle BW, Cosgrove DM III. Surgical and long-term antifungal therapy for fungal prosthetic valve endocarditis. Ann Thorac Surg 1995;60:538– 43.
Pathologic Finding of Restenosis in Stent-Implanted Right Ventricle– Pulmonary Artery Extracardiac Conduit Shintaro Nemoto, MD, Akira Sakai, MD, Yutaka Miyoshi, MD, Kiyomitsu Yasuhara, MD, and Masashi Seguchi, MD Departments of Cardiovascular Surgery and Pediatric Cardiology, Seirei Hamamatsu General Hospital, Shizuoka, Japan
We describe an excised specimen of a stent-implanted valved equine pericardial extracardiac conduit in the right heart. It appears from careful pathologic examination that the stent acted as a nidus for thrombus formation followed by thick neo-intimal development over the stent, which caused restenosis. Restenosis occurred despite anticoagulation. (Ann Thorac Surg 1999;68:1411–3) © 1999 by The Society of Thoracic Surgeons Accepted for publication March 25, 1999. Address reprint requests to Dr Nemoto, Laboratories of Cardiac Molecular and Cellular Physiology, Baylor College of Medicine, Department of Medicine—Cardiology, Veterans Affairs Medical Center, Room 243, Bldg 110, 2002 Holcombe Blvd, Houston, TX 77030.
0003-4975/99/$20.00 PII S0003-4975(99)00736-5
1412
CASE REPORT NEMOTO ET AL RESTENOSIS OF STENTED EXTRACARDIAC CONDUIT
Ann Thorac Surg 1999;68:1411–3
A
lthough stent implantation has improved the outcome of the use of right ventricle–pulmonary artery conduits, a major problem with restenosis still exists. We present the case of a patient in whom a stent that had not fully expanded served as the nidus for severe restenosis.
A male baby was born with tetralogy of Fallot, pulmonary atresia, and aortopulmonary window. When he was 6 months old, total repair was performed using a handmade equine pericardial extracardiac conduit (14 mm in diameter) with a fabricated trileaflet valve. Three years after repair, the conduit was severely stenosed, and balloon dilation was attempted. The pressure gradient across the conduit was reduced from 90 mm Hg to 42 mm Hg. One year later, there was major restenosis (gradient, 72 mm Hg). A second balloon dilation was not successful. When the patient was 5 years of age, a balloon-expandable stent (Palmaz-Schatz, Johnson & Johnson Interventional Systems) was successfully implanted. The gradient decreased from 70 mm Hg to 30 mm Hg. Aspirin (2 mg 䡠 kg⫺1 䡠 day⫺1) was administered after this operation. One year after the implantation, the patient had a high fever and complete atrioventricular block. Methicillinresistant Staphylococcus aureus was detected in blood cultures. An echocardiogram showed severe restenosis of the conduit. After signs of infection had disappeared with antibiotic therapy, conduit replacement was performed. With the patient under cold cardioplegic arrest, the right ventricular outflow tract was reconstructed by the technique of Cerfolio and colleagues [1]. Briefly, both the side and posterior parts of the fibrous tissue surrounding the conduit were preserved. A handmade monocuspid polytetrafluoroethylene patch was anastomosed to the preserved fibrous tissue, distal pulmonary artery stump, and proximal right ventricular incision. The patient made a good recovery. A postoperative echocardiogram showed a substantial reduction in the right ventricular to left ventricular pressure ratio (0.45). The pathologic findings were as follows: Grossly, a continuous fibrous peel was present on the surface of the stent and in the space between the stent and the conduit wall (Fig 1). This peel was the major cause of conduit stenosis. The stent had not been uniformly deployed throughout the conduit, and this resulted in the space in which the peel developed. The stent along with thick granular fibrin seemed to be floating in the right ventricular cavity at the proximal end of the conduit (Fig 2). Histologic examination of the fibrous peel revealed similar fibrous connective tissue proliferation with hyalinosis.
Fig 1. Cross section of excised conduit at distal anastomosis level. A continuous fibrous peel was present on the surface of the stent and in the space between the stent and the conduit wall, and it constituted the major cause of conduit stenosis. (graduation ⫽ 1 mm.)
with full expansion of the stent, a fibrous peel developed in the Dacron conduit, which was excised [2]. Thus, even full stent deployment may not prevent this process. In contrast, Ando and coworkers [5] reported that degenerated valve leaflets and external compression by the sternum were the main causes of stenosis in a valved equine pericardial conduit. Intimal peel formation was not prominent in the absence of a stent. It appears from our findings and those of others that the stent acts as a nidus for thrombus formation, which is followed by development of a thick neo-intima over the stent. This possible mechanism of stenosis is different from that reported by Powell and colleagues [6] in which recurrent obstruction of stent-implanted homografts in the right ventricle–pulmonary artery position was caused by external compression and progressive stenosis outside the stented region. In conclusion, although the indications for stenting
Comment The pathologic finding in this case of stenosis may differ from the findings reported by Almagor [2], Hosking [3], and their associates in stented conduits. One characteristic of the stenosis in those reports was progressive thickening of the fibrous peel because of organization of thrombus between the peel and the Dacron conduit [4]. However, even
Fig 2. Cross section of excised conduit at proximal anastomosis level. The stent had not been uniformly deployed, and this created the space between it and the conduit wall. The stent along with thick granular fibrin seemed to be floating in the right ventricular cavity.
Ann Thorac Surg 1999;68:1413– 4
CASE REPORT COWL ET AL CATAMENIAL PNEUMOTHORAX
1413
stenosed right ventricle–pulmonary artery conduits are still evolving, it should be emphasized that an incompletely deployed stent can serve as a nidus for thrombus formation and progressive obstruction, even when anticoagulants are used. We thank Dr Hiroshi Kobayashi of Seirei Hamamatsu General Hospital and Dr Yasuko Tomizawa of Tokyo Women’s Medical College for histologic studies and discussion.
References 1. Cerfolio RJ, Danielson GK, Puga FJ, et al. Results of an autologous tissue reconstruction for replacement of obstructed extracardiac conduits. J Thorac Cardiovasc Surg 1995;110:1359– 66. 2. Almagor Y, Prevosti LG, Bartorelli AL, et al. Balloon expandable stent implantation in stenotic right heart valved conduits. J Am Coll Cardiol 1990;16:1310– 4. 3. Hosking MC, Williams WG, Freedom RM, et al. Intravascular stent prosthesis for right ventricular outflow obstruction. J Am Coll Cardiol 1992;20:373– 80. 4. Agarwal KC, Edwards WD, Feldt RH, Danielson GK, Puga FJ, McGoon DC. Clinicopathological correlates of obstructed right-sided porcine-valved extracardiac conduits. J Thorac Cardiovasc Surg 1981;81:591– 601. 5. Ando M, Imai Y, Takanashi Y, Hoshino S, Seo K, Terada M. Fate of trileaflet equine pericardial extracardiac conduit used for the correction of anomalies having pulmonic ventricle– pulmonary arterial discontinuity. Ann Thorac Surg 1997;64: 154– 8. 6. Powell AJ, Lock JE, Keane JF, Perry SB. Prolongation of RV-PA conduit life span by percutaneous stent implantation. Intermediate-term results. Circulation 1995;92:3282– 8.
Visualization of Diaphragmatic Fenestration Associated With Catamenial Pneumothorax Clayton T. Cowl, MD, MS, William F. Dunn, MD, and Claude Deschamps, MD Division of Pulmonary and Critical Care Medicine and Section of General Thoracic Surgery, Mayo Medical Center and Mayo Graduate School of Medicine, Rochester, Minnesota
Catamenial pneumothorax is a rare entity of unknown etiology characterized by recurrent accumulation of air in the thoracic space during or preceding menstruation. We documented the presence of a diaphragmatic fenestration during thoracoscopy, lending support for hypotheses involving diaphragmatic defects as possible avenues of air collection in the thorax. (Ann Thorac Surg 1999;68:1413– 4) © 1999 by The Society of Thoracic Surgeons Accepted for publication March 26, 1999. Address reprint requests to Dr Cowl, Division of Pulmonary and Critical Care Medicine, Mayo Medical Center, 200 First St SW, Rochester, MN 55905; e-mail: [email protected].
© 1999 by The Society of Thoracic Surgeons Published by Elsevier Science Inc
Fig 1. Chest radiograph revealed complete right-sided lung collapse in this otherwise healthy 39-year-old woman.
A
lthough cases of spontaneous pneumothorax are relatively common, recurrent pneumothoraces associated with menstruation are rare. Catamenial pneumothoraces, coined from the Greek root meaning monthly [1], were first described in the late 1950s by Maurer and colleagues [2], but its physiologic mechanism remains unclear. A 39-year-old woman was referred for evaluation of recurrent right-sided pneumothoraces. She was in excellent health 12 years before presentation with no history of pelvic endometriosis before she developed episodes of chest discomfort characterized by a “dull ache” in the right anterosuperior chest. At first, the pain episodes resolved spontaneously. Gradually, however, the pain, dyspnea, and a sensation of a “fluid motion” in her chest persisted. Right-sided apical pneumothoraces were confirmed radiographically several times each year, and, in retrospect, usually at the onset of menstruation. Although frequent, the pneumothoraces were never more than 15% of the hemithoracic volume and the patient had not previously undergone needle or chest tube decompression. Trial therapies with danazol, leuprolide, and medroxyprogesterone acetate were unsuccessful. Three weeks before presentation the patient noted increasing dyspnea and thoracic pain on exertion, then experienced right-sided neck pressure associated with right lung collapse (Fig 1). Video-assisted thoracoscopic exploration of the right pleural cavity was subsequently performed. A 1-cm circular diaphragmatic fenestration was identified (Fig 2) and sutured closed, followed by mechanical pleurodesis. No intrathoracic endometriosis, blebs, or bullae were visualized. Serum prostaglandin levels were normal. Postoperative recovery was uneventful and she remained asymptomatic 9 months after her procedure. 0003-4975/99/$20.00 PII S0003-4975(99)00735-3