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J Neurol Neurosurg Psychiatry 1982; 45:457– 460 Skedros DG, Cass SP, Hirsch BE, et al. Beta-2 transferrin assay in clinical management of cerebral spinal fluid and perilymphatic fluid leaks. J Otolaryngol 1993; 22:341–344 Da Silva VF, Shamji FM, Reid RH, et al. Subarachnoidpleural fistula complicating thoracotomy: case report and review of the literature. Neurosurgery 1987; 20:802– 805 Hofstetter KR, Bjelland JC, Patton DD, et al. Detection of bronchopleural-subarachnoid fistula by radionuclide myelography: case report. J Nucl Med 1977; 18:981–983 Labadie EL, Hamilton RH, Lundell DC, et al. Hypoliquorreic headache and pneumocephalus caused by thoracosubarachnoid fistula. Neurology 1977; 27:993–995 Lovaas ME, Castillo RG, Deutschman CS. Traumatic subarachnoid-pleural fistula. Neurosurgery 1985; 17:650 – 652 Lenter C, ed. Cerebrospinal fluid. In: Geigy scientific tables. Caldwell, NJ: West Ciba-Geigy, 1981; 165–177 Cardiac Decortication (Epicardiectomy) for Occult Constrictive Cardiac Physiology After Left Extrapleural Pneumonectomy* John G. Byrne, MD, FCCP; Alexandros N. Karavas, MD; Yolonda L. Colson, MD; Raphael Bueno, MD, FCCP; William G. Richards, PhD; David J. Sugarbaker, MD, FCCP; and Samuel Z. Goldhaber, MD, FCCP Constrictive cardiac physiology typically does not occur in the absence of parietal pericardium. However, we report eight patients who, after left extrapleural pneumonectomy and removal of the parietal pericardium for malignancy, presented with dyspnea, jugular venous distension, and peripheral or generalized edema unresponsive to diuretics. Cardiac decortication (epicardiectomy) was performed whereby a thickened peel encasing the heart was surgically excised, resulting in vigorous contraction and expansion of the heart. In one patient, decortication occurred early after pneumonectomy and was incomplete. Acute signs of inflammation were present, and recurrence necessitated repeat decortication. When patients present with dyspnea, hepatojugular reflux, and peripheral edema refractory to diuretics, constrictive cardiac physiology should be considered in the differential diagnosis, even in the absence of parietal pericardium. (CHEST 2002; 122:2256 –2259) Key words: cardiac decortication; epicardiectomy; pneumonectomy Abbreviations: EPP ⫽ extrapleural pneumonectomy; POD ⫽ postoperative day; RV ⫽ right ventricular xtrapleural pneumonectomy of the left lung includes E removal of the lung, parietal pleura, parietal pericar- dium, and diaphragm.1 Therefore, constrictive cardiac physiology due to pericarditis would not seem possible postoperatively. We report eight patients who nonetheless acquired occult constrictive cardiac physiology due to epicardial constriction, despite left extrapleural pneumonectomy (EPP) for underlying cancer. In all cases, fibrous material grew around and encased the heart. These patients required reoperation for epicardial decortication to alleviate dyspnea and peripheral edema refractory to diuretics. Materials and Methods We undertook a retrospective chart review of the preoperative presentations, clinical evaluations that revealed constrictive physiology, operative procedures, and patient outcomes. Results In the period from February 1997 to January 2000, 133 patients underwent EPP. Seven patients (5%) acquired constrictive cardiac physiology after left EPP for malignant mesothelioma, and one patient acquired constrictive cardiac physiology after left intrapericardial pneumonectomy for non-small cell lung carcinoma. The mesothelioma was epithelial in six patients and epithelial/sarcomatoid in one patient. The parietal pericardium was entirely excised in seven patients and partially excised in one patient. Pericardial and/or diaphragm reconstruction was accomplished using polytetrafluoroethylene (Gore-Tex; W.L. Gore & Associates; Flagstaff, AZ). Adjunct therapy included preoperative chemotherapy and radiation in one patient and intraoperative heated chemotherapy in four patients. No talc was used in any patients. After a median interval of 3.3 months (range, 1.6 to 18.9 *From the Cardiac Surgery (Drs. Byrne and Karavas), Thoracic Surgery (Drs. Colson, Bueno, Richards, and Sugarbaker), and Cardiovascular Divisions (Dr. Goldhaber), Brigham and Women’s Hospital, Boston, MA. Presented at the American Heart Association Scientific Sessions, Anaheim, CA, November 11–14, 2001. Manuscript received November 30, 2001; revision accepted June 6, 2002. Correspondence to: John G. Byrne, MD, FCCP, Division of Cardiac Surgery, Brigham and Women’s Hospital, 75 Francis St, Boston, MA; e-mail: [email protected] 2256 Downloaded From: http://publications.chestnet.org/pdfaccess.ashx?url=/data/journals/chest/21985/ on 05/07/2017 Selected Reports months) following pneumonectomy, patients were readmitted with symptoms and signs consistent with constrictive cardiac physiology. Symptoms always included dyspnea, jugular venous distension, hepatojugular reflux, and peripheral leg edema, all unresponsive to diuretics. One patient required dialysis because of anasarca. In the absence of parietal pericardium, medical-care providers did not initially attribute the symptoms to constrictive cardiac physiology (Fig 1). Details that led to the diagnosis of constrictive cardiac physiology in these eight patients are presented in Table 1. CT scan was performed in four patients, but it was not conclusive and did not help further in the establishment of the diagnosis. MRI, performed in one patient, revealed fibrous bands encasing the heart. All patients were re-explored via a left thoracotomy. Intraoperative findings in each case included a “leatherlike” scar covering uniformly the entire surface of the heart and restricting the heart from its normal contraction and relaxation. Dissection in the plane between the fibrous tissue and the myocardium (epicardiectomy) resulted in decortication of the entire myocardium with minimal bleeding. Peeling off the fibrous tissue restored vigorous contraction and expansion of the heart. In one patient, operated on 47 days after the initial pneumonectomy, the tissue was too adherent to peel off entirely without risking injury to the left anterior descending coronary artery. Therefore, the anterior portion of the fibrous peel was left in situ. Another patient developed right ventricular (RV) free-wall rupture on coughing in preparation for extubation in the operating room. She required immediate re-exploration and institution of cardiopulmonary bypass, with arterial cannulation via the left ventricular apex advanced through the aortic valve and venous drainage initially via the rent in the right ventricle and later through the right atrium after further dissection. The RV rent was repaired with bovine pericardium, and the patient had an unremarkable recovery. Details of histopathology and clinical follow-up are provided in Table 2. One patient died of cachexia and heart failure on postoperative day (POD) 14, despite successful decortication. One patient, who could only be partially decorticated, acquired a relapsing constrictive epicarditis requiring hospital readmission and reoperative decortication on POD 42 and POD 81 after the primary decortication. He died 10 days after the last procedure as a consequence of progressive hemodynamic deterioration. The other six patients recovered fully from a cardiac aspect. Three patients died of the primary cancer between 101 days and 132 days after decortication, and the other three patients are alive 20, 26, and 50 months after decortication. Discussion Pericardium consists of two layers: (1) an outer fibrous parietal layer with collagen fibers interlaced with extensive elastic fibers, and (2) an inner visceral serous layer composed of a single layer of mesothelial cells. In 1913, Sauerbruch2 performed the first decortication for constrictive parietal pericarditis and showed that the myocardium regained full function after the thickened pericardial layer was removed. Nissen and Schweizer3 reported that in almost all patients with constrictive pericardial disease, both the parietal and visceral layers of the pericardium thicken.3 They operated on and successfully decorticated the first patient described with epicardial constriction in the presence of a normal parietal pericardium. In our series, decortication was performed by creating a plane between the myocardium and the thickened peel, Figure 1. Angiographic diastolic pressures in eight patients (in mm Hg). RA a ⫽ right atrium a wave; RV dia ⫽ right ventricular diastolic pressure; PA dia ⫽ pulmonary artery diastolic pressure; PCWP mean ⫽ mean pulmonary capillary wedge pressure; LV dia ⫽ left ventricular diastolic pressure. www.chestjournal.org CHEST / 122 / 6 / DECEMBER, 2002 Downloaded From: http://publications.chestnet.org/pdfaccess.ashx?url=/data/journals/chest/21985/ on 05/07/2017 2257 Table 1—Clinical Presentations* Patient No. Intervals Clinical Manifestations 1 568 d after left EPP Anasarca requiring dialysis 2 103 d after LIPP Dyspnea, confusion, gait disturbance 3 113 d after left EPP Edema, dyspnea 4 108 d after left EPP 5 85 d after left EPP Edema, ascites, dyspnea, nausea and vomiting, syncope, seizure Anasarca, dyspnea, hepatic congestion 6 92 d after left EPP Edema, dyspnea 7 60 d after left EPP Edema, dyspnea, cardiac decompensation 8 47 d after left EPP Edema, ascites, dyspnea Echo Findings Prior to Decortication Restrictive ventricular motion, abnormal septal motion, ejection fraction 55% Lack of myocardial expansion, abnormal septal motion, ejection fraction 50% Limited expansion of the heart, abnormal septal motion, ejection fraction 55% Abnormal RV dynamics, abnormal septal motion, ejection fraction 50% Right ventricle restricted, abnormal septal motion, ejection fraction 50% Normal left ventricular/RV function; enlarged atria, ejection fraction 50% Mild-to-moderate RV dysfunction, abnormal septal motion, ejection fraction 50% *LIPP ⫽ left intrapericardial pneumonectomy. essentially performing an epicardiectomy. This procedure was accomplished with minimal bleeding. In one patient only, decorticated only 47 days after pneumonectomy (the earliest time interval in our series), we could not safely separate the fibrous tissue from the heart without causing bleeding, so decortication was limited to the inferolateral region of the myocardium. This inability to safely dissect the rind is most likely attributed to the time interval between pneumonectomy and decortication. We believe that inflammation was still active, because the tissue was highly vascularized and fibrin organization had not been completed. The patient subsequently relapsed and required reoperative decortication. All other patients were operated after a period of 2 months, usually between 2 to 4 months after pneumonectomy. This seems to be the appropriate timing, late enough to allow the inflammation to subside but also early enough to detect possible local metastasis. In the patient who experienced RV free-wall rupture, we believe that the RV decortication was likely too aggressive, and this, in addition to the absence of the epicardium (after decortication), weakened the RV free wall, making it susceptible to tearing. Therefore, on the right ventricle in particular, we suggest that decortication should be conservative. In our effort to detect precipitating factors for this unusual clinical manifestation, we could not identify any component of the adjunct therapy or intraoperative approach at the time of the EPP that would differentiate patients who acquired epicardial scarring from those who did not. Some received chemotherapy and all patients had reconstruction of the diaphragm using polytetrafluoroethylene. Patients with mesothelioma or lung cancer are seen postoperatively at 2 weeks, 6 weeks, 3 months, and then every 3 months for 2 years. At each visit, they undergo physical examination, chest radiography and, if abnormal- Table 2—Histologic Findings and Outcomes Patient No. Histopathologic Findings 1 Dense fibroconnective tissue, tumor positive 2 Extensive fibrosis, chronic inflammation, tumor negative Dense fibroconnective tissue, focal hemosiderin, tumor negative Dense fibrous tissue with erosion, granulation, fibrinous exudate, tumor negative Dense fibrous tissue, chronic inflammation, hemosiderin, tumor negative Fibrous tissue with chronic inflammation, tumor positive Metastatic malignant mesothelioma, tumor positive (Report from repeat decortication) fibrous tissue with acute and chronic inflammation, fibrin, deposition, granulation tissue 3 4 5 6 7 8 Follow-up Status as of August 5, 2001 Required reoperation right atrial thrombectomy on POD 13 No further hemodynamic compromise Death on POD 157 No further hemodynamic compromise Alive on POD 534 No further hemodynamic compromise Alive on POD 1439 No follow-up Alive on POD 782 No further hemodynamic compromise Death on POD 132 Death due to cachexia and heart failure Required two repeat decortications Death on POD 14 Death on POD 89 2258 Downloaded From: http://publications.chestnet.org/pdfaccess.ashx?url=/data/journals/chest/21985/ on 05/07/2017 Death on POD 101 Selected Reports ities are identified, echocardiography and chest CT. We now pay particular attention to the signs and symptoms of right heart failure and have adopted a low threshold to perform echocardiography. Since this is the first report of this clinical manifestation, we do not recommend echocardiography unless clinical signs and symptoms of right heart failure are present. Echocardiography is very sensitive in detecting constrictive physiology, but findings should be confirmed by cardiac catheterization, which will show the typical pressure equalization and also establish the indication for operation. CT scan was insensitive in detecting morphologic abnormalities, while MRI, in the one occasion used, revealed clearly the fibrous bands encasing the heart. CT scan is used primarily for the detection of metastatic disease. Conclusion Constrictive cardiac physiology can occur even in the absence of the parietal pericardium. Constrictive epicarditis evolves from an early phase with highly vascularized tissue that is difficult to dissect to a late phase in which the tissue is less vascularized and easy to excise. When patients present with dyspnea, hepatojugular reflux, and peripheral edema refractory to diuretics, constrictive cardiac physiology should be considered in the differential diagnosis, even in the absence of parietal pericardium. References 1 Sugarbaker DJ, Flores RM, Jaklitsch MT, et al. Resection margins, extrapleural nodal status, and cell type determine postoperative long-term survival in trimodality therapy of malignant pleural mesothelioma: results in 183 patients. Thorac Cardiovasc Surg 1999; 117:54 – 63 2 Sauerbruch F. Die Chirurgie des Herzens und seines Beutels. In: Sauerbruch F, ed. Die Chirurgie der Brustorgane. 2nd ed. Berlin, Germany: J Springer, 1920; 141–308 3 Nissen R, Schweizer W. Constrictive epicarditis [in German]. Thoraxchir Vask Chir 1968; 16:296 –300 Bacterial Endocarditis and Functional Mitral Stenosis* A Report of Two Cases and Brief Literature Review Irving Y. Tiong, MD; Gian M. Novaro, MD; Brian Jefferson, MD; Michael Monson, MD; Nicholas Smedira, MD, FCCP; and Marc S. Penn, MD, PhD Mitral valve endocarditis typically results in mitral regurgitation. However, endocarditis leading to functional mitral stenosis is uncommon and, when present, fungal organisms are typically implicated. Thus, obstructive-type bacterial endocarditis due to large vegetations blocking the mitral valve orifice is www.chestjournal.org a rare occurrence, with approximately 20 reported cases in the literature. We report on two patients with bacterial endocarditis and severe functional mitral stenosis requiring emergent surgery. Additionally, this is the first report of vancomycin-resistant enterococcus causing endocarditis and functional mitral stenosis. The discussion emphasizes the hemodynamic instability of these patients and need for early surgical intervention. (CHEST 2002; 122:2259 –2262) Key words: bacterial endocarditis; echocardiography; enterococcal infection; mitral valve stenosis; vegetations Abbreviations: MRSA ⫽ methicillin-resistant Staphylococcus aureus; TEE ⫽ transesophageal echocardiography; VRE ⫽ vancomycin-resistant enterococcus alvular dysfunction in infective endocarditis implies V the development of a new regurgitant lesion. The occurrence of an obstructive or “functionally” stenotic valvular condition in the setting of endocarditis is rare and often goes clinically unsuspected until late in the course of the disease.1– 8 Moreover, suspicion of this condition usually surfaces only in cases of fungal endocarditis.9,10 Importantly, when present, the management of such a scenario is medically challenging and uniformly warrants emergent surgery. We describe two cases of bacterial endocarditis presenting with functional mitral stenosis due to obstructive vegetations at the mitral valve orifice. Novel to our report are the microorganisms involved in this “obstructive-type” of endocarditis, namely methicillinresistant Staphylococcus aureus (MRSA) and vancomycinresistant enterococcus (VRE). A discussion on the management of these patients and a brief review of the literature is provided. Case Reports Case 1 A 54-year-old African-American woman presented with fevers, respiratory distress, and mental status changes. Her medical history was significant for end-stage renal disease with secondary hyperparathyroidism, long-standing hypertension, and IV drug abuse. The week prior to hospital admission, she missed several sessions of hemodialysis, becoming progressively lethargic, and was brought by her family to the emergency department. On presentation, her temperature was 39.3°C, BP was 97/65 mm Hg, and heart rate was 120 beats/min. Physical examination was remarkable for jugular venous distension, an apical holosystolic murmur that radiated to the axilla, a short apical diastolic murmur, and bilateral pulmonary crackles up to the mid-lung fields. *From the Department of Cardiovascular Medicine (Drs. Novaro, Jefferson, Monson, and Penn), Internal Medicine (Dr. Tiong), and Cardiothoracic Surgery (Dr. Smedira), The Cleveland Clinic Foundation, Cleveland, OH. Manuscript received March 21, 2002; revision accepted June 21, 2002. Correspondence to: Marc S. Penn, MD, PhD, Departments of Cardiovascular Medicine and Cell Biology, NC10, Cleveland Clinic Foundation, 9500 Euclid Ave, Cleveland, OH 44195; e-mail: [email protected] CHEST / 122 / 6 / DECEMBER, 2002 Downloaded From: http://publications.chestnet.org/pdfaccess.ashx?url=/data/journals/chest/21985/ on 05/07/2017 2259