mothorax for pulmonary tuberculosis. In 1971 she developed a bronchopleural fistula, with Aspergillus infection of the pleura and the appearance of a "fungous ball." Bronchopleural fistulae as a late complication of therapeutic pneumothorax preceded Aspergillus infection of the pleura in all of the cases in which operation was not performed, as reported by Krakowski et al? and Voisin et al." The actual cause of the bronchopleural fistula is uncertain in our patient, since it was not associated with positive mycobacterial cultures; four of the patients reported by Voisin et al 8 also had no evidence of active tuberculosis. The development of a "pleural aspergilloma" has also been previously described. While in our patient it was difficult to determine radiologically whether the fungus ball was in a lung cavity or in the pleural space, the latter seems likely, for no lung cavity was seen in the chest x-ray film of 1967. Likewise in the other cases reported, Aspergillus infection of the pleura seemed to be a result of pleural disease rather than of rupture of a lung cavity into the pleura. All treatment in this case was intrapleural with no systemic therapy so that a parenchymallesion would not have been reached. In our patient, instillation of antifungal agents into the pleura led to apparently permanent eradication of the organisms and of the fungus ball. Since the bronchopleural fistula remained, and since the patient seemed a poor risk for major surgical repair, it was decided to create an Eloesser flap for longterm drainage of the pleural space. With this treatment, permanent closure of the bronchopleural flstula appears to have

occurred.

1 Salfelder K, Capretti C, Hartung M, et al: Dos casos poco comunes de aspergilosis pulmonary pleural. Mycopathol Mycol AppI14:78, 1961 2 Barlow D: Aspergillosis complicating pulmonary tuberculosis. Proc R Soc Moo 47 :877, 1954 3 Monod 0, Dieudonne P, Tardieu P: Les aspergilloses pulmonaries postoperatoires. J Fr Moo Chir Thorae 18: 579,1964 4 Golebiowski AK: Pleural aspergillosis following resection for pulmonary tuberculosis. Tubercle 39: In, 1958 5 Sitkowski W, Piotrowski A, Kryszkiewicz T: Aspergillus fumigatus Iako przyczyna powiklania pooperacyjnego. ( AspergiUus fumigatus as the cause of postoperative complications). Gruzlica 27: 1069, 1959 6 Sochocky S: Infection of pneumonectomy space with AspergiUus fumigatus treated by nystatin. Dis Chest 36: 554, 1959 7 Krakowka P, Rowinska E, Halweg H: Infection of the pleura by Aspergillus fumigafus. Thorax 25 :245, 1970 8 Voisin C, Sergeant YH, Wallaert MC, et al: L'aspergillose pleurale ( Aspects etiologiques, Anatomo-cliniques et therapeutiques. A propos de 15 observations) . Rev Tuberc Pneumol33:477, 1969 9 Irani FA, Dolovieh J, Newhouse MT: Bronchopulmonary and pleural aspergillosis. Am Rev Resp Dis 103:552, 1971 10 Eloesser L: Of an operation for tuberculous empyema. Ann Thorac Surg 8:355, 1969

98 MAULL, Me ELVEIN

Infarcted cExtralobar Sequestratien · •

Pulmo~ary

Kimball I. Matdl, M.D.,·· and Richard B. McEloein, "M.D.t

A 32-year-old man who presented with symptoms 8111gestiDI empyema was foud at thoracotomy to have Infarction of an eDndo.... pulmonary sequestration. He w. completely reUeved by esdsion of the sequestration after securiDg the systemic vascular pedicle which bad undergone torsion. Recopition of the BDOmalo.. bloody supply and other colllenital anomalies In such cases Is emphasized.

P

ulmonary sequestration is a manifestation of disturbed embryonic development with nonfunctioning lung tissue receiving its vascular supply from the systemic circulation. Two forms of pulmonary sequestration are described, intralobar and extralobar. Infarcted pulmonary sequestration of either type has not been described previously. This is a report of a patient with an infarcted extralobar pulmonary sequestration, an unusual complication of an uncommon entity.

CASE REPoRT A 32-year-old white man presented with a two-week history of intermittent, severe, pleuritic, nonradiating anterior pain in the left side of his chest. His initial treatment with analgesics was ineffective, and a chest x-ray film 11 days prior to admission showed a left lower-lobe infiltrate and pleural effusion. The patient was started on therapy with antibiotics and expectorants when he developed fever and chills. No therapeutic response occurred. His pain grew more severe, and the patient had increasing dyspnea, insomnia, and fatigability. His past history was unremarkable. Physical examination revealed an acutely ill and mildly tachypneic man complaining of pain in the left side of his chest. The thorax was of normal configuration with dullness to percussion over the lower left portion of the thorax. Breath sounds were absent in this same area, with diminished breath sounds superiorly. The right side of the thorax was normal. Laboratory data on admission included a hematocrit reading of 39 percent and a leukocyte count of 12,BOO/cu mm with 71 percent segmented neutrophils and 5 percent stab forms. Chest roentgenogram showed what was thought to be an enlarged left pleural effusion. Thoracocentesis was perfonned, but no fluid or purulent material was obtained. On the second hospital day, left thoracotomy was performed. At operation, BOO ml of slightly serosanguineous sterile fluid was removed. A thickened fibrinous peel was identified over the atelectatic left lower lobe, diaphragm, and chest wall. A discrete mass of purplish-red firm tissue was found posterolateral to the left lower lobe. It had a pleural covering and a single pedicle which had twisted upon itseH. The ·From the Department of Surgery, Good Samaritan Hospital, and the University of Kentucky Medical Center, Lexington. Resident/Surgery, University of Kentu cky Medieal ··Chief 1 Center. tclinical Associate Professor of Surgery, University of Kentucky Medical Center. Reprint reqtle8t8: Dr. McElvein, 2121 Nicholasville Rd., lexington, Kentuckf/40503

CHEST, 68: 1, JULY, 1975

vascular supply originating from the descending aorta above the diaphragm was secured, and the specimen was removed. A decortication was performed, The patient's hospital course was uncomplicated. He was discharged on the seventh postoperative day, and most notable was the prompt relief of pain and dyspnea postoperatively. Pathologic Findings

The sequestered lobe measured 6 X 8 X 6 cm and weighed 60 gm. It was composed of mottled, purplish-red finn tissue with a single artery entering by the pedicle, the lumen of which was filled with a thrombus. Microscopically, it was characterized by hemorrhage and necrosis. Recognizable lung tissue was interspersed with bronchioles. The pathologic diagnosis was infarcted pulmonary sequestration.

foregut duplication or communication are the most commonly encountered entities. None was found in the present case.

REFERENCES 1 Pryce DM: Lower accessory pulmonary artery with intralobar sequestration of lung. J Path Bact 58:457, 1946 2 Boyden EA: Bronchogenic cyst and theory of intralobar sequestration. J Thorac Surg 35: 605, 1958 3 Eaker AB, Hannon JL, French SU: Pulmonary sequestration: A review of the English literature with a report of four cases. Am J Surg 95:31, 1958 4 Smith ZA: Some controversial aspects of intralobar sequestration of the lung. Surg Gynecol Obstet 114:57, 1962 5 Carter R: Pulmonary sequestration. Ann Thorac Surg 7:68, 1969

DISCUSSION

Extralobar pulmonary sequestration was first described in 1861 by Rokitansky and Rektorzik. Although first considered an accessory lobe, Pryce's' clear description of intralobar pulmonary sequestration in 1946 served to consolidate the two entities. The pathogenesis remained in doubt and continues to be a source of considerable discussion. Several reviews are exhaustive in this regard.v" A summary of the important differences between intralobar and extralobar pulmonary sequestration is found in the Table 1 (modified from Carter") . The most important concern of the surgeon when dealing with pulmonary sequestration is an awareness of the anomalous systemic vascular supply originating from the aorta which may arise above or below the diaphragm. Serious hemorrhage and even death have been reported when the condition is not recognized at surgery. The treabnent of extralobar pulmonary sequestration is excision. The high incidence of associated anomalies, approaching 40 percent in some series, stresses the need for complete intrathoracic exploration at the time of operation. Pulmonary agenesis, eventration or diaphragmatic hernia, ectopic pancreatic tissue, and Table I--Claaraelerime. of I ntralobcrr and Es.ralobar Pulmonary Seque.aratioru

Feature

Intralobar

Site

Localized to posterior Found above or below basilar segments; no diaphragm; hasseparate separatepleural pleural investment investment

Extralobar

Laterality

60% on left

Arterialsupply

Fromaorta; often Fromsystemic or pullarge, above or below monary artery; usually diaphragm small and variable

Venous drainage

Pulmonaryveins

Azygos, hemiazyg08, or portal venous system

A&cIooiated anomalies

Rare

Frequentand serious

Presentation

Recurrent infection

Asymptomatic; incidental radiographic finding

CHEST, 68: 1, JULY, 1975

90% on left

Demonstration of Ebstein's Anomaly by Simultaneous Catheter-Tip Localization of the Tricuspid Valve and Right Coronary Artery Visualization: A N·ew Method* Richard E. Kerber, M.D., Melvin L. Marcus, M.D., and Paul M. Wolfson, D.O.

The diagnosis of EbsteiD's anomaly bas tnditioally been made by angiocardiography and confirmed by simultaneous intracardiac electrocardiographic IIIId pressure recordiDp. These techniques may result in faile positive or negative tests. A new method is proposed, whereby the right COnMIary artery is' used as an anglographic marker for the tricuspid 811Dulus and a pressure catheter simultaneously marks the position of the tricuspid valve. In the right anterior obUque position the tip of the pressure catheter should be just under the right coronary artery as the tricuspid leaftets close In systole, indicating the 80maal relatioDship of the tricuspid leaftets and annulus. In EbstelD's anomaly the tip of the catheter extends weD past the coronary artery in systole, demonstrating the characteristic displacement of the atCachments of the tricuspid valve downward toward the right ventricular apex. anomaly a well described congenital malE bstein's formation of the tricuspid valve; its characteristic is

feature is abnormal attachment of the tricuspid leaflets to the right ventricular wall. l - 4 This can generally be demonstrated by selective right ventricular angiography.4 Intracardiac electrode catheters have also been extensively used to confirm the diagnosis. 5 ,6 However, o From

the Cardiovascular Center Department of Internal Medicine, University of Iowa and Veterans Administration Hospitals, Iowa City Iowa. Supported in part by National Heart and Lung Institute Grant No. HL-OI4388 and in part by NHLI Grant No. HL"()5729. Reprint requests: Dr. Kerber, University Hospitals, Iowa City

52242

DEMONSTRATION OF EBSTEIN'S ANOMALY 99