the fourth intercostal space, 2 inches inside the midclavicular

line. The first and second heart sounds were normal. A

continuous murmur was heard along the right sternal border. Breath sounds were diminished over the wall of the right side of the chest. The patient's electrocardiogram revealed right axis deviation and mild ventricular hypertrophy. A chest roentgenogram and barium swallow suggested hypoplastic right lung and a cardiac silhouette located in the right side of the chest (Fig 1). The clinical impression was patent ductus arteriosus with hypoplastic right lung. Cardiac catheterization and angiographie studies revealed patent ductus arteriosus which was entered from the pulmonary artery. The right pulmonary artery appeared hypoplastic and the left normal. The presence of the patent ductus arteriosus was further demonstrated by left ventricular cineangiogram (Fig 2). The right upper pulmonary vein entered the right atrium. Arterial oxygen saturation was 92 volumes percent, suggesting mild intrapulmonary shunting. Pulmonary arterial pressure was mildly elevated to 41/26 mm Hg. The patent ductus arteriosus was approached via a median sternotomy. The pericardium was opened, and ductal closure was achieved with two ligatures of heavy silk. The postoperative course was uneventful DISCUSSION

Fifty-nine . cases of patent ductus arteriosus were found on Schechter'sl review of 414 patients with congenital subtractive bronchopulmonic maHonnation. Six of this group underwent ductal closure. The usual surviving patient with an absent lung and persistent patent ductus arteriosus has the left lung absent and the patent ductus on the left. Closure of such a patent ductus is easily achieved through the left side of the chest. 2 ,4 In two patients with absent left lung and right-sided patent ductus arteriosus, the patent ductus was approached via bilateral anterior thoracotomy and via right anterolateral thoracotomy by Swan et aI. 5 . The feasibility and safety of an approach to the patent ductus arteriosus via a median sternotomy was demonstrated both by Kirklin and Silvera and by McGoon a in their experience with patients undergoing open-heart surgery. An approach via median sternotomy was chosen for our patient in order to avoid opening the left pleural space and compressing the single left lung during the ductal closure. Closure of a patent ductus arteriosus is indicated in the presence of a single pulmonary artery. This is especially important if there is pulmonary hypertension, as in our patient. The overloaded pulmonary circuit is exposed to the risk of pulmonary vascular disease if the left-to-right shunt is allowed to persist.

REF'ERENCFS 1 Schechter DC: Congenital absence or deficiency of lung tissue. Ann Thorac Surg 6:286, 1971 2 Nicks R: Agenesis of the lung with persistent ductus arteriosus. Thorax 12: 140, 1957 Closure of patent ductus during open heart 3 McGoon ~: surgery. J Thorae Cardiovasc Surg 48:456, 19644 Jimenez-Martinez M, Perez-Alvarez J, Perez-Trevino C, et al: Agenesis of the lung with patent ductus arteriosus

786 KILPATRICK, KOONTZ, NELSON

during open heart surgery. J Thorae Cardiovasc Surg 50:59,1965 5 Swan H., Owens JC, Pool PE, et al: Absent left pulmonary artery and right-sided patent ductus arteriosus. Arch Surg 87:196; 1963 6 Kirklin]W, Silver AW: Technique of exposing the ductus arteriosus prior to establishing extracorporeal circulation. Proc Staff Meetings Mayo Coo 33:423, 1958

Nocardia Infedion in a Bronchopulmonary Sequestration * MAl George R. Kilpatrick, /r., MC, USA;·· MAl C. H. Koontz, MC, USA;·· and COL Roald A. Nelson, MC, USA, F.C.C.P.

_III,

A C8Ie of iDtnIob8r broaehopubDoamy _ II reported ill wbleb the sequestered hmg W8S lDfeeted with Noeardia sp. Although reeurreDt PYOIeDk lDfeetlo.. .e. commOD in palmoamy tIdI Is tbe 8nt ease report with documented noem-diOllL Bronchopulmonary sequestration is an uncommon disorder. In fact, in our 600-bed referral hospital, there have been only 21 documented cases spanning the last two decades. A striking finding in our experience and in a review of the literature is the signfficant number of patients with this condition who have a history of recurrent pulmonary infections, cough, expectoration, and fever for many years before the diagnosis of sequestration is suspected or proved. 1 For this reason, even though bronchopulmonary sequestration is rare, it is important for clinicians to be aware of this entity and how these cases may present clinically. We present herein the first reported case of recurrent nocardiosis involving a sequestered area of lung. CASEREroRT On a routine chest x-ray film in October 1972, a 26-yearold asymptomatic white man was found to have multiple large pulmonary nodules in the right lower lobe (Fig 1A). There was no past history of recurrent pulmonary infections, and previous chest x-ray films were reported as normal. He had served overseas tours in Europe and southeast Asia, but had been in the United States for one year prior to this chest x-ray examination. Examination of the lungs revealed localized 8ne rAles over the right lower lung field posteriorly, along with percussion dullness. Findings from the remainder of the physical examination, including careful palpation of the testicles and lymph nodes, were normal. The hematocrit, white blood cell count, urinal~ and blood chemistry studies were normal, except for a slightly ·From the Department of Medicine and Pulmonary Disease Service, Fitzsimons Army Medical Center, Denver. The opinions and assertions contained herein are the private views of the authors and do not necessarily reflect the position of the Department of the Army or the Deparbnent of Defense. • • Subspecialty Residents in Pulmonary Disease. Reprint requests: Col. Nelson, Fitzsimons Anny Medical Center, Denver 80240

CHEST, 69: 6, JUNE, 1976

elevated lactic dehydrogenase level of 250 international units ( IU ) (normal, 90 to 200 IU). The erythrocytic sedimentation rate was 34 mm/hr. The results of serum protein electrophoresis and quantitative immunoglobulin levels were also normal. Sputum cultures for bacteria, fungi, and acidfast bacilli were negative. Skin tests with purified protein derivative of tuberculin (5 tuberculin units), histoplasmin, and coccidioidin were negative; however, mumps and Monilia skin tests were positive. Liver and brain scans were normal. Studies of pulmonary function demonstrated a mild restriction in the vital capacity and mild arterial hypoxemia. Tomograms of the pulmonary nodules showed no evidence of calci6cation or cavitation. A percutaneous needle aspiration of one of the pulmonary nodules was performed under fluoroscopic guidance. Ten milliliters of yellow-tan, thick purulent material was aspirated, and culture of this material grew Nocardia sp. (This culture was originally reported as N In-asiliensis, with speciation on the basis of casein hydrolysis, good growth on 0.4 percent gelatin media, and characteristic morphology; however, these tests do not fuHlll the present minimum criteria for differentiating between N In-asiliensis and the more common N asteroides.2 ,3· The isolate, however, was clearly a Nocardia species.) Following the percutaneous needle lung aspiration, the patient developed empyema which required a chest tube for eight days. His temperature spiked to 38.3°C (loo.9°F), but he gradually became afebrile over several days. He initially received intravenous therapy with penicillin, but this was changed to 6 gm of sulfadiazine per day in divided doses·after culturing Nocardia sp. The sulfadiazine dose was reduced to 4 gm/day to maintain a serum sulfadiazine level of 12 to 15 mg/100 mi. The pulmonary nodular infiltrates gradually cleared over the next six months, leaving minimal fibrotic changes (Fig IB ). The patient received treatment with sulfadiazine until the middle of July 1973 (for a total of nine months of therapy). In September 1973 the patient developed chills, fever, malaise, myalgias, headaches, and pain in the right side of the chest, but there was no sputum production. A chest x-ray film revealed a recurrence of the large pulmonary nodules in the right lower lobe. This film was almost identical to the initial film talcen one year previously. Flexible fiberoptic bronchoscopic examination revealed normal bronchial ana-

tomic findings. Endobronchial brushings and sputum specimens failed to grow Nocardia or any other pathogens. The patient was treated for suspected recurrent nocardiosis with sulfadiazine (4 gm/ day) and ampicillin (2 gm/day) in divided doses. Findings from a spinal tap and brain scan were normal. The pulmonary nodular infiltrates again gradually cleared over six months, leaving minimal fibrotic changes. Treatment with ampicillin was discontinued after eight months of therapy. Sulfadiazine was continued, maintaining serum sulfadiazine levels in the range of 10 to 15 mg/loo ml. In October 1974 the patient was hospitalized again for further diagnostic studies because of a suspected structural abnormality of the right lower lobe. He was asymptomatic, and the nocardial infection appeared well controlled by sulfadiazine therapy. A retrograde aortic arteriogram (Fig lC) revealed a large artery arising from the descending thoracic aorta and supplying an area of the right lower lobe with venous drainage from this sequestered lung via the pulmonary vein. Bronchograms of the right lower lobe revealed lack of filling of the peripheral bronchi to this sequestered area. The patient subsequently underwent right thoracotomy and right lower lobectomy to remove the sequestered area. His postoperative course has been uneventful with no evidence of recurrent pulmonary infection. DISCUSSION

Although recurrent infections are common in patients with bronchopuhnonary sequestrations, this is the first reported patient who was infected with Nocardia. The infections usually are presumed to be bacterial pneumonias or nonspecific puhnonary abscesses. There have been several case reports of Mycobacterium tuberculosis in sequestered lung but no other reports of granulomatous infections, to our knowledge. 4 •5 Another unique aspect of this case is that when the patient had a recurrent infection in September 1973, his chest radiograph was nearly identical to his initial film in October 1972, with multiple large nodules in the right lower lobe. This pattern strongly suggested a structural abnormality of the lung, such as sequestration. Nocardiosis is uncommon in the United States but occurs in similar frequency in normal hosts and in per-

Fl .URE 1 (left). Multipl pulmonary nodules i.n right low r lob. lB. (center). I aring of pulmonary nodule aft r ix month of sulfadiazine therapy. Ie (right). ortograrn demonstrating large systemic artery to sequestered area of right lower lobe.

CHEST, 69: 6, JUNE, 1976

NOCARDIA IIIFECnON IN BRONCHOPULMONARY SEQUESTRAnON 787

sons significantly compromised due to malignant neoplasms, immunosuppressive therapy, or collagen vascular disease. 3 The patient presented in this report is a man with apparently nonnal immunologic status whose nocardial infection initially represented probable saprophytic low-grade infection in the sequestered lung. It was only after needle aspiration of the nocardial abscess when empyema developed that the infection became clinically evident. His overall prognosis should be good in that the nocardial infection was controlled with sulfadiazine and ampicillin therapy and there was never evidence of dissemination. Surgical removal of the sequestration effectively alleviated the source of recurrent infection. Of the four pathogenic species of Nocardia, N asteroidea accounts for more than 99 percent of cases of nocardiosis in the United States. There have been only five reported cases of pulmonary or disseminated disease due to N brasiliensis in this country. 2,6 Nocardia asteroides, as well as other nocardial species, are common saprophytes of decaying organic material and are frequently isolated from soil. Man-to-man or animal-toanimal transmission has never been demonstrated. The usual portal of entry is the respiratory tract, most frequently leading to a chronic pulmonary infection. 3, 1 Bronchopulmonary sequestration is an anomaly characterized by partial or complete separation of one segment of the lung from the remainder of the lung. This sequestered lung receives its blood supply from the systemic circulation, generally from an arterY or arteries originating from the thoracic or upper abdominal aorta. Ext;ralobar bronchopulmonary sequestration denotes complete separation of one area of lung from the remaining lung, usually with encasement in its own pleura and venous drainage to the hemiazygous system. Intralobar sequestration is more common and implies a partial developmental separation of a portion of one lobe from its remaining part, with venous drainage generally into pulmonary veins. Bronchopulmonary sequestration is most often diagnosed in young adults before the age of 30 years. It is more commonly found in men than in women, with the sex ratio being 1.5: 1 in the intralobar type and 3: 1 in the extralobar . t,xpe. Some patients are asymptomatic when the lesion is Drst detected after a routine chest x-ray film; however, most patients have had symptoms of recurrent pulmonary infections, often for many years before the diagnosis is made. This condition is strongly suggested on the basis of characteristic roentgenographic findings.! Sequestrations usually involve the medial and posterior basal segments of the lower lobes. When multilobular or cystic lesions, therefore, occur in either lower lobe adjacent to the diaphragm and the posterior chest wall and are located medially on the posteroanterior projection, bronchopulmonary sequestration should be strongly considered. This diagnosis was considered in this case, but the definitive evaluation was diverted with the development of empyema secondary to Nocardia infection. The most important definitive diagnostic procedure is

788 VAlDYA, BERGER, GRANADA

the aortogram which demonstrates an aberrant artery or arteries entering the sequestered area of lung. s This procedure is not only valuable in establishing the diagnosis but is also helpful to the thoracic surgeon planning the operation in that it shows the number, the caliber, and the location of abnormal vessels. The prognosis of this condition without surgery is not known, for there are few cases that have been observed untreated. 9 ,lO The only treabnent for this condition is the surgical removal of the affected lung. This should be easily accomplished by an experienced thoracic surgical team, and the procedure should be curative.

REFERENCES 1 Wier JA, Nelson RA: Congenital vascular anomalies of the lung. In Practice of Medicine. New York, Harper and Rowe, 1972, pp 14-16 2 Berd D: Nocardia b1'ariliensis infection in the United States: A report of nine cases and a review of the literature. Am J elin Pathol 60:254-258, 1973 3 Palmer DL, Harvey RL, Wheeler JIe: Diagnostic and therapeutic consideration in Noetmlio 4fIe1'Oides infedion. Medicine 53:391-401, 1974 4 Schachter EN, Karpick RJ: Bronchopulmonary sequestration and pulmonary tuberculosis. Chest 62:331-333, 1972 5 Lobenwein-Weinegg E: Intralobare bronchopulmonale sequestration and tuberculose. Radiol Coo BioI 41:466-

474, 1972 6 Diamond RD, Bennett JE: Disseminated Noctmlla brariliensis infection. Arch Intern Med 131:735-736, 1973 7 Murray JF, Finegold SM, Froman 5, et al: The changing spectrum of nocardiosis: A review and presentation of nine cases. Am Rev Respir Dis 83:315-330, 1961 8 Ranninger Ie, Valvassori GE: Angiographic diagnosis of intralobar pulmonary sequestration. Am J Roentgenol 92: 540-546, 1964 9 Holstein P, Hielms E: Bronchopulmonary sequestration. J Thorac Cardiovasc Surg 65:462-466, 1973 10 Marks C, Wiener SN, Reydman M: Pulmonary sequestration. Chest 61:253-257, 1972

Pulmonary Infection due to

Torulopsis glaIJrafa* Report of a Case Treeted with Rucytosine Gita N. Vaidya, M.D.; Herbert W. Berget', M.D., F.C.C.P.; and Margarito G. GrantJda, M.D.

w.

A C8Ie of pulmoaary infection due to TorulofIriI gltJbrata in a 43-year-old lDan without other known palmoaary or systemic disease Is deserlbed. The dlaposll made frOID multiple positive sputum euItures.. After treatm.ellt with 8ucytosine for 21 montlls, there w.- llUll'ked

sym,.

°From the Pulmonary Section, Deparbnent of Medicine, Mount Sinai Hospital Services, City Hospital Center at Elmhurst, NY, and the Mount Sinai School of Medicine, City University of New York, New York. Reprint requests: Dr. Vaidya, Mt. Sinai Hospital Services, 79-01 Broadway, Elmhurst, New Y01'k 11373

CHEST, 69: 6, JUNE, 1976

Nocardia infection in a bronchopulmonary sequestration.

A case of intralobar bronchopulmonary sequestration is reported in which the sequestered lung was infected with Nocardia sp. Although recurrent pyogen...
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