706 CASE REPORT SANTORO ET AI. Ann Thorac Surg ANOMALOUS CORONARY ARTERY AND COARCTATION 1995;60:706-8

discharged home and remains well with no evidence of local, systemic, or device infection at 6-month follow-up.

C o m m e n t

Implantable cardioverter-defibrillator implantat ion has become a common procedure, with more than 50,000 devices placed since 1980. Although infection rates as high as 20% have been reported [3], most large series describe an incidence of infection of about 2% [1, 4, 5]. Although many cases are obvious, clinical presentat ion can be subtle. In these situations, imaging studies such as gallium scans [6] or computed tomography [7] may be helpful. As in this case, the gallium scan may also suggest infection on the sensing electrodes or patches, a finding that may be missed with i n d i u m - I l l leukocyte scans [8].

The present case also illustrates the importance of complete removal of all components when the diagnosis of infection is made. Even relatively small portions of retained epicardial sensing electrodes led to persistent problems over an 18-month period. Apparent successful managemen t of device infection has been reported with local irrigation only [2[ or with removal of the generator only [1]. However, many patients with less-than-complete extraction have required subsequent operative removal [5] or have died with evidence of undra ined purulence in the patch components [8]. With the proliferation of non- thoracotomy devices, endocardial lead extraction may also be advisable, although in our patient no communi- cating infection with the t ransvenous ICD was identified and this device was retained. At present, complete re- moval of all components represents the preferred strat- egy for management of infected cardioverter-defibrillators.

References

1. Siclari F, Klein H, Troster HJ, Borst HG. Infectious complica- tions after AICD implantation [Abstract]. PACE 1990;13:547.

2. Taylor RL, Cohen DJ, Widman LE, Chilton RJ, O'Rourke RA. Infection of an implantable cardioverter defibrillator: man- agement without removal of the device in selected cases. PACE 1990;13:1352-5.

3. Barbola J, Denes P, Ezri MD, Hauser RG, Serry C, Goldin MD. The automatic implantable cardioverter defibrillator. Clinical experience, complications and follow-up in 25 patients. Arch Intern Med 1988;148:70-6.

4. Winkle RA, Mead RH, Ruder MA, et al. Long-term outcome with the automatic implantable cardioverter-defibrillator. J Am Coll Cardiol 1989;13:1353-61.

5. Wunderly D, Maloney J, Edel T, McHen D, M, McCarthy PM. Infections in implantable cardioverter defibrillator patients. PACE 1990;13:1360-4.

6. Kelly PA, Wallace S, Tucker B, et al. Postoperative infection with the automatic implantable cardioverter defibrillator: clinical presentation and use of the gallium scan in diagnosis. PACE 1988;11:1220-5.

7. Almassi GH, Olinger GN, Troup PJ, Chapman PD, Goodman LR. Delayed infection of the automatic implantable cardio- verter-defibrillator. Current recognition and management. J Thorac Cardiovasc Surg 1988;95:908-11.

8. Bakker PFA, Hauer RNW, Wever EFD. Infections involving implanted cardioverter defibrillator devices. PACE 1992;15: 654-8.

Origin of Both Coronary Arteries From the Pulmonary Artery and Aortic Coarctation Giuseppe Santoro, MD, Duccio di Carlo, MD, Adriano Carotti, MD, Roberto Formigari, MD, Renata Boldrini, MD, Cesare Bosman, MD, and Luigi Ballerini, MD

Departments of Pediatric Cardiology, Cardiac Surgery, and Pathology, Ospedale Bambino Gesh, Rome, Italy

Single trunk anomalous origin of both coronary arteries from the pulmonary artery is a rare congenital cardiac anomaly. We report on 2 cases of its association with aortic coarctation, the diagnosis of which in living pa- tients is very difficult. We think that the possibility of this anatomic arrangement should always be considered in patients with isolated aortic coarctation whose clinical condition seems impaired rather than improved after an apparently successful coarctectomy.

(Ann Thorac Surg 1995;60:706-8)

S ingle t runk anomalous origin of both coronary arter- ies from the pulmonary artery is a rare malformation

[1, 2l in which the entire myocardial blood supply derives from the pulmonary artery. This anomaly is incompatible with life except in the presence of high pulmonary arterial oxygen saturation or pressure to provide ade- quate myocardial perfusion. Diagnosis has been usually made on autopsy, and only a few cases have been identified during life because of associated malforma- tions [3].

In this article we report on 2 cases of association of anomalous origin of both coronary arteries by a single t runk from the pulmonary artery and isolated aortic coarctation.

Case Reports

Patient 1

A l -month-o ld infant was referred for cardiologic exam- ination because of failure to thrive and congestive heart failure. Electrocardiography showed sinus tachycardia but did not show ST-T ischemic changes suggestive of anomalous coronary artery origin. Echocardiography showed a severe aortic coarctation and mild global hy- pokinesia of the left ventricle; the patient underwent urgent coarctation repair by subclavian flap operation. The postoperative course was complicated by difficulty of weaning from mechanical ventilation. Repeat echocardi- ography showed deteriorated left ventricular function, and eventually the infant died of intractable ventricular

Accepted for publication March 8, 1995.

Address reprint requests to Dr di Carlo, Department of Cardiac Surgery, Ospedale Bambino Gesu, P.zza S. Onofrio, 4, 00165 Rome, Italy.

© 1995 by The Society of Thoracic Surgeons 0003-4975195159.50 0003-4975(95)00311-8

Ann Thorac Surg CASE REPORT SANTORO ET A L 707 1995;60:706-8 ANOMALOUS CORONARY ARTERY AND COARCTATION

failure. On autopsy an anomalous origin of both coronary arteries from pu lmonary ar tery via a single t runk was diagnosed.

Patient 2

A 2-month-old infant was admit ted in congestive heart failure after a coarctation repair per formed in another inst i tut ion 15 days before. Clinical examinat ion revealed hepa tomega ly and bilateral pu lmonary rales. Electrocar- d iography showed sinus tachycardia (180 beats /min) , and diffuse ischemic ST-T changes. On echocard iography an extremely poorly functioning left ventricle was seen, wi thout evidence of residual obstruction at the site of previous coarctation repair. Cardiac catheterization was under taken, which showed an anomalous origin of both coronary arteries from the right pu lmonary artery via a single t runk (Fig 1). Surgical correction with re implanta- tion of the coronary arteries and right pu lmonary ar tery per icardial patch reconstruct ion was performed, but the infant could not be weaned from card iopulmonary by- pass and died in the operat ing room. Autopsy confirmed the angiocardiographic diagnosis and showed fibroelas- tosis and severe ischemic changes of the myocardium. The coronary ostium was patent at the re implanta t ion site.

C o m m e n t

Single t runk anomalous origin of both coronary arteries from the pu lmonary artery is a rare cardiac malforma- tion, often d iagnosed only on autopsy. It may occur as an isolated anomaly or in association with other cardiac malformations, which may influence the t ime of onset of symptoms and the clinical course. The prognosis is bet ter in pat ients with associated cardiac malformat ions with increased pu lmonary artery oxygen saturat ion or pres- sure [3, 5, 8] than in those with isolated anomaly. In case of associated malformations, the diagnosis is difficult because the clinical and electrocardiographic pictures are domina ted by the associated lesions. Several cardiac anomal ies have been descr ibed in association with the anomalous origin of both coronary arteries from the pu lmonary ar tery [4-9]. In the association with isolated aortic coarctation, the coronary ar tery perfusion strictly depends on the degree of pu lmonary hyper tens ion which, in turn, is correlated with aortic coarctation in the neonatal age. In fact, in our patients, a sudden drop of left ventr icular afterload after coarctectomy must have re- sul ted in a lower pu lmonary artery pressure and reduced coronary ar tery perfusion.

In our institution, cardiac catheterization and angiog- raphy are not rout inely per formed in the preopera t ive evaluation of pat ients with isolated aortic coarctation, even in the presence of impai red left ventr icular function. In our experience, a mi ld - to -modera te left ventr icular hypokines ia is not unusual in s imple coarctation, and it is found to improve after relief of the aortic obstruction. The postoperat ive course of our patients, conversely, was character ized by clinical and ins t rumenta l evidence of

B

Fig, 1. Pulmonary angiography in anteroposterior (A) and lateral (B) views, showing the anomalous origin of the single trunk coronary artery (arrow).

deter iorat ion of the myocardia l function. After the first case, which remained und iagnosed until autopsy, the correct diagnosis was suspected in the second pat ient and cardiac catheterizat ion confirmed it.

In conclusion, the possibi l i ty of coronary ar tery anom- aly should be considered in pat ients with isolated aortic coarctation whose clinical condit ion seems aggravated, ra ther than improved, by an apparen t ly successful coar- tectomy. This course of events in the neonata l per iod or early months of life should suggest the anomalous origin of both coronary arteries. Early diagnosis is essential to avoid the occurrence of ventr icular f ibroelastosis and irreversible myocardia l failure, which preclude surgical repair.

708 CASE REPORT USU1 El AI, Ann Thorac Surg MVR AFTER CABG ~Av'ITH BILATERAL ITA 1995;60:708-9

References

1. Roberts WO. Anomalous origin of both coronary arteries from the pulmonary artery. Am J Cardiol 1962;10:595-9.

2. Heifetz S, Rabinowitz M, Mueller K, Virmani R. Total anom- alous origin of the coronary arteries from the pulmonary artery. Pediatr Cardiol 1986;7:11-6.

3. Goldblatt E, Adam APS, Ross IK, Savage JP, Morris LL. Single trunk anomalous origin of both coronary arteries from the pulmonary artery: diagnosis and surgical management. J Thorac Cardiovasc Surg 1984;87:59-65.

4. Colmes RA, Sideris CI. Anomalous origin of both coronary arteries from the pulmonary trunk. Myocardial infarction in otherwise normal heart. Am J Cardiol 1963;12:263-9.

5. Feldt RH, Ongley PA, Titus JL. Total coronary arterial circu- lation from pulmonary artery with survival to age seven. Report of a case. Mayo Clin Proc 1965;40:539-43.

6. Ogden JA. Origin of a single coronary artery from the pul- monary artery. Am Heart J 1969;78:251-3.

7. Monselise MB, Vlodaver Z, Neufeld HW. Single coronary artery. Origin from the pulmonary trunk in association with ventricular septal defect. Chest 1970;58:613-6.

8. D'Alessandro LC, Di Lorenzo M. Coronaria unica con origine dall'arteria polmonare sinistra in un uomo di 39 anni ancora vivente, affetto da "truncus arteriosus communis." G Ital Cardiol 1976;6:939-45.

9. Sennari E, Sato Y, Matsuoko Y, Yamamoto K, Okishirna T, Hayakawa K. A case report of anomalous origin of a single coronary artery from the pulmonary artery associated with multiple cardiac malformations. Jpn Circ J 1982;46:329-33.

Mitral Valve Replacement via Right Thoracotomy After Coronary Arterial Grafting Akihiko Usui, MD, Mitsuo Kawamura, MD, Michiaki Hibi, MD, Katsuhiko Yoshida, MD, Fumihiko Murakami, MD, and Jinichi lwase, MD

Division of Cardiac Surgery, Owari Prefectural Hospital, Ichinomiya, Japan

Mitral valve replacement was performed through a right thoracotomy using femorofemoral bypass under pro- found systemic hypothermia in a 62-year-old man who had undergone coronary artery bypass grafting using both internal thoracic arteries. The right thoracotomy approach minimizes the risk of injury to the arterial grafts, and deep hypothermia obviates the need to inter- rupt the grafts to administer cardioplegia. This technique provides excellent exposure of the mitral valve while minimizing the operative risk.

(Ann Thorac Surg 1995;60:708-9)

R eoperation through a median sternotomy is difficult when the internal thoracic arteries (ITAs) have been

used for coronary revascularization. The grafts may be

Accepted for publication March 7, 1995.

Address reprint requests to Dr Usui, 2-903 Umegaoka, Tenpaku-ku, Nagoya, Japan 468.

injured when the chest is entered or during the dissec- tion of adhesions. Further, administrat ion of cardiople- gia necessitates interruption of the arterial grafts. Cohn and colleagues [1] have reported the successful re- replacement of a mitral valve through a right thoracot- omy using femorofemoral bypass and profound systemic hypothermia without aortic cross-clamping or cardiople- gia. We applied this technique to a patient who suffered mitral valve regurgitation after coronary revasculariza- tion using both ITAs. A right thoracotomy avoids injury to the grafts, and deep hypothermia obviates the need for cardioplegia. This technique provides excellent exposure of the mitral valve while minimizing the operative risk.

A 62-year-old man suffered a large anteroseptal myocar- dial infarction in March 1993. Coronary angiography revealed severe stenosis of the left anterior descending coronary artery, the diagonal branch, and the circumflex artery. Left ventriculography demonstrated severe hypo- kinesis of the anteroseptal wall and mild mitral valve regurgitation. Coronary bypass grafting was performed in May 1993. Bilateral ITA grafts to the left anterior descending and circumflex arteries were created, and a vein graft was anastomosed to the diagonal branch. Postoperative angiography revealed that all grafts were patent, with no exacerbation of mitral regurgitation. The patient was asymptomatic at the time of discharge. How- ever, shortness of breath and palpitations developed in September 1993. Ultrasonic echocardiography revealed severe mitral regurgitation with anterior leaflet prolapse and moderate tricuspid valve regurgitation. The patient became progressively more symptomatic despite medi- cal management . Cardiac catheterization demonstrated severe pulmonary hypertension (pulmonary wedge pres- sure of 24 mm Hg, and pulmonary systolic arterial pressure of 78 mm Hg with a systolic arterial pressure of 88 mm Hg). The cardiac index was 1.68 L. rain 1. m 2 in April 1994.

The patient was placed in a 45-degree right lateral decubitus position. External defibrillation pads were placed on the left anterior and posterior chest wall before the incision was made, and a right anterolateral thora- cotomy was created through the fourth intercostal space. The superior vena cava was identified above the pericar- dium, cannulated directly with a metal- t ipped cannula, and encircled with a vascular tape. The inferior vena cava was taped below the pericardium. The right inguinal vessels were exposed in the groin, and a long inferior vena caval cannula and an arterial infusion cannula were inserted. The pericardium was identified and opened just above the right atrium, avoiding injury to the phrenic nerve. A vent tube was placed through the right upper pulmonary vein. The aorta was not cross-clamped, nor was cardioplegia administered. The patient was placed on cardiopulmonary bypass with a perfusion index of 2.5 L/min and cooled to 20°C, as measured nasopharyn- geally. Fibrillation was induced, and the right atr ium was opened longitudinally. The atriotomy was extended to the upper atrial septum, around the right atrial append-

© 1995 by The Society of Thoracic Surgeons 0003-4975/95/$9.50 0003-4975(95)00312-9