Volum* 122 Numbrr
and an oral anticoagulant). At 9 months’ follow-up, she remains totally asymptomatic, and two coronary angiograms, respectively performed at 2 and 7 months (Fig. 2), showed first partial and then complete angiographic healing of the LM dissection. In the literature, we found only four cases of spontaneous LM coronary dissection that were diagnosed antemortem.3-6 All were young women, and two were in the postpartum period. Treatment was always surgical.3‘6 Three of these patients survived, with a follow-up of 1.5 to 13 months. As far as we know, the patient reported here constitutes the first case of mid-term survival without surgical repair after spontaneous LM coronary artery dissection with complete angiographic resolution. The dissection cannot have been caused by the catheter because of the length of the left main stem and the location of the lesion in its distal part. A vascular collagen disease cannot be excluded, but the etiology of most spontaneous coronary artery dissections is still unclear. Their treatment is difficult, because of their rarity and the uncertain long-term prognosis. Emergency revascularization is the only solution for patients with ongoing or recurrent ischemia of a large myocardial area caused by an obstructive coronary dissection. However, partial or total resolution of coronary dissections has already been documented, and some authors have reported favorable results with medical therapy alone.2 Others used treatment with percutaneous transluminal coronary angioplasty, and others again recommended systematic bypass grafting.5 The most suitable strategy for patients with no evidence of persistent ischemia and no significant angiographic stenosis at the time of the angiogram remains an open question. Bypass grafting of the highly patent but fragile vesselsinvolved carries a high risk of rapid graft occlusion, and probably of further dissection at the distal anastomosis site. The therapeutic option must be considered with the greatest caution with respect to the LM coronary artery. In the patient reported here, an already severely depressed LV function provided an additional reason for medical therapy. However, the patient’s evolution suggests that in the absence of residual ischemia, spontaneous healing of the dissection and a favorable midterm outcome can be obtained without surgical repair.
REFERENCES
1.
2.
3.
4. 5. 6.
Brief Communications
6
Bulkley BH, Roberts WC. Dissectinganeurysm (hematoma) limited to coronary artery: a clinicopathologicstudy of six patients. Am J Med 1973;55:747-56. DeMaio SJ, Kinsella SH, Silverman ME. Clinical courseand long-term prognosis of spontaneous coronary artery dissection. Am J Cardiol 1989;64:471-4. Vicari R, Eybel C, Monson D. Survival following spontaneous coronary artery dissection:surgicalrepair by extrusion of intramural hematoma. AM HEART J 1986;111:593-4. Molloy P, Ablett M, AndersonK. Left main stem coronaryartery dissection.Br Heart J 1980;53:705-8. Thayer J, Healy R, Maggs P. Spontaneous coronary artery dissection.Ann Thorac Surg 1987;44:97-107. RazaviM. Unusual forms of coronaryartery disease.In: Best A, ed. Clevelandclinic cardiovascularconsultations.Philadelphia: F.A. Davis Co., 1975:42.
Coronary artery-left cardiac surgery
1759
ventricle fistulas after
Victor A. Jebara, MD, Antoine Sarkis, MD,a Christophe Acar, MD, Serban Mihaileanu, MD, Patrice Dervanian, MD, Jean Noel Fabiani, MD, Alain Deloche, MD, Charles Pauly Laubry,* and Alain Carpentier, MD, PhD. Paris, France
Acquired coronary cardiac fistulas (CCF) are rare and are usually secondary to cardiac trauma. They most frequently drain into the right ventricle (RV). Only 3 % drain into the left ventricle (LV).l, 2 Management of coronary artery-left ventricular (CALV) fistulas is not well established. We recently encountered three cases of CALV fistulas following cardiac surgical procedures. The purpose of this study is to analyze the pathogenesis of this disease as well as the diagnostic and management protocols. Case No. 1. A 57-year-old woman was referred for double valve replacement (aortic plus mitral). She was in New York Heart Association (NYHA) functional class III and her work-up showed severe stenosis of both the aortic and the mitral orifices. A preoperative coronary angiogram was normal. Surgery was conducted in the standard fashion and two pericardial bioprosthesis (Carpentier-Edwards, Baxter Healthcare Corp., Edwards Division, Santa Ana, Calif.) were seated. Twenty-four hours postoperatively a 316 diastolic murmur was heard at the level of the third intercostal space parasternally. An aortic perivalvular leak was suspected and echo-Doppler was not conclusive, since the conditions of the examination were difficult. The patient was taken back to the operating room. During surgery the aortic valve was well seated and no dehiscence or leak could be found; nevertheless, this prosthesis was replaced by the same type of valve. The next day the diastolic murmur persisted. An aortogram was obtained. It confirmed the absence of perivavular leak and established the diagnosis of a fistula between the second septal artery and the LV cavity, which did not exist on the preoperative film (Fig. 1). The postoperative course was uneventful and the patient was discharged on the twenty-first postoperative day. Eighteen months later, a new coronary angiogram showed absence of the fistula (Fig. 1). The patient was still asymptomatic and doing well. Case No. 2. A 78year-old woman presented with unstable angina. Coronary angiography showed 90 % stenosis of the left anterior descending (LAD) and the circumflex arteries. A triple bypass was performed: left internal mammary to the LAD, right internal mammary (free graft) to the first obtuse marginal branch, and the radial artery to
Fromthe Departmentof Cardiovascular “the Department of Cardiology, HBpital Reprint Surgery, 414133156
requests: Victor A Jebara, MD, HBpital Broussais, 96 rue Didot,
Surgery, HBpital Saint Joseph. Department 75014 Paris,
Broussais;
of Cardiovascular France.
and
1760
December 199, American Heart Journal
Brief Communications
a
Fig. 1. a, Postoperative coronary angiogram showing extravasation of contrast (arrow) from the second septal branch into the left ventricle. b, Eighteen months later the fistula was occluded. the second obtuse marginal branch. The postoperative course was uneventful. Routine control coronary angiography on day 15 showed three patent grafts and a CCF between the second septal branch and the LV. The patient was totally asymptomatic. Repeat control angiography 7 months later while the patient was still asymptomatic and well showed that the fistula was occluded. Case No. 3. A 17-year-old man presented with severe congenital aortic stenosis associated with LV hypertrophy predominant in the subvalvular septum. He underwent two percutaneous balloon valvuloplasties with early restenosis, and was referred to surgery. An aortic valve replacement was undertaken with a No. 19 St. Jude Medical prosthesis (St. Jude Medical Inc., St. Paul, Minn.) and an extensive septal myectomy was performed. Postoperatively, thirddegree atrioventricular (AV) block occurred requiring insertion of a permanent pacemaker. On day 15 control transthoracic color-coded Doppler echocardiography showed in addition to the “physiologic” regurgitation on the St Jude prosthesis an abnormal jet emerging from the septum at the zone of the myectomy and flowing into the
LV cavity (Fig. 2). Ten cubic centimeters of hand-agitat,ed saline were injected through a peripheral intravenous catheter during two-dimensional echocardiographic imaging. The contrast cloud clearly defines the right ventricle without any sign suggestive of intracardiac shunt, thus excluding the presence of a vent,ricular septal defect. The diagnosis of coronary artery-left ventricle fistula was therefore established based on the following criteria: (1) origin of the flow at a definite distance from the aort,ic root, thus eliminating paravalvular leak or ruptured sinus of Valsalva; (2) direction of the flow toward the LV cavity; (3) exclusive diastolic flow documented by M-mode colorcoded Doppler and high velocity (3.6 m/set) of this flow recorded by continuous wave Doppler. The patient was discharged 1 month postoperatively. Three months later, he was asymptomatic and doing well. Acquired CCF are usually posttraumatic.‘,” In 1987 we2 were able to find 30 cases of posttraumatic CCF reported in the literature. Most of the CCF in these cases (27 of 30) drained into the right cardiac chambers. Only one drained into the left ventricle.:’ Acquired CCF are rarely iatrogenic. Coronary artery-right ventricle fistulas have been reported after endomyocardial biopsies in cardiac transplant recipients.4 Cardiac surgery represents another cause of iatrogenie CCF illustrated in our three patients. The mechanism of the CCF in patients No. 1 and 2 is probably secondary to the LV venting technique. This technique consists of introducing a specially designed multiperforated 16F needle through the anterior surface of the RV, across the septum and into the LV (Fig. 3). Because of its simplicity and reliability this technique, introduced by Guilmet in 1983, was widely adopted in France and in other centers worldwide. While he is perforating the septum the physician can easily injure a septal branch and can therefore create a CCF. However, if care is taken to place the needle perpendicular to the septum and toward the apex, no major septal branches should be encountered. This approach has been used with satisfaction in over 6000 cases at our institution. The CCF in case No. 3 is a result of the extensive septal myectomy that was performed. Diagnosis of acquired CCF can be suspected on auscultation when a new murmur is heard along the left border of the sternum. It is usually continuous when the CCF drains into the RV, whereas it is purely diastolic when the CCF drains into the LV. Coronary angiography used to be the unique examination to diagnose CCF, as in patients No. 1 and 2. Recently, color-coded Doppler echocardiography has been proved to be an efficient diagnostic tool in CCF.“-7 This technique was used in case No. 3 and established the diagnosis of coronary artery-left ventricle fistula by showing a jet of mosaic signals emerging from the septum into the LV. Continuous wave (CW) Doppler showed a high velocity diastolic flow with the morphologic characteristics of an aortic insufhciency with two main differences. (1) When the CW probe is looking through the apex, a negative spectrum of velocities is recorded. (2) There exists a free protodiastolic interval corresponding to the isovolumic relaxation of the LV, which is absent in the case of aortic insufficiency. Furthermore, these echocardiographic characteristics, com-
Volume 122 Number 6
Brief Communications
176 1
Fig. 2. Color flow mapping two-dimensional Doppler examination. Parasternal long-axis view in diastole. The abnormal flow (A) can be seen as a mosaic jet emerging from the septum (S) into the left ventricle (LV). Normal LV filling (B). LA, Left atrium.
bined wibh venous contrast echocardiography, definitely exclude the diagnosis of a ventricular septal defect. Management of acquired CALV fistulas is not well established. While it is generally accepted that the majority of coronary artery-to-right ventricular (CARV) fistulas should be operated, the need for surgery in patients with CALV fistulas remains controversial. This is mainly because of the paucity of cases reported. In fact, contrary to CARV fistulas, which have a continuous high pressure-tolow pressure flow, fistulas that drain into the LV have an exclusive diastolic flow. Therefore acquired CCF are not a homogeneous group and do not encompass the same natural history. In our three cases we decided to postpone surgical closure of these fistulas. At 18 and 7 months, respectively, patients No. 1 and 2 remained completely asymptomatic and both fistulas closed spotaneously. On the other hand, in case No. 3 the fistula remained unchanged at 3 months with no ventricular or coronary artery dilatation. Recently, Marek et al7 reported the case of an asymptomatic 67-year-old patient in whom, 6 years after aortic valve replacement, color-coded Doppler echocardiography showed the presence of a coronary artery-left ventricular fistula. Neither the LV nor the left coronary arteries were found to be dilated, and the flow through the fistula was reported to be minimal. In conclusion, CALV fistulas do not seem to progress like most CARV fistulas, which constitute left-to-right shunts responsible for dilatation of the coronary artery and ultimately leading to congestive heart failure. CALV fistulas tend to close spontaneously or to decrease significantly with time and do not require surgical treatment.
Fig. 3. Schema showing the venting technique ble for the fistula in patients No. 1 and 2.
responsi-
REFERENCES
1. Lowe The tients Surg 2. Jebara ulaire vast
JE, Adams DH, Cummings RG, Wesly RLR, Philips HR. natural history and recommended management of pawith traumatic coronary artery fistulas. Ann Thorac 1983;36:295-305. VA, Corbi P, Dreyfus G, et al. Fistule coronaroventriciatrogenique postoperatoire. Ann Chir Thorac Cardio1989;43:643-5.
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December 1891 American Heart Journal
Brief Communications
3. Cheng TO, Adkins PC. Traumatic aneurysm of left anterior descending coronary artery with fistulous opening into the left ventricle and left ventricular aneurysm after stab wound af chest. Am J Cardiol 1973;31:384. 4. Lefevre T, Gosselin G, Carrier M, Leung TK, Dyrda I, Bonan
R. Fistules coronaroventriculaires droites chez le transplant6 cardiaque. Une complication de la biopsie endomyocardique. Arch Ma1 Coeur 1990;83:1713-8. 5I. Lau
KC,
Ng YM.
Evaluation
of coronary
color-coded Doppler echocardiography. 689-91. 6. Sanders
SP, Parness
IA, Colan
arterial
fistula
by
Am J Cardioll989;64:
SD. Recognition
of abnormal
connections of coronary arteries with the use of Doppler color flow mapping. J Am Co11 Cardiol 1989;13:922-6. 7. Marek
A, Rey
JL,
Tribouilloy
C, et al. Fistule
coronaroven-
triculaire gauche iatrogene postoperatoire. Mise en evidence par le Doppler bidimensionnel B codage couleur. Arch Ma1 Coeur
1991;84:419-23.
Variant angina in single coronary artery Kwang
Kon Koh,
MD. Kyunggi-do,
Korea
By definition isolated single coronary artery arises from the aortic trunk by a single coronary ostium and supplies the entire heart regardless of its distribution. This is a rare congenital anomaly and occurs as an isolated finding in approximately 0.024 D/Cof the popu1ation.l In about 40 !‘() of the reported cases it is associated with other congenital anomalies.“* 3 Diagnosis was first made in a living patient by angiography by Halperin et al4 in 1967. It was said that typical angina did not occur with single coronary artery in the absence of a coexisting coronary artery or aortic stenosis. The clinical significance of the isolated single coronary artery anomaly depends on the adequacy of its orifice and on whether there is any acute angulation or compromise of the major branches as they traverse the base of the heart passing between or around the great vessels. Angina pectoris has been reported to occur due to hypoplasia, acute angulation of an artery, or atherosclerosis of the proximal vessel.” However, angina pectoris by coronary spasm has not been reported up to now in a single coronary artery.‘i I recent witnessed angina1 attacks by coronary spasm in a 26-year-old woman with an isolated single coronary artery. A 26-year-old woman visited our hospital on February 2, 1990, for evaluation of frequent chest pain, first noted 3 years previously. The patient had severe substernal discomfort during face washing or brushing her teeth in the early morning, with a duration of several minutes with a frequency of three to six occurrences. Angina was relieved by rest or by sublingual nitroglycerin tablets. There was no From the Division of Cardiology, Department University and Inha General Hospital.
of Internal
Medicine,
Reprint requests: Kwang Kon Koh, MD, Division of Cardiology, ment of Internal Medicine, Inha University and Inha General 3309-327 Tae Pyeong-dong, Seong Nam-shi, Kyunggi-do, Korea 4/4/33143
Inha
DepartHospital, 461-192.
Fig. 1. Type LB* single coronary artery (Smith-Lipton classification). Right anterior oblique (A) and left anterior (B). Left coronary angiography oblique cranial projection revealed about 40”rm stenosis of the mid left anterior descending artery and the proximal right coronary artery just branching from the left anterior descending artery.
chest pain during exercise. The patient had no previous history of hypertension, diabetes mellitus, or cigarette smoking. Serum lipoprotein electrophoresis showed a normal pattern. An electrocardiogram at rest was normal. Echocardiography showed no associated congenital anomaly. The patient completed 10 metabolic equivalents (METS) of an exercise test (Bruce protocol) and had 1 mm of horizontal ST segment depression in lead II, III, aVF, and Vs on a postexercise electrocardiogram with chest pain. Coronary angiography demonstrated that the right coronary artery originated from the mid left anterior descending artery and ran in front of the pulmonary artery (Lipt.on and Smith classification type LII*).l, 3 Left coronary angiography revealed about 40”; stenosis of the mid left anterior descending artery and the proximal right coronary artery just branching from the left anterior descending artery, but the patient did not complain of chest pain and electrocardiography showed no important ST-T segment changes (Fig. 1). A vasoactive substance was not infused to aggravate the spasm. The reason was that if acetylcholine