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A CASE OF AORTICO-RIGHT VENTRICULAR FISTULA FOLLOWING A CLOSED CHEST INJURY
A CASE OF AORTICO-RIGHT VENTRICULAR FISTULA FOLLOWING A CLOSED CHEST I N J U R Y Frank Gerbode, M.D.*
R. E. Hackett, F.R.C.S., Zelman Freeman,
G. T. Benness, M.B., B.S.,** and J. Bruce Johnston, MS., F.R.C.S., Sydney,
M.R.C.P.,
F.R.A.C.S.,
Australia
A
N aortico-cardiac fistula is a rare sequel of chest injury. So far as the au thors are aware, all such cases have occurred following either a penetrat ing injury of the chest or as a complication of surgery in the neighborhood of the aortic valve ring, as, for instance, in the repair of a high ventricular septal defect or repair of the defect in Pallot's tetralogy. The present case report describes a patient who developed an aortico-right ventricular fistula following a closed chest injury. This was diagnosed preoperatively and successfully repaired with the use of extracorporeal circulation. PASE REPORT On Nov. 3, 1962, K. M., 33 years of age, a saxophone player in a T.V. orchestra, was driving home early in the morning when he momentarily fell asleep. H e lost control of his car which left the road and struck a telegraph pole, the impact of which threw him forcibly against the steering wheel. On admission to Eastern Suburbs Hospital he was conscious but did not remember t h e accident and complained of difficulty in breathing to such an extent t h a t he felt he was going to die. He also complained of pain in the lower costal region on both sides and a painful left knee. On examination he was pale but was not s\veating and the pulse was slow. The blood pressure was 90/60 mm. Hg. A p a r t from a slight scratch over his left knee there were no other apparent injuries. Because of t h e possibility of injury to the heart an electrocardiogram was taken. This showed a nodal rhythm with a pattern suggesting a recent posterior infarction. There was marked S-T elevation in the chest leads compatible w i t h epicardial damage (Fig. 1). A chest x-ray film made on admission showed no evidence of injury to the bony cage of the thorax but the heart was enlarged. An additional electrocardiogram t a k e n 13 hours after injury showed A-V dissociation with rapid restoration in t h e level of the S-T segments toward normal. The next day his general condition had improved. Examination revealed a small bruise over the left pectoral muscle about the size of a ten cent piece. The blood pressure was steady at 140/70 mm. H g and t h e heart sounds were clear. Another electrocardiogram (48 hours after the accident) showed a rapid evolution of t h e T-wave changes resembling those seen in myocardial infarction. These changes, however, usually take several days to deFrom the Department of Cardio-Thoracic Surgery, University of New South Wales, Sydney, Australia. Received for publication April 22, 1964. •Guest Professor of Surgery, University of New South Wales, Sydney, Australia. ••Director, Department of Radiology, St. Vincent's Hospital, Sydney, Australia. 1016
Vol. 48, No. 6 December, 1964
AORTICO-RIGHT VENTRICULAR
1017
FISTULA
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Fig. 1.—Electrocardiogram that shows nodal rhythm and a pattern consistent with recent posterior infarction and epicardial damage.
AVR
AVL
AVF
pig. 2.—Electrocardiogram that shows nodal rhythm with terminal vector anterior, 48 hours after injury.
1018
GERBODE E T AL.
J. Thoracic and Cardiovas. Surg.
Fig. 3.—X-ray fllm taken the day after admission. Note haziness at 1. base of left lung. Part of this may be due to rupture of the pericardium causing- dislocation of the heart into the left pleural cavity and part to acute right ventricular dilatation.
velop (Fig. 2). A chest roentgenogram revealed a slight increase in the heart size. The serum glutamic oxalocetic transaminase was 224 King units and the serum lactic dehydrogenase was 1,850 units. He remained well until the fourth day following the accident when his respirations became labored. On examination his pulse was collapsing in quality and of the para doxical variety. The blood pressure was 160/70 mm. H g and t h e venous pressure was raised up to the ear lobes. A Grade 2 continuous murmur could be heard in t h e second and third left intercostal spaces (Fig. 3). A chest roentgenogram revealed gross enlarge ment of the heart to the left with the apex touching the chest wall (Fig. 4 ) . H e was then digitalized with digoxin and put on a maintenance dose of 0.25 mg. twice daily. I t was now apparent t h a t his injury had produced an abnormal shunt so he was trans ferred for further t r e a t m e n t to Prince H e n r y Hospital. A pericardial aspiration was performed as the chest x-ray study and electrocardio gram suggested an effusion. No fluid was found. Over the next few days his general con dition gradually improved. The continuous murmur persisted, although it waxed and waned in intensity. Repeated chest roentgenograms showed a gradual diminution in heart size. On November 12, cardiac catheterization showed a rise in arterial oxygen saturation in the right ventricle. Cineangiography carried out at St. Vincent's Hospital showed a dominant left coronary artery (Fig. 5, a) with retrograde tilling of the right coronary artery as far as the marginal branch (Fig. 5, 6 ) . Anastomotic vessels passed from the origin of the left to the origin of the right coronary artery but the origin of the right coronary artery could not be demonstated. The intervenlricular septum and the aortic valve were
Vol. 48, No. 6 December, 1964
AORTICO-RIGHT VENTRICULAR F I S T U L A
1019
I I I I I M I I I I I I I I I I I I I I I I I I I II I I K).2 Se
L.S.E. (H.F.)
-
'
'
M.A.
Pig. 4.—Phonocardiogram shows continuous murmur at left sternal edge (L. S. E.). First and second heart sounds are best seen at mitral area (M. A.).
Pig. 5.—a, Angiogram with selective injection into the left coronary artery in the L. A. O. projection shows a normal dominant left coronary artery. &, Angiogram shows retrograde filling of the right coronary artery and its marginal branch (8:00 o'clock in the picture) from an injection into the left coronary artery. In addition, intercoronary anastomoses from the left coronary artery to the origin of the right coronary artery are seen (10:00 o'clock in the picture), c, Aortogram, L. A. O. projection, shows a normal aortic valve, the origin of the left coronary artery, and a fistula from the right sinus of Valsalva into the right ventricle.
intact. The catheter passed into a fistula arising from the right coronary sinus and t h e aortic injection showed a connection from the right coronary sinus to a false aneurysm and then to the right ventricle (Fig. 5, c). The patient was discharged from the hospital on November 21, when all symptoms had disappeared. The continuous murmur persisted, although the heart size was normal. Digitalis therapy was no longer required. Serial electrocardiograms are shown in Fig. 6. A vectorcardiogram confirmed both the right bundle branch block and the posterior (inferior) infarction (Fig. 7 ) . The p a t i e n t was re-admitted later for operation which was performed on March 4, 1963.
2
3 AVR
AVL
AVF
VI
V2
V3
V4
V5
V6
Fig. 6.—Serial electrocardiograms from Nov. 6, 1962 to May 6, 1963. Note change from nodal rhythm to sinus rhythm and also the develop ment of right bundle branch block pattern which has persisted and is still present 9 months after operation. Note also T inversion across chest which is not a part of the usual right bundle branch block pattern but probably reflects epicardial injury.
$%ÀY
ό NOV '62 8 NOV '62
1
Fig·. 7.—Frank vectorcardiogram made in March, 1963. Note rightward and anterior terminal element in frontal and horizontal planes. Note upward displacement of early vectors in sagittal plane indicative of posterior infarction.
OPERATION
On opening the thorax through a sternal splitting incision, it was found that the blood pressure had a tendency to fall with manipulation of the heart. The heart seemed to be much more toward the left side than would have been expected. This was found to be due to the presence of a large hole in the peri cardium, just posterior to the phrenic nerve. This allowed the entire apex of the heart up to the pulmonary veins to prolapse into the left pleural space and un doubtedly accounted for the peculiar behavior of the blood pressure. It was noted that when the heart was held up in a better position the blood pressure improved. On examination of the anterior surface of the heart near the upper portion of the right ventricle and right coronary artery, a scar was found which ob literated all the ordinary markings. A thrill was palpable on the right side of the front of the right ventricle and there was also a thrill in the right atrium, adjacent to this area. A blood sample was taken from the right atrium but it seemed to have the same oxygen content as the superior vena caval blood. Bypass was established in the usual manner and a decompressing drain was inserted into the left atrium from the right side. The aorta was then dis sected out and the patient was cooled to 30° C. The right ventricle was opened transversely between two small coronary vessels. A tract was found on the septal wall of the right ventricle just above the insertion of the tricuspid valve. On opening this tract a white, thin, scarred area, measuring 2.5 cm. in diameter, was found and in the center of this there was a 4 mm. opening into the base of the aorta. On release of the aortic clamp there was a gush of blood through the hole into the right ventricle. The opening was closed by approximating the adjacent scarred muscle with a double continuous row of 3-0 silk, with a superimposed Dacron patch (Fig. 8). This effectively secured the weak area at the base of the aorta in the region of
1022
GERBODE ET AL.
J. Thoracic and
Cardiovas. Surg.
Fig. 8.—Lesion, at operation and details of repair.
the right coronary sinus and on release of the aortic clamp there was no fur ther leak from the aorta. On examination of the tricuspid valve, a tear, 2 cm. in length, was found in the anterior leaflet. This probably also occurred during the initial injury. The rent was closed with a continuous arterial suture. While repairing the tricuspid valve it was noted that there was a patent foramen ovale and this was closed with two interrupted sutures. On rewarming, spontaneous defibrillation occurred and a good strong heart beat resulted. Air was aspirated from the left ventricle, and, after the right ventriculotomy was closed with a double row of sutures, decannulation was done in the usual way. When the heart fell into the left pleural space through the big rent in the pericardium, a fall of 15 mm. in effective arterial blood pressure occurred. The lower two thirds of the rent was therefore repaired with continuous and inter rupted silk. The heart was now held in a normal position. The anterior peri cardium was not closed. Two drains were inserted, one in each pleural space. There was no thrill or bruit discernible at the end of the procedure. The thorax was closed in the usual fashion. Postoperatively the patient made an uninterrupted recovery and was dis charged on April 29, completely free from symptoms and with no abnormal
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Fig. 9.—Roentgenogram of chest (March 28, 1963) made following surgical treatment.
physical signs. The roentgenogram of the chest was normal (Fig. 9). On fol low-up on November 11, 1963, he was completely normal, felt well, and was able to run without distress. DISCUSSION
Although the condition is rare, the possibility of a ruptured sinus of Valsalva should always be borne in mind in any patient who is relatively young, previously in good health, and who suddenly develops an episode of chest pain, dyspnea, a continuous murmur, and a collapsing pulse. Examination will usu ally show a hyperactive heart with enlargement to the left and a right ven tricular heave. A thrill is often present and the murmur is usually continuous but will vary in position according to the location of the fistula. Usually it is maximal in the second and third left intercostal spaces. In most instances the condition is predisposed to by a congenital lack in continuity between the media of the aorta and the annulus fibrosus of the aortic ring. The aneurysm may ai-ise from any of the sinuses at the root of the aorta, but usually arises from the right coronary and adjacent two thirds of the noncoronary sinus.
1024
GERBODE ET AL.
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Aneurysms may also develop secondarily in such conditions as syphilitic aortitis, mycotic aortitis, bacterial endocarditis, atherosclerosis or, occasionally, trauma. An aneurysm so formed is symptomless until it ruptures, and the rupture may occur into any one of the numerous adjacent intraperieardial structures to which the aortic ring is closely related. Rupture into the right ventricle will result in right heart failure and functional tricuspid incompetence. If rupture occurs into the pulmonary artery the downhill course is much more rapid. Some times rupture takes place into the superior vena cava or right atrium and pro duces acute venous engorgement, which, although severe, has a relatively bet ter outlook. Fistulas have also occurred into the left atrium, 1 the left ventricle, 2 and into the pericardial sac.3 It is important to make the diagnosis as the natural prognosis is poor and the lesion can now readily be repaired. There have been numerous case reports and reviews in the literature of such operations, 4,5 but the number of opera tions on patients who develop fistulas after trauma is distinctly rare. King and Shumacker 6 reported the case history of a boy, 15 years of age, who had sustained a gunshot wound. This penetrated the right ventricle and entered the aorta and was then swept along into the left common iliac artery. This fistula was successfully repaired with the use of bypass. A similar case was observed by Gerbode.7 Smyth and his associates8 reported the case of a 22-year-old Negro male who developed a fistula following a stab wound which had entered the right ventricle close to the pulmonary valve ring. After closure of the right ventricle at an emergency thoraeotomy, a coarse thrill was noted in the region of the right ventricular wound, together with a low diastolic pres sure. Thirty-two days later a communication between the right sinus of Valsalva and the right ventricle was repaired with the use of bypass. Nowlan and co-authors9 reported a case of right ventricular aortic fistula 48 hours after a stab wound and this also was successfully repaired 10 days later. Swanepoel and colleagues10 described another case of a fistula between the aorta and the right atrium which occurred after a stab wound. During the operative repair of the ventricular septal defect in Fallot's tetralogy, the sutures are placed very near the aortic sinuses which have on their lateral walls only a thin layer of muscle and it is possible to produce a fistula either at the time of the operation or as a result of some pressure or ischémie necrosis in the region of the sutures following operation. Agustsson and co-workers11 described the cases of 2 patients, 1 of whom developed a fistula 4 months after the direct suture of a ventricular septal defect. Therefore, after surgery of this type, an unexplained continuous murmur or persistent heart failure should suggest the possibility of this complication. The type of repair required depends on the cause. In general, the con genital type, in which the media is either absent or deficient for a considerable distance away from the fistula, will usually require a patch to close the open ing and support the inadequate aortic wall. In the acquired type, the media may be preserved intact except at the area of the fistula, and the surrounding wall should, therefore, be able to accept and hold sutures.
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December, 1964
The timing of the operation is also important. Many of these patients im prove after a few days and may even be symptomless for weeks or months but this does not alter the prognosis which is ultimately poor. However, it may be wiser to defer operation to allow the patient to improve so that an elective pro cedure can be undertaken at some future date with relatively little risk. SUMMARY
A case is reported of a patient who developed a traumatic myocardial in farction following a closed chest injury, in whom a fistula developed from the right coronary sinus of Valsalva to the right ventricle. This was diagnosed preoperatively and successfully repaired with the use of cardiac bypass. The pa tient made a complete and perfect recovery. As far as the authors are aware, this is the first case of this type so far reported in the literature. REFERENCES 1. Heiner, D. C , Hara, M., and White, H . J . : Cardio-aortic Fistulae and Aneurysms of the Sinus of Valsalva in Infancy: A Report of an Aortic Left Atrial Communica tion Indistinguishable From a Ruptured Aneurysm of the Aortic Sinus, Pediatrics 27: 415, 1961. 2. Warthen, R. O. : Congenital Aneurysm of t h e Right Anterior Sinus of Valsalva ( I n terventricular Aneurysm) With Spontaneous Rupture Into the Left Ventricle, Am. Heart J . 37: 975, 1949. 3. Weiss, L.: Rupture of an Aneurysm of t h e Right Aortic Sinus (of Valsalva), Brit. H e a r t J . 19: 138, 1957. 4. Gerbode, F . , Osborn, J . J., Johnston, J . B.. and Kerth, W. J . : Ruptured Aneurysms of t h e Aortic Sinuses of Valsalva, Ani. J . Surg. 102: 268, 1961. 5. Sawyers, J . L., Adams, J . E., a n d Scott, H . W.: Surgical Treatment for Aneurysms of t h e Aortic Sinuses W i t h Aortico-atrial Fistula: Experimental a n d Clinical Study, Surgery 4 1 : 26, 1957. 6. King, H., a n d Shumacker, H. B., J r . : Surgical Repair of a Traumatic Aortic Right V e n t r i c u l a r F i s t u l a , J . THORACIC SURG. 3 5 : 734, 1958.
7. Gerbode, F . : Discussion of Sawyers et al.s 8. Smyth, N . P . D., Adkins, P . C., Kelser, G. A., and Calatayud, J . : Traumatic Aortic Right Ventricular Fistula, Surg., Gvnec. & Obst. 109: 566, 1959. 9. Nowlan, J . A., Steiger, Z., Bicoff, J . P., Fell, E. A., and Tobin, J . R.: Traumatic Aortic Right Ventricular Fistula, J . A. M. A. 181: 159, 1962. 10. Swanepoel, A., Schrire, V., Nellen, M., Vogepoel, L., and Barnard, C : Traumatic Aortico Right Atrial Fistula: Report of a Case Corrected by Operation, Am. H e a r t J . 6 1 : 120, 1961. 11. Agustsson, M. H., Weinberg, M., Gasul, B. M., Fell, E . H., Arcilla, R. A., Bicoff, J . P., Steiger, Z., and I w a T.: Fistulas Following Corrective Operations for Ven t r i c u l a r Septal Defects
43: 166, 1962.
and T e t r a l o g y of F a l l o t J . THORACIC & CARDIOVAS. SURG.
R. E. Hackett, F.R.C.S., Zelman Freeman,
G. T. Benness, M.B., B.S.,** and J. Bruce Johnston, MS., F.R.C.S., Sydney,
M.R.C.P.,
F.R.A.C.S.,
Australia
A
N aortico-cardiac fistula is a rare sequel of chest injury. So far as the au thors are aware, all such cases have occurred following either a penetrat ing injury of the chest or as a complication of surgery in the neighborhood of the aortic valve ring, as, for instance, in the repair of a high ventricular septal defect or repair of the defect in Pallot's tetralogy. The present case report describes a patient who developed an aortico-right ventricular fistula following a closed chest injury. This was diagnosed preoperatively and successfully repaired with the use of extracorporeal circulation. PASE REPORT On Nov. 3, 1962, K. M., 33 years of age, a saxophone player in a T.V. orchestra, was driving home early in the morning when he momentarily fell asleep. H e lost control of his car which left the road and struck a telegraph pole, the impact of which threw him forcibly against the steering wheel. On admission to Eastern Suburbs Hospital he was conscious but did not remember t h e accident and complained of difficulty in breathing to such an extent t h a t he felt he was going to die. He also complained of pain in the lower costal region on both sides and a painful left knee. On examination he was pale but was not s\veating and the pulse was slow. The blood pressure was 90/60 mm. Hg. A p a r t from a slight scratch over his left knee there were no other apparent injuries. Because of t h e possibility of injury to the heart an electrocardiogram was taken. This showed a nodal rhythm with a pattern suggesting a recent posterior infarction. There was marked S-T elevation in the chest leads compatible w i t h epicardial damage (Fig. 1). A chest x-ray film made on admission showed no evidence of injury to the bony cage of the thorax but the heart was enlarged. An additional electrocardiogram t a k e n 13 hours after injury showed A-V dissociation with rapid restoration in t h e level of the S-T segments toward normal. The next day his general condition had improved. Examination revealed a small bruise over the left pectoral muscle about the size of a ten cent piece. The blood pressure was steady at 140/70 mm. H g and t h e heart sounds were clear. Another electrocardiogram (48 hours after the accident) showed a rapid evolution of t h e T-wave changes resembling those seen in myocardial infarction. These changes, however, usually take several days to deFrom the Department of Cardio-Thoracic Surgery, University of New South Wales, Sydney, Australia. Received for publication April 22, 1964. •Guest Professor of Surgery, University of New South Wales, Sydney, Australia. ••Director, Department of Radiology, St. Vincent's Hospital, Sydney, Australia. 1016
Vol. 48, No. 6 December, 1964
AORTICO-RIGHT VENTRICULAR
1017
FISTULA
SfüIffiltiM i i l l l i i l l i l AVR i^mmiiiiiiiliiiiii
iffflfffiffilSW
filiÜHP
II
111 fô Hffli >» H i1 m1 31 lui1 i |
pi
ÎÏKU'tUî
1! h
AV L
LU
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AV F
JBB!
4§!B V5
USÉ tt:îj|la
Fig. 1.—Electrocardiogram that shows nodal rhythm and a pattern consistent with recent posterior infarction and epicardial damage.
AVR
AVL
AVF
pig. 2.—Electrocardiogram that shows nodal rhythm with terminal vector anterior, 48 hours after injury.
1018
GERBODE E T AL.
J. Thoracic and Cardiovas. Surg.
Fig. 3.—X-ray fllm taken the day after admission. Note haziness at 1. base of left lung. Part of this may be due to rupture of the pericardium causing- dislocation of the heart into the left pleural cavity and part to acute right ventricular dilatation.
velop (Fig. 2). A chest roentgenogram revealed a slight increase in the heart size. The serum glutamic oxalocetic transaminase was 224 King units and the serum lactic dehydrogenase was 1,850 units. He remained well until the fourth day following the accident when his respirations became labored. On examination his pulse was collapsing in quality and of the para doxical variety. The blood pressure was 160/70 mm. H g and t h e venous pressure was raised up to the ear lobes. A Grade 2 continuous murmur could be heard in t h e second and third left intercostal spaces (Fig. 3). A chest roentgenogram revealed gross enlarge ment of the heart to the left with the apex touching the chest wall (Fig. 4 ) . H e was then digitalized with digoxin and put on a maintenance dose of 0.25 mg. twice daily. I t was now apparent t h a t his injury had produced an abnormal shunt so he was trans ferred for further t r e a t m e n t to Prince H e n r y Hospital. A pericardial aspiration was performed as the chest x-ray study and electrocardio gram suggested an effusion. No fluid was found. Over the next few days his general con dition gradually improved. The continuous murmur persisted, although it waxed and waned in intensity. Repeated chest roentgenograms showed a gradual diminution in heart size. On November 12, cardiac catheterization showed a rise in arterial oxygen saturation in the right ventricle. Cineangiography carried out at St. Vincent's Hospital showed a dominant left coronary artery (Fig. 5, a) with retrograde tilling of the right coronary artery as far as the marginal branch (Fig. 5, 6 ) . Anastomotic vessels passed from the origin of the left to the origin of the right coronary artery but the origin of the right coronary artery could not be demonstated. The intervenlricular septum and the aortic valve were
Vol. 48, No. 6 December, 1964
AORTICO-RIGHT VENTRICULAR F I S T U L A
1019
I I I I I M I I I I I I I I I I I I I I I I I I I II I I K).2 Se
L.S.E. (H.F.)
-
'
'
M.A.
Pig. 4.—Phonocardiogram shows continuous murmur at left sternal edge (L. S. E.). First and second heart sounds are best seen at mitral area (M. A.).
Pig. 5.—a, Angiogram with selective injection into the left coronary artery in the L. A. O. projection shows a normal dominant left coronary artery. &, Angiogram shows retrograde filling of the right coronary artery and its marginal branch (8:00 o'clock in the picture) from an injection into the left coronary artery. In addition, intercoronary anastomoses from the left coronary artery to the origin of the right coronary artery are seen (10:00 o'clock in the picture), c, Aortogram, L. A. O. projection, shows a normal aortic valve, the origin of the left coronary artery, and a fistula from the right sinus of Valsalva into the right ventricle.
intact. The catheter passed into a fistula arising from the right coronary sinus and t h e aortic injection showed a connection from the right coronary sinus to a false aneurysm and then to the right ventricle (Fig. 5, c). The patient was discharged from the hospital on November 21, when all symptoms had disappeared. The continuous murmur persisted, although the heart size was normal. Digitalis therapy was no longer required. Serial electrocardiograms are shown in Fig. 6. A vectorcardiogram confirmed both the right bundle branch block and the posterior (inferior) infarction (Fig. 7 ) . The p a t i e n t was re-admitted later for operation which was performed on March 4, 1963.
2
3 AVR
AVL
AVF
VI
V2
V3
V4
V5
V6
Fig. 6.—Serial electrocardiograms from Nov. 6, 1962 to May 6, 1963. Note change from nodal rhythm to sinus rhythm and also the develop ment of right bundle branch block pattern which has persisted and is still present 9 months after operation. Note also T inversion across chest which is not a part of the usual right bundle branch block pattern but probably reflects epicardial injury.
$%ÀY
ό NOV '62 8 NOV '62
1
Fig·. 7.—Frank vectorcardiogram made in March, 1963. Note rightward and anterior terminal element in frontal and horizontal planes. Note upward displacement of early vectors in sagittal plane indicative of posterior infarction.
OPERATION
On opening the thorax through a sternal splitting incision, it was found that the blood pressure had a tendency to fall with manipulation of the heart. The heart seemed to be much more toward the left side than would have been expected. This was found to be due to the presence of a large hole in the peri cardium, just posterior to the phrenic nerve. This allowed the entire apex of the heart up to the pulmonary veins to prolapse into the left pleural space and un doubtedly accounted for the peculiar behavior of the blood pressure. It was noted that when the heart was held up in a better position the blood pressure improved. On examination of the anterior surface of the heart near the upper portion of the right ventricle and right coronary artery, a scar was found which ob literated all the ordinary markings. A thrill was palpable on the right side of the front of the right ventricle and there was also a thrill in the right atrium, adjacent to this area. A blood sample was taken from the right atrium but it seemed to have the same oxygen content as the superior vena caval blood. Bypass was established in the usual manner and a decompressing drain was inserted into the left atrium from the right side. The aorta was then dis sected out and the patient was cooled to 30° C. The right ventricle was opened transversely between two small coronary vessels. A tract was found on the septal wall of the right ventricle just above the insertion of the tricuspid valve. On opening this tract a white, thin, scarred area, measuring 2.5 cm. in diameter, was found and in the center of this there was a 4 mm. opening into the base of the aorta. On release of the aortic clamp there was a gush of blood through the hole into the right ventricle. The opening was closed by approximating the adjacent scarred muscle with a double continuous row of 3-0 silk, with a superimposed Dacron patch (Fig. 8). This effectively secured the weak area at the base of the aorta in the region of
1022
GERBODE ET AL.
J. Thoracic and
Cardiovas. Surg.
Fig. 8.—Lesion, at operation and details of repair.
the right coronary sinus and on release of the aortic clamp there was no fur ther leak from the aorta. On examination of the tricuspid valve, a tear, 2 cm. in length, was found in the anterior leaflet. This probably also occurred during the initial injury. The rent was closed with a continuous arterial suture. While repairing the tricuspid valve it was noted that there was a patent foramen ovale and this was closed with two interrupted sutures. On rewarming, spontaneous defibrillation occurred and a good strong heart beat resulted. Air was aspirated from the left ventricle, and, after the right ventriculotomy was closed with a double row of sutures, decannulation was done in the usual way. When the heart fell into the left pleural space through the big rent in the pericardium, a fall of 15 mm. in effective arterial blood pressure occurred. The lower two thirds of the rent was therefore repaired with continuous and inter rupted silk. The heart was now held in a normal position. The anterior peri cardium was not closed. Two drains were inserted, one in each pleural space. There was no thrill or bruit discernible at the end of the procedure. The thorax was closed in the usual fashion. Postoperatively the patient made an uninterrupted recovery and was dis charged on April 29, completely free from symptoms and with no abnormal
Vol. 48. No. 6 December, 1964
AORTICO-RIGHT VENTRICULAR F I S T U L A
1023
Fig. 9.—Roentgenogram of chest (March 28, 1963) made following surgical treatment.
physical signs. The roentgenogram of the chest was normal (Fig. 9). On fol low-up on November 11, 1963, he was completely normal, felt well, and was able to run without distress. DISCUSSION
Although the condition is rare, the possibility of a ruptured sinus of Valsalva should always be borne in mind in any patient who is relatively young, previously in good health, and who suddenly develops an episode of chest pain, dyspnea, a continuous murmur, and a collapsing pulse. Examination will usu ally show a hyperactive heart with enlargement to the left and a right ven tricular heave. A thrill is often present and the murmur is usually continuous but will vary in position according to the location of the fistula. Usually it is maximal in the second and third left intercostal spaces. In most instances the condition is predisposed to by a congenital lack in continuity between the media of the aorta and the annulus fibrosus of the aortic ring. The aneurysm may ai-ise from any of the sinuses at the root of the aorta, but usually arises from the right coronary and adjacent two thirds of the noncoronary sinus.
1024
GERBODE ET AL.
J. Thoracic and Cardiovas. Surg.
Aneurysms may also develop secondarily in such conditions as syphilitic aortitis, mycotic aortitis, bacterial endocarditis, atherosclerosis or, occasionally, trauma. An aneurysm so formed is symptomless until it ruptures, and the rupture may occur into any one of the numerous adjacent intraperieardial structures to which the aortic ring is closely related. Rupture into the right ventricle will result in right heart failure and functional tricuspid incompetence. If rupture occurs into the pulmonary artery the downhill course is much more rapid. Some times rupture takes place into the superior vena cava or right atrium and pro duces acute venous engorgement, which, although severe, has a relatively bet ter outlook. Fistulas have also occurred into the left atrium, 1 the left ventricle, 2 and into the pericardial sac.3 It is important to make the diagnosis as the natural prognosis is poor and the lesion can now readily be repaired. There have been numerous case reports and reviews in the literature of such operations, 4,5 but the number of opera tions on patients who develop fistulas after trauma is distinctly rare. King and Shumacker 6 reported the case history of a boy, 15 years of age, who had sustained a gunshot wound. This penetrated the right ventricle and entered the aorta and was then swept along into the left common iliac artery. This fistula was successfully repaired with the use of bypass. A similar case was observed by Gerbode.7 Smyth and his associates8 reported the case of a 22-year-old Negro male who developed a fistula following a stab wound which had entered the right ventricle close to the pulmonary valve ring. After closure of the right ventricle at an emergency thoraeotomy, a coarse thrill was noted in the region of the right ventricular wound, together with a low diastolic pres sure. Thirty-two days later a communication between the right sinus of Valsalva and the right ventricle was repaired with the use of bypass. Nowlan and co-authors9 reported a case of right ventricular aortic fistula 48 hours after a stab wound and this also was successfully repaired 10 days later. Swanepoel and colleagues10 described another case of a fistula between the aorta and the right atrium which occurred after a stab wound. During the operative repair of the ventricular septal defect in Fallot's tetralogy, the sutures are placed very near the aortic sinuses which have on their lateral walls only a thin layer of muscle and it is possible to produce a fistula either at the time of the operation or as a result of some pressure or ischémie necrosis in the region of the sutures following operation. Agustsson and co-workers11 described the cases of 2 patients, 1 of whom developed a fistula 4 months after the direct suture of a ventricular septal defect. Therefore, after surgery of this type, an unexplained continuous murmur or persistent heart failure should suggest the possibility of this complication. The type of repair required depends on the cause. In general, the con genital type, in which the media is either absent or deficient for a considerable distance away from the fistula, will usually require a patch to close the open ing and support the inadequate aortic wall. In the acquired type, the media may be preserved intact except at the area of the fistula, and the surrounding wall should, therefore, be able to accept and hold sutures.
Vol. 48. No. 6
AORTICO-RIGHT VENTRICULAR F I S T U L A
1025
December, 1964
The timing of the operation is also important. Many of these patients im prove after a few days and may even be symptomless for weeks or months but this does not alter the prognosis which is ultimately poor. However, it may be wiser to defer operation to allow the patient to improve so that an elective pro cedure can be undertaken at some future date with relatively little risk. SUMMARY
A case is reported of a patient who developed a traumatic myocardial in farction following a closed chest injury, in whom a fistula developed from the right coronary sinus of Valsalva to the right ventricle. This was diagnosed preoperatively and successfully repaired with the use of cardiac bypass. The pa tient made a complete and perfect recovery. As far as the authors are aware, this is the first case of this type so far reported in the literature. REFERENCES 1. Heiner, D. C , Hara, M., and White, H . J . : Cardio-aortic Fistulae and Aneurysms of the Sinus of Valsalva in Infancy: A Report of an Aortic Left Atrial Communica tion Indistinguishable From a Ruptured Aneurysm of the Aortic Sinus, Pediatrics 27: 415, 1961. 2. Warthen, R. O. : Congenital Aneurysm of t h e Right Anterior Sinus of Valsalva ( I n terventricular Aneurysm) With Spontaneous Rupture Into the Left Ventricle, Am. Heart J . 37: 975, 1949. 3. Weiss, L.: Rupture of an Aneurysm of t h e Right Aortic Sinus (of Valsalva), Brit. H e a r t J . 19: 138, 1957. 4. Gerbode, F . , Osborn, J . J., Johnston, J . B.. and Kerth, W. J . : Ruptured Aneurysms of t h e Aortic Sinuses of Valsalva, Ani. J . Surg. 102: 268, 1961. 5. Sawyers, J . L., Adams, J . E., a n d Scott, H . W.: Surgical Treatment for Aneurysms of t h e Aortic Sinuses W i t h Aortico-atrial Fistula: Experimental a n d Clinical Study, Surgery 4 1 : 26, 1957. 6. King, H., a n d Shumacker, H. B., J r . : Surgical Repair of a Traumatic Aortic Right V e n t r i c u l a r F i s t u l a , J . THORACIC SURG. 3 5 : 734, 1958.
7. Gerbode, F . : Discussion of Sawyers et al.s 8. Smyth, N . P . D., Adkins, P . C., Kelser, G. A., and Calatayud, J . : Traumatic Aortic Right Ventricular Fistula, Surg., Gvnec. & Obst. 109: 566, 1959. 9. Nowlan, J . A., Steiger, Z., Bicoff, J . P., Fell, E. A., and Tobin, J . R.: Traumatic Aortic Right Ventricular Fistula, J . A. M. A. 181: 159, 1962. 10. Swanepoel, A., Schrire, V., Nellen, M., Vogepoel, L., and Barnard, C : Traumatic Aortico Right Atrial Fistula: Report of a Case Corrected by Operation, Am. H e a r t J . 6 1 : 120, 1961. 11. Agustsson, M. H., Weinberg, M., Gasul, B. M., Fell, E . H., Arcilla, R. A., Bicoff, J . P., Steiger, Z., and I w a T.: Fistulas Following Corrective Operations for Ven t r i c u l a r Septal Defects
43: 166, 1962.
and T e t r a l o g y of F a l l o t J . THORACIC & CARDIOVAS. SURG.