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Asian Cardiovasc Thorac Ann 2001;9:332-333
© 2001 Asia Publishing EXchange Pte Ltd

CASE STUDY

Severe Mechanical Hemolysis After Complete Occlusion of Arterial Duct

Ümrah Aydogan, MD, Türkan Tansel, MD1

Department of Pediatrics
1 Department of Cardiovascular Surgery Istanbul University School of Medicine Istanbul, Turkey
For reprint information contact: Ümrah Aydogan, MD Tel: 90 532 612 4719 Fax: 90 212 631 4170 email: uaydogan{at}turk.net Department of Pediatrics, Istanbul University School of Medicine, Çapa, Istanbul 34390, Turkey.

   ABSTRACT
TOP
 ABSTRACT
INTRODUCTION
 CASE REPORT
 DISCUSSION
 REFERENCES
 
Streptokinase was administered for femoral artery thrombosis in a 14-month-old girl after complete occlusion of an arterial duct with a Jackson coil. Hemoglobinuria started after femoral artery patency was achieved. Ductal shunting was unimpeded 35 hours after cessation of streptokinase. The problem was resolved by implantation of a second coil.


   INTRODUCTION
TOP
 ABSTRACT
 INTRODUCTION
CASE REPORT
 DISCUSSION
 REFERENCES
 
A rare but serious complication of transcatheter occlusion of a patent ductus arteriosus (PDA) is mechanical hemolysis; its incidence was 0.5% in a multicenter study.1 Fragmentation of red blood cells is caused by hitting the occluding device at high velocity when shunted through an incompletely occluded arterial duct.


   CASE REPORT
TOP
 ABSTRACT
 INTRODUCTION
 CASE REPORT
DISCUSSION
 REFERENCES
 
A 14-month-old girl with frequent lower respiratory tract infections, fatigue during feeding, and growth retardation (8.1 kg, 73 cm) was referred for investigation of a cardiac murmur. Physical and echocardiographic findings were compatible with a moderate sized PDA. The diameter of the duct was measured as 5.5 mm at the pulmonary artery end, and the shape of the wide ampulla was type A. She was admitted for elective transcatheter PDA occlusion. An aortogram confirmed the echocardiographic data. The standard occlusion procedure with a Jackson coil (William Cook, Braeverskoy, Denmark) with 4 loops of 8 mm in diameter was performed via the venous route using 5F Seldinger sheaths (Cordis Corp., Miami, FL, USA) as described elsewhere.2 A post-procedure aortogram revealed complete occlusion of the duct (Figure 1AGo). The duration of arterial catheterization was less than 40 minutes. Signs of ischemia were noted the next day and the patient began to cry and point to her right leg, although color Doppler ultrasonography revealed blood flow at 35 m•sec-1 in the superficial femoral artery. Streptokinase was started with a bolus dose of 4,000 U•kg-1 and a maintenance dose of 1,000 U•kg-1•h-1, because of its more rapid thrombolytic effect compared to heparin. The maintenance dose was raised to 2,000 U•kg-1•h-1 9 hours later because physical and laboratory findings did not change. Bleeding from both inguinal puncture sites occurred in the 10th hour of streptokinase treatment, with the return of distal pulses in the right lower extremity; thus the streptokinase infusion was stopped. Severe hemoglobinuria was observed 4 hours later. It was hoped that with elimination of the drug from the plasma, hemolysis would stop in a few hours, with spontaneous reocclusion of the PDA. Immediate repeat catheterization was considered dangerous as the thrombolytic effect of streptokinase persisted. However, the hemoglobinuria did not resolve and erythrocyte transfusion was necessary because of severe anemia. On the 4th day after the occlusion procedure and 35 hours after cessation of streptokinase, hemoglobinuria still persisted. Color Doppler echocardiography demonstrated multiple jets through the duct into the pulmonary artery. A second occlusion procedure was undertaken. Left femoral artery access failed because of a streptokinase-related hematoma from a previous introduction attempt. The right femoral artery was catheterized again, at a point 1 cm above the original entry site. The aortogram revealed a moderate left-to-right ductal shunt (Figure 1BGo). Another Jackson coil, 3 mm in diameter with 4 loops, was implanted into the original one. Complete occlusion was demonstrated by angiography. A recurrence of femoral thrombosis resolved within 3 hours with another streptokinase infusion, and the patient was discharged 2 days later.




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  		Figure 1. (A) Aortogram demonstrating complete occlusion of the arterial duct in the first occlusion procedure. (B) Aortography after streptokinase treatment demonstrating a moderate sized ductal shunt.

 

   DISCUSSION
TOP
 ABSTRACT
 INTRODUCTION
 CASE REPORT
 DISCUSSION
REFERENCES
 
Femoral artery thrombosis is the most frequent com-plication of retrograde arterial cardiac catheterization, particularly in neonates and infants. The use of prophylactic heparinization to avoid this problem is controversial. However, PDA coil occlusion is a relatively short procedure and in order to abolish residual ductal leakage by thrombosis, prophylactic heparin administra-tion is usually withheld. It is recommended that heparin infusion be started if the distal pulses are not palpable a few hours after the arterial catheterization, whether or not prophylactic heparin was administered.3 However, in a patient such as ours, the risk of recanalization of the PDA must be considered. If the physical findings have not changed by the next day, most investigators currently prefer fibrinolytic therapy as the next step rather than surgical embolectomy.4 Streptokinase is eliminated from plasma with a half-life of 18 to 30 minutes. The biological half-life is more prolonged, ranging from 82 to 184 minutes. Plasminogen concentrations usually return to normal within 24 hours after cessation of thrombolytic therapy.5 When these data were taken into consideration, it was expected that ductal leakage would cease in a few hours due to thrombosis around the coil. Unfortunately, it persisted.

Several approaches to the problem of hemolysis after PDA occlusion with coils have been described, including early coil reocclusion, transcatheter removal of the original coil and implantation of a second one, and waiting for spontaneous cessation of hemolysis.6–8 The latter approach is possible only in cases of mild hemolysis. When hemolysis is persistent and severe, residual flow must be halted as soon as possible so as to avoid renal damage and administration of blood products. To the best of our knowledge, mechanical hemolysis after complete occlusion of a PDA has not been reported previously. This experience illustrates the risk of hemolysis despite a completely occluded PDA if thrombolytic therapy for femoral thrombosis is started. It should be kept in mind that ductal shunting may not stop even 35 hours after cessation of thrombolytic therapy and no options other than a conservative approach are available in this period.


   REFERENCES
TOP
 ABSTRACT
 INTRODUCTION
 CASE REPORT
 DISCUSSION
 REFERENCES
 

  1. Tynan M. Transcatheter occlusion of persistent arterial duct: report of the European Registry. Lancet 1992; 340:1062–6.[Medline]

  2. Tometzki AJ, Arnold R, Peart I, Sreeram N, Abdulhamed JM, Godman MJ, et al. Transcatheter occlusion of the patent ductus arteriosus with Cook detachable coils. Heart 1996;76:531–5.[Abstract/Free Full Text]

  3. Ino T, Benson LN, Freedom RM, Barker GA, Aipursky A, Rowe RD. Thrombolytic therapy for femoral artery thrombosis after pediatric cardiac catheterization. Am Heart J 1988;15:633–9.

  4. Aydogan Ü, Cantez T, Dindar A, Tanman B, Ertugrul T, Omeroglu R. Fibrinolytic therapy for femoral arterial thrombosis after cardiac catheterization in infants and children. J Invas Cardiol 1992;4:445–7.

  5. Chintagumpala MM, Steuber CP. Anticoagulant and thrombolytic agents. In: Garson A Jr, Bricker JT, Fisher DJ, Neish SR, editors. The science and practice of pediatric cardiology. 2nd ed. Baltimore: Williams & Wilkins, 1998:2541–52.

  6. Cheung YF, Leung MP, Chau KT. Early implantation of multiple spring coils for severe haemolysis after incomplete transcatheter occlusion of persistent arterial duct. Heart 1997;77:477–8.[Abstract/Free Full Text]

  7. Henry G, Danilowicz D, Verme R. Severe hemolysis following partial coil occlusion of patent ductus arteriosus. Cathet Cardiovasc Diagn 1996;39:371–2.[Medline]

  8. Lee C, Hsieh K, Huang T, Choong C. Spontaneous resolution of hemolysis after partial coil occlusion of ductus arteriosus. Pediatr Cardiol 1999;20:371–2.[Medline]





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