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J Thorac Cardiovasc Surg 1999;117:1230-1231
© 1999 Mosby, Inc.

LETTERS TO THE EDITOR

A successful treatment of serous leakage from a polytetrafluoroethyleneBlalock-Taussig shunt with intravenous fibrinogen administration

Department of Surgery
Institute of Clinical Medicine
Universityof Tsukuba
1-1-1 Tennoh-dai
305-8575 Tsukuba-city, Japan

To the Editor:

Occasionally, excessive serous fluid leakage occurs as a complicationafter the modified polytetrafluoroethylene (PTFE) Blalock-Taussig (B-T) shunt.The estimated frequency of this complication is reported as approximately20%, ^1,2 but the mechanism is not clearly defined. We present a case of serousleakage through a modified B-T shunt treated with intravenous fibrinogen administrationand describe our hypothesis about the role of fibrinolysis in this problem.

A 3-year-old boy (11.5 kg) with single right ventricle and pulmonarystenosis was admitted for the treatment of cyanosis and exercise intolerance.A blood test revealed polycythemia with a hemoglobin level of 20.8 g/dL. Wedecided not to proceed directly to a Fontan procedure because of the lackof direct measurement of pulmonary arterial pressure and pulmonary vascularresistance (the catheter did not go through the narrow ventricular outflowtract). As a preparation for a Fontan-type procedure, the boy underwent aleft modified B-T shunt with a 5-mm PTFE shunt. Anticoagulation was initiatedon the first postoperative day with a continuous heparin infusion (200 units/kgper day; Novo Heparin, Hoechst Marion Roussel, AG, Frankfurt, Germany) andan oral administration of ticlopidine hydrochloride (5 mg/kg per day; Panaldine,Dai-Ichi, Inc, Tokyo, Japan). On the third postoperative day, the chest tubedrainage increased appreciably and the anticoagulation therapy was discontinued.The drainage continued for more than a week, with excessive serous fluid lossesaveraging 700 mL daily. The blood test revealed a marked decrease in totalplasma protein (4.2 g/dL) and plasma fibrinogen (93.6 mg/dL) levels. The massiveleakage continued and plasma protein and fibrinogen did not increase despitethe infusion of packs of frozen plasma. We administered fibrinogen (heatedand freeze-dried human fibrinogen; Fibrinogen HT, Yoshitomi, Inc, Osaka, Japan)intravenously, 200 mg/kg per day, for 2 days. The leakage stopped instantlyand the fibrinogen level increased to more than 300 mg/dL. The chest tubewas removed and the patient was discharged after a catheterization. Anticoagulationtherapy was never resumed.

Three weeks later, the boy was readmitted with dyspnea, chest pain,and increasing cyanosis. A chest x-ray film revealed a massive pleural effusionon the left side, and a chest tube was inserted. Chest drainage continuedfor several days, with excessive serous fluid losses and decreased plasmaprotein (4.4 g/dL) and fibrinogen (71.3 mg/dL) levels. Fibrinogen (200 mg/kgper day for 2 days) was administered again, and the fluid leakage stoppedcompletely within 2 days. On the basis of a high D-dimer level (736 ng/mL)detected after the second fibrinogen administration, we speculated that thefibrinolytic system had been greatly amplified in this cyanotic patient. Therefore,we started an oral medication of tranexamic acid (Transamin-G; Dai-Ichi, Inc,Tokyo, Japan) 30 mg/kg daily as an antifibrinolytic therapy. Additional prophylacticfibrinogen infusion was achieved twice at 2-week intervals. Since then, theplasma fibrinogen level has been within normal range (no less than 170 mg/dL),and the plasma D-dimer level has been maintained at a relatively lower level(200 to 600 ng/mL), leaving the patient in stable condition without any fluidleakage. Neither thrombocytopenia nor bleeding tendency was observed. No thromboticevents occurred. A bidirectional Glenn shunt was successfully done 6 monthsafter the B-T shunt.

LeBlanc and associates ²first described excessive fluid leakage after a modified B-T shunt. Boltonand Cannon ³ suggested thatrapid wetting of the graft with organic solvents or a high blood flow withfaulty formation of fibrin may be responsible for the leakage. However, themechanism still remains unclear. In most of the cases previously described,conservative management failed and surgical interventions were required. Maitlandand coworkers ⁴ reported 2cases in which leakage was controlled with intraluminal fibrin glue. Noyezand Daenen ¹ reported thecase of a patient treated with fibrin glue and collagen fleece wrapping. Intravenousfibrinogen administration was first described by Suzuki and associates ⁵ as an effective conservative managementof the leakage. Human fibrinogen products were originally developed for congenitalfibrinogenopenia and are commercially available in Germany and Japan. Accordingto their report, 7 patients were successfully treated with fibrinogen administration(150-200 mg/kg per day for 3 or 4 days), and in 2 of those cases the plasmafibrinogen level was much lower than the normal range at the beginning ofthe leakage. As we described, the fact that plasma fibrinogen level decreasedand plasma D-dimer level increased at the beginning of the fluid losses maybe an important clue to the mechanism of leakage. We speculate that the fibrinolyticsystem could be greatly amplified in some cyanotic patients with polycythemiaagainst great thrombotic tendency owing to high blood viscosity. A probableexplanation is that the fibrin cross-linking step and newly formed fibrinclots in a PTFE graft wall could be easily impaired and degraded when thisoverwhelming fibrinolytic regulation occurs. Once the strong fibrinolysisbegins, a large amount of fibrinogen may need to be supplied until newly formedcross-linked fibrin is tightened and subsequent clot formation is completed.Platelet function of patients with polycythemia may play another importantrole in this problem. In our preliminary data of the patients, very weak aggregationand poor -granule release response have been observed. We hypothesizethat both strong fibrinolytic response and weakened platelet function couldbe the physiologic adaptation phenomenon against the great thrombotic risk.Intravenous fibrinogen administration is effective and the combination offibrinogen and antifibrinolytic therapy may be the best conservative managementavailable for serous leakage through PTFE so far. Further investigations areneeded to prove our hypothesis.

12/8/97767

References

1. Noyez L, Daenen W. The modified polytetrafluoroethyleneBlalock-Taussig shunt: case report of an unusual complication. J Thorac CardiovascSurg 1987;94:634-5.[Abstract]
   
2. LeBlanc J, Albus R, Williams WG, Moes CA,Wilson G, Freedom RM, et al. Serous fluid leakage: a complication followingthe modified Blalock-Taussig shunt. J Thorac Cardiovasc Surg 1984;88:259-62.[Abstract]
   
3. Bolton W, Cannon JA. Seroma formation associatedwith PTFE vascular grafts used as arteriovenous fistulae. Dialysis Transplant 1981;10:60-6.
   
4. Maitland A, Williams WG, Coles JG, FreedomRM, Trusler GA. A method of treating serous fluid leak from a polytetrafluoroethyleneBlalock-Taussig shunt. J Thorac Cardiovasc Surg 1985;90:791-3.[Abstract]
   
5. Suzuki T, Fukuda T, Katohgi T, Oda Y. Effectivenessof fibrinogen to avoid serous fluid leakage through polytetrafluoroethylene(PTFE) tubular graft. J Jpn Assoc Thorac Surg 1990;38:596-600.
   

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