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J Thorac Cardiovasc Surg 1999;118:953-954
© 1999 Mosby, Inc.

BRIEF COMMUNICATIONS

UNILATERAL RIGHT PULMONARY THROMBOENDARTERECTOMY FOR CHRONIC EMBOLISM: A SUCCESSFUL PROCEDURE IN AN INFANT

Virginie Lambert, MDa, Philippe Durand, MDb, Denis Devictor, MDb, Claude Planché, MDa, Alain Serraf, MDa, Le Plessis Robinson and Le Kremlin Bicêtre, France

From the Pediatric Cardiology and Cardiac Surgery, Marie-Lannelongue Hospital,a Le Plessis Robinson, and Pediatric Intensive Care Unit, University Hospital of Bicêtre,b Le Kremlin Bicêtre, France.

Address for reprints: Virginie Lambert, MD, Pediatric Cardiology and Cardiac Surgery, Marie-Lannelongue Hospital, 133 Avenue de la Résistance, 92350 Le Plessis Robinson, France.

Pulmonary embolism is a rare disorder in newborn infants, usually related to central venous catheter thrombosis,Go 1 and is a source of significant morbidity and death. Although surgical embolectomy has been described as a salvage procedure in acute pulmonary embolism,Go 2 to the best of our knowledge salvage thromboendarterectomy for chronic pulmonary embolism has not yet been reported in an infant.

Clinical summary.
A male infant weighing 930 gm was delivered vaginally at 26 weeks’ gestation of a mother treated with dexamethasone during pregnancy because of congenital hyperplasia of the adrenal gland. At birth, respiratory distress syndrome mandated mechanical ventilation. Arterial and venous umbilical lines up to the second day of life were replaced by a Broviac catheter in the superior vena cava. From day 2 to day 30, bronchopulmonary dysplasia progressively appeared on thoracic radiography. On day 30, hepatomegaly and edema of the legs occurred without any evidence of thrombosis in the right atrium and inferior vena cava on echocardiography. During the following weeks, several attempts at extubation were made but failed after a few days because of progressive deterioration in respiratory status. At the age of 6 months (weight 4.2 kg), refractory hypoxemia necessitating ventilation with 100% oxygen was imputed to a multimicrobial pneumonopathy. Echocardiography showed right atrial and ventricular dilatation, suprasystemic pulmonary pressure, and a nearly complete occlusion of the right pulmonary artery (RPA) by a mass at its origin(Fig 1). No thrombus was seen in the heart or the venae cavae. The diagnosis of massive RPA embolism was suspected and confirmed by venous angiography. The infant was treated twice with a 1 mg/kg dose of recombinant tissue-type plasminogen activator without effect on the thrombus. Progressive profound hypoxia necessitated more aggressive mechanical ventilation, and because of hemodynamic deterioration surgical embolectomy was undertaken.



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  		Fig. 1. Echocardiography: the short-axis view of the great arteries shows an obstruction of the RPA by a bright, rounded mass. The MPA and LPA appear dilated. RPA, Right pulmonary artery; MPA, main pulmonary artery; LPA, left pulmonary artery.

 
At the operation, a calcified thrombus extended from the ostium of the RPA distally to the RPA branches. A thromboendarterectomy was then performed to extract fragments up to the intermediate and superior lobular pulmonary artery. Histologic examination showed old, fibrous, calcified clots. The immediate postoperative course disclosed severe pulmonary vascular reactivity and persisting suprasystemic pressure. The hemodynamic status improved progressively and the infant could be extubated 8 days later. Heparin infusion was continued, with a high dose up to 700 IU/kg in 24 hours being necessary to provide partial thromboplastin times within therapeutic ranges. To prevent rethrombosis, we administered low-molecular-weight heparin subcutaneously for 6 weeks. Subsequent investigations showed normal levels of protein C, protein S, and antithrombin III. At discharge, echocardiography showed a distal RPA stenosis and a right ventricular systolic pressure of 50 mm Hg. The RPA became more stenotic 3 months later and necessitated percutaneous dilatation(Fig 2), which reduced the mean pressure in the main and the left pulmonary arteries from 56 to 30 mm Hg. At the age of 1 year, which was 6 months after the operation, the infant had recovered well and showed uneventful development free from oxygen therapy.



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  		Fig. 2. Pulmonary angiography performed 3 months after thromboendarterectomy. A, The RPA is stenosed immediately after branching at the end of the arteriotomy; the SLB is still obstructed by clot. B, After dilatation, the vascular lumen is regular and the RPA appears thrombus free except for SLB. RPA, Right pulmonary artery; SLB, superior lobular branch.

 
Discussion.
Pulmonary embolism is a rare disorder in pediatric patients. The Canadian RegistryGo 1 established an incidence of 0.86 per 10000 hospital admissions, which is significantly lower than for hospitalized adults. This incidence is probably underestimated because of the infant’s better physiologic tolerance, which delays or even masks the diagnosis. In a review of autopsy cases of pulmonary embolism in children, the antemortem diagnosis was considered in only one third of patients.Go 3 In our patient, pulmonary symptoms were first attributed to preterm delivery with frequent bronchopulmonary dysplasia and nosocomial pneumonopathy, and signs of right ventricular failure at the time of thrombus migration were unrecognized. In children, in contrast to adults, a deep vein thrombosis and pulmonary embolism are always explained by a predisposing factor. In the first year of life, the most common cause is the use of central venous lines.Go 1 The long-term use of a central venous line was probably the cause of thrombus formation in our patient, who otherwise had no coagulation disorders. The risk is even increased in preterm infants because of a relatively low flow through the catheter, the size of the catheter relative to the vessel, and a decreased ability to lyse thrombi.Go 4 That fact warrants frequent echocardiographic examinations in these patients to allow direct imaging of the catheter, intracardiac or pulmonary arterial thrombi, or indirect indices of increased right ventricular volume or pressure. Even though rare, the diagnosis of chronic pulmonary embolism should be suspected in an infant with unexplained pulmonary symptoms or right ventricular failure, especially in preterm infants or children who have a prolonged need for a central line.

Because experience is limited in the pediatric population, management of pulmonary embolism is extrapolated from guidelines for adults. Heparin infusion and thrombolytic therapy are the treatments of choice. In the presence of hemodynamic deterioration caused by massive pulmonary embolism, surgical embolectomy should be discussed.Go 2 In our case, the attempt at thrombolysis failed and surgical embolectomy was considered as a salvage procedure. Surgical removal of pulmonary artery or right-sided intracardiac thrombi has been successfully described several times in children,Go Go 1,2 but the mortality rate is high in premature infants weighing less than 1500 g.Go 4 A pulmonary embolectomy alone would not have provided adequate relief of the obstruction in our patient, and a pulmonary thromboendarterectomy as described in adultsGo 5 was necessary to remove distal organized thrombus and obtain hemodynamic improvement. To the best of our knowledge, this operation has not previously been reported in children. The histologic examination supports the observations of the Canadian RegistryGo 1: with a follow-up of 6 months to 3 years, the majority of children had incomplete resolution of their thrombotic event, possibly related to an impairment of endogenous fibrinolysis shown by studies in vitro.

In adults, chronic embolism results in pulmonary hypertension and carries a poor prognosis. The long-term consequences of pulmonary embolism in children are unknown, and chronic thromboembolic pulmonary hypertension probably is underdiagnosed. Surgical correction by pulmonary thromboendarterectomy is feasible in low-weight children and yields a good result. Thromboendarterectomy should be discussed in symptomatic children to decrease pulmonary pressure and improve gas exchange, especially in preterm infants with hypoxia related to bronchopulmonary dysplasia.

References

  1. Andrew M, David M, Adams M, Kaiser A, Anderson R, Barnard D, et al. Venous thromboembolic complications (VTE) in children: first analyses of the Canadian Registry of VTE. Blood 1994;83:1251-7.[Abstract/Free Full Text]
  2. Gamillscheg A, Nurnberg JH, Alexi-Meskishvili V, Werner H, Abdul-Kaliq H, Uhlemann F, et al. Surgical emergency embolectomy for the treatment of fulminant pulmonary embolism in a preterm infant. J Pediatr Surg 1997;32:1516-8.[Medline]
  3. Buck JR, Connors RH, Coon WW, Weintraub WH, Wesley JR, Coran AG. Pulmonary embolism in children. J Pediatr Surg 1981;16:385-91.[Medline]
  4. Alkalay AL, Mazkereth R, Santulli T, Pomerance JJ. Central venous line thrombosis in premature infants: a case management and literature review. Am J Perinatol 1993;10:323-7.[Medline]
  5. Dartevelle P, Fadel E, Chapelier A, Macchiarini P, Cerrina J, Leroy-Ladurie F, Parquin F, et al. Surgical treatment of chronic pulmonary thromboembolic disease. Sang Thromb Vaisseaux 1997;9:241-50.
Received for publication June 22, 1999. Accepted for publication June 29, 1999.


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