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Thrombosis of the abdominal aorta in a newborn: Case report and review of literature
T h r o m b o s i s of the A b d o m i n a l A o r t a in a N e w b o r n : Case R e p o r t and R e v i e w of L i t e r a t u r e By Jos6 Maria Pereira de Godoy, Carlos Henrique De Marchi, Marciali Gongalves Fonseca Silva, Maria Carmen Monteiro Carvalho, Airton Moscardini, and Jane Cristina Medeiros S#o Paulo, Brazil
The case of a newborn infant w h o presented thrombosis in the abdominal portion of the aorta, which resulted in the complete occlusion is reported. Diagnosis was made using Doppler echocardiography. The initial therapeutic approach was clinical support, but as the thrombi started to increase in size (secondary thrombosis) and evolved to the infrarenal terminal aorta, anticoagulation with heparin was indicated.
The total disappearance of both the secondary thrombosis and the embolus was evidenced by a subsequent echocardiography after the anticoagulation therapy. J Pediatr Surg 38:Ell. Copyright 2003, Elsevier Science (USA). All rights reserved.
HROMBOSIS is a rare event in infants. However, many conditions can contribute to an increased risk of formation of thrombi during the neonatal period. The most common cause of arterial thromboembolism in infants is related to the use of catheters. This includes both cardiac and peripheral catheterization. 1 Conditions such as Takayasu's arteritis, transplanted arteries, Kawasaki's disease, and congenital forms of cardiac diseases are reported as causes of embolism. 2 The incidence of thrombotic complications after cardiac catheterization via the femoral artery, in the absence of anticoagulation, is approximately 40%. Infants younger than 10 years have a higher incidence than older infants. Prevention with the use of aspirin does not significantly reduce the risk of arterial thrombosis. However, the use of 100 to 150 U/kg of heparin reduces the risk from 40% to approximately 8%. The use of a single 50 U/kg dose of heparin can be as effective as a single 100 U/kg dose when it is given immediately after puncture. 2 The main symptoms and signs of arterial occlusion in newborn infants are a reduction in temperature, perfusion of the skin, and an absence of pulse at the extremities. However, newborn infants with occlusion of the aorta can be asymptomatic. 2-4 Patients with Kawasaki's disease benefit in the acute phase of the illness, at about 14 days, with high doses of aspirin (from 80 to 100 mg/kg/d). Shortening of the limb and intermittent claudication are long-term complications as a consequence of thrombosis after femoral catheterization in infants. 2 The aims of this study were to report the development of a case of thrombosis in the abdominal portion of the aorta in a newborn infant and to review published papers relating to this subject.
CASE REPORT
T
Journal of Pediatric Surgery, Vol 38, No 4 (April), 2003:E11
INDEX WORDS: Arterial thrombosis, aorta, newborn.
The case of a newborn boy is who was born wifl0out apparent problems and who was released from hospitaJ breas/feeding is reported. He was well until floe fourflo day after birflo when he started to refuse to eat and had reduced bowel movements. His mofloer noticed a reduction in floe quantity of urine and float fl0e urine was darker. On fl0e 10fl0 postnatal day, he was hospitalized wifl0 dehydration, anuda, anemia, and increased levels of urea and creafinine. He tmderwent rehydration and antibiotic fl0erapy; however, renal failure developed, and he was submitted to peritoneal diaJysis. An ultrasonic renal examination dearly showed spacing of fl0e renal capsules, however, wifl0out evidence of anatomic alterations. Ofl0er abdominal aJteratJons, such as flarombosis of file abdominal aoFta, were not detected. On file 18ill day of life, he began to saxfferfrom edema, and a sys~_ohcmurmur was auscultated. Until tiffs point, fl0e child did not present clinical alterations, but when fl0e physicaJ exaxnination evidenced a caxdiac aJteratJon, Doppler echocardiography was performed, which showed cardiac dilation wifl0fl0e presence of a mass in fl0e left ventricle and Atrial SeptaJ Defect (ASD) of 4 ram. The approach in relation to fl0e mass was expectant because fl0e infant was aJready within fl0e therapeutic guidelines of anticoagulation, float is, fl0e activated partjaj thromboplastin time (APTF) was altered wifl0out prior treatment. The infaJlt evolved well wifl0 tiffs treatment. Twenty-five days postnatally, the temperature of the lower limbs dropped, and the absence of a femoral pulse led to a diagnosis of thrombosis or embolism by Doppler in the abdominal portion of the aorta with distal occlusion, (Fig 1). An expectant conduct was chosen accompanied by Doppler examinations when, at 55 days, there was an increase in the thrombus in the aorta artery, and the intracardiac thrombus disappeared. Therapy of anticoagulation using heparin then was chosen maintaining the APTT between 70 and 90. The secondary thrombosis gradually disappeared. A total break-up of the thrombus occurred at 15 days of anticoagulation treatment, and, after this period,
From the Departments of Cardiology and Vascular Surgery and Pediatrics, S6o Josd do Rio Preto University School of Medicine, S6o Paulo, Brazil Address reprint requests to Josd Maria Pereira de Godoy, MD, Rua Floriano Peixoto, 2950, S6o Josd do Rio Preto, SP, Brazil, CEP, 15010-020. Copyright 2003, Elsevier Science (USA). All rights reserved. 1531-5037/03/3804-0035530. 00/0 doi: 10.1053/jpsu.2003.50149 11
12
DE GODOY ET AL
Fig 1. Thrombosis in the abdominal aorta.
the heparinwas suspended(Fig 2). The thrombosisimproved,the distal pulses returned, and the renal function normalized. DISCUSSION
In this study, the presence of thrombosis and occlusion of the distal aorta are reported in an infant. The chosen treatment was expectant owing to the general state of health of the infant and because the coagulogram was altered (anticoagulated) suggesting that the evolution of the disease was within the therapeutic range. However, monitoring with echocardiography was performed and
Fig 2. Thrombosis gradually disappeared.
because of secondary thrombosis, the therapy was changed to anticoagulation using heparin. There is no defined conduct in relation to trealment of thrombosis and thromboembolism of newborn infants. Published data are limited to case studies and a small series of cases. Treatment must be considered on a case-by-case basis. Alternatives include clinical observation, anticoagulation, thrombolytic therapy, and surgical thrombectomy. 1-4 In asymptomatic cases, clinical observation may be indicated, but there are no randomized studies that assess the results. When the choice of clinical observation has been made, it is advisable to monitor the thrombosis, and, if it starts to worsen, anticoagulation should be initiated. Conventional anticoagulation using heparin can be used, although, again, clinical data are scarce. One series of a few cases suggests that the treatment is efficient in the solution of thrombosis of newborn infants. The indicated period of anticoagulation is from 10 to 14 days administered endovenously and maintaining the levels of antifactor from 0.3 to 0.7 U/mL. These data are extrapolated from the treatment of adults. 5 Low-molecular-weight heparin is another option of anticoagulation, and this treatment offers some advantages compared with conventional heparin. 6 These advantages include subcutaneous application, lower incidence of bleeding, and greater laboratory control, which, in adults, is not always monitored. Thrombolytic therapy is another option, but there are few publications relating to this, and they only deal with case studies. 7 The most aggressive therapy is surgical thrombectomy, which has been described as successful in the few cases published. 8 The study shows that using an anticoagulant such as heparin was the better option than expectant treatment to resolve the abdominal aorta thrombosis. Adherence of the clot to the intima normally consolidates after 24 hours; the possibility of secondary thrombosis and its consequences suggest a precocious therapeutic approach. The therapy of thrombosis and thromboembolism in newborn infants must be considered on a case-by-case basis where the risks and benefits of each case must be analyzed during the evolution of the disease.
REFERENCES
1. MichelsonAD, BovilE, MonagleP, et al: Antithrombotictherapy in children. Chest 114:748S-769S, 1998 2. HusseinHJ, Trowiszsch E, Becker J, et al: Treatmentof neonatal thrombus formation with recombinant tissue plasminogen activator: Six years experienceand review of the literature. Arch Dis Child Fetal Neonatal 85:18-22, 2001 3. Godoy JMP, Marco LA: Trombose em rec6m-nascido.Pediatria 19:134-138, 1997 4. Manco-JohnsonMJ: Diagnosisand managementof thrombosisin the perinatalperiod. Semin Perinatol 14:393-402, 1990
5. EdstromCS, ChristensenRD: Evaluationand treatmentof thrombosis in the neonatalintensivecare unit. Clin Perinatol27:623-641, 2000 6. KlingerG, HellmannJ, DanemanA: Severeaorticthrombosisin the neonate--Successfultreatmentwith low-moleculax-weighthepaxin:Two case reports and reviewof the literature.Am J Perinatol17:151-158,2000 7. AhluwaliaJS, Kelsall AW, Diederich S, et al: Successful treatment of aortic thrombosis after umbilical catheterization with tissue plasminogenactivator. Acta Paediatr 83:1215-1217, 1994 8. Colbum MD, Gelabert HA, Quinones-BaldrichW: Neonatal aortic thrombosis. Surgery 111:21-28, 1992
The case of a newborn infant w h o presented thrombosis in the abdominal portion of the aorta, which resulted in the complete occlusion is reported. Diagnosis was made using Doppler echocardiography. The initial therapeutic approach was clinical support, but as the thrombi started to increase in size (secondary thrombosis) and evolved to the infrarenal terminal aorta, anticoagulation with heparin was indicated.
The total disappearance of both the secondary thrombosis and the embolus was evidenced by a subsequent echocardiography after the anticoagulation therapy. J Pediatr Surg 38:Ell. Copyright 2003, Elsevier Science (USA). All rights reserved.
HROMBOSIS is a rare event in infants. However, many conditions can contribute to an increased risk of formation of thrombi during the neonatal period. The most common cause of arterial thromboembolism in infants is related to the use of catheters. This includes both cardiac and peripheral catheterization. 1 Conditions such as Takayasu's arteritis, transplanted arteries, Kawasaki's disease, and congenital forms of cardiac diseases are reported as causes of embolism. 2 The incidence of thrombotic complications after cardiac catheterization via the femoral artery, in the absence of anticoagulation, is approximately 40%. Infants younger than 10 years have a higher incidence than older infants. Prevention with the use of aspirin does not significantly reduce the risk of arterial thrombosis. However, the use of 100 to 150 U/kg of heparin reduces the risk from 40% to approximately 8%. The use of a single 50 U/kg dose of heparin can be as effective as a single 100 U/kg dose when it is given immediately after puncture. 2 The main symptoms and signs of arterial occlusion in newborn infants are a reduction in temperature, perfusion of the skin, and an absence of pulse at the extremities. However, newborn infants with occlusion of the aorta can be asymptomatic. 2-4 Patients with Kawasaki's disease benefit in the acute phase of the illness, at about 14 days, with high doses of aspirin (from 80 to 100 mg/kg/d). Shortening of the limb and intermittent claudication are long-term complications as a consequence of thrombosis after femoral catheterization in infants. 2 The aims of this study were to report the development of a case of thrombosis in the abdominal portion of the aorta in a newborn infant and to review published papers relating to this subject.
CASE REPORT
T
Journal of Pediatric Surgery, Vol 38, No 4 (April), 2003:E11
INDEX WORDS: Arterial thrombosis, aorta, newborn.
The case of a newborn boy is who was born wifl0out apparent problems and who was released from hospitaJ breas/feeding is reported. He was well until floe fourflo day after birflo when he started to refuse to eat and had reduced bowel movements. His mofloer noticed a reduction in floe quantity of urine and float fl0e urine was darker. On fl0e 10fl0 postnatal day, he was hospitalized wifl0 dehydration, anuda, anemia, and increased levels of urea and creafinine. He tmderwent rehydration and antibiotic fl0erapy; however, renal failure developed, and he was submitted to peritoneal diaJysis. An ultrasonic renal examination dearly showed spacing of fl0e renal capsules, however, wifl0out evidence of anatomic alterations. Ofl0er abdominal aJteratJons, such as flarombosis of file abdominal aoFta, were not detected. On file 18ill day of life, he began to saxfferfrom edema, and a sys~_ohcmurmur was auscultated. Until tiffs point, fl0e child did not present clinical alterations, but when fl0e physicaJ exaxnination evidenced a caxdiac aJteratJon, Doppler echocardiography was performed, which showed cardiac dilation wifl0fl0e presence of a mass in fl0e left ventricle and Atrial SeptaJ Defect (ASD) of 4 ram. The approach in relation to fl0e mass was expectant because fl0e infant was aJready within fl0e therapeutic guidelines of anticoagulation, float is, fl0e activated partjaj thromboplastin time (APTF) was altered wifl0out prior treatment. The infaJlt evolved well wifl0 tiffs treatment. Twenty-five days postnatally, the temperature of the lower limbs dropped, and the absence of a femoral pulse led to a diagnosis of thrombosis or embolism by Doppler in the abdominal portion of the aorta with distal occlusion, (Fig 1). An expectant conduct was chosen accompanied by Doppler examinations when, at 55 days, there was an increase in the thrombus in the aorta artery, and the intracardiac thrombus disappeared. Therapy of anticoagulation using heparin then was chosen maintaining the APTT between 70 and 90. The secondary thrombosis gradually disappeared. A total break-up of the thrombus occurred at 15 days of anticoagulation treatment, and, after this period,
From the Departments of Cardiology and Vascular Surgery and Pediatrics, S6o Josd do Rio Preto University School of Medicine, S6o Paulo, Brazil Address reprint requests to Josd Maria Pereira de Godoy, MD, Rua Floriano Peixoto, 2950, S6o Josd do Rio Preto, SP, Brazil, CEP, 15010-020. Copyright 2003, Elsevier Science (USA). All rights reserved. 1531-5037/03/3804-0035530. 00/0 doi: 10.1053/jpsu.2003.50149 11
12
DE GODOY ET AL
Fig 1. Thrombosis in the abdominal aorta.
the heparinwas suspended(Fig 2). The thrombosisimproved,the distal pulses returned, and the renal function normalized. DISCUSSION
In this study, the presence of thrombosis and occlusion of the distal aorta are reported in an infant. The chosen treatment was expectant owing to the general state of health of the infant and because the coagulogram was altered (anticoagulated) suggesting that the evolution of the disease was within the therapeutic range. However, monitoring with echocardiography was performed and
Fig 2. Thrombosis gradually disappeared.
because of secondary thrombosis, the therapy was changed to anticoagulation using heparin. There is no defined conduct in relation to trealment of thrombosis and thromboembolism of newborn infants. Published data are limited to case studies and a small series of cases. Treatment must be considered on a case-by-case basis. Alternatives include clinical observation, anticoagulation, thrombolytic therapy, and surgical thrombectomy. 1-4 In asymptomatic cases, clinical observation may be indicated, but there are no randomized studies that assess the results. When the choice of clinical observation has been made, it is advisable to monitor the thrombosis, and, if it starts to worsen, anticoagulation should be initiated. Conventional anticoagulation using heparin can be used, although, again, clinical data are scarce. One series of a few cases suggests that the treatment is efficient in the solution of thrombosis of newborn infants. The indicated period of anticoagulation is from 10 to 14 days administered endovenously and maintaining the levels of antifactor from 0.3 to 0.7 U/mL. These data are extrapolated from the treatment of adults. 5 Low-molecular-weight heparin is another option of anticoagulation, and this treatment offers some advantages compared with conventional heparin. 6 These advantages include subcutaneous application, lower incidence of bleeding, and greater laboratory control, which, in adults, is not always monitored. Thrombolytic therapy is another option, but there are few publications relating to this, and they only deal with case studies. 7 The most aggressive therapy is surgical thrombectomy, which has been described as successful in the few cases published. 8 The study shows that using an anticoagulant such as heparin was the better option than expectant treatment to resolve the abdominal aorta thrombosis. Adherence of the clot to the intima normally consolidates after 24 hours; the possibility of secondary thrombosis and its consequences suggest a precocious therapeutic approach. The therapy of thrombosis and thromboembolism in newborn infants must be considered on a case-by-case basis where the risks and benefits of each case must be analyzed during the evolution of the disease.
REFERENCES
1. MichelsonAD, BovilE, MonagleP, et al: Antithrombotictherapy in children. Chest 114:748S-769S, 1998 2. HusseinHJ, Trowiszsch E, Becker J, et al: Treatmentof neonatal thrombus formation with recombinant tissue plasminogen activator: Six years experienceand review of the literature. Arch Dis Child Fetal Neonatal 85:18-22, 2001 3. Godoy JMP, Marco LA: Trombose em rec6m-nascido.Pediatria 19:134-138, 1997 4. Manco-JohnsonMJ: Diagnosisand managementof thrombosisin the perinatalperiod. Semin Perinatol 14:393-402, 1990
5. EdstromCS, ChristensenRD: Evaluationand treatmentof thrombosis in the neonatalintensivecare unit. Clin Perinatol27:623-641, 2000 6. KlingerG, HellmannJ, DanemanA: Severeaorticthrombosisin the neonate--Successfultreatmentwith low-moleculax-weighthepaxin:Two case reports and reviewof the literature.Am J Perinatol17:151-158,2000 7. AhluwaliaJS, Kelsall AW, Diederich S, et al: Successful treatment of aortic thrombosis after umbilical catheterization with tissue plasminogenactivator. Acta Paediatr 83:1215-1217, 1994 8. Colbum MD, Gelabert HA, Quinones-BaldrichW: Neonatal aortic thrombosis. Surgery 111:21-28, 1992