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CC BY 4.0 · Journal of Child Science 2021; 11(01): e212-e215
DOI: 10.1055/s-0041-1733873
Case Report

Pulmonary Embolism in a Critically Ill Infant with Univentricular Parallel Circulation

Kazuhiro Shoya 
1   Department of Critical Care and Emergency Medicine, Tokyo Metropolitan Children's Medical Center, Tokyo, Japan
,
Jun Maeda
2   Department of Cardiology, Tokyo Metropolitan Children's Medical Center, Tokyo, Japan
,
Hiroki Nagamine
2   Department of Cardiology, Tokyo Metropolitan Children's Medical Center, Tokyo, Japan
,
Akihiro Shimotakahara
3   Department of Surgery, Tokyo Metropolitan Children's Medical Center, Tokyo, Japan
,
Yukihiro Yoshimura
4   Department of Cardiovascular Surgery, Tokyo Metropolitan Children's Medical Center, Tokyo, Japan
,
Osamu Saito
1   Department of Critical Care and Emergency Medicine, Tokyo Metropolitan Children's Medical Center, Tokyo, Japan
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Abstract

A 3-month-old infant patient with hypoplastic left heart syndrome diagnosed in the prenatal period required long-term intensive care for refractory chylothorax and chylous ascites after undergoing bilateral pulmonary artery banding at age 6 days. Weaning from mechanical ventilation was difficult due to massive edema, and a central venous catheter was required because enteral feeding was hampered by the refractory chyle leakage, for which surgery was ineffective. On the evening after central venous catheter replacement was performed, his respiratory condition suddenly deteriorated; cardiac ultrasound revealed that the left pulmonary arterial blood flow had decreased, and enhanced computed tomography demonstrated a left pulmonary embolism (PE), which was identified as the cause of a sudden decrease in the pulmonary blood flow. The patient died due to refractory septic shock at age 5 months. PEs in children, especially patients with cyanotic congenital heart disease, are difficult to diagnose because of their atypical presentation. Current diagnostic criteria are based on adult guidelines, and a few factors in the criteria, such as tachycardia and hypoxia, are difficult to apply for pediatric cyanotic patients with intracardiac or extracardiac right-to-left shunt. In fact, those criteria have lower specificities in children. In conclusion, the present case suggested that a sudden decrease in the pulmonary blood flow can aid the physicians in diagnosing PE in patients with cyanotic congenital heart disease. We need more pediatric cases and evidence of PE in children to make a PE guideline, which is specific to pediatric patients including cyanotic congenital heart disease.

Keywords

pulmonary embolism - decreased pulmonary blood flow - hypoplastic left heart syndrome - cyanotic congenital heart disease - univentricular parallel circulation

Authors' Contributions

K.S. drafted the manuscript; J.M. and O.S. revised the manuscript and made scientific contributions; J.M., H.N., A.S., Y.Y., and O.S. managed the patient; all the authors read and approved the final manuscript.




Publication History

Received: 17 March 2021

Accepted: 14 June 2021

Article published online:
31 July 2021

© 2021. The Author(s). This is an open access article published by Thieme under the terms of the Creative Commons Attribution License, permitting unrestricted use, distribution, and reproduction so long as the original work is properly cited. (https://creativecommons.org/licenses/by/4.0/)

Georg Thieme Verlag KG
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  • References

  • 1 Andrew M, David M, Adams M. et al. Venous thromboembolic complications (VTE) in children: first analyses of the Canadian Registry of VTE. Blood 1994; 83 (05) 1251-1257
    CrossrefPubMedGoogle Scholar
  • 2 Biss TT, Brandão LR, Kahr WH, Chan AK, Williams S. Clinical features and outcome of pulmonary embolism in children. Br J Haematol 2008; 142 (05) 808-818
    CrossrefPubMedGoogle Scholar
  • 3 van Ommen CH, Heijboer H, Büller HR, Hirasing RA, Heijmans HS, Peters M. Venous thromboembolism in childhood: a prospective two-year registry in The Netherlands. J Pediatr 2001; 139 (05) 676-681
    CrossrefPubMedGoogle Scholar
  • 4 Stein PD, Kayali F, Olson RE. Incidence of venous thromboembolism in infants and children: data from the National Hospital Discharge Survey. J Pediatr 2004; 145 (04) 563-565
    CrossrefPubMedGoogle Scholar
  • 5 Dijk FN, Curtin J, Lord D, Fitzgerald DA. Pulmonary embolism in children. Paediatr Respir Rev 2012; 13 (02) 112-122
    PubMedGoogle Scholar
  • 6 Byard RW. Fatal embolic events in childhood. J Forensic Leg Med 2013; 20 (01) 1-5
    CrossrefPubMedGoogle Scholar
  • 7 Patocka C, Nemeth J. Pulmonary embolism in pediatrics. J Emerg Med 2012; 42 (01) 105-116
    CrossrefPubMedGoogle Scholar
  • 8 Male C, Julian JA, Massicotte P, Gent M, Mitchell L. PROTEKT Study Group. Significant association with location of central venous line placement and risk of venous thrombosis in children. Thromb Haemost 2005; 94 (03) 516-521
    Article in Thieme ConnectPubMedGoogle Scholar
  • 9 Brandão LR, Labarque V, Diab Y, Williams S, Manson DE. Pulmonary embolism in children. Semin Thromb Hemost 2011; 37 (07) 772-785
    Article in Thieme ConnectPubMedGoogle Scholar
  • 10 Bernstein D, Coupey S, Schonberg SK. Pulmonary embolism in adolescents. Am J Dis Child 1986; 140 (07) 667-671
    PubMedGoogle Scholar
  • 11 Biss TT, Brandão LR, Kahr WHA, Chan AK, Williams S. Clinical probability score and D-dimer estimation lack utility in the diagnosis of childhood pulmonary embolism. J Thromb Haemost 2009; 7 (10) 1633-1638
    CrossrefPubMedGoogle Scholar
  • 12 Rajpurkar M, Warrier I, Chitlur M. et al. Pulmonary embolism-experience at a single children's hospital. Thromb Res 2007; 119 (06) 699-703
    CrossrefPubMedGoogle Scholar
  • 13 Hennelly KE, Baskin MN, Monuteuax MC. et al. Detection of pulmonary embolism in high-risk children. J Pediatr 2016; 178: 214-218.e3
    CrossrefPubMedGoogle Scholar
  • 14 Ramiz S, Rajpurkar M. Pulmonary embolism in children. Pediatr Clin North Am 2018; 65 (03) 495-507
    CrossrefPubMedGoogle Scholar
  • 15 Rajpurkar M, Williams S, Goldenberg NA. et al. Results of a multinational survey of diagnostic and management practices of thromboembolic pulmonary embolism in children. Thromb Res 2019; 183: 98-105
    CrossrefPubMedGoogle Scholar
  • 16 Zaidi AU, Hutchins KK, Rajpurkar M. Pulmonary embolism in children. Front Pediatr 2017; 5: 170
    CrossrefPubMedGoogle Scholar
  • 17 Henzler T, Barraza Jr JM, Nance Jr JW. et al. CT imaging of acute pulmonary embolism. J Cardiovasc Comput Tomogr 2011; 5 (01) 3-11
    CrossrefPubMedGoogle Scholar
  • 18 Lee EY, Tse SKS, Zurakowski D. et al. Children suspected of having pulmonary embolism: multidetector CT pulmonary angiography–thromboembolic risk factors and implications for appropriate use. Radiology 2012; 262 (01) 242-251
    CrossrefPubMedGoogle Scholar
  • 19 Tang CX, Schoepf UJ, Chowdhury SM, Fox MA, Zhang LJ, Lu GM. Multidetector computed tomography pulmonary angiography in childhood acute pulmonary embolism. Pediatr Radiol 2015; 45 (10) 1431-1439
    CrossrefPubMedGoogle Scholar