Simultaneous Pulmonary Embolism and Ischemic Stroke in a Patient with Cor Biloculare after Glenn Anastomosis

Authors

  • Sipei Pan Department of Neurology, the First Affiliated Hospital of Wenzhou Medical University, Wenzhou, China
  • Zhangyang Wang Department of Neurology, Huashan Hospital, Fudan University, Shanghai, China
  • Xiang Wei Department of Neurology, the First Affiliated Hospital of Wenzhou Medical University, Wenzhou, China
  • Hai Tang Department of Neurology, the Affiliated Hospital of Xuzhou Medical University, Xuzhou, China
  • Zhengzheng Li Department of Neurology, the First Affiliated Hospital of Wenzhou Medical University, Wenzhou, China
  • Yang Zhang Department of Neurology, the First Affiliated Hospital of Wenzhou Medical University, Wenzhou, China
  • Saijun Zhou Department of Neurology, the First Affiliated Hospital of Wenzhou Medical University, Wenzhou, China
  • Nan Zhang Department of Neurology, the First Affiliated Hospital of Wenzhou Medical University, Wenzhou, China

DOI:

https://doi.org/10.1532/hsf.2054

Keywords:

Cor biloculare, Glenn anastomosis, thrombosis

Abstract

Background: Cor biloculare, two-chambered heart due to the absence of atrial and ventricular septa, is a rare congenital heart anomaly. For Cor biloculare and other cardiac defects with single ventricle physiology, Glenn anastomosis has been developed as a palliative procedure. Thrombosis secondary to Glenn anastomosis in the patient with Cor biloculare could pose a serious threat to the survival, and has never been reported before.

Case report: We report the case of a 27-year-old patient, with past history of Glenn anastomosis that was performed 7 years ago for the palliation of Cor biloculare. She presented with pulmonary embolism and ischemic stroke simultaneously at 7 days after Cesarean section. Due to her critical status, systemic anticoagulation with low-molecular-weight heparin was started immediately, followed by lifelong warfarin therapy. Pulmonary embolism regressed and neurological symptoms were considerably diminished after the anticoagulation treatment.

Conclusion: This case illuminates the potential risk of thrombotic events in this patient cohort and demonstrates that anticoagulation therapy is an effective, secure, and appropriate for the management of this disease.

References

Bonnel AR. 2015. Thrombolytics for late superior caval vein thrombus in a patient with tricuspid atresia and single-lung Glenn anastomosis. Cardiol in Young 1:1-5.

Forbes TJ, Rosenthal GL, Jr RG, Ott DA, Feltes TF. 1997. Risk factors for life-threatening cavopulmonary thrombosis in patients undergoing bidirectional superior cavopulmonary shunt: an exploratory study. Am Heart J 134:865-71.

Gunta S, Kamath S. 2012. A case of pulmonary embolism and stroke in a 16-year-old girl. WMJ 111:58-60.

Iannoli ED, Eaton MP, Shapiro JR. 2005. Bidirectional glenn shunt surgery using lepirudin anticoagulation in an infant with heparin-induced thrombocytopenia with thrombosis. Anesthes Analg 101:74-6, table of contents.

Jonas RA. 1994. Indications and timing for the bidirectional Glenn shunt versus the fenestrated Fontan circulation. J Thorac Cardiovasc Surg 108:522-4.

Kopf GS, Laks H, Stansel HC, Hellenbrand WE, Kleinman CS, Talner NS. 1990. Thirty-year follow-up of superior vena cava-pulmonary artery (Glenn) shunts. J Thorac Cardiovasc Surg 100:670-1.

Koutlas TC, Harrison JK, Bashore TM, O'Laughlin MP, Tripp ME, Gaynor JW. 1996. Late conduit occlusion after modified Fontan procedure with classic Glenn shunt. Ann Thorac Surg 62:261-2.

Kozicka UA, Kowalski M, Hoffman P. 2015. Thrombosis within Glenn anastomosis. J Rare Cardiovasc Dis 2:89-91.

Liu M, Menzoian JO. 2015. A patient with multiple paradoxical emboli. J Vasc Surg 63:1085-7.

Odegard KC, Jr MG, Dinardo JA, et al. 2002. Coagulation abnormalities in patients with single-ventricle physiology precede the Fontan procedure J Thorac Cardiovasc Surg 123:459-65.

Omar HR, Huang C, Miller JH, Mangar D, Kabemba A, Camporesi EM. 2013. Simultaneous pulmonary embolism and cerebrovascular stroke. Herz 38:884-6.

Oski JA, Canter CE, Spray TL, Kan JS, Cameron DE, Murphy AM. 1996. Embolic stroke after ligation of the pulmonary artery in patients with functional single ventricle. Am Heart J 132:836-40.

Pennington DG, Nouri S, Ho J, et al. 1981. Glenn shunt: long-term results and current role in congenital heart operations. Ann Thorac Surg 31:532-9.

Ravn HB, Hjortdal VE, Stenbog EV, et al. 2001. Increased platelet reactivity and significant changes in coagulation markers after cavopulmonary connection. Heart 85:61-5.

Temple WW, Bloor CM. 1981. Cor biloculare and associated malformations. Virchows Archiv 391:345-56.

Wang YB, Fu XH, Gu XS. 2014. Dextrocardia with cor biloculare in a 44 Year old woman. J Case Reports 4:245-8.

Wiszniewska M, Bytowska A. 2013. Ischemic stroke due to postpartum angiopathy complicated by pulmonary embolism with favorable outcome. Acta Clinica Croatica 52:267.

Yuan SM, Jing H. 2009. Palliative procedures for congenital heart defects. Archiv Cardiovas Dis 102:549-57.

Published

2019-05-08

How to Cite

Pan, S., Wang, Z., Wei, X., Tang, H., Li, Z., Zhang, Y., Zhou, S., & Zhang, N. (2019). Simultaneous Pulmonary Embolism and Ischemic Stroke in a Patient with Cor Biloculare after Glenn Anastomosis. The Heart Surgery Forum, 22(3), E180-E182. https://doi.org/10.1532/hsf.2054

Issue

Vol. 22 No. 3 (2019)

Section

Article
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