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Published in: Journal of Medical Case Reports 1/2018

Open Access 01-12-2018 | Case report

Two potentially lethal conditions of probable immune origin occurring in a pregnant woman: a case report

Authors: H. M. Senanayake, M. Patabendige

Published in: Journal of Medical Case Reports | Issue 1/2018

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Abstract

Background

Thrombotic thrombocytopenic purpura and peripartum cardiomyopathy are potentially lethal complications of pregnancy. We describe a case in which both of these developed in the same patient. The etiologies of both conditions remain uncertain, but they share immune hyperreactivity as a possible cause.

Case presentation

A 33-year-old Lankan primigravida gave birth at 38 weeks of gestation by cesarean section when she presented with right-sided abdominal pain and a provisional diagnosis of appendicitis. Her pain persisted postoperatively, and on the second postoperative day, she physicaly collapsed suddenly with abdominal distention. Immediate laparotomy revealed generalized oozing from the peritoneum resulting in hemoperitoneum and intestinal hemorrhage. Her laboratory reports showed microangiopathic hemolytic anemia and thrombocytopenia. She also had elevated liver enzyme, lactate dehydrogenase, and creatinine concentrations. A diagnosis of thrombotic thrombocytopenic purpura was made. After a steady recovery, she was discharged from the hospital on the 16th postoperative day, but 12 hours later, she was readmitted with acute-onset progressively worsening shortness of breath. Echocardiography confirmed peripartum cardiomyopathy. She was treated with a bromocriptine and heart failure regimen. At 6 weeks postpartum, her laboratory test results and cardiac function had improved.

Conclusions

A possible autoimmune association might have caused both conditions in our patient. This case report serves as a warning message that pregnant women with one possible condition with autoimmune association could go on to develop other similar conditions.
Literature
1.
go back to reference Gaddam S, Pablani L, Chainani V, Kavuda RR. Complete recovery of ischemic cardiomyopathy from thrombotic thrombocytopenic purpura. Clin Med Insights Cardiol. 2011;5:29–33.CrossRefPubMedPubMedCentral Gaddam S, Pablani L, Chainani V, Kavuda RR. Complete recovery of ischemic cardiomyopathy from thrombotic thrombocytopenic purpura. Clin Med Insights Cardiol. 2011;5:29–33.CrossRefPubMedPubMedCentral
2.
go back to reference Rock GA, Shumak KH, Buskard NA, et al. Comparison of plasma exchange with plasma infusion in the treatment of thrombotic thrombocytopenic purpura. N Engl J Med. 1991;325:393–7.CrossRefPubMed Rock GA, Shumak KH, Buskard NA, et al. Comparison of plasma exchange with plasma infusion in the treatment of thrombotic thrombocytopenic purpura. N Engl J Med. 1991;325:393–7.CrossRefPubMed
3.
go back to reference Sadler JE, Moake JL, Miyata T, George JN. Recent advances in thrombotic thrombocytopenic purpura. In: Hematology Am Soc Hematol Educ Program; 2004. p. 407–23. Sadler JE, Moake JL, Miyata T, George JN. Recent advances in thrombotic thrombocytopenic purpura. In: Hematology Am Soc Hematol Educ Program; 2004. p. 407–23.
4.
go back to reference Blumberg N, Heal JM, Gettings KF, Phipps RP, Masel D, Refaai MA, et al. An association between decreased cardiopulmonary complications transfusion-related acute lung injury and transfusion-associated circulatory overload and implementation of universal leukoreduction of blood transfusions. Transfusion. 2010;50:2738–44.CrossRefPubMedPubMedCentral Blumberg N, Heal JM, Gettings KF, Phipps RP, Masel D, Refaai MA, et al. An association between decreased cardiopulmonary complications transfusion-related acute lung injury and transfusion-associated circulatory overload and implementation of universal leukoreduction of blood transfusions. Transfusion. 2010;50:2738–44.CrossRefPubMedPubMedCentral
5.
go back to reference James N, George MD. Thrombotic thrombocytopenic purpura. N Engl J Med. 2006;354:1927–35.CrossRef James N, George MD. Thrombotic thrombocytopenic purpura. N Engl J Med. 2006;354:1927–35.CrossRef
6.
go back to reference Scully M, Hunt BJ, Benjamin S, et al. Guidelines on the diagnosis and management of thrombotic thrombocytopenic purpura and other thrombotic microangiopathies. Br J Haematol. 2012;158:323–35.CrossRefPubMed Scully M, Hunt BJ, Benjamin S, et al. Guidelines on the diagnosis and management of thrombotic thrombocytopenic purpura and other thrombotic microangiopathies. Br J Haematol. 2012;158:323–35.CrossRefPubMed
7.
go back to reference Crowther MA, Heddle N, Hayward CPM, et al. Splenectomy done during hematologic remission to prevent relapse in patients with thrombotic thrombocytopenic purpura. Ann Intern Med. 1996;125:294–6.CrossRefPubMed Crowther MA, Heddle N, Hayward CPM, et al. Splenectomy done during hematologic remission to prevent relapse in patients with thrombotic thrombocytopenic purpura. Ann Intern Med. 1996;125:294–6.CrossRefPubMed
8.
go back to reference Ono T, Mimuro J, Madoiwa S, et al. Severe secondary deficiency of von Willebrand factor-cleaving protease (ADAMTS13) in patients with sepsis-induced disseminated intravascular coagulation: its correlation with development of renal failure. Blood. 2006;107:528–34.CrossRefPubMed Ono T, Mimuro J, Madoiwa S, et al. Severe secondary deficiency of von Willebrand factor-cleaving protease (ADAMTS13) in patients with sepsis-induced disseminated intravascular coagulation: its correlation with development of renal failure. Blood. 2006;107:528–34.CrossRefPubMed
9.
go back to reference Manea M, Karpman D. Molecular basis of ADAMTS13 dysfunction in thrombotic thrombocytopenic purpura. Pediatr Nephrol. 2009;24(3):447–58.CrossRefPubMed Manea M, Karpman D. Molecular basis of ADAMTS13 dysfunction in thrombotic thrombocytopenic purpura. Pediatr Nephrol. 2009;24(3):447–58.CrossRefPubMed
10.
go back to reference Feys HB, Liu F, Dong N, Pareyn I, Vauterin S, et al. ADAMTS-13 plasma level determination uncovers antigen absence in acquired thrombotic thrombocytopenic purpura and ethnic differences. J Thromb Haemost. 2006;4(5):955–62.CrossRefPubMed Feys HB, Liu F, Dong N, Pareyn I, Vauterin S, et al. ADAMTS-13 plasma level determination uncovers antigen absence in acquired thrombotic thrombocytopenic purpura and ethnic differences. J Thromb Haemost. 2006;4(5):955–62.CrossRefPubMed
11.
go back to reference Coppo P, Bengoufa D, Veyradier A, et al. Severe ADAMTS13 deficiency in adult idiopathic thrombotic microangiopathies defines a subset of patients characterized by various autoimmune manifestations, lower platelet count, and mild renal involvement. Medicine (Baltimore). 2004;83:233–44.CrossRef Coppo P, Bengoufa D, Veyradier A, et al. Severe ADAMTS13 deficiency in adult idiopathic thrombotic microangiopathies defines a subset of patients characterized by various autoimmune manifestations, lower platelet count, and mild renal involvement. Medicine (Baltimore). 2004;83:233–44.CrossRef
12.
go back to reference Fu LX, Magnusson Y, Bergh CH, Liljeqvist JA, Waagstein F, et al. Localization of a functional autoimmune epitope on the muscarinic acetylcholine receptor-2 in patients with idiopathic dilated cardiomyopathy. J Clin Invest. 1993;91:1964–8.CrossRefPubMedPubMedCentral Fu LX, Magnusson Y, Bergh CH, Liljeqvist JA, Waagstein F, et al. Localization of a functional autoimmune epitope on the muscarinic acetylcholine receptor-2 in patients with idiopathic dilated cardiomyopathy. J Clin Invest. 1993;91:1964–8.CrossRefPubMedPubMedCentral
13.
go back to reference Sterin-Borda L, Gorelik G, Borda E. Chagasic IgG bingding with cardiac muscarinic cholinergic receptors modifies cholinergic-mediated cellular transmembrane signals. Clin Immunol Immunopathol. 1991;61:389–97.CrossRef Sterin-Borda L, Gorelik G, Borda E. Chagasic IgG bingding with cardiac muscarinic cholinergic receptors modifies cholinergic-mediated cellular transmembrane signals. Clin Immunol Immunopathol. 1991;61:389–97.CrossRef
14.
go back to reference Selle T, Renger I, Labidi S, Bultmann I, Hilfiker-Kleiner D. Reviewing peripartum cardiomyopathy: current state of knowledge. Future Cardiol. 2009;5:175–89.CrossRefPubMed Selle T, Renger I, Labidi S, Bultmann I, Hilfiker-Kleiner D. Reviewing peripartum cardiomyopathy: current state of knowledge. Future Cardiol. 2009;5:175–89.CrossRefPubMed
15.
go back to reference Liu J, Wang Y, Chen M, Zhao W, Wang X, Wang H, Zhang Z, Zhang J, Xu L, Chen J, Yang X, Zhang L. The correlation between peripartum cardiomyopathy and autoantibodies against cardiovascular receptors. PLoS One. 2014;9(1):e86770.CrossRefPubMedPubMedCentral Liu J, Wang Y, Chen M, Zhao W, Wang X, Wang H, Zhang Z, Zhang J, Xu L, Chen J, Yang X, Zhang L. The correlation between peripartum cardiomyopathy and autoantibodies against cardiovascular receptors. PLoS One. 2014;9(1):e86770.CrossRefPubMedPubMedCentral
16.
go back to reference Ansari AA, Fett JD, Carraway RE, Mayne AE, Onlamoon N, Sundstrom JB. Autoimmune mechanisms as the basis for human peripartum cardiomyopathy. Clin Rev Allergy Immunol. 2002;23(3):301–24.CrossRefPubMed Ansari AA, Fett JD, Carraway RE, Mayne AE, Onlamoon N, Sundstrom JB. Autoimmune mechanisms as the basis for human peripartum cardiomyopathy. Clin Rev Allergy Immunol. 2002;23(3):301–24.CrossRefPubMed
17.
go back to reference Sliwa K, Hilfiker-Kleiner D, Petrie MC, Mebazaa A. Current state of knowledge on aetiology, diagnosis, management, and therapy of peripartum cardiomyopathy: a position statement from the Heart Failure Association of the European Society of Cardiology Working Group on peripartum cardiomyopathy. Eur J Heart Fail. 2010;12(8):767–78.CrossRefPubMed Sliwa K, Hilfiker-Kleiner D, Petrie MC, Mebazaa A. Current state of knowledge on aetiology, diagnosis, management, and therapy of peripartum cardiomyopathy: a position statement from the Heart Failure Association of the European Society of Cardiology Working Group on peripartum cardiomyopathy. Eur J Heart Fail. 2010;12(8):767–78.CrossRefPubMed
18.
19.
20.
go back to reference Cosmai EM, Puzis L, Tsai HM, Lian EC. Thrombocytopenic purpura and cardiomyopathy in pregnancy reversed by combined plasma exchange and infusion. Eur J Haematol. 2002;68(4):239–42.CrossRefPubMed Cosmai EM, Puzis L, Tsai HM, Lian EC. Thrombocytopenic purpura and cardiomyopathy in pregnancy reversed by combined plasma exchange and infusion. Eur J Haematol. 2002;68(4):239–42.CrossRefPubMed
Metadata
Title
Two potentially lethal conditions of probable immune origin occurring in a pregnant woman: a case report
Authors
H. M. Senanayake
M. Patabendige
Publication date
01-12-2018
Publisher
BioMed Central
Published in
Journal of Medical Case Reports / Issue 1/2018
Electronic ISSN: 1752-1947
DOI
https://doi.org/10.1186/s13256-018-1701-4

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