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. 2015 Feb 13:15:3.
doi: 10.1186/s12878-015-0023-7. eCollection 2015.

Case report: paroxysmal cold hemoglobinuria presenting during pregnancy

Affiliations

Case report: paroxysmal cold hemoglobinuria presenting during pregnancy

Andrea O Akpoguma et al. BMC Hematol. .

Abstract

Background: Paroxysmal cold hemoglobinuria is caused by a biphasic IgG autoantibody that triggers complement-mediated intravascular hemolysis. Paroxysmal cold hemoglobinuria has not previously been reported to occur in association with pregnancy.

Case presentation: We report a case of an 18 year old female who presented in early pregnancy with acute hemolytic anemia and a positive Donath-Landsteiner antibody test. She was diagnosed with paroxysmal cold hemoglobinuria and treated supportively. Her hemolysis resolved within 6 weeks. Because maternal IgG autoantibodies can cross the placenta, the patient was monitored closely throughout her pregnancy for recurrence. The outcome of the pregnancy was successful, with no evidence of neonatal anemia or hemolysis.

Conclusion: This patient had a classic presentation of paroxysmal cold hemoglobinuria with rapid onset of hemolytic anemia that resolved spontaneously. To our knowledge, this is the first report of paroxysmal cold hemoglobinuria presenting during pregnancy.

Keywords: Donath-Landsteiner antibody; Hemolytic anemia; Paroxysmal cold hemoglobinuria; Pregnancy.

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Figures

Figure 1
Figure 1
Donath-Landsteiner antibody test. Hemolysis of donor erythrocytes was observed when patient serum (tube 7) or a mixture of patient serum and normal serum (to provide complement; tube 9) was incubated at 0°C for 30 minutes, followed by 37°C for 60 minutes. Little or no hemolysis was seen when patient serum or mixed serum was incubated at 0°C for 90 minutes (tubes 1 and 3) or 37°C for 90 minutes (tubes 4 and 6). No hemolysis was observed in control tubes containing normal serum only (tubes 2, 5, 8).
Figure 2
Figure 2
Time course of hemoglobin and lactic acid dehydrogenase (LDH) values.

References

    1. Bass GF, Tuscano ET, Tuscano JM. Diagnosis and classification of autoimmune hemolytic anemia. Autoimmun Rev. 2014;13(4–5):560–4. doi: 10.1016/j.autrev.2013.11.010. - DOI - PubMed
    1. Sokol RJ, Booker DJ, Stamps R. Paroxysmal cold hemoglobinuria and the elusive Donath-Landsteiner antibody. Immunohematology. 1998;14(3):109–12. - PubMed
    1. Sokol RJ, Hewitt S. Autoimmune hemolysis: a critical review. Crit Rev Oncol Hematol. 1985;4(2):125–54. doi: 10.1016/S1040-8428(85)80013-5. - DOI - PubMed
    1. Sokol RJ, Hewitt S, Booker DJ. Erythrocyte autoantibodies, autoimmune haemolysis, and myelodysplastic syndromes. J Clin Pathol. 1989;42(10):1088–91. doi: 10.1136/jcp.42.10.1088. - DOI - PMC - PubMed
    1. Sanford KW, Roseff SD. Detection and significance of Donath-Landsteiner antibodies in a 5-year-old female presenting with hemolytic anemia. Lab Medicine. 2010;41(4):209–12. doi: 10.1309/LM1E9Z2QZEREPAI. - DOI

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