Recurrent Donath-Landsteiner hemolytic anemia: a pediatric case report

Transfusion. 2017 Jun;57(6):1401-1406. doi: 10.1111/trf.14032. Epub 2017 Mar 31.

Abstract

Background: Paroxysmal cold hemoglobinuria (PCH) is a form of autoimmune hemolytic anemia caused by the Donath-Landsteiner antibody (D-L antibody). In children, this is typically a transient immune-mediated hemolysis that follows a viral illness and does not recur. Recurrent acute or chronic PCH due to D-L antibody is very rare.

Case report: We have reported a unique case of recurrent PCH in a 5-year-old boy with two acute episodes of hemolysis separated by 21 months of hematologic remission. Each episode was severe requiring red blood cell transfusions, intravenous methylprednisolone, and intravenous immunoglobulin during the second episode. Testing identified recurrence of the D-L antibody with the classic anti-P biphasic hemolysis.

Conclusion: This demonstrates that PCH can be a recurrent disease in the pediatric population (in the absence of syphilis) with the classical D-L antibody.

Publication types

  • Case Reports

MeSH terms

  • Anemia, Hemolytic, Autoimmune / diagnosis*
  • Anemia, Hemolytic, Autoimmune / pathology
  • Anemia, Hemolytic, Autoimmune / therapy
  • Child, Preschool
  • Erythrocyte Transfusion
  • Female
  • Hemoglobinuria, Paroxysmal / diagnosis
  • Hemoglobinuria, Paroxysmal / pathology
  • Hemoglobinuria, Paroxysmal / therapy
  • Hemolysis / physiology
  • Humans
  • Immunoglobulins / administration & dosage
  • Immunoglobulins / therapeutic use
  • Male
  • Methylprednisolone / administration & dosage
  • Methylprednisolone / therapeutic use

Substances

  • Immunoglobulins
  • Methylprednisolone

Supplementary concepts

  • Donath-Landsteiner hemolytic anemia