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A 2-month-old boy with hemolytic anemia and reticulocytopenia following intravenous immunoglobulin therapy for Kawasaki disease: a case report and literature review

  • Kim, Na Yeon (Department of Pediatrics, CHA Bundang Medical Center, CHA University) ;
  • Kim, Joon Hwan (Department of Pediatrics, CHA Bundang Medical Center, CHA University) ;
  • Park, Jin Suk (Department of Pediatrics, CHA Bundang Medical Center, CHA University) ;
  • Kim, Soo Hyun (Department of Obstetrics and Gynecology, CHA Gangnam Medical Center, CHA University) ;
  • Cho, Yeon Kyung (Department of Obstetrics and Gynecology, CHA Gangnam Medical Center, CHA University) ;
  • Cha, Dong Hyun (Department of Obstetrics and Gynecology, CHA Gangnam Medical Center, CHA University) ;
  • Kim, Ki Eun (Department of Pediatrics, CHA Gangnam Medical Center, CHA University) ;
  • Kang, Myung Suh (Department of Laboratory Medicine, CHA Bundang Medical Center, CHA University) ;
  • Lim, Kyung Ah (Department of Pediatrics, CHA Gangnam Medical Center, CHA University) ;
  • Sheen, Youn Ho (Department of Pediatrics, CHA Gangnam Medical Center, CHA University)
  • 투고 : 2014.07.22
  • 심사 : 2014.10.03
  • 발행 : 2016.11.15

초록

Herein, we report a rare case of hemolytic anemia with reticulocytopenia following intravenous immunoglobulin therapy in a young infant treated for Kawasaki disease. A 2-month-old boy presented with fever lasting 3 days, conjunctival injection, strawberry tongue, erythematous edema of the hands, and macular rash, symptoms and signs suggestive of incomplete Kawasaki disease. His fever resolved 8 days after treatment with aspirin and high dose infusion of intravenous immunoglobulin. The hemoglobin and hematocrit decreased from 9.7 g/dL and 27.1% to 7.4 g/dL and 21.3%, respectively. The patient had normocytic hypochromic anemia with anisocytosis, poikilocytosis, immature neutrophils, and nucleated red blood cells. The direct antiglobulin test result was positive, and the reticulocyte count was 1.39%. The patient had an uneventful recovery. However, reticulocytopenia persisted 1 month after discharge.

키워드

참고문헌

  1. Newburger JW, Fulton DR. Kawasaki disease. Curr Opin Pediatr 2004;16:508-14. https://doi.org/10.1097/01.mop.0000137796.23813.64
  2. Gulhan B, Kesici S, Beken S, Cilsal E, Kale G, Alehan D, et al. Varying clinical features of Turkish Kawasaki disease patients. Turk J Pediatr 2012;54:1-6.
  3. Berard R, Whittemore B, Scuccimarri R. Hemolytic anemia following intravenous immunoglobulin therapy in patients treated for Kawasaki disease: a report of 4 cases. Pediatr Rheumatol Online J 2012;10:10. https://doi.org/10.1186/1546-0096-10-10
  4. Takahashi M, Mason W, Lewis AB. Regression of coronary aneurysms in patients with Kawasaki syndrome. Circulation 1987;75:387-94. https://doi.org/10.1161/01.CIR.75.2.387
  5. Gordon DJ, Sloan SR, de Jong JL. A pediatric case series of acute hemolysis after administration of intravenous immunoglobulin. Am J Hematol 2009;84:771-2. https://doi.org/10.1002/ajh.21544
  6. Conley CL, Lippman SM, Ness PM, Petz LD, Branch DR, Gallagher MT. Autoimmune hemolytic anemia with reticulocytopenia and erythroid marrow. N Engl J Med 1982;306:281-6. https://doi.org/10.1056/NEJM198202043060507
  7. Liesveld JL, Rowe JM, Lichtman MA. Variability of the erythropoietic response in autoimmune hemolytic anemia: analysis of 109 cases. Blood 1987;69:820-6.
  8. Conley CL, Lippman SM, Ness P. Autoimmune hemolytic anemia with reticulocytopenia. A medical emergency. JAMA 1980;244:1688-90. https://doi.org/10.1001/jama.1980.03310150024022
  9. Van De Loosdrecht AA, Hendriks DW, Blom NR, Smit JW, De Wolf JT, Vellenga E. Excessive apoptosis of bone marrow erythroblasts in a patient with autoimmune haemolytic anaemia with reticulocytopenia. Br J Haematol 2000;108:313-5. https://doi.org/10.1046/j.1365-2141.2000.01867.x
  10. Mangan KF, Besa EC, Shadduck RK, Tedrow H, Ray PK. Demonstration of two distinct antibodies in autoimmune hemolytic anemia with reticulocytopenia and red cell aplasia. Exp Hematol 1984;12:788-93.