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Clinical Medicine & Research
Volume 2, Number 4 : 228 -232
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© 2004 Marshfield Clinic
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Clinical Overview

Hydrops Fetalis Caused by Homozygous {alpha}-Thalassemia and Rh Antigen Alloimmunization

Report of a Survivor and Literature Review

Divya-Devi Joshi, MD, FAAP

Pediatric Hematology/Oncology, Marshfield Clinic, Marshfield, Wisconsin

H. James Nickerson, MD

Pediatric Hematology/Oncology, Marshfield Clinic, Marshfield, Wisconsin

Michael J. McManus, MD

Pediatric Hematology/Oncology, Marshfield Clinic, Marshfield, Wisconsin

REPRINT REQUESTS: Divya-Devi Joshi, MD, FAAP, Pediatric Hematology/Oncology, Marshfield Clinic, 1000 North Oak Avenue, Marshfield, WI 54449, Telephone: 715-389-3050, Fax: 715-389-4746, Email: joshi.divya-devi{at}marshfieldclinic.org

Hematologic causes of hydrops fetalis include homozygous {alpha}-thalassemia and immune hemolytic anemias. We report the case of a boy with hydrops fetalis who had homozygous {alpha}-thalassemia and alloimmune hemolytic anemia due to anti-E and anti-C blood group antibodies. He received intrauterine red blood cell transfusions and postnatal chronic transfusion with iron chelation therapy. A non-myeloablative sibling stem cell transplant failed. He is now 5 years and 6 months of age, hypothyroid with short stature, but in overall good health. He is one of the oldest reported homozygous {alpha}-thalassemia survivors and, to our knowledge, the only survivor with immune- and nonimmune-induced hydrops fetalis.

Key Words: Hydrops fetalis • alpha-Thalassemia • Blood transfusion, intrauterine • Iron chelating agents • Survival • Blood transfusion/methods






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