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Peer-reviewedCC BY-NC 4.0Neonatology

Hemolytic Jaundice and Anemia With Methemoglobinemia in Dizygotic Newborn Twins With Hb Evans: Case Report

  • Alina Saldarriaga1
  • Bailey B. Zeiler2
  • Rachel Brennan3
  • Timothy M. Bahr2,4
  • Robin K. Ohls2
  • Patrick G. Gallagher5
  • Robert D. Christensen2,4
5 affiliations
  1. 1Division of Neonatology, Department of Pediatrics, Logan Health, Kalispell, MT, USA
  2. 2Division of Neonatology, Department of Pediatrics, University of Utah Health, Salt Lake City, UT, USA
  3. 3Division of Pediatric Hematology/Oncology, Logan Health, Kalispell, MT, USA
  4. 4Women and Newborns Research, Intermountain Health, Murray, UT, USA
  5. 5Department of Pediatrics, Center for Perinatal Research, Nationwide Children’s Hospital, Ohio State University, Columbus, OH, USA
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Abstract

Introduction: It is highly unusual for a newborn infant to present in the first days after birth with cyanosis secondary to methemoglobinemia and with hemolytic anemia and jaundice. This combination suggests the possibility of an unstable hemoglobin variant.

Case Presentation: We describe dizygotic twins, both with this presentation, where whole-genome sequencing identified hemoglobin Evans (a point missense substitution in HBA2, the alpha2-globin gene) and found no other co-inherited variants known to be associated with neonatal methemoglobinemia, jaundice, or anemia. Herein, we report on four affected members of this family. We also review the 6 previously published reports of Hb Evans. We explain the pathogenesis and review available treatments.

Conclusion: Hb Evans might be more common than the small number of published cases would indicate. The presentation of neonatal cyanosis secondary to methemoglobinemia accompanied by hemolytic anemia and jaundice should raise the possibility of an unstable hemoglobin variant such as Hb Evans.

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Cite

BibTeXRIS
BibTeX
@article{saldarriaga2026hemolytic,
  title = {{Hemolytic Jaundice and Anemia With Methemoglobinemia in Dizygotic Newborn Twins With Hb Evans: Case Report}},
  author = {Alina Saldarriaga and Bailey B. Zeiler and Rachel Brennan and Timothy M. Bahr and Robin K. Ohls and Patrick G. Gallagher and Robert D. Christensen},
  journal = {Neonatology},
  year = {2026},
  month = oct,
  doi = {10.1159/neo/adsag021},
  url = {https://doi.org/10.1159/neo/adsag021},
  note = {CC BY-NC 4.0},
}
RIS
TY  - JOUR
TI  - Hemolytic Jaundice and Anemia With Methemoglobinemia in Dizygotic Newborn Twins With Hb Evans: Case Report
AU  - Alina Saldarriaga
AU  - Bailey B. Zeiler
AU  - Rachel Brennan
AU  - Timothy M. Bahr
AU  - Robin K. Ohls
AU  - Patrick G. Gallagher
AU  - Robert D. Christensen
PY  - 2026
DA  - 2026/10/10
JO  - Neonatology
DO  - 10.1159/neo/adsag021
UR  - https://doi.org/10.1159/neo/adsag021
AB  - Introduction: It is highly unusual for a newborn infant to present in the first days after birth with cyanosis secondary to methemoglobinemia and with hemolytic anemia and jaundice. This combination suggests the possibility of an unstable hemoglobin variant.  Case Presentation: We describe dizygotic twins, both with this presentation, where whole-genome sequencing identified hemoglobin Evans (a point missense substitution in HBA2, the alpha2-globin gene) and found no other co-inherited variants known to be associated with neonatal methemoglobinemia, jaundice, or anemia. Herein, we report on four affected members of this family. We also review the 6 previously published reports of Hb Evans. We explain the pathogenesis and review available treatments.  Conclusion: Hb Evans might be more common than the small number of published cases would indicate. The presentation of neonatal cyanosis secondary to methemoglobinemia accompanied by hemolytic anemia and jaundice should raise the possibility of an unstable hemoglobin variant such as Hb Evans.
SN  - 1661-7800
SN  - 1661-7819
ER  -