A Variable Clinical Presentation of Hemoglobin City of Hope
Hemoglobin City of Hope (Hb-COH), NC_000011.9(NM_000518.5):c.208G > A; NP_000509.1:p.(Gly70Ser), has rarely been described. The presentation ranges from asymptomatic heterozygosity to significant anemia in patients carrying an additional pathogenic variant in β-globin. To elucidate the clinical spectrum of Hb-COH, we analyzed 31 individuals carrying the variant, including, for the first time, homozygous individuals. Seven patients who were compound heterozygous for Hb-COH and an additional variant in β-globin, presented with mild-to-severe microcytic anemia and elevated hemoglobin-A2. Three (43%) of these also had elevated fetal hemoglobin, but none required blood transfusions. Seven patients coinherited Hb-COH with an -α<sup>3.7</sup>-deletion (NG_000006.1:g.34247_38050del), their presentation ranged from mild microcytic anemia to normal blood counts. Three homozygous and 14 heterozygous individuals for Hb-COH had normal blood counts. Most Hb-COH alleles whose origin was traceable were from Ashkenazi Jews (70.4%). To conclude, while isolated Hb-COH appears asymptomatic even in the homozygous state, it may cause significant anemia when coinherited with an additional pathogenic variant in β-globin. Understanding the full impact of Hb-COH is crucial for optimal patient management and for genetic counseling.
Authors
- Orly Dgany
- Oded Gilad (ORCID: https://orcid.org/0000-0002-0033-3425)
- Shai Izraeli (ORCID: https://orcid.org/0000-0002-6938-2540)
- Orna Steinberg‐Shemer (ORCID: https://orcid.org/0000-0002-1680-2388)
- Tanya Krasnov
- Tracie Alison Goldberg (ORCID: https://orcid.org/0000-0002-8120-0439)
- Sharon Noy‐Lotan (ORCID: https://orcid.org/0009-0001-0986-3641)
- Joanne Yacobovich (ORCID: https://orcid.org/0000-0003-3763-5694)
- Vardiella Meiner (ORCID: https://orcid.org/0000-0001-6284-9657)
- Hannah Tamary (ORCID: https://orcid.org/0000-0003-4745-3448)
- Dafna Brik Simon (ORCID: https://orcid.org/0000-0002-5216-1135)
- Dvora Filon
Institutions
- Tel Aviv University (IL)
- Hebrew University of Jerusalem (IL)
- Sheba Medical Center (IL)
- Hadassah Medical Center (IL)
- Rabin Medical Center (IL)
- Schneider Children's Medical Center (IL)
- Edmond and Lily Safra Children's Hospital (IL)
Publication Details
- Journal
- Clinical Genetics
- Published
- 2024-12-18
- DOI
- https://doi.org/10.1111/cge.14675
- Citations
- 1
- Primary Topic
- Hemoglobinopathies and Related Disorders
- Type
- article
- Field-Weighted Citation Impact
- 0.44