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.

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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
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article

A Variable Clinical Presentation of Hemoglobin City of Hope

Orly Dgany, Oded Gilad, Shai Izraeli, Orna Steinberg‐Shemer et al.
1 citations
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Hemoglobinopathies and Related Disorders
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article

A Variable Clinical Presentation of Hemoglobin City of Hope

Orly Dgany, Oded Gilad, Shai Izraeli, Orna Steinberg‐Shemer, Tanya Krasnov, Tracie Alison Goldberg, Sharon Noy‐Lotan, Joanne Yacobovich, Vardiella Meiner, Hannah Tamary, Dafna Brik Simon, Dvora Filon
article en
1 citations

Abstract

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.

Clinical GeneticsVol. 107(5)
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)
Openalex Percentile: Top 29%
Hemoglobinopathies and Related Disorders
0.44
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