Plasma heme pool compartmentalization is linked to pathophysiology in Sickle Cell Disease

Heme toxicity plays a central role in the pathophysiology of Sickle Cell Disease (SCD), contributing to severe complications such as vaso-occlusion and acute chest syndrome. The continuous release of hemoglobin and heme from increased intravascular hemolysis can exceed the capacity of protective scavenger proteins, leading to heme accumulation in plasma. Interactions with various binding partners result in the formation of different plasma heme species and the compartmentalization of the plasma heme pool. In an observational biomarker study, we used novel bioanalytical assays to quantify plasma heme species in 36 stable-state SCD patients and 36 age, sex, and ethnicity-matched controls. Our results revealed substantially different compartmentalization of plasma heme, despite similar levels of total plasma heme in SCD patients (50 µmol/L) and controls (43 µmol/L). Using a correlation analysis across 85 biomarkers, we examined the association of specific heme species with SCD pathophysiology. Hemopexin-accessible heme (HAH) emerged as a refined indicator of heme burden linked to pathways driving severe SCD complications. A strong inverse correlation was observed between HAH and hemopexin (R = -0.73, p < 0.001), suggesting that hemopexin deficiency contributes to elevated HAH levels. Accurate characterization of clinically relevant plasma heme species and understanding their effects on SCD pathophysiology is essential for the development of new targeted therapies.

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Journal
PLoS ONE
Published
2026-03-26
DOI
https://doi.org/10.1371/journal.pone.0343527
Primary Topic
Hemoglobinopathies and Related Disorders
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article
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article

Plasma heme pool compartmentalization is linked to pathophysiology in Sickle Cell Disease

Alan G. Haynes, Lucas Veuthey, Stefanie Graeter, Francesco Grandoni et al.
PLoS ONE
Hemoglobinopathies and Related Disorders
article

Plasma heme pool compartmentalization is linked to pathophysiology in Sickle Cell Disease

Alan G. Haynes, Lucas Veuthey, Stefanie Graeter, Francesco Grandoni, Emmanuel Levrat, Daniel Couto, Justine Brodard, Linet Njue, Thomas Gentinetta, Alexander Schaub, Cesare Medri, Ioannis Chanias, Gregory J. Kato, Marlies Illi, Mathilde Gavillet, Alicia Rovó, Moritz Saxenhofer, Ramona Merki, Sandra Mena Perez, Nicole Hadorn, Christelle Chirlias, Mila Keiser, Lorenzo Alberio, Kaveh Samii, Kathrin Susann Bieri, Anna Schnell, Jacqueline Adam, Ana-Patricia Batista Mesquita, Gerasimos Tsilimidos, Anne Angelillo-Scherrer
article en

Abstract

Heme toxicity plays a central role in the pathophysiology of Sickle Cell Disease (SCD), contributing to severe complications such as vaso-occlusion and acute chest syndrome. The continuous release of hemoglobin and heme from increased intravascular hemolysis can exceed the capacity of protective scavenger proteins, leading to heme accumulation in plasma. Interactions with various binding partners result in the formation of different plasma heme species and the compartmentalization of the plasma heme pool. In an observational biomarker study, we used novel bioanalytical assays to quantify plasma heme species in 36 stable-state SCD patients and 36 age, sex, and ethnicity-matched controls. Our results revealed substantially different compartmentalization of plasma heme, despite similar levels of total plasma heme in SCD patients (50 µmol/L) and controls (43 µmol/L). Using a correlation analysis across 85 biomarkers, we examined the association of specific heme species with SCD pathophysiology. Hemopexin-accessible heme (HAH) emerged as a refined indicator of heme burden linked to pathways driving severe SCD complications. A strong inverse correlation was observed between HAH and hemopexin (R = -0.73, p < 0.001), suggesting that hemopexin deficiency contributes to elevated HAH levels. Accurate characterization of clinically relevant plasma heme species and understanding their effects on SCD pathophysiology is essential for the development of new targeted therapies.

PLoS ONEVol. 21(3)
University of Bern (CH), University Hospital of Bern (CH), Fribourg Development Agency (CH), University Hospital of Geneva (CH), CSL (United States) (US), Kantonsspital Aarau (CH), CSL (Switzerland) (CH), Geneva College (US), University of Lausanne (CH)
Openalex Percentile: Top 7%
Hemoglobinopathies and Related Disorders
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