Evidence map›Paper›PMID 41886505›Full record

ArticlePloS one2026

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

Moritz Saxenhofer, Daniel Couto, Sandra Mena Perez, Marlies Illi, Justine Brodard, Linet Njue, Ioannis Chanias, Cesare Medri, Gerasimos Tsilimidos, Nicole Hadorn and 20 more

Abstract read
In one paragraph

Article in PloS one, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

0numbers the graph read from it
0cells of the map it votes in
0citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

0 citing papers in PubMed.

No citing paper in PubMed yet.

4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

30 authors.

Moritz SaxenhoferCSL Behring, CSL Biologics Research Centre, Bern, Switzerland.ORCID https://orcid.org/0000-0002-5995-5026
Daniel CoutoCSL Behring, CSL Biologics Research Centre, Bern, Switzerland.ORCID https://orcid.org/0000-0003-0223-1497
Sandra Mena PerezCSL Behring, CSL Biologics Research Centre, Bern, Switzerland.
Marlies IlliCSL Behring, CSL Biologics Research Centre, Bern, Switzerland.
Justine BrodardDepartment of Hematology and Central Hematology Laboratory, Inselspital, Bern University Hospital, Bern, Switzerland.
Linet NjueDepartment of Hematology and Central Hematology Laboratory, Inselspital, Bern University Hospital, Bern, Switzerland.
Ioannis ChaniasDepartment of Hematology and Central Hematology Laboratory, Inselspital, Bern University Hospital, Bern, Switzerland.
Cesare MedriDepartment of Hematology and Central Hematology Laboratory, Inselspital, Bern University Hospital, Bern, Switzerland.
Gerasimos TsilimidosService and Central Laboratory of Haematology, Department of Oncology and Department of Laboratory Medicines and Pathology, Lausanne University Hospital (CHUV) and University of Lausanne (UNIL), Lausanne, Switzerland.
Nicole HadornDepartment of Hematology and Central Hematology Laboratory, Inselspital, Bern University Hospital, Bern, Switzerland.
Christelle ChirliasService and Central Laboratory of Haematology, Department of Oncology and Department of Laboratory Medicines and Pathology, Lausanne University Hospital (CHUV) and University of Lausanne (UNIL), Lausanne, Switzerland.
Ana-Patricia Batista MesquitaService and Central Laboratory of Haematology, Department of Oncology and Department of Laboratory Medicines and Pathology, Lausanne University Hospital (CHUV) and University of Lausanne (UNIL), Lausanne, Switzerland.
Lucas VeutheyService and Central Laboratory of Haematology, Department of Oncology and Department of Laboratory Medicines and Pathology, Lausanne University Hospital (CHUV) and University of Lausanne (UNIL), Lausanne, Switzerland.
Anna SchnellCSL Behring, CSL Biologics Research Centre, Bern, Switzerland.ORCID https://orcid.org/0000-0003-0378-7809
Stefanie GraeterCSL Behring, CSL Biologics Research Centre, Bern, Switzerland.
Gregory J KatoCSL Behring, Clinical Research & Development, King of Prussia, Pennsylvania, United States of America.
Jacqueline AdamCSL Behring, CSL Biologics Research Centre, Bern, Switzerland.
Ramona MerkiOncology and Hematology Department, Aarau Cantonal Hospital, Aarau, Switzerland.
Emmanuel LevratDepartment of Hemato-Oncology, Hôpital Cantonal Fribourg, Fribourg, Switzerland.
Kaveh SamiiClinical Hematology Unit, Geneva University Hospitals (HUG), Geneva, Switzerland.
Mila KeiserDepartment of Hematology and Central Hematology Laboratory, Inselspital, Bern University Hospital, Bern, Switzerland.
Kathrin Susann BieriUniversity of Bern, Department of Clinical Research, Bern, Switzerland.
Alan G HaynesUniversity of Bern, Department of Clinical Research, Bern, Switzerland.
Anne Angelillo-ScherrerDepartment of Hematology and Central Hematology Laboratory, Inselspital, Bern University Hospital, Bern, Switzerland.
Lorenzo AlberioService and Central Laboratory of Haematology, Department of Oncology and Department of Laboratory Medicines and Pathology, Lausanne University Hospital (CHUV) and University of Lausanne (UNIL), Lausanne, Switzerland.
Francesco GrandoniService and Central Laboratory of Haematology, Department of Oncology and Department of Laboratory Medicines and Pathology, Lausanne University Hospital (CHUV) and University of Lausanne (UNIL), Lausanne, Switzerland.
Mathilde GavilletService and Central Laboratory of Haematology, Department of Oncology and Department of Laboratory Medicines and Pathology, Lausanne University Hospital (CHUV) and University of Lausanne (UNIL), Lausanne, Switzerland.
Thomas GentinettaCSL Behring, CSL Biologics Research Centre, Bern, Switzerland.
Alicia RovóDepartment of Hematology and Central Hematology Laboratory, Inselspital, Bern University Hospital, Bern, Switzerland.ORCID https://orcid.org/0000-0002-7757-4753
Alexander SchaubCSL Behring, CSL Biologics Research Centre, Bern, Switzerland.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

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.

Indexed as

Anemia, Sickle CellHemeAdolescentAdultBiomarkersCase-Control StudiesFemaleHemopexinHumansMaleVaso-Occlusive CrisesYoung AdultBiomarkersHemeHemopexin

Identifiers

PMID41886505
PMCPMC13020781

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.