{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,20]],"date-time":"2026-07-20T12:20:38Z","timestamp":1784550038275,"version":"3.55.0"},"reference-count":33,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2026,3,31]],"date-time":"2026-03-31T00:00:00Z","timestamp":1774915200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"},{"start":{"date-parts":[[2026,3,31]],"date-time":"2026-03-31T00:00:00Z","timestamp":1774915200000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Br J Haematol"],"published-print":{"date-parts":[[2026,5]]},"abstract":"<jats:title>Summary<\/jats:title>\n                  <jats:p>\n                    Hereditary spherocytosis (HS) is a hereditary haemolytic anaemia, caused by pathogenic variants in genes encoding red blood cell membrane proteins. Osmotic gradient ektacytometry evaluates red cell deformability and hydration and is increasingly used in the diagnosis of HS. We retrospectively evaluated laboratory data from 233 HS patients focusing on osmotic gradient ektacytometry parameters, including two novel parameters, O\n                    <jats:sub>min<\/jats:sub>\n                    \u2010width and O\n                    <jats:sub>max<\/jats:sub>\n                    \u2010width. We found that the maximum elongation index (EI\n                    <jats:sub>max<\/jats:sub>\n                    ), representing the maximum deformability, was decreased in\n                    <jats:italic>SPTB<\/jats:italic>\n                    relative to\n                    <jats:italic>SLC4A1<\/jats:italic>\n                    and\n                    <jats:italic>SPTA1<\/jats:italic>\n                    (0.509 vs. 0.557 and 0.564, both\n                    <jats:italic>p<\/jats:italic>\n                    \u2009&lt;\u20090.01). Hydration was most affected in\n                    <jats:italic>SLC4A1<\/jats:italic>\n                    , with the lowest median O\n                    <jats:sub>hyper<\/jats:sub>\n                    (416\u2009mOsm\/kg). The novel parameters also showed differences: O\n                    <jats:sub>min<\/jats:sub>\n                    \u2010width was higher in\n                    <jats:italic>SPTB<\/jats:italic>\n                    and\n                    <jats:italic>ANK1<\/jats:italic>\n                    compared to\n                    <jats:italic>SLC4A1<\/jats:italic>\n                    and\n                    <jats:italic>SPTA1<\/jats:italic>\n                    (\n                    <jats:italic>p<\/jats:italic>\n                    \u2009&lt;\u20090.05). O\n                    <jats:sub>max<\/jats:sub>\n                    \u2010width was lowest in\n                    <jats:italic>SLC4A1<\/jats:italic>\n                    (92\u2009mOsm\/kg). We found that non\u2010missense variants, opposed to missense variants, were associated with decreased deformability in\n                    <jats:italic>SPTB<\/jats:italic>\n                    and\n                    <jats:italic>ANK1<\/jats:italic>\n                    subgroups as well as with decreased hydration in the\n                    <jats:italic>ANK1<\/jats:italic>\n                    subgroup specifically (O\n                    <jats:sub>hyper<\/jats:sub>\n                    missense 472\u2009mOsm\/kg vs. non\u2010missense 436\u2009mOsm\/kg). Lastly, when classifying disease severity based on reticulocyte production index, we found that O\n                    <jats:sub>min<\/jats:sub>\n                    , EI\n                    <jats:sub>max<\/jats:sub>\n                    , area under the curve (AUC), O\n                    <jats:sub>min<\/jats:sub>\n                    \u2010width and O\n                    <jats:sub>max<\/jats:sub>\n                    \u2010width differed between mild and moderate patients (all\n                    <jats:italic>p<\/jats:italic>\n                    \u2009&lt;\u20090.05). Our findings suggest that osmotic gradient ektacytometry provides additional information on HS pathophysiology and clinical severity.\n                  <\/jats:p>","DOI":"10.1111\/bjh.70457","type":"journal-article","created":{"date-parts":[[2026,3,31]],"date-time":"2026-03-31T08:34:57Z","timestamp":1774946097000},"page":"1785-1796","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["In\u2010depth analysis of osmotic gradient ektacytometry parameters across different genotypes in hereditary spherocytosis"],"prefix":"10.1111","volume":"208","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-6033-2945","authenticated-orcid":false,"given":"Jonathan R. A.","family":"de Wilde","sequence":"first","affiliation":[{"name":"Red Blood Cell Research Group, Central Diagnostic Laboratory\u2010Research, University Medical Centre Utrecht Utrecht University  Utrecht The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6678-2071","authenticated-orcid":false,"given":"Geoffrey Z. L.","family":"Kuppens","sequence":"additional","affiliation":[{"name":"Center for Benign Haematology, Thrombosis and Haemostasis, Van Creveldkliniek, University Medical Centre Utrecht Utrecht University  Utrecht The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maryse E.","family":"Boesveld","sequence":"additional","affiliation":[{"name":"Red Blood Cell Research Group, Central Diagnostic Laboratory\u2010Research, University Medical Centre Utrecht Utrecht University  Utrecht The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4258-3836","authenticated-orcid":false,"given":"Annelies J.","family":"van Vuren","sequence":"additional","affiliation":[{"name":"Department of Internal Medicine Ziekenhuisgroep Twente  Almelo The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2867-2581","authenticated-orcid":false,"given":"Wouter W.","family":"van Solinge","sequence":"additional","affiliation":[{"name":"Red Blood Cell Research Group, Central Diagnostic Laboratory\u2010Research, University Medical Centre Utrecht Utrecht University  Utrecht The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2466-953X","authenticated-orcid":false,"given":"Esm\u00e9","family":"Waanders","sequence":"additional","affiliation":[{"name":"Department of Genetics University Medical Centre Utrecht  Utrecht The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3934-7189","authenticated-orcid":false,"given":"Eduard J.","family":"van Beers","sequence":"additional","affiliation":[{"name":"Center for Benign Haematology, Thrombosis and Haemostasis, Van Creveldkliniek, University Medical Centre Utrecht Utrecht University  Utrecht The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8306-8726","authenticated-orcid":false,"given":"Minke A. E.","family":"Rab","sequence":"additional","affiliation":[{"name":"Red Blood Cell Research Group, Central Diagnostic Laboratory\u2010Research, University Medical Centre Utrecht Utrecht University  Utrecht The Netherlands"},{"name":"Department of Haematology Erasmus Medical Centre Rotterdam  Rotterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9685-1755","authenticated-orcid":false,"given":"Marije","family":"Bartels","sequence":"additional","affiliation":[{"name":"Center for Benign Haematology, Thrombosis and Haemostasis, Van Creveldkliniek, University Medical Centre Utrecht Utrecht University  Utrecht The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8236-8292","authenticated-orcid":false,"given":"Richard","family":"van Wijk","sequence":"additional","affiliation":[{"name":"Red Blood Cell Research Group, Central Diagnostic Laboratory\u2010Research, 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