{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,14]],"date-time":"2026-10-14T09:48:46Z","timestamp":1791971326810,"version":"4.3.4"},"reference-count":59,"publisher":"Ovid Technologies (Wolters Kluwer Health)","issue":"2","license":[{"start":{"date-parts":[[2026,5,1]],"date-time":"2026-05-01T00:00:00Z","timestamp":1777593600000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0"}],"content-domain":{"domain":["lww.com","ovid.com"],"crossmark-restriction":true},"short-container-title":[],"abstract":"<jats:p>\n                    Inherited bone marrow failure syndromes (IBMFSs) are rare, genetically heterogeneous disorders characterized by BMF with possible increased risk of myeloid malignancies. Recent advances in genomic diagnostic techniques have identified a new IBMFS, including\n                    <jats:italic toggle=\"yes\">Oncostatin M<\/jats:italic>\n                    (\n                    <jats:italic toggle=\"yes\">OSM<\/jats:italic>\n                    ) deficiency, which is an autosomal recessive condition linked to early-onset BMF with possible predisposition to myelodysplastic syndrome (MDS). OSM is a cytokine in the interleukin-6 family and regulates hematopoiesis through the OSM receptor and leukemia inhibitory factor receptor-mediated signaling, activating pathways essential for hematopoietic stem cell progenitor proliferation and differentiation. The reported cases from consanguineous families presented with early-onset cytopenias of variable severity, and some patients developed MDS. In patients with biallelic variants in\n                    <jats:italic toggle=\"yes\">OSM<\/jats:italic>\n                    , hematologic improvement was observed following eltrombopag in two reported patients, while transfusion dependency and high-risk cytogenetics required hematopoietic stem cell transplantation.\n                    <jats:italic toggle=\"yes\">OSM<\/jats:italic>\n                    deficiency is rare; its mechanistic and clinical parallels with other IBMFS underscore its importance. This review outlines the emerging evidence on OSM\u2019s function in hematopoiesis and its deficiency as a newly recognized IBMFS with a potential predisposition to MDS, highlighting the need for early genetic diagnosis and tailored treatment strategies.\n                  <\/jats:p>","DOI":"10.4103\/hemoncstem.hemoncstem-d-26-00005","type":"journal-article","created":{"date-parts":[[2026,10,14]],"date-time":"2026-10-14T09:00:08Z","timestamp":1791968408000},"page":"e0005","update-policy":"https:\/\/doi.org\/10.1097\/lww.0000000000001000","source":"Crossref","is-referenced-by-count":0,"title":["Oncostatin M deficiency: A newly recognized inherited bone marrow failure syndrome with potential predisposition to myeloid malignancy"],"prefix":"10.4103","volume":"19","author":[{"given":"Alfadil","family":"Haroon","sequence":"first","affiliation":[{"name":"Adult Hematology, Stem Cell Transplant and Cellular Therapy Section, Cancer Center of Excellence, King Faisal Specialist Hospital and Research Center, Riyadh, Saudi Arabia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Riad El","family":"Fakih","sequence":"additional","affiliation":[{"name":"Adult Hematology, Stem Cell Transplant and Cellular Therapy Section, Cancer Center of Excellence, King Faisal Specialist Hospital and Research Center, Riyadh, Saudi Arabia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Abdullah H.","family":"Alfalah","sequence":"additional","affiliation":[{"name":"Precision Medicine Laboratory Department, Genomic Medicine Center of Excellence (GMCoE), King Faisal Specialist Hospital and Research Center"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sateesh","family":"Maddirevula","sequence":"additional","affiliation":[{"name":"Precision Medicine Laboratory Department, Genomic Medicine Center of Excellence (GMCoE), King Faisal Specialist Hospital and Research Center"},{"name":"College of Medicine, Alfaisal University, Riyadh, 11533, Saudi Arabia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mahmoud","family":"Aljurf","sequence":"additional","affiliation":[{"name":"Adult Hematology, Stem Cell Transplant and Cellular Therapy Section, Cancer Center of Excellence, King Faisal Specialist Hospital and Research Center, Riyadh, Saudi Arabia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Syed Osman","family":"Ahmed","sequence":"additional","affiliation":[{"name":"Adult Hematology, Stem Cell Transplant and Cellular Therapy Section, Cancer Center of Excellence, King Faisal Specialist Hospital and Research Center, Riyadh, Saudi Arabia"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"276","published-online":{"date-parts":[[2026,5]]},"reference":[{"key":"R1-20261014","doi-asserted-by":"crossref","first-page":"526","DOI":"10.1111\/bjh.14535","article-title":"The genomics of inherited bone marrow failure: from mechanism to the 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