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Here we describe a patient presenting with elevated IgE levels, atopic eczema, chronic pulmonary aspergillosis, and bone fractures, homozygous for a rare missense variant in the oncostatin M receptor (OSMR) gene. We demonstrate that the patient OSMR V436D variant is deleterious and leads to decreased OSMR surface expression and impaired OSM-induced STAT3 phosphorylation. Blood profiling showed elevated IgE-expressing plasmablasts and peripheral T follicular helper cells and atypical memory B cells. A germinal center model revealed a B cell\u2013extrinsic defect in concordance with a largely fibroblast-confined phenotype. Finally, reconstitution of patient fibroblasts led to functional complementation of the cellular phenotype. We propose OSMR deficiency as a novel genetic etiology of AR-HIES, resembling the clinical and immunological phenotype of STAT3 AD-HIES, but mediating its phenotypic profile mainly in the stromal rather than hematopoietic compartment.<\/jats:p>","DOI":"10.70962\/jhi.20250119","type":"journal-article","created":{"date-parts":[[2026,2,3]],"date-time":"2026-02-03T07:21:50Z","timestamp":1770103310000},"update-policy":"https:\/\/doi.org\/10.70962\/jhi.crossmarkpolicy","source":"Crossref","is-referenced-by-count":2,"title":["Oncostatin M receptor deficiency as a novel candidate genetic cause of autosomal recessive hyper-IgE syndrome"],"prefix":"10.70962","volume":"2","author":[{"ORCID":"https:\/\/orcid.org\/0009-0006-6090-1996","authenticated-orcid":true,"given":"Sisse","family":"Andersen","sequence":"first","affiliation":[{"id":[{"id":"https:\/\/ror.org\/01aj84f44","id-type":"ROR","asserted-by":"publisher"}],"name":"Aarhus University 1 Department of Biomedicine, , Aarhus,","place":["Denmark"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8744-1615","authenticated-orcid":true,"given":"Kristian","family":"Assing","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/00ey0ed83","id-type":"ROR","asserted-by":"publisher"}],"name":"Odense University Hospital 2 Department of Clinical Immunology, , Odense,","place":["Denmark"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0002-9704-5442","authenticated-orcid":true,"given":"Julie","family":"Jensen","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/01aj84f44","id-type":"ROR","asserted-by":"publisher"}],"name":"Aarhus University 1 Department of Biomedicine, , Aarhus,","place":["Denmark"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0853-0893","authenticated-orcid":true,"given":"Line Dahlerup","family":"Rasmussen","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/00ey0ed83","id-type":"ROR","asserted-by":"publisher"}],"name":"Odense University Hospital 3 Department of Infectious Diseases, , Odense,","place":["Denmark"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6382-9906","authenticated-orcid":true,"given":"Christian B.","family":"Laursen","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/00ey0ed83","id-type":"ROR","asserted-by":"publisher"}],"name":"Odense University Hospital 4 Department of Pulmonology, , Odense,","place":["Denmark"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4841-5187","authenticated-orcid":true,"given":"Christoffer D.","family":"Dellgren","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/00ey0ed83","id-type":"ROR","asserted-by":"publisher"}],"name":"Odense University Hospital 5 Department of Clinical genetics, , Odense,","place":["Denmark"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3932-8762","authenticated-orcid":true,"given":"Dani\u00ebla Maria","family":"Hinke","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/01aj84f44","id-type":"ROR","asserted-by":"publisher"}],"name":"Aarhus University 1 Department of Biomedicine, , Aarhus,","place":["Denmark"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5409-045X","authenticated-orcid":true,"given":"S\u00f8ren E.","family":"Degn","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/01aj84f44","id-type":"ROR","asserted-by":"publisher"}],"name":"Aarhus University 1 Department of Biomedicine, , Aarhus,","place":["Denmark"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1853-9704","authenticated-orcid":true,"given":"Trine H.","family":"Mogensen","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/01aj84f44","id-type":"ROR","asserted-by":"publisher"}],"name":"Aarhus University 1 Department of Biomedicine, , Aarhus,","place":["Denmark"]},{"name":"Aarhus University Hospital 6 Department of Infectious Diseases, , Aarhus,","place":["Denmark"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"291","published-online":{"date-parts":[[2026,3,3]]},"reference":[{"key":"2026030315263908400_bib1","doi-asserted-by":"publisher","first-page":"277","DOI":"10.1016\/j.iac.2008.01.005","article-title":"The hyper-IgE syndromes","volume":"28","author":"Freeman","year":"2008","journal-title":"Immunol. 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