{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,1]],"date-time":"2026-09-01T23:52:39Z","timestamp":1788306759430,"version":"build-2803163510"},"reference-count":46,"publisher":"MDPI AG","issue":"4","license":[{"start":{"date-parts":[[2025,2,18]],"date-time":"2025-02-18T00:00:00Z","timestamp":1739836800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["IJMS"],"abstract":"<jats:p>Schimke immuno-osseous dysplasia (SIOD) is a hereditary autosomal-recessive multi-system disorder with early mortality. It has variable clinical presentations, mainly characterised by disproportional short stature, steroid-resistant nephrotic syndrome, spondyloepiphyseal dysplasia, and T-cell immunodeficiency. In the majority of cases, SIOD is caused by pathogenic sequence variants (PSVs) in the SMARCAL1 gene that encodes protein involved in chromatin remodelling. SIOD is an ultra-rare condition, with an incidence of ~1 per 1\u20133 million live births; data on its genetic and clinical features are scarce. We conducted a retrospective study of 21 paediatric patients with SIOD diagnosed in our centre during the years 2003\u20132023. The most common extra-renal clinical features were short stature, osseous dysplasia, multiple stigmas, and leukopenia. Proteinuria of varying severity was observed in 16 cases. The five-year overall survival rate (OS) was 89% (95% CI 77\u2013100%), and the ten-year OS was 10%. Next-generation sequencing (NGS) revealed the following PSVs in SMARCAL1 in 19 patients: c.355_500del, c.2542G&gt;T, c.2290C&gt;T, c.2562del, c.2533_2534del, c.1582A&gt;C, c.1933C&gt;T, c.1010T&gt;C, c.1736C&gt;T, c.2070dup, c.2551A&gt;T, c.2149_2150dup, c.939delC, and c.1451T&gt;A; the most common was c.2542G&gt;T, resulting in premature translation termination (p.E848*), and it was found in 14 patients either in a homozygous (four patients) or compound-heterozygous (10 patients) state. According to microsatellite analysis, it is a \u201cfounder mutation\u201d in Russia.<\/jats:p>","DOI":"10.3390\/ijms26041744","type":"journal-article","created":{"date-parts":[[2025,2,18]],"date-time":"2025-02-18T12:16:37Z","timestamp":1739880997000},"page":"1744","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":4,"title":["Genetic and Clinical Features of Schimke Immuno-Osseous Dysplasia: Single-Centre Retrospective Study of 21 Unrelated Paediatric Patients over a Period of 20 Years"],"prefix":"10.3390","volume":"26","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-1615-2044","authenticated-orcid":false,"given":"Anastasiia","family":"Milovanova","sequence":"first","affiliation":[{"name":"Federal State Autonomous Institution \u201cNational Medical Research Center of Children\u2019s Health\u201d of the Ministry of Health of the Russian Federation, 119991 Moscow, Russia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3131-331X","authenticated-orcid":false,"given":"Petr","family":"Ananin","sequence":"additional","affiliation":[{"name":"Federal State Autonomous Institution \u201cNational Medical Research Center of Children\u2019s Health\u201d of the Ministry of Health of the Russian Federation, 119991 Moscow, Russia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tatiana","family":"Vashurina","sequence":"additional","affiliation":[{"name":"Federal State Autonomous Institution \u201cNational Medical Research Center of Children\u2019s Health\u201d of the Ministry of Health of the Russian Federation, 119991 Moscow, Russia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Olga","family":"Zrobok","sequence":"additional","affiliation":[{"name":"Federal State Autonomous Institution \u201cNational Medical Research Center of Children\u2019s Health\u201d of the Ministry of Health of the Russian Federation, 119991 Moscow, Russia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Svetlana","family":"Dmitrienko","sequence":"additional","affiliation":[{"name":"Federal State Autonomous Institution \u201cNational Medical Research Center of Children\u2019s Health\u201d of the Ministry of Health of the Russian Federation, 119991 Moscow, Russia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alla","family":"Ryaposova","sequence":"additional","affiliation":[{"name":"Federal State Autonomous Institution \u201cNational Medical Research Center of Children\u2019s Health\u201d of the Ministry of Health of the Russian Federation, 119991 Moscow, Russia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elena","family":"Tsygina","sequence":"additional","affiliation":[{"name":"Federal State Autonomous Institution \u201cNational Medical Research Center of Children\u2019s Health\u201d of the Ministry of Health of the Russian Federation, 119991 Moscow, Russia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alexander","family":"Pushkov","sequence":"additional","affiliation":[{"name":"Federal State Autonomous Institution \u201cNational Medical Research Center of Children\u2019s Health\u201d of the Ministry of Health of the Russian Federation, 119991 Moscow, Russia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1423-0379","authenticated-orcid":false,"given":"Ilya","family":"Zhanin","sequence":"additional","affiliation":[{"name":"Federal State Autonomous Institution \u201cNational Medical Research Center of Children\u2019s Health\u201d of the Ministry of Health of the Russian Federation, 119991 Moscow, Russia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9354-6824","authenticated-orcid":false,"given":"Daria","family":"Chudakova","sequence":"additional","affiliation":[{"name":"Federal State Autonomous Institution \u201cNational Medical Research Center of Children\u2019s Health\u201d of the Ministry of Health of the Russian Federation, 119991 Moscow, Russia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aliy","family":"Asanov","sequence":"additional","affiliation":[{"name":"Department of Medical Genetics, N.V. Sklifosovsky Institute of Clinical Medicine, I.M. Sechenov First Moscow State Medical University, 115552 Moscow, Russia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4905-1303","authenticated-orcid":false,"given":"Olga","family":"Shchagina","sequence":"additional","affiliation":[{"name":"Research Centre for Medical Genetics, 115522 Moscow, Russia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aleksander","family":"Polyakov","sequence":"additional","affiliation":[{"name":"Research Centre for Medical Genetics, 115522 Moscow, Russia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andrey","family":"Fisenko","sequence":"additional","affiliation":[{"name":"Federal State Autonomous Institution \u201cNational Medical Research Center of Children\u2019s Health\u201d of the Ministry of Health of the Russian Federation, 119991 Moscow, Russia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kirill","family":"Savostyanov","sequence":"additional","affiliation":[{"name":"Federal State Autonomous Institution \u201cNational Medical Research Center of Children\u2019s Health\u201d of the Ministry of Health of the Russian Federation, 119991 Moscow, Russia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6301-9313","authenticated-orcid":false,"given":"Alexey","family":"Tsygin","sequence":"additional","affiliation":[{"name":"Federal State Autonomous Institution \u201cNational Medical Research Center of Children\u2019s Health\u201d of the Ministry of Health of the Russian Federation, 119991 Moscow, Russia"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2025,2,18]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"1088","DOI":"10.1016\/S0140-6736(71)90400-4","article-title":"Chondroitin-6-sulphaturia, defective cellular immunity, and nephrotic syndrome","volume":"298","author":"Schimke","year":"1971","journal-title":"Lancet"},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1007\/s004310050001","article-title":"Manifestations and treatment of Schimke immuno-osseous dysplasia: 14 new cases and a review of the literature","volume":"159","author":"Boerkoel","year":"2000","journal-title":"Eur. 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