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Frequent co\u2010occurrence of neuroblastic tumors have fueled suspicion of an autoimmune paraneoplastic neurological syndrome (PNS); however, specific anti\u2010neural autoantibodies, a hallmark of PNS, have not been identified. Our objective is to determine if an autoimmune paraneoplastic etiology underlies ROHHAD.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>Immunoglobulin G (IgG) from pediatric ROHHAD patients (n\u00a0=\u00a09), non\u2010inflammatory individuals (n\u00a0=\u00a0100) and relevant pediatric controls (n\u00a0=\u00a025) was screened using a programmable phage display of the human peptidome (PhIP\u2010Seq). Putative ROHHAD\u2010specific autoantibodies were orthogonally validated using radioactive ligand binding and cell\u2010based assays. Expression of autoantibody targets in ROHHAD tumor and healthy brain tissue was assessed with immunohistochemistry and mass spectrometry, respectively.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Autoantibodies to ZSCAN1 were detected in ROHHAD patients by PhIP\u2010Seq and orthogonally validated in 7\/9 ROHHAD patients and 0\/125 controls using radioactive ligand binding and cell\u2010based assays. Expression of ZSCAN1 in ROHHAD tumor and healthy human brain tissue was confirmed.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Interpretation<\/jats:title>\n                    <jats:p>Our results support the notion that tumor\u2010associated ROHHAD syndrome is a pediatric PNS, potentially initiated by an immune response to peripheral neuroblastic tumor. ZSCAN1 autoantibodies may aid in earlier, accurate diagnosis of ROHHAD syndrome, thus providing a means toward early detection and treatment. This work warrants follow\u2010up studies to test sensitivity and specificity of a novel diagnostic test. Last, given the absence of the ZSCAN1 gene in rodents, our study highlights the value of human\u2010based approaches for detecting novel PNS subtypes. ANN NEUROL 2022;92:279\u2013291<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/ana.26380","type":"journal-article","created":{"date-parts":[[2022,4,25]],"date-time":"2022-04-25T04:10:04Z","timestamp":1650859804000},"page":"279-291","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":56,"title":["<scp>ZSCAN1<\/scp>\n                    Autoantibodies Are Associated with Pediatric Paraneoplastic\n                    <scp>ROHHAD<\/scp>"],"prefix":"10.1002","volume":"92","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7764-6529","authenticated-orcid":false,"given":"Caleigh","family":"Mandel\u2010Brehm","sequence":"first","affiliation":[{"name":"Department of Biochemistry and Biophysics University of California  San Francisco CA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Leslie A.","family":"Benson","sequence":"additional","affiliation":[{"name":"Department of Neurology Harvard Medical School  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