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Given the growing evidence for the involvement of loss of the Y chromosome (LoY) in male diseases including cancer, we investigated Y chromosome status in NB. Male NB tumor samples from a Swedish cohort, analyzed using Cytoscan HD SNP\u2010microarray, were selected. Seventy NB tumors were analyzed for aneuploidy of the Y chromosome, and these data were correlated with other genetic, biological, and clinical parameters. LoY was found in 21% of the male NB tumors and it was almost exclusively found in those with high\u2010risk genomic profiles. Furthermore, LoY was associated with increased age at diagnosis and enriched in tumors with 11q\u2010deletion and activated telomere maintenance mechanisms. In contrast, tumors with an <jats:italic>MYCN<\/jats:italic>\u2010amplified genomic profile retained their Y chromosome. The understanding of LoY in cancer is limited, making it difficult to conclude whether LoY is a driving event in NB or function of increased genomic instability. Gene expression analysis of Y chromosome genes in male NB tumors showed low expression of certain genes correlating with worse overall survival. <jats:italic>KDM5D<\/jats:italic>, encoding a histone demethylase stands out as an interesting candidate for further studies. LoY has been shown to impact the epigenomic layer of autosomal loci in nonreproductive tissues, and <jats:italic>KDM5D<\/jats:italic> has been reported as downregulated and\/or associated with poor survival in different malignancies. Further studies are needed to explore the mechanisms and functional consequences of LoY in NB.<\/jats:p>","DOI":"10.1002\/gcc.23260","type":"journal-article","created":{"date-parts":[[2024,7,19]],"date-time":"2024-07-19T12:14:35Z","timestamp":1721391275000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":12,"title":["Loss of Chromosome Y in Neuroblastoma Is Associated With High\u2010Risk Disease, 11q\u2010Deletion, and Telomere Maintenance"],"prefix":"10.1002","volume":"63","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-5250-2163","authenticated-orcid":false,"given":"Anna","family":"Djos","sequence":"first","affiliation":[{"name":"Department of Laboratory Medicine, Institute of Biomedicine, Sahlgrenska Academy University of Gothenburg  Gothenburg Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Johanna","family":"Svensson","sequence":"additional","affiliation":[{"name":"Department of Laboratory 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