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The cellular distribution of CLRN1 in the retina remains uncertain, either because its expression levels are low or because its epitopes are masked. Indeed, in the adult mouse retina, <jats:italic>Clrn1<\/jats:italic> mRNA is developmentally downregulated, detectable only by RT\u2010PCR. In this study we used the highly sensitive RNAscope <jats:italic>in situ<\/jats:italic> hybridization assay and single\u2010cell RNA\u2010sequencing techniques to investigate the distribution of <jats:italic>Clrn1<\/jats:italic> and <jats:italic>CLRN1<\/jats:italic> in mouse and human retina, respectively. We found that <jats:italic>Clrn1<\/jats:italic> transcripts in mouse tissue are localized to the inner retina during postnatal development and in adult stages. The pattern of <jats:italic>Clrn1<\/jats:italic> mRNA cellular expression is similar in both mouse and human adult retina, with <jats:italic>CLRN1<\/jats:italic> transcripts being localized in M\u00fcller glia, and not photoreceptors. We generated a novel knock\u2010in mouse with a hemagglutinin (HA) epitope\u2010tagged CLRN1 and showed that CLRN1 is expressed continuously at the protein level in the retina. Following enzymatic deglycosylation and immunoblotting analysis, we detected a single CLRN1\u2010specific protein band in homogenates of mouse and human retina, consistent in size with the main CLRN1 isoform. Taken together, our results implicate M\u00fcller glia in USH3 pathology, placing this cell type to the center of future mechanistic and therapeutic studies to prevent vision loss in this disease. \u00a9 2019 The Authors. <jats:italic>The Journal of Pathology<\/jats:italic> published by John Wiley &amp; Sons Ltd on behalf of Pathological Society of Great Britain and Ireland.<\/jats:p>","DOI":"10.1002\/path.5360","type":"journal-article","created":{"date-parts":[[2019,10,18]],"date-time":"2019-10-18T03:19:01Z","timestamp":1571368741000},"page":"195-204","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":32,"title":["Clarin\u20101 expression in adult mouse and human retina highlights a role of M\u00fcller glia in Usher syndrome"],"prefix":"10.1002","volume":"250","author":[{"given":"Lei","family":"Xu","sequence":"first","affiliation":[{"name":"Department of Ophthalmology University of Florida  Gainesville FL 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