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In this study, we fine\u2010mapped this region through whole\u2010genome sequencing of five MTRDHH2 heterozygous carriers and 95 non\u2010carriers from various ovine breeds. We identified a single base pair duplication within the <jats:italic>SLC33A1<\/jats:italic> gene, leading to a frameshift mutation and a premature stop codon (p.Arg246Alafs*3). SLC33A1 encodes a transmembrane transporter of acetyl\u2010coenzyme A that is crucial for cellular metabolism. To investigate the lethality of this mutation in homozygous MTR sheep, we performed at\u2010risk matings using artificial insemination (AI) between heterozygous <jats:italic>SLC33A1<\/jats:italic> variant carriers (<jats:italic>SLC33A1_dupG<\/jats:italic>). Pregnancy was confirmed 15\u2009days post\u2010AI using a blood test measuring interferon Tau\u2010stimulated MX1 gene expression. Ultrasonography between 45 and 60\u2009days post\u2010AI revealed a 12% reduction in AI success compared with safe matings, indicating embryonic\/fetal loss. This was supported by the <jats:italic>MX1<\/jats:italic> differential expression test suggesting fetal losses between 15 and 60\u2009days of gestation. We also observed a 34.7% pre\u2010weaning mortality rate in 49 lambs born from at\u2010risk matings. Homozygous <jats:italic>SLC33A1_dupG<\/jats:italic> lambs accounted for 47% of this mortality, with deaths occurring mostly within the first 5\u2009days without visible clinical signs. Therefore, appropriate management of <jats:italic>SLC33A1_dupG<\/jats:italic> with an allele frequency of 0.04 in the MTR selection scheme would help increase overall fertility and lamb survival.<\/jats:p>","DOI":"10.1111\/age.13459","type":"journal-article","created":{"date-parts":[[2024,6,24]],"date-time":"2024-06-24T04:07:07Z","timestamp":1719202027000},"page":"644-657","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["A single base pair duplication in the <i>SLC33A1<\/i> gene is associated with fetal losses and neonatal lethality in Manech T\u00eate Rousse dairy sheep"],"prefix":"10.1111","volume":"55","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-4770-0867","authenticated-orcid":false,"given":"Maxime","family":"Ben\u00a0Braiek","sequence":"first","affiliation":[{"name":"GenPhySE Universit\u00e9 de Toulouse, INRAE, ENVT  Castanet\u2010Tolosan 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