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We describe a maternally transmitted PM (77 CGG) that was passed to a son (103 CGG), and to a daughter (220\u20131822 CGG), who were affected with FXTAS and FXS, respectively. The male with the PM showed low\u2010level mosaicism for normal size of 30 and 37 CGG. This male had two offspring: one female mosaic for PM and FM (56, 157, &gt;200 CGG) and another with only a 37 CGG allele detected in multiple tissues, neither with a clinical phenotype. The female with the 37 CGG allele showed normal levels of <jats:italic>FMR1<\/jats:italic> methylation and mRNA and passed this 37 CGG allele to one of her daughters, who was also unaffected. These findings show that post\u2010zygotic paternal retraction can lead to low\u2010level mosaicism for normal size alleles, with these normal alleles being functional when passed over two generations.<\/jats:p>","DOI":"10.1002\/ajmg.a.62500","type":"journal-article","created":{"date-parts":[[2021,9,21]],"date-time":"2021-09-21T07:52:15Z","timestamp":1632210735000},"page":"304-309","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Paternal retraction of a fragile X allele to normal size, showing normal function over two generations"],"prefix":"10.1002","volume":"188","author":[{"given":"Essra","family":"Bartlett","sequence":"first","affiliation":[{"name":"Victorian Clinical Genetics Services Murdoch Children's Research Institute, Royal Children's Hospital  Melbourne Victoria Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alison 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