{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,30]],"date-time":"2026-04-30T02:20:34Z","timestamp":1777515634213,"version":"3.51.4"},"reference-count":25,"publisher":"Wiley","issue":"2-3","license":[{"start":{"date-parts":[[2005,6,7]],"date-time":"2005-06-07T00:00:00Z","timestamp":1118102400000},"content-version":"vor","delay-in-days":5240,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Am. J. Med. Genet."],"published-print":{"date-parts":[[1991,2]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Although most males with the fragile\u2010X [fra(X)] syndrome do not reproduce, there are 2 published pedigrees that include affected males who have daughters and who thus appear to have transmitted the fragile\u2010X chromosome to their progeny. In addition, one published fra(X) pedigree includes an apparently normal male who expresses cytogenetically the fra(X) site at high frequency and who has 3 daughters. In the 6 daughters of these 3 males, there is little or no cytogenetic expression of the fra(X). I interpret these pedigrees within the context of my X\u2010inactivation imprinting model of the fra(X) syndrome (<jats:italic>Genetics<\/jats:italic> 117:587\u2013599): the cytogenetic manifestation of the imprinted state of the mutant fra(X) chromosome [high percentage of cytogenetic expression] is no longer present in daughters of imprinted males. I propose that the imprinted state is erased when an imprinted fragile\u2010X chromosome is passed through a male. Such erasure in the gender opposite to the gender that established the imprint is in accord with other examples of chromosome imprinting in mammals. Additional data from unpublished fra(X) pedigrees are requested.<\/jats:p>","DOI":"10.1002\/ajmg.1320380247","type":"journal-article","created":{"date-parts":[[2005,6,12]],"date-time":"2005-06-12T04:09:12Z","timestamp":1118549352000},"page":"391-395","source":"Crossref","is-referenced-by-count":12,"title":["Possible Erasure of the Imprint on a Fragile X Chromosome When Transmitted by a Male"],"prefix":"10.1002","volume":"38","author":[{"given":"Charles D.","family":"Laird","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2005,6,7]]},"reference":[{"key":"e_1_2_1_2_1","doi-asserted-by":"publisher","DOI":"10.1038\/315496a0"},{"key":"e_1_2_1_3_1","doi-asserted-by":"publisher","DOI":"10.1038\/253165a0"},{"key":"e_1_2_1_4_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.1320140412"},{"key":"e_1_2_1_5_1","doi-asserted-by":"crossref","first-page":"1429","DOI":"10.1093\/genetics\/45.10.1429","article-title":"The controlling element in sex chromosome behavior in Sciara","volume":"45","author":"Crouse HV","year":"1960","journal-title":"Genetics"},{"key":"e_1_2_1_6_1","doi-asserted-by":"publisher","DOI":"10.1146\/annurev.ge.17.120183.001103"},{"key":"e_1_2_1_7_1","doi-asserted-by":"crossref","first-page":"587","DOI":"10.1093\/genetics\/117.3.587","article-title":"Proposed mechanism of inheritance and expression of the human fragile\u2010X syndrome of mental retardation","volume":"117","author":"Laird CD","year":"1987","journal-title":"Genetics"},{"key":"e_1_2_1_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/0168-9525(87)90268-X"},{"key":"e_1_2_1_9_1","first-page":"696","article-title":"Two progenitor cells for human oogonia inferred from the pattern of chromosome imprinting in the fragile\u2010X syndrome","volume":"46","author":"Laird CD","year":"1990","journal-title":"Am J Hum Genet"},{"key":"e_1_2_1_10_1","doi-asserted-by":"publisher","DOI":"10.1038\/324161a0"},{"key":"e_1_2_1_11_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.1320270219"},{"key":"e_1_2_1_12_1","first-page":"1341","volume-title":"Mendelian Inheritance in Man: Catalogs of Autosomal Dominant, Autosomal Recessive, and X\u2010linked Phenotypes","author":"McKusick VA","year":"1988"},{"key":"e_1_2_1_13_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.20.6.476"},{"key":"e_1_2_1_14_1","unstructured":"Moric\u2010PetrovicS(1989) Personal communication."},{"key":"e_1_2_1_15_1","doi-asserted-by":"publisher","DOI":"10.1146\/annurev.ge.20.120186.000545"},{"key":"e_1_2_1_16_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.1320210413"},{"key":"e_1_2_1_17_1","doi-asserted-by":"publisher","DOI":"10.1038\/328248a0"},{"key":"e_1_2_1_18_1","doi-asserted-by":"publisher","DOI":"10.1038\/328251a0"},{"key":"e_1_2_1_19_1","doi-asserted-by":"publisher","DOI":"10.1007\/BF00291644"},{"key":"e_1_2_1_20_1","doi-asserted-by":"publisher","DOI":"10.1016\/0092-8674(87)90330-8"},{"key":"e_1_2_1_21_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(89)91908-9"},{"key":"e_1_2_1_22_1","doi-asserted-by":"publisher","DOI":"10.1007\/BF00273654"},{"key":"e_1_2_1_23_1","doi-asserted-by":"publisher","DOI":"10.1007\/BF00283690"},{"key":"e_1_2_1_24_1","doi-asserted-by":"publisher","DOI":"10.1038\/327725a0"},{"key":"e_1_2_1_25_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.1320300169"},{"key":"e_1_2_1_26_1","doi-asserted-by":"crossref","unstructured":"YuW\u2010D WengerSL SteeleMW(1990) X\u2010chromosome imprinting in the Fragile\u2010X syndrome.Hum Genet(in press).","DOI":"10.1007\/BF00193580"}],"container-title":["American Journal of Medical Genetics"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fajmg.1320380247","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/ajmg.1320380247","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,8,30]],"date-time":"2023-08-30T12:54:16Z","timestamp":1693400056000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/ajmg.1320380247"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[1991,2]]},"references-count":25,"journal-issue":{"issue":"2-3","published-print":{"date-parts":[[1991,2]]}},"alternative-id":["10.1002\/ajmg.1320380247"],"URL":"https:\/\/doi.org\/10.1002\/ajmg.1320380247","archive":["Portico"],"relation":{},"ISSN":["0148-7299","1096-8628"],"issn-type":[{"value":"0148-7299","type":"print"},{"value":"1096-8628","type":"electronic"}],"subject":[],"published":{"date-parts":[[1991,2]]}}}