{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,5,13]],"date-time":"2025-05-13T14:21:34Z","timestamp":1747146094316,"version":"3.40.5"},"reference-count":26,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2022,12,20]],"date-time":"2022-12-20T00:00:00Z","timestamp":1671494400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["American J of Med Genetics Pt A"],"published-print":{"date-parts":[[2023,3]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Autosomal dominant, sex\u2010limited inheritance is a distinct mode of transmission that should not be conflated with X\u2010linked inheritance. From animal studies, we know that sex\u2010limited inheritance implies the chance to \u201cturn off\u201d some genes in either males or females, in order to meliorate the phenotype, for example, by improving the fecundity. In this way, sex\u2010limited genes play an important role in the evolution of diverse species of animals. In human genetics, however, the biological significance of sex\u2010limited genes is unknown until today. When screening the literature, we found, thus far, three human examples of sex\u2010limited transmission. Autosomal dominant, male\u2010limited inheritance has meticulously been studied in a particular form of precocious puberty. Limitation to females was described in autosomal dominant lymphedema of the CESLR1 type, being underpinned by convincing molecular findings. Another example is white lentiginosis of Grosshans that shows clinical evidence of such mode of transmission although molecular findings are lacking as yet. In the animal kingdom, autosomal dominant sex\u2010limited inheritance is a well\u2010established phenomenon that has extensively been studied in various species such as butterflies, damselflies, fish (cichlids), and birds. Hence, at this point in time, it seems likely that other human examples of this mode of inheritance have previously been reported or will be published in the future.<\/jats:p>","DOI":"10.1002\/ajmg.a.63073","type":"journal-article","created":{"date-parts":[[2022,12,20]],"date-time":"2022-12-20T23:47:00Z","timestamp":1671580020000},"page":"684-689","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Autosomal dominant inheritance with sex\u2010limited manifestation: An unusual mode of transmission in humans and animals"],"prefix":"10.1002","volume":"191","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-6338-1766","authenticated-orcid":false,"given":"Rudolf","family":"Happle","sequence":"first","affiliation":[{"name":"Department of Dermatology, Medical Center University of Freiburg  Freiburg Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0094-2674","authenticated-orcid":false,"given":"Kilian","family":"Eyerich","sequence":"additional","affiliation":[{"name":"Department of Dermatology, Medical Center University of Freiburg  Freiburg Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2022,12,20]]},"reference":[{"key":"e_1_2_10_2_1","doi-asserted-by":"publisher","DOI":"10.1210\/jcem-22-11-1095"},{"key":"e_1_2_10_3_1","doi-asserted-by":"publisher","DOI":"10.1038\/hdy.1990.42"},{"key":"e_1_2_10_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-3476(85)80460-1"},{"key":"e_1_2_10_5_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.38.11.761"},{"key":"e_1_2_10_6_1","doi-asserted-by":"publisher","DOI":"10.1111\/cge.13622"},{"key":"e_1_2_10_7_1","doi-asserted-by":"publisher","DOI":"10.1080\/13887890.2005.9748252"},{"key":"e_1_2_10_8_1","doi-asserted-by":"publisher","DOI":"10.1046\/J.1420-9101.2003.00569.x"},{"key":"e_1_2_10_9_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13221-016-0035-5"},{"key":"e_1_2_10_10_1","first-page":"7","article-title":"La lentiginose blanche [The white lentiginosis]","volume":"121","author":"Grosshans E.","year":"1994","journal-title":"Annales de Dermatologie et de V\u00e9n\u00e9r\u00e9ologie"},{"key":"e_1_2_10_11_1","doi-asserted-by":"publisher","DOI":"10.1111\/jdv.17878"},{"key":"e_1_2_10_12_1","doi-asserted-by":"publisher","DOI":"10.1007\/978-3-030-89937-0"},{"key":"e_1_2_10_13_1","doi-asserted-by":"publisher","DOI":"10.1038\/75997"},{"key":"e_1_2_10_14_1","doi-asserted-by":"publisher","DOI":"10.1163\/jmg.32.7.553"},{"key":"e_1_2_10_15_1","doi-asserted-by":"publisher","DOI":"10.1111\/cge.12388"},{"key":"e_1_2_10_16_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.annder.2015.01.017"},{"key":"e_1_2_10_17_1","doi-asserted-by":"publisher","DOI":"10.1111\/bjd.15089"},{"key":"e_1_2_10_18_1","doi-asserted-by":"publisher","DOI":"10.1159\/000350040"},{"key":"e_1_2_10_19_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM198408233110807"},{"key":"e_1_2_10_20_1","doi-asserted-by":"publisher","DOI":"10.1038\/365652a0"},{"issue":"6","key":"e_1_2_10_21_1","first-page":"147","article-title":"Aplasia of the M\u00fcllerian system: Evidence for probable sex\u2010limited autosomal dominant inheritance","volume":"14","author":"Shokeir M. 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