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No Atrxnull males (Atrx null/Y;GATA1-cre +/− ) were recovered at birth, indicating that the absence of Atrx results in embryonic lethality. This finding was unexpected, since human ATR-X patients clearly survive to adulthood (see Discussion). Embryos were dissected at 7.5, 8.5, and 9.5 dpc and genotyped by PCR analysis of DNA extracted from yolk sac or total embryo (Figure 4B and Protocol S1). Atrxnull males were present at expected mendelian ratios (~25%) at both 7.5 dpc and 8.5 dpc (Table 1). However, by 9.5 dpc, depletion was observed both in the number of Atrxnull males (7%) and in the total number of males recovered (31%). No Atrxnull males were recovered after 9.5 dpc. Thus the absence of Atrx gives rise to embryonic lethality in mice before 9.5 dpc."}

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{"project":"craft-ca-core-dev","denotations":[{"id":"T3543","span":{"begin":12,"end":16},"obj":"PR:000004503"},{"id":"T3544","span":{"begin":21,"end":25},"obj":"NCBITaxon:10088"},{"id":"T3545","span":{"begin":40,"end":46},"obj":"SO:0000359"},{"id":"T3546","span":{"begin":56,"end":60},"obj":"PR:000004503"},{"id":"T3547","span":{"begin":64,"end":68},"obj":"SO:0000359"},{"id":"T3548","span":{"begin":75,"end":80},"obj":"GO:0007618"},{"id":"T3549","span":{"begin":97,"end":102},"obj":"PR:000007857"},{"id":"T3550","span":{"begin":125,"end":129},"obj":"PR:000004503"},{"id":"T3551","span":{"begin":135,"end":140},"obj":"PR:000007857"},{"id":"T3552","span":{"begin":155,"end":159},"obj":"PR:000004503"},{"id":"T3553","span":{"begin":171,"end":175},"obj":"PR:000004503"},{"id":"T3554","span":{"begin":183,"end":188},"obj":"PR:000007857"},{"id":"T3555","span":{"begin":217,"end":222},"obj":"GO:0007567"},{"id":"T3556","span":{"begin":255,"end":259},"obj":"PR:000004503"},{"id":"T3557","span":{"begin":271,"end":280},"obj":"UBERON:0000922"},{"id":"T3558","span":{"begin":327,"end":332},"obj":"NCBITaxon:9606"},{"id":"T3559","span":{"begin":337,"end":338},"obj":"GO:0000805"},{"id":"T3560","span":{"begin":367,"end":376},"obj":"UBERON:0000113"},{"id":"T3561","span":{"begin":395,"end":402},"obj":"UBERON:0000922"},{"id":"T3562","span":{"begin":495,"end":503},"obj":"UBERON:0001040"},{"id":"T3563","span":{"begin":513,"end":519},"obj":"UBERON:0000922"},{"id":"T3564","span":{"begin":549,"end":553},"obj":"PR:000004503"},{"id":"T3565","span":{"begin":718,"end":722},"obj":"PR:000004503"},{"id":"T3566","span":{"begin":791,"end":795},"obj":"PR:000004503"},{"id":"T3567","span":{"begin":856,"end":860},"obj":"PR:000004503"},{"id":"T3568","span":{"begin":875,"end":884},"obj":"UBERON:0000922"},{"id":"T3569","span":{"begin":898,"end":902},"obj":"NCBITaxon:10088"}],"text":"To generate Atrxnull mice, heterozygous floxed females (Atrx WT/flox) were mated with homozygous GATA1-cre transgenic males (Atrx WT/Y;GATA1-cre +/+ ). No Atrxnull males (Atrx null/Y;GATA1-cre +/− ) were recovered at birth, indicating that the absence of Atrx results in embryonic lethality. This finding was unexpected, since human ATR-X patients clearly survive to adulthood (see Discussion). Embryos were dissected at 7.5, 8.5, and 9.5 dpc and genotyped by PCR analysis of DNA extracted from yolk sac or total embryo (Figure 4B and Protocol S1). Atrxnull males were present at expected mendelian ratios (~25%) at both 7.5 dpc and 8.5 dpc (Table 1). However, by 9.5 dpc, depletion was observed both in the number of Atrxnull males (7%) and in the total number of males recovered (31%). No Atrxnull males were recovered after 9.5 dpc. Thus the absence of Atrx gives rise to embryonic lethality in mice before 9.5 dpc."}