X. Geng, L. Chen, R. Srinivasan, R. J. Kylat, M. Witte, R. Erickson
{"title":"小鼠中Sos1突变缺乏胚胎纯合子或成年杂合子淋巴表型,纯合子Cx47突变缺乏淋巴胚胎表型。","authors":"X. Geng, L. Chen, R. Srinivasan, R. J. Kylat, M. Witte, R. Erickson","doi":"10.2458/lymph.5405","DOIUrl":null,"url":null,"abstract":"We have studied the lymphatic phenotypes of 2 mutations, known to cause abnormalities of lymphatics in humans, in mice. The Cx47 R260C mutation (variably penetrant in humans heterozygous for it and causing limb lymphedema) had an adult mouse phenotype of hyperplasia and increased lymph nodes only in homozygous condition but we did not find any anatomical phenotype in day 16.5 homozygous embryos. Mice harboring the Sos1 mutation E846K (causing Noonan's in man which occasionally shows lymphatic dysplasia) had no adult heterozygous phenotype in lymphatic vessel appearance and drainage (homozygotes are early embryonic lethals) while day 16.5 heterozygous embryos also had no detectable anatomical phenotype.","PeriodicalId":51122,"journal":{"name":"Lymphology","volume":"55 3 1","pages":"129-134"},"PeriodicalIF":0.7000,"publicationDate":"2022-11-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"1","resultStr":"{\"title\":\"Lack of embryonic homozygous or adult heterozygous lymphatic phenotypes for a Sos1 mutation and lack of lymphatic embryonic phenotypes for a homozygous Cx47 mutation in mice.\",\"authors\":\"X. Geng, L. Chen, R. Srinivasan, R. J. Kylat, M. Witte, R. Erickson\",\"doi\":\"10.2458/lymph.5405\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"We have studied the lymphatic phenotypes of 2 mutations, known to cause abnormalities of lymphatics in humans, in mice. The Cx47 R260C mutation (variably penetrant in humans heterozygous for it and causing limb lymphedema) had an adult mouse phenotype of hyperplasia and increased lymph nodes only in homozygous condition but we did not find any anatomical phenotype in day 16.5 homozygous embryos. Mice harboring the Sos1 mutation E846K (causing Noonan's in man which occasionally shows lymphatic dysplasia) had no adult heterozygous phenotype in lymphatic vessel appearance and drainage (homozygotes are early embryonic lethals) while day 16.5 heterozygous embryos also had no detectable anatomical phenotype.\",\"PeriodicalId\":51122,\"journal\":{\"name\":\"Lymphology\",\"volume\":\"55 3 1\",\"pages\":\"129-134\"},\"PeriodicalIF\":0.7000,\"publicationDate\":\"2022-11-22\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"1\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Lymphology\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.2458/lymph.5405\",\"RegionNum\":4,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q4\",\"JCRName\":\"IMMUNOLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Lymphology","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.2458/lymph.5405","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q4","JCRName":"IMMUNOLOGY","Score":null,"Total":0}
Lack of embryonic homozygous or adult heterozygous lymphatic phenotypes for a Sos1 mutation and lack of lymphatic embryonic phenotypes for a homozygous Cx47 mutation in mice.
We have studied the lymphatic phenotypes of 2 mutations, known to cause abnormalities of lymphatics in humans, in mice. The Cx47 R260C mutation (variably penetrant in humans heterozygous for it and causing limb lymphedema) had an adult mouse phenotype of hyperplasia and increased lymph nodes only in homozygous condition but we did not find any anatomical phenotype in day 16.5 homozygous embryos. Mice harboring the Sos1 mutation E846K (causing Noonan's in man which occasionally shows lymphatic dysplasia) had no adult heterozygous phenotype in lymphatic vessel appearance and drainage (homozygotes are early embryonic lethals) while day 16.5 heterozygous embryos also had no detectable anatomical phenotype.
期刊介绍:
The Journal contains original articles, special features (see below), and information regarding the International Society of Lymphology. It seeks original papers dealing with clinical and basic studies of the lymphatic system and its disorders including related fields. Articles are accepted for external review and publication on the condition that they are contributed to Lymphology only and that no substantial part has been or will be published elsewhere.