T. W. Hansen, M. Seip, C. D. de Verdier, Å. Ericson
{"title":"红细胞嘧啶5′-核苷酸酶缺乏症。报告2例新病例,并复习文献。","authors":"T. W. Hansen, M. Seip, C. D. de Verdier, Å. Ericson","doi":"10.1111/J.1600-0609.1983.TB01518.X","DOIUrl":null,"url":null,"abstract":"We present 2 Norwegian siblings who we believe to be the first cases of pyrimidine 5'-nucleotidase deficiency diagnosed in the Scandinavian countries. In addition to the features presented by previously published cases, our patients exhibited signs of intravascular haemolysis with haemoglobinuria and increased loss of iron in the urine. The loss of iron has made iron medication necessary from time to time. A kidney biopsy from the oldest patient revealed accumulation of iron in the epithelium of the proximal tubuli. The kidneys were considerably enlarged in both patients, but with normal function tests.","PeriodicalId":21489,"journal":{"name":"Scandinavian journal of haematology","volume":"29 1","pages":"122-8"},"PeriodicalIF":0.0000,"publicationDate":"2009-04-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"17","resultStr":"{\"title\":\"Erythrocyte pyrimidine 5'-nucleotidase deficiency. Report of 2 new cases, with a review of the literature.\",\"authors\":\"T. W. Hansen, M. Seip, C. D. de Verdier, Å. Ericson\",\"doi\":\"10.1111/J.1600-0609.1983.TB01518.X\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"We present 2 Norwegian siblings who we believe to be the first cases of pyrimidine 5'-nucleotidase deficiency diagnosed in the Scandinavian countries. In addition to the features presented by previously published cases, our patients exhibited signs of intravascular haemolysis with haemoglobinuria and increased loss of iron in the urine. The loss of iron has made iron medication necessary from time to time. A kidney biopsy from the oldest patient revealed accumulation of iron in the epithelium of the proximal tubuli. The kidneys were considerably enlarged in both patients, but with normal function tests.\",\"PeriodicalId\":21489,\"journal\":{\"name\":\"Scandinavian journal of haematology\",\"volume\":\"29 1\",\"pages\":\"122-8\"},\"PeriodicalIF\":0.0000,\"publicationDate\":\"2009-04-24\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"17\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Scandinavian journal of haematology\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.1111/J.1600-0609.1983.TB01518.X\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"\",\"JCRName\":\"\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Scandinavian journal of haematology","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.1111/J.1600-0609.1983.TB01518.X","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"","JCRName":"","Score":null,"Total":0}
Erythrocyte pyrimidine 5'-nucleotidase deficiency. Report of 2 new cases, with a review of the literature.
We present 2 Norwegian siblings who we believe to be the first cases of pyrimidine 5'-nucleotidase deficiency diagnosed in the Scandinavian countries. In addition to the features presented by previously published cases, our patients exhibited signs of intravascular haemolysis with haemoglobinuria and increased loss of iron in the urine. The loss of iron has made iron medication necessary from time to time. A kidney biopsy from the oldest patient revealed accumulation of iron in the epithelium of the proximal tubuli. The kidneys were considerably enlarged in both patients, but with normal function tests.