{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,13]],"date-time":"2026-05-13T00:06:59Z","timestamp":1778630819725,"version":"3.51.4"},"reference-count":19,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2005,6,3]],"date-time":"2005-06-03T00:00:00Z","timestamp":1117756800000},"content-version":"vor","delay-in-days":9650,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Am. J. Med. Genet."],"published-print":{"date-parts":[[1979,1]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>The prevalence at birth of anencephaly or spina bifida cystica among sibs of individuals born in British Columbia with these defects was studied using routinely collected records of morbidity and mortality within the province. The population prevalence of these neural\u2010tube defects in British Columbia is approximately 1.55 per thousand births, lower than in other areas where family studies have been carried out. A total of 1,028 affected individuals born in British Columbia during the period 1952\u20131970 was ascertained from health and vital documents collected on an ongoing basis by the Division of Vital Statistics of the British Columbia Ministry of Health. Information on the families of these cases was acquired using computer\u2010linked groupings of British Columbia marriage, birth, and stillbirth records.<\/jats:p><jats:p>The risk of affected individuals among the sibs was 2.4%, about 15 times the population prevalence at birth. The prevalence among subsequent sibs of the first affected individual in a family was 2.1%. There was no difference in risk when various factors that influence the population incidence of these conditions were considered. The risk of recurrence of either of these anomalies after two previously affected sibs was 4.8%, or double the risk after one affected sib.<\/jats:p><jats:p>From these results and those of a recent New York survey of sib risks of neural\u2010tube anomalies it would seem that the risk of anencephaly and spina bifida cystica to sibs of affected cases in North America is approximately half the risk in Great Britain, and it is this lower risk of 2% that should be used for genetic counseling in North America.<\/jats:p>","DOI":"10.1002\/ajmg.1320030409","type":"journal-article","created":{"date-parts":[[2005,6,11]],"date-time":"2005-06-11T13:57:50Z","timestamp":1118498270000},"page":"377-387","source":"Crossref","is-referenced-by-count":52,"title":["Sib risks of anencephaly and spina bifida in British Columbia"],"prefix":"10.1002","volume":"3","author":[{"given":"Mary L.","family":"McBride","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Edmond A.","family":"Murphy","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2005,6,3]]},"reference":[{"key":"e_1_2_1_2_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.5.2.81"},{"key":"e_1_2_1_3_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.10.3.209"},{"key":"e_1_2_1_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(67)91240-8"},{"key":"e_1_2_1_5_1","first-page":"288","article-title":"Anencephalus in Canada, 1934\u201370","volume":"100","author":"Elwood JM","year":"1974","journal-title":"Am J Epidemiol"},{"key":"e_1_2_1_6_1","unstructured":"ElwoodJM McBrideML(in press): Contrasting effects of maternal fertility and birth rank on the occurrence of neural tube defects.J Epidemiol Comm Health."},{"key":"e_1_2_1_7_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM197602122940704"},{"key":"e_1_2_1_8_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.15.2.101"},{"key":"e_1_2_1_9_1","first-page":"565","article-title":"Spina bifida cystica and anencephalus in twins","volume":"22","author":"Lorber J","year":"1977","journal-title":"Z Kinderchir Grenzgeb"},{"key":"e_1_2_1_10_1","first-page":"322","article-title":"The British Columbia registry of handicapped children and adults: Evolutionary changes over twenty years","volume":"66","author":"Lowry RB","year":"1975","journal-title":"Can J Public Health"},{"key":"e_1_2_1_11_1","doi-asserted-by":"publisher","DOI":"10.1126\/science.138.3540.593"},{"key":"e_1_2_1_12_1","doi-asserted-by":"crossref","first-page":"577","DOI":"10.1542\/peds.47.3.577","article-title":"Anencephaly and spina bifida in Israel","volume":"47","author":"Naggan L","year":"1971","journal-title":"Pediatrics"},{"key":"e_1_2_1_13_1","first-page":"335","article-title":"Record linking. The design of efficient systems for linking records into individual and family histories","volume":"19","author":"Newcombe HB","year":"1967","journal-title":"Am J Hum Genet"},{"key":"e_1_2_1_14_1","volume-title":"Medical Genetics: Principles and Practice","author":"Nora JJ","year":"1974"},{"key":"e_1_2_1_15_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1469-8749.1972.tb02645.x"},{"key":"e_1_2_1_16_1","doi-asserted-by":"crossref","first-page":"6","DOI":"10.1111\/j.1469-8749.1968.tb04824.x","article-title":"The outcome of pregnancies before and after the birth of infants with nervous system malformations","volume":"15","author":"Smithells RW","year":"1968","journal-title":"Dev Med Child Neurol"},{"key":"e_1_2_1_17_1","first-page":"87","volume-title":"Registers for the Detection and Prevention of Genetic Disease","author":"Trimble BK","year":"1976"},{"key":"e_1_2_1_18_1","doi-asserted-by":"publisher","DOI":"10.1002\/tera.1420170111"},{"key":"e_1_2_1_19_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.2.3.161"},{"key":"e_1_2_1_20_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(68)90709-5"}],"container-title":["American Journal of Medical Genetics"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fajmg.1320030409","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/ajmg.1320030409","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,11,12]],"date-time":"2023-11-12T23:36:55Z","timestamp":1699832215000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/ajmg.1320030409"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[1979,1]]},"references-count":19,"journal-issue":{"issue":"4","published-print":{"date-parts":[[1979,1]]}},"alternative-id":["10.1002\/ajmg.1320030409"],"URL":"https:\/\/doi.org\/10.1002\/ajmg.1320030409","archive":["Portico"],"relation":{},"ISSN":["0148-7299","1096-8628"],"issn-type":[{"value":"0148-7299","type":"print"},{"value":"1096-8628","type":"electronic"}],"subject":[],"published":{"date-parts":[[1979,1]]}}}