{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,17]],"date-time":"2026-09-17T14:57:41Z","timestamp":1789657061933,"version":"4.0.1"},"reference-count":26,"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":7611,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Am. J. Med. Genet."],"published-print":{"date-parts":[[1984,8]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>We surveyed 264 persons (132 carriers, 132 matched noncarriers) screened for Tay\u2010Sachs heterozygosity during 1974\u201376 in a program directed at senior high school students in Montreal. Among 198 who apparently received the questionnaire in 1982, the response rate was 42% (38 carriers, 45 noncarriers; age range 21\u201326 yr). Respondents and nonrespondents had no apparent demographic differences. Of eight unable to remember their genotype only one was a carrier (these persons were excluded from the study). The subjects were: single (75%), married (20%), engaged (3%), divorced (1%); 32% of carriers were engaged or married vs 16% of noncarriers. (There were no carrier couples in our sample, but one such couple, who married after being screened in the high school program, requested amniocentesis in 1981.) Only three of the 12 spouses or fianc\u00e9(s) of carriers have not been tested (vs 3 of 6 noncarrier partners). Only 19% of carriers now attach any \u201cworry\u201d to heterozygosity (vs 46% at the earlier time of test disclosure, P = 0.001); carriers with spouses or fianc\u00e9(e)s are less \u201cworried\u201d than unattached carriers. Only 3% of carriers claim they would change marriage plansif their fianc\u00e9(e) was also a carrier. Carriers and noncarriers uniformly approve (96%) genetic screening for themselves and for other mutant genotypes; 92% of carriers and 95% of noncarriers approve being screened in high school. These findings indicate that Canadians screened in high school: 1) have largely positive attitudes toward genetic screening long after the experience, and 2) are making appropriate use of the test result.<\/jats:p>","DOI":"10.1002\/ajmg.1320180424","type":"journal-article","created":{"date-parts":[[2005,6,11]],"date-time":"2005-06-11T19:47:16Z","timestamp":1118519236000},"page":"769-778","source":"Crossref","is-referenced-by-count":71,"title":["A private view of heterozygosity: Eight\u2010year follow\u2010up study on carriers of the Tay\u2010Sachs gene detected by high school screening in montreal"],"prefix":"10.1002","volume":"18","author":[{"given":"Susan","family":"Zeesman","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Carol L.","family":"Clow","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lola","family":"Cartier","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Charles R.","family":"Scriver","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"John M.","family":"Opitz","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"James F.","family":"Reynolds","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2005,6,3]]},"reference":[{"key":"e_1_2_1_2_1","volume-title":"Tay\u2010Sachs disease in Lebanese Canadians, and a Tay\u2010Sachss screening program in the French\u2010Canadian population","author":"Andermann E","year":"1977"},{"key":"e_1_2_1_2_2","first-page":"161","volume":"18","journal-title":"Prog Clin Biol Res"},{"key":"e_1_2_1_3_1","first-page":"345","article-title":"Knowledge and attitudes towards Tay\u2010Sachs disease among a college student population","volume":"54","author":"Austein CF","year":"1981","journal-title":"Yale J Biol Med"},{"key":"e_1_2_1_4_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM197411282912206"},{"key":"e_1_2_1_5_1","doi-asserted-by":"crossref","unstructured":"BeckerMH(1974):The health belief model and personal health behaviour. Health Educ Monogr 2 (winter).","DOI":"10.1177\/109019817400200407"},{"key":"e_1_2_1_6_1","doi-asserted-by":"publisher","DOI":"10.2105\/AJPH.72.12.1386"},{"key":"e_1_2_1_7_1","first-page":"537","article-title":"Tay\u2010Sachs screening: Motives for participating and knowledge of genetics and probability","volume":"28","author":"Childs B","year":"1976","journal-title":"Am J Hum Genet"},{"key":"e_1_2_1_8_1","first-page":"550","article-title":"Tay\u2010Sachs screening: Social and psychological impact","volume":"28","author":"Childs B","year":"1976","journal-title":"Am J Hum Genet"},{"key":"e_1_2_1_9_1","first-page":"381","article-title":"The adolescent copes with genetic screening: A study of Tay\u2010Sachs screening among high\u2010school students","volume":"18","author":"Clow CL","year":"1977","journal-title":"Prog Clin Biol Res"},{"key":"e_1_2_1_10_1","doi-asserted-by":"crossref","first-page":"86","DOI":"10.1542\/peds.59.1.86","article-title":"Knowledge about and attitudes toward genetic screening among high\u2010school students: The Tay\u2010Sachs experience","volume":"59","author":"Clow CL","year":"1977","journal-title":"Pediatrics"},{"key":"e_1_2_1_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/0037-7856(77)90030-0"},{"key":"e_1_2_1_12_1","doi-asserted-by":"crossref","first-page":"131","DOI":"10.1542\/peds.59.1.131","article-title":"Genetic screening: For better or for worse","volume":"59","author":"Holtzman NA","year":"1977","journal-title":"Pediatrics"},{"key":"e_1_2_1_13_1","first-page":"447","article-title":"Screening for reproductive counselling: Social, ethical and medicolegal issues in the Tay\u2010Sachs disease experience","volume":"103","author":"Kaback MM","year":"1982","journal-title":"Prog Clin Biol Res"},{"key":"e_1_2_1_14_1","doi-asserted-by":"publisher","DOI":"10.2105\/AJPH.68.11.1116"},{"issue":"6","key":"e_1_2_1_15_1","first-page":"145","article-title":"Sociologic studies in human genetics. 1. Compliance factors in a voluntary heterozygote screening program","author":"Kaback MM","year":"1974","journal-title":"Birth Defects"},{"key":"e_1_2_1_16_1","doi-asserted-by":"publisher","DOI":"10.1080\/19485565.1975.9988157"},{"key":"e_1_2_1_17_1","volume-title":"Genetic Screening. Programs, Principles and Research","author":"National Academy of Sciences (National Research Council)","year":"1975"},{"key":"e_1_2_1_18_1","first-page":"945","volume-title":"The Metabolic Basis of Inherited Disease","author":"O'Brien JS","year":"1983"},{"key":"e_1_2_1_19_1","doi-asserted-by":"crossref","first-page":"523","DOI":"10.1093\/clinchem\/27.4.523","article-title":"Screening for the Tay\u2010Sachscarrier. 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