Abstract
Cystic fibrosis (CF) has an incidence of one in 2,636 livebirths and a carrier rate of one in 26 inhabitants in Brittany. One objective of a major enquiry among parents having a CF child as well as CF adolescents and adults was to evaluate the reproductive behavior of 124 couples attending a CF care center. Knowledge of recurrence risk resulted in deciding against further progeny or in reducing the number of children (average number of children: 1.96; ideal mean number of children: 3.7). Thirty-five percent adopted or changed their method of contraception after the birth of their affected child, but the change was due to the birth of the CF child in only 14.3% of the couples. Prenatal diagnosis (PD) was favored by 95.1%, and 41.2% had used it; 68.6% were in favor of pregnancy interruption for CF and 76.2% would interrupt the pregnancy should PD reveal that their fetus had CF. All 123 respondents thought that genetic counseling was useful, but only 87.1% knew of its availability. Our results are quite different from those previously published. Although results could be population-specific, one cannot exclude the fact that they reflect a change of attitudes among parents, the other studies being much older.
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References
Al-Jader LN, Goodchild MC, Ryley HC, Harper PS (1990) Attitudes of parents of cystic fibrosis children towards neonatal screening and antenatal diagnosis. Clin Genet 38:460–465
Boat, Welsh MJ, Beaudet AL (1989) Cystic fibrosis. In: Scriver CR, Beaudet AL, Sly WS, Valle D (eds) The metabolic basis of inherited disease. McGraw-Hill, New York, pp 2649–2680
Borgo G, Fabiano T, Perobelli S, Mastella G (1992) Effect of introducing prenatal diagnosis on the reproductive behaviour of families at risk for cystic fibrosis. A cohort study. Prenat Diagn 12:821–830
Boué J, Muller F, Simon-Bouy B, Faure C, Boué A (1991) Consequences of prenatal diagnosis of cystic fibrosis on the reproductive attitudes of parents of affected children. Prenat Diagn 11:209–214
De Braekeleer M, Murray F, Daigneault J, Allard C, Simard F, Aubin G (1994) Disease knowledge and reproductive attitudes of parents having a child with cystic fibrosis. Ann Genet 37:89–92
De Braekeleer M, Bellis G, Gaimard M, Rault G, Milot M, Simard F (2000a) Mucoviscidose et fécondité des couples porteurs du gène au Saguenay-Lac-Saint-Jean (Québec, Canada). Population 55:359–366
De Braekeleer M, Bellis G, Rault G, Allard C, Milot M, Simard F (2000b) Reproductive attitudes of couples having a child with cystic fibrosis in Saguenay-Lac-Saint-Jean (Quebec, Canada). Ann Genet 43:93–97
De Braekeleer M, Bellis G, Rault G (2003) Disease knowledge among cystic fibrosis families in Brittany (France). J Cystic Fibrosis 2:S93
Evers-Kiebooms G, Van den Berghe H (1979) Impact of genetic counseling: a review of published follow-up studies. Clin Genet 15:465–474
Evers-Kiebooms G, Denayer L, Cassiman JJ, Van den Berghe H (1988) Family planning decisions after the birth of a cystic fibrosis child. The impact of prenatal diagnosis. Scand J Gastroenterol 143 [Suppl]:38–46
Evers-Kiebooms G, Denayer L, Van den Berghe H (1990) A child with cystic fibrosis. II. Subsequent family planning decisions, reproduction and use of prenatal diagnosis. Clin Genet 37:207–215
Férec C, Audrezet MP, Mercier B, Guillermit H, Moullier P, Quere I, Verlingue C (1992) Detection of over 98% cystic fibrosis mutations in a Celtic population. Nat Genet 1:188–191
Gillet D (2002) Aspects démographiques de la mucoviscidose. Etude des phénomènes de nuptialité, fécondité et mortalité. Université de Bretagne Occidentale, Brest
Jedlicka-Köhler I, Götz M, Eichler I (1994) Utilization of prenatal diagnosis for cystic fibrosis over the past seven years. Pediatrics 94:13–16
Kaback, Ziplin D, Boyd P, Cantor R (1984a) Attitudes towards prenatal diagnosis of cystic fibrosis among parents of affected children. In: Lawsons D (ed) Cystic fibrosis: horizons. Wiley, Chichester, pp 15–28
Kaback M, Zippin D, Boyd P, Cantor R (1984b) Cystic fibrosis: impact on reproductive decisions and parental attitudes toward prenatal diagnosis (PND). Am J Hum Genet 36:57S
Kraus EM, Brettler DB (1988) Assessment of reproductive risks and intentions by mothers of children with hemophilia. Am J Med Genet 31:259–267
Kyle D, Cummins C, Evans S (1988) Factors affecting the uptake of screening for neural tube defect. Br J Obstet Gynaecol 95:560–564
Léridon H (2001) Femmes et hommes face au désir d’enfant. In: Knibiehler Y (ed) Maternité. Affaire privée, affaire publique. Paris, Bayard, pp 49–59
Meryash DL, Abuelo D (1988) Counseling needs and attitudes toward prenatal diagnosis and abortion in fragile-X families. Clin Genet 33:349–355
Phelan (1983) Screening for cystic fibrosis. In: Meadow SR (ed) Recent advances in paediatrics. Churchill Levinstone, Edinburgh, pp 103–120
Rona RJ, Beech R, Mandalia S, Donnai D, Kingston H, Harris R, Wilson O, Axtell C, Swan AV, Kavanagh F (1994) The influence of genetic counselling in the era of DNA testing on knowledge, reproductive intentions and psychological wellbeing. Clin Genet 46:198–204
Scotet V, De Braekeleer M, Roussey M, Rault G, Parent P, Dagorne M, Journel H, Lemoigne A, Codet JP, Catheline M, David V, Chaventré A, Duguépéroux I, Verlingue C, Quéré I, Mercier B, Audrezet MP, Férec C (2000) Neonatal screening for cystic fibrosis in Brittany, France: assessment of 10 years’ experience and impact on prenatal diagnosis. Lancet 356:789–794
Scotet V, Audrezet MP, Roussey M, Rault G, Blayau M, De Braekeleer M, Férec C (2003) Impact of public health strategies on the birth incidence of cystic fibrosis in Brittany. Hum Genet 113:280–285
Steele MW, Rosser L, Rodnan JB, Bryce M (1986) Effect of sibship position on reproductive behavior of couples after the birth of a genetically handicapped child. Clin Genet 30:328–334
Super M (1987) Genetic counselling and antenatal diagnosis of cystic fibrosis. J R Soc Med 80 [Suppl 15]:13–15
Ten Kate LP (1977) A method for analysing fertility of heterozygotes for autosomal recessive disorders, with special reference to cystic fibrosis, Tay-Sachs disease and phenylketonuria. Ann Hum Genet 40:287–297
Thomson E (1997) Couple childbearing desires, intentions, and births. Demography 34:343–354
Toulemon L, Léridon H (1999) La famille idéale: combien d’enfants, à quel âge? INSEE Première 652:1–4
Wertz DC, Rosenfield JM, Janes SR, Erbe RW (1991) Attitudes toward abortion among parents of children with cystic fibrosis. Am J Public Health 81:992–996
Wertz DC, Janes SR, Rosenfield JM, Erbe RW (1992) Attitudes toward the prenatal diagnosis of cystic fibrosis: factors in decision making among affected families. Am J Hum Genet 50:1077–1085
Acknowledgements
The study was supported in part by grants from the “Fondation du Centre Hélio-Marin” in Roscoff through the “Plan d’Action Régional pour la Mucoviscidose (PARM)” in Brittany and the “Programme Hospitalier de Recherche Clinique (PHRC).”
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De Braekeleer, M., Rault, G. & Bellis, G. Reproductive attitudes of couples having a child with cystic fibrosis in Brittany (France). J Hum Genet 49, 285–289 (2004). https://doi.org/10.1007/s10038-004-0147-2
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DOI: https://doi.org/10.1007/s10038-004-0147-2
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