Open AccessDevelopment of a fragile X syndrome ( FXS ) knowledge scale: towards a modified multidimensional measure of informed choice for FXS population carrier screening
Author(s) -
Ames Alice G.,
Jaques Alice,
Ukoumunne Obioha C.,
Archibald Alison D.,
Duncan Rony E.,
Emery Jon,
Metcalfe Sylvia A.
Publication year - 2015
Publication title -
health expectations
Language(s) - English
Resource type - Journals
SCImago Journal Rank - 1.314
H-Index - 74
eISSN - 1369-7625
pISSN - 1369-6513
DOI - 10.1111/hex.12009
Subject(s) - fragile x syndrome , cronbach's alpha , scale (ratio) , population , psychology , construct (python library) , medicine , family medicine , clinical psychology , applied psychology , computer science , psychometrics , psychiatry , environmental health , quantum mechanics , programming language , physics
Background Genetic carrier screening is increasingly possible for many conditions, but it is important to ensure decisions are informed. The multidimensional measure of informed choice ( MMIC ) is a quantitative instrument developed to evaluate informed choice in prenatal screening for D own syndrome, measuring knowledge, attitudes and uptake. To apply the MMIC in other screening settings, the knowledge scale must be modified. Objective To develop and validate a modified MMIC knowledge scale for use with women undergoing carrier screening for fragile X syndrome ( FXS ). Setting and participants Responses to MMIC items were collected through questionnaires as part of a FXS carrier screening pilot study in a preconception setting in M elbourne, A ustralia. Design Ten knowledge scale items were developed using a modified D elphi technique. Cronbach's alpha and factor analysis were used to validate the new FXS knowledge scale. We summarized the knowledge, attitudes and informed choice status based on the modified MMIC . Results Two hundred and eighty‐five women were recruited, 241 eligible questionnaires were complete for analysis. The FXS knowledge scale items measured one salient construct and were internally consistent (alpha = 0.70). 71% (172/241) of participants were classified as having good knowledge, 70% (169/241) had positive attitudes and 27% (65/241) made an informed choice to accept or decline screening. Discussion and conclusions We present the development of a knowledge scale as part of a MMIC to evaluate informed choice in population carrier screening for FXS . This can be used as a template by other researchers to develop knowledge scales for other conditions for use in the MMIC .