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001-es BibID:	BIBFORM101855
Első szerző:	Meijer, Caroline R.
Cím:	Prediction models for celiac disease development in children from high-risk families : data from the PreventCD cohort / Meijer Caroline R., Auricchio Renata, Putter Hein, Castillejo Gemma, Crespo Paula, Gyimesi Judit, Hartman Corina, Kolacek Sanja, Koletzko Sibylle, Korponay-Szabo Ilma, Ojinaga Eva Martinez, Polanco Isabel, Ribes-Koninckx Carmen, Shamir Raanan, Szajewska Hania, Troncone Riccardo, Villanacci Vincenzo, Werkstetter Katharina, Mearin M. Luisa
Dátum:	2022
ISSN:	0016-5085
Megjegyzések:	Background and aims: Screening for celiac disease (CD) is recommended in children with affected first-degree relatives (FDR). However, the frequency of screening and at what age remain unknown. Aims: to detect variables influencing the risk of CD-development and develop and validate clinical prediction models to provide individualized screening advice. Methods: Analysis of prospective data from the ten years follow-up of the PreventCD-birth cohort involving 944 genetically predisposed children with CD-FDR. Variables significantly influencing the CD-risk were combined to determine a risk score. Landmark analyses were performed at different ages. Prediction models were created by multivariable Cox proportional hazards regression analyses, backward elimination and Harrell's c-index for discrimination. Validation was done using data from the independent NeoCel cohort. Results: In March 2019, the median follow-up was 8.3 years (22 days-12.0 years); 135/944 children developed CD (mean age 4.3years (1.1-11.4). CD developed significantly more often in girls (p=0.005) and in HLA-DQ2 homozygous individuals (8-year cumulative incidence 35.4% versus maximum of the other HLA-risk groups 18.2% [P<0.001]). The effect of homozygosity DR3-DQ2/DR7-DQ2 on CD-developing was only present in girls (interaction p=0.04). The prediction models showed good fitting in the validation cohort (Cox regression 0.81(0.54)). To calculate a personalized risk of CD-development and provide screening advice, we designed the Prediction application https://hputter.shinyapps.io/preventcd/. Conclusion: Children with CD-FDR develop CD early in life, and their risk depends on gender, age and HLA-DQ:all factors which are important for a sound screening advice. These children should be screened early in life, including HLA-DQ2/8-typing, and if genetically predisposed to CD, should get further personalized screening advice using our Prediction app.
Tárgyszavak:	Orvostudományok Klinikai orvostudományok idegen nyelvű folyóiratközlemény külföldi lapban folyóiratcikk celiac disease
Megjelenés:	Gastroenterology. - 163 : 2 (2022), p. 426-436. -
További szerzők:	Auricchio, Renata Putter, Hein Castillejo, Gemma (gyermekgyógyász, gasztroenterológus) Crespo-Escobar, Paula Gyimesi Judit Hartman, Corina Kolaček, Sanja Koletzko, Sibylle Korponay-Szabó Ilma (1959-) (gyermekgyógyász) Ojinaga, Eva Martinez Polanco, Isabel Ribes-Koninckx, Carmen Shamir, Raanan (gyermekgyógyász) Szajewska, Hania (gyermekgyógyász) Troncone, Riccardo Villanacci, Vincenzo Werkstetter, Katharina (gyermekgyógyász, gasztroenterológus) Mearin, Maria Luisa
Pályázati támogatás:	OTKA-101788 OTKA TAMOP 2.2.11/1/KONV-2012-0023 TÁMOP
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