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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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001-es BibID:	BIBFORM056540
Első szerző:	Vriezinga, Sabine Lisa
Cím:	Randomized feeding intervention in infants at high risk for celiac disease / S. L. Vriezinga, R. Auricchio, E. Bravi, G. Castillejo, A. Chmielewska, P. Crespo Escobar, S. Kolaček, S. Koletzko, I. R. Korponay-Szabo, E. Mummert, I. Polanco, H. Putter, C. Ribes-Koninckx, R. Shamir, H. Szajewska, K. Werkstetter, L. Greco, J. Gyimesi, C. Hartman, C. Hogen Esch, E. Hopman, A. Ivarsson, T. Koltai, F. Koning, E. Martinez-Ojinaga, C. te Marvelde, A. Mocic Pavic, J. Romanos, E. Stoopman, V. Villanacci, C. Wijmenga, R. Troncone, M. L. Mearin
Dátum:	2014
ISSN:	0028-4793
Megjegyzések:	BACKGROUND A window of opportunity has been suggested for reducing the risk of celiac disease by introducing gluten to infants at 4 to 6 months of age. METHODS We performed a multicenter, randomized, double-blind, placebo-controlled dietaryintervention study involving 944 children who were positive for HLA-DQ2 or HLADQ8 and had at least one first-degree relative with celiac disease. From 16 to 24 weeks of age, 475 participants received 100 mg of immunologically active gluten daily, and 469 received placebo. Anti-transglutaminase type 2 and antigliadin antibodies were periodically measured. The primary outcome was the frequency of biopsy-confirmed celiac disease at 3 years of age. RESULTS Celiac disease was confirmed by means of biopsies in 77 children. To avoid underestimation of the frequency of celiac disease, 3 additional children who received a diagnosis of celiac disease according to the 2012 European Society for Pediatric Gastroenterology, Hepatology, and Nutrition diagnostic criteria (without having undergone biopsies) were included in the analyses (80 children; median age, 2.8 years; 59% were girls). The cumulative incidence of celiac disease among patients 3 years of age was 5.2% (95% confidence interval [CI], 3.6 to 6.8), with similar rates in the gluten group and the placebo group (5.9% [95% CI, 3.7 to 8.1] and 4.5% [95% CI, 2.5 to 6.5], respectively; hazard ratio in the gluten group, 1.23; 95% CI, 0.79 to 1.91). Rates of elevated levels of anti-transglutaminase type 2 and antigliadin antibodies were also similar in the two study groups (7.0% [95% CI, 4.7 to 9.4] in the gluten group and 5.7% [95% CI, 3.5 to 7.9] in the placebo group; hazard ratio, 1.14; 95% CI, 0.76 to 1.73). Breast-feeding, regardless of whether it was exclusive or whether it was ongoing during gluten introduction, did not significantly influence the development of celiac disease or the effect of the intervention. CONCLUSIONS As compared with placebo, the introduction of small quantities of gluten at 16 to 24 weeks of age did not reduce the risk of celiac disease by 3 years of age in this group of high-risk children. (Funded by the European Commission and others; PreventCD Current Controlled Trials number, ISRCTN74582487.)
Tárgyszavak:	Orvostudományok Klinikai orvostudományok idegen nyelvű folyóiratközlemény külföldi lapban
Megjelenés:	New England Journal Of Medicine 371 : 14 (2014), p. 1304-1315. -
További szerzők:	Auricchio, Renata Bravi, Enzo (biológus) Castillejo, Gemma (gyermekgyógyász, gasztroenterológus) Chmielewska, Anna Crespo-Escobar, Paula Kolaček, Sanja Koletzko, Sibylle Korponay-Szabó Ilma (1959-) (gyermekgyógyász) Mummert, Eckart Polanco, Isabel Putter, Hein Ribes-Koninckx, Carmen Shamir, Raanan (gyermekgyógyász) Szajewska, Hania (gyermekgyógyász) Werkstetter, Katharina (gyermekgyógyász, gasztroenterológus) Greco, Luigi Gyimesi Judit Hartman, Corina Hogen Esch, Caroline Hopman, Erica Ivarsson, Anneli Koltai Tünde Koning, Frits Martinez-Ojinaga, Eva Marvelde, Chantal te Pavic, Ana Romanos, Jihane Stoopman, Els Villanacci, Vincenzo Wijmenga, Cisca Troncone, Riccardo Mearin, Maria Luisa
Pályázati támogatás:	TÁMOP-2.2.11/1/KONV-2012-0023 TÁMOP 101788 OTKA
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001-es BibID:	BIBFORM072050
Első szerző:	Werkstetter, Katharina
Cím:	Accuracy in Diagnosis of Celiac Disease Without Biopsies in Clinical Practice / Werkstetter Katharina J., Korponay-Szabó Ilma Rita, Popp Alina, Villanacci Vincenzo, Salemme Marianna, Heilig Gabriele, Lillevang Sren Thue, Mearin Maria Luisa, Ribes-Koninckx Carmen, Thomas Adrian, Troncone Riccardo, Filipiak Birgit, Mäki Markku, Gyimesi Judit, Najafi Mehri, Dolinsek Jernej, Dydensborg Sander Stine, Auricchio Renata, Papadopoulou Alexandra, Vécsei Andreas, Szitanyi Peter, Donat Ester, Nenna Rafaella, Alliet Philippe, Penagini Francesca, Garnier-Lengliné Hélene, Castillejo Gemma, Kurppa Kalle, Shamir Raanan, Hauer Almuthe Christine, Smets Francoise, Corujeira Susana, van Winckel Myriam, Buderus Stefan, Chong Sonny, Husby Steffen, Koletzko Sibylle, ProCeDE study group
Dátum:	2017
ISSN:	0016-5085
Megjegyzések:	BACKGROUND & AIMS:The guidelines of the European Society of Pediatric Gastroenterology, Hepatology, and Nutrition allow for diagnosis of celiac disease without biopsies in children with symptoms and levels of immunoglobulin A against tissue-transglutaminase (TGA-IgA) 10-fold or more the upper limit of normal (ULN), confirmed by detection of endomysium antibodies (EMA) and positivity for HLA-DQ2/DQ8. We performed a large, international prospective study to validate this approach.METHODS:We collected data from consecutive pediatric patients (18 years or younger) on a gluten-containing diet who tested positive for TGA-IgA from November 2011 through May 2014, seen at 33 pediatric gastroenterology units in 21 countries. Local centers recorded symptoms; measurements of total IgA, TGA, and EMA; and histopathology findings from duodenal biopsies. Children were considered to have malabsorption if they had chronic diarrhea, weight loss (or insufficient gain), growth failure, or anemia. We directly compared central findings from 16 antibody tests (8 for TGA-IgA, 1 for TGA-IgG, 6 for IgG against deamidated gliadin peptides, and 1 for EMA, from 5 different manufacturers), 2 HLA-DQ2/DQ8 tests from 2 manufacturers, and histopathology findings from the reference pathologist. Final diagnoses were based on local and central results. If all local and central results were concordant for celiac disease, cases were classified as proven celiac disease. Patients with only a low level of TGA-IgA (threefold or less the ULN) but no other results indicating celiac disease were classified as no celiac disease. Central histo-morphometry analyses were performed on all other biopsies and cases were carefully reviewed in a blinded manner. Inconclusive cases were regarded as not having celiac disease for calculation of diagnostic accuracy. The primary aim was to determine whether the nonbiopsy approach identifies children with celiac disease with a positive predictive value (PPV) above 99% in clinical practice. Secondary aims included comparing performance of different serological tests and to determine whether the suggested criteria can be simplified.RESULTS:Of 803 children recruited for the study, 96 were excluded due to incomplete data, low level of IgA, or poor-quality biopsies. In the remaining 707 children (65.1% girls; median age, 6.2 years), 645 were diagnosed with celiac disease, 46 were found not to have celiac disease, and 16 had inconclusive results. Findings from local laboratories of TGA-IgA 10-fold or more the ULN, a positive result from the test for EMA, and any symptom identified children with celiac disease (n = 399) with a PPV of 99.75 (95% confidence interval [CI], 98.61-99.99); the PPV was 100.00 (95% CI, 98.68-100.00) when only malabsorption symptoms were used instead of any symptom (n = 278). Inclusion of HLA analyses did not increase accuracy. Findings from central laboratories differed greatly for patients with lower levels of antibodies, but when levels of TGA-IgA were 10-fold or more the ULN, PPVs ranged from 99.63 (95% CI, 98.67-99.96) to 100.00 (95% CI, 99.23-100.00).CONCLUSIONS:Children can be accurately diagnosed with celiac disease without biopsy analysis. Diagnosis based on level of TGA-IgA 10-fold or more the ULN, a positive result from the EMA tests in a second blood sample, and the presence of at least 1 symptom could avoid risks and costs of endoscopy for more than half the children with celiac disease worldwide. HLA analysis is not required for accurate diagnosis. Clinical Trial Registration no: DRKS00003555.
Tárgyszavak:	Orvostudományok Klinikai orvostudományok idegen nyelvű folyóiratközlemény külföldi lapban celiac
Megjelenés:	Gastroenterology 153 : 4 (2017), p. 924-935. -
További szerzők:	Korponay-Szabó Ilma (1959-) (gyermekgyógyász) Popp, Alina Villanacci, Vincenzo Salemme Marianna Heilig, Gabriele Lillevang, Sren Thue Mearin, Maria Luisa Ribes-Koninckx, Carmen Thomas, Adrian Troncone, Riccardo Filipiak, Birgit Mäki, Markku Gyimesi Judit Najafi, Mehri Dolinśek, Jernej Dydensborg Sander, Stine Auricchio, Renata Papadopoulou, Alexandra Vécsei Andreas Szitanyi Péter Donat, Ester Nenna, Rafaella Alliet, Philippe Penagini, Francesca Garnier-Lengliné, Hélene Castillejo, Gemma (gyermekgyógyász, gasztroenterológus) Kurppa, Kalle Shamir, Raanan (gyermekgyógyász) Hauer, Almuthe Christine Smets, Francoise Corujeira, Susana van Winckel, Myriam Buderus, Stefan Chong, Sonny Husby, Steffen Koletzko, Sibylle ProCeDE study group
Pályázati támogatás:	OTKA-101788 OTKA
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