30 results on '"Cilleruelo ML"'
Search Results
2. Factors Associated With Decision to Treat or Not to Treat Helicobacter pylori Infection in Children: Data From the EuroPedHp Registry.
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Le Thi TG, Werkstetter K, Kotilea K, Bontems P, Cabral J, Cilleruelo ML, Kori M, Barrio J, Homan M, Kalach N, Lima R, Tavares M, Urruzuno P, Misak Z, Urbonas V, and Koletzko S
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- Humans, Child, Female, Male, Adolescent, Europe epidemiology, Child, Preschool, Comorbidity, Infant, Helicobacter Infections drug therapy, Helicobacter Infections epidemiology, Helicobacter pylori drug effects, Anti-Bacterial Agents therapeutic use, Registries
- Abstract
Background: European and North-American guidelines on management of H. pylori infection in children provide the option not to treat even if the infection is endoscopically confirmed. We used data from the EuroPedHp Registry to identify factors associated with therapy decisions., Methods: We included treatment-naïve patients reported between 2017 and 2020 from 30 centers in 17 European countries. Multivariable logistic regression identified factors including comorbidities within and outside the gastrointestinal (GI) tract influencing the decision for or against therapy., Results: Of 1165 patients (52% females, median age 12.8), 28% (321/1165) reported any alarm symptom, 26% (307/1165) comorbidities, and 16% (192/1165) did not receive eradication treatment. Therapy was initiated less often in children having any GI comorbidity (57%, n = 181), particularly in those with eosinophilic esophagitis (60%, n = 35), inflammatory bowel disease (54%, n = 28), and celiac disease (43%, n = 58), compared to those with non-GI (86%, n = 126) or no comorbidity (89%, n = 858), despite similar frequencies of alarm and non-alarm symptoms, ulcers, erosions, and nodular gastritis. Patients with GI and without comorbidities remained more likely untreated in high versus low H. pylori prevalence countries (p < 0.0001). In children without comorbidities, factors favoring therapy included older age, being overweight, having symptoms, erosions, antral nodularity, and available antibiotic susceptibility results., Conclusion: In this cohort, H. pylori-infected children with GI comorbidities compared to no comorbidity showed 75% reduced chance of receiving eradication therapy. We found no evidence supporting different management strategies in infected patients with GI comorbidities compared to all pediatric patients with endoscopically proven H. pylori infection., (© 2024 The Author(s). Helicobacter published by John Wiley & Sons Ltd.)
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- 2024
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3. Transcultural adaptation to Spain of the CDPQOL questionnaire specific for coeliac children.
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Barrio-Torres J, Sebastián-Viana T, Fernández-Pérez C, Pérez-Fernández C, Martínez-Escribano B, Gutierrez-Junquera C, Román E, and Cilleruelo ML
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- Humans, Child, Male, Female, Spain, Adolescent, Surveys and Questionnaires, Reproducibility of Results, Factor Analysis, Statistical, Cross-Cultural Comparison, Celiac Disease psychology, Quality of Life, Translations, Psychometrics methods
- Abstract
This study aimed to translate, cross-culturally adapt, and validate the CDPQOL questionnaire, a coeliac disease (CD)-specific paediatric health-related quality of life (HRQoL) instrument (CDPQOL), in Spanish children with CD. The CDPQOL questionnaire has two versions for children aged 8-12 and 13-18. Translation and linguistic validation were performed following an international consensus process. Internal consistency was calculated using Cronbach's alpha and McDonald's omega coefficients, and convergent validity was assessed with average variance extracted (AVE). Confirmatory factor analysis (CFA) and exploratory factor analysis (EFA), when necessary, were carried out to assess the construct validity. A total of 235 children were included. In the 8-12 age group, a change in the distribution of items to a new structure of three dimensions (negative emotions, food feelings and social interaction) was required. In this new model, CFA supported the fit of the model (χ
2 /gl = 1.79, RMSEA = 0.077 (IC 95% 0.05-0.100), CFI = 0.969, TLI 0.960, SRMR = 0.081) and Cronbach's alpha and McDonald's omega coefficients were > 0.7 in all three dimensions. In the 13-18 age group, CFA showed that all fit indexes were acceptable (χ2 /gl = 1.702, RMSEA = 0.102 (IC 95% 0.077-0.127), p < 0.001, CFI = 0.956, TLI = 0.947, SRMR = 0.103) and Cronbach's alpha and McDonald's omega coefficients were > 0.7 in all three dimensions, except for uncertainty dimension. Conclusions: The Spanish version of the CDPQOL questionnaire is a useful instrument to assess quality of life in coeliac children whose native language was Spanish spoken in Spain, with changes in item distribution in the younger age group questionnaire. What is Known: • The first specific questionnaire for coeliac children, Dutch Coeliac Disease Questionnaire (CDDUX), which focuses on diet, was translated into Spanish and validated allowing to evaluate the HRQoL of Spanish coeliac children. • Spanish Children and parents feel the disease had no substantial negative impacts on patient HRQoL using this questionnaire, similar to that observed with other countries. What is New: • The age specific for CD children (CDPQOL) was elaborated in the USA and focuses on other aspects not evaluated by CDDUX such as emotional and social issues related to living with CD. • The CDPQOL was translated into Spanish and validated allowing it to be used to assess Spanish coeliac children's QoL., (© 2024. The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature.)- Published
- 2024
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4. Helicobacter pylori infection found during upper endoscopy performed for the diagnosis of celiac, inflammatory bowel diseases, and eosinophilic esophagitis: A multicenter pediatric European study.
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Kotilea K, Romano C, Miele E, Kindermann A, Dolstra Y, Misak Z, Urbonas V, Sykora J, Urruzuno P, Krauthammer A, Rogalidou M, Dimakou K, Zangen T, Roma E, Zellos A, Cilleruelo ML, M'Rini M, Bontems P, Sahin Y, Tavares M, Shahinyan T, Vuletic B, Kalach N, and Kori M
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- Humans, Male, Female, Child, Retrospective Studies, Adolescent, Europe epidemiology, Prevalence, Endoscopy, Gastrointestinal, Child, Preschool, Helicobacter Infections diagnosis, Helicobacter Infections epidemiology, Helicobacter Infections drug therapy, Eosinophilic Esophagitis epidemiology, Eosinophilic Esophagitis diagnosis, Inflammatory Bowel Diseases epidemiology, Inflammatory Bowel Diseases diagnosis, Inflammatory Bowel Diseases microbiology, Helicobacter pylori isolation & purification, Celiac Disease diagnosis, Celiac Disease epidemiology
- Abstract
Background: Helicobacter pylori may be found during upper gastrointestinal endoscopy (UGE) performed to diagnose celiac disease (CeD), inflammatory bowel disease (IBD), and eosinophilic esophagitis (EoE). We aimed to describe the frequency of H. pylori in children undergoing UGE for CeD, IBD, and EoE and the number of children receiving eradication treatment., Materials and Methods: A retrospective multicenter study from 14 countries included pediatric patients diagnosed with CeD, IBD, and EoE between January 2019 and December 2021., Data Collected: age, gender, hematologic parameters, endoscopic, histologic, and H. pylori culture results, and information on eradication treatment., Results: H. pylori was identified in 349/3890 (9%) children [167 (48%) male, median 12 years (interquartile range 8.1-14.6)]. H. pylori was present in 10% (173/1733) CeD, 8.5% (110/1292) IBD and 7.6% (66/865) EoE patients (p = NS). The prevalence differed significantly between Europe (Eastern 5.2% (28/536), Southern 3.8% (78/2032), Western 5.6% (28/513)) and the Middle East 26.6% (215/809) [odds ratio (OR) 7.96 95% confidence interval (CI) (6.31-10.1) p < 0.0001]. Eradication treatment was prescribed in 131/349 (37.5%) patients, 34.6% CeD, 35.8% IBD, and 56.1% EoE. Predictors for recommending treatment included erosions/ulcers [OR 6.45 95% CI 3.62-11.47, p < 0.0001] and nodular gastritis [OR 2.25 95% CI 1.33-3.81, p 0.003]. Treatment rates were higher in centers with a low H. pylori prevalence (<20%) [OR 3.36 95% CI 1.47-7.66 p 0.004]., Conclusions: Identifying H. pylori incidentally during UGE performed for the most common gastrointestinal diseases varies significantly among regions but not among diseases. The indications for recommending treatment are not well defined, and less than 40% of children received treatment., (© 2024 John Wiley & Sons Ltd.)
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- 2024
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5. Correction: Health-related quality of life using specific and generic questionnaires in Spanish coeliac children.
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Barrio J, Cilleruelo ML, Román E, and Fernández C
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- 2023
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6. "Geographical distribution of risk genotypes in pediatric patients with celiac disease in Spain".
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Sánchez-Valverde F, Martínez-Ojinaga E, Donat E, Bodas A, Bandrés E, Torres R, Ibáñez B, Cilleruelo ML, Castillejo G, Pérez-Solis D, Ochoa C, Eizaguirre FJ, García S, García JI, Barrio J, Vecino R, Miranda MDC, Juste M, Salazar JC, Armas H, Ortigosa L, Urruzuno P, García Z, Balmaseda E, Martinez-Costa C, La Orden E, Codoñer P, Roca A, Trillo C, Sebastian M, García R, Peña-Quintana L, Barros P, Soria M, García R, Pérez-Moneo B, Polanco I, Ribes C, and Román E
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- Humans, Child, Spain epidemiology, Genetic Predisposition to Disease, Alleles, Genotype, Haplotypes, HLA-DQ beta-Chains genetics, HLA-DQ alpha-Chains genetics, HLA-DQ Antigens genetics, Celiac Disease genetics
- Abstract
Celiac disease is strongly associated with HLA DQ, specifically with haplotypes. DRB1*03-DQA1*05:01/DQB1*02:01 (DQ2.5),DRB1*07-DQA1*02:01/DQB1*02:02 (DQ2.2), DRB1*11-DQA1*05:05/DQB1*03:01 (DQ7.5), and DRB1*04-DQA1*03:01/DQB1*03:02 (DQ8). The distribution of these risk haplotypes in patients with celiac disease is different in the geographical areas investigated. A high frequency of DRB1*07- DQA1*02:01/DQB1*02:02 (DQ2.2) and DRB1*11-DQA1*05:05/DQB1*03:01 (DQ7.5), has been described in Southern Europe. We analyzed 2102 confirmed CD cases with information on both DQB1* alelles and their distribution by geographical area in Spain. According to the presence of this haplotype in one or two chromosomes, the genotype is classified in: DQ2 homozygous, DQ2 heterozygous (cis or trans), DQ8 homozygous, DQ8/DQ2.5, DQ 2.2 homozygous and genotype known as "half DQ2". Two different patterns of risks related to CD were identified. In the Basque Country and Navarre, the Mediterranean Area (Aragon, Catalonia, Valencia, Balearic Islands, and Murcia), the South of Spain (Andalucía and Extremadura), and the Canary Islands, higher frequency of DQ2.5 trans, and more than 80% of DQ2.5/DQ2.2 homozygosis were described. The Cantabrian Coast (Cantabria, Asturias, and Galicia) and Central Areas (Castilla-León and Castilla-La Mancha) showed a higher percentage of DQ2.5/DQ2.5 homozygosis and a lower DQ2.5 in trans frequency, as in Northern Europe. Madrid has an intermediate model between the two described above. 17 cases (0.8%) did not carry any CD risk haplotypes., Competing Interests: Declaration of Competing Interest The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper., (Copyright © 2023 American Society for Histocompatibility and Immunogenetics. Published by Elsevier Inc. All rights reserved.)
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- 2023
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7. Coeliac Disease Case-Control Study: Has the Time Come to Explore beyond Patients at Risk?
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Castillejo G, Ochoa-Sangrador C, Pérez-Solís D, Cilleruelo ML, Donat E, García-Burriel JI, Sánchez-Valverde F, Garcia-Calatayud S, Eizaguirre FJ, Martinez-Ojinaga E, Barros P, Leis R, Salazar JC, Barrio J, Peña-Quintana L, Luque V, Polanco I, Ribes C, and Roman E
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- Child, Humans, Case-Control Studies, Transglutaminases, Mass Screening, Immunoglobulin A, Autoantibodies, Celiac Disease diagnosis
- Abstract
The worldwide prevalence of asymptomatic coeliac disease (CD) is increasing, which is in part due to the routine screening of children with risk factors. Both symptomatic and asymptomatic patients with CD are at risk of long-term complications. The objective of this study was to compare the clinical characteristics of asymptomatic and symptomatic children at the time of CD diagnosis. A case-control study was conducted using data from a cohort of 4838 CD patients recruited from 73 centers across Spain between 2011 and 2017. A total of 468 asymptomatic patients (cases) were selected and matched by age and sex with 468 symptomatic patients (controls). Clinical data, including any reported symptoms, as well as serologic, genetic, and histopathologic data were collected. No significant differences were found between the two groups in most clinical variables, nor in the degree of intestinal lesion. However, the asymptomatic patients were taller (height z-score -0.12 (1.06) vs. -0.45 (1.19), p < 0.001) and were less likely to have anti transglutaminase IgA antibodies ≥ 10 times the upper normal limit (66.2% vs. 758.4%, p = 0.002). Among the 37.1% of asymptomatic patients who were not screened for CD due to the absence of risk factors, only 34% were truly asymptomatic, while the remaining 66% reported non-specific CD-related symptoms. Therefore, expanding CD screening to any child who undergoes a blood test could reduce the burden of care for some children, as many of those considered asymptomatic reported non-specific CD-related symptoms.
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- 2023
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8. Correlation of Anti-Tissue Transglutaminase Antibodies With the Mucosal Changes and IgA Status of Children With Celiac Disease.
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Donat E, Roca M, Castillejo G, Sánchez-Valverde F, García-Burriel JI, Martínez-Ojinaga E, Eizaguirre FJ, Barrio J, Cilleruelo ML, Pérez-Solís D, Ochoa-Sangrador C, Vecino-López R, Miranda-Cid MDC, García-Calatayud S, Torres-Peral R, Juste M, Armas H, Barros-García P, Leis R, Solaguren R, Salazar JC, García-Romero R, Ortigosa L, Peña-Quintana L, Urruzuno P, Codoñer-Franch P, Garcia-Casales Z, Masiques ML, Galicia-Poblet G, Crehuá-Gaudiza E, Balmaseda E, Rubio-Santiago J, Polanco-Allué I, Román-Riechmann E, and Ribes-Koninckx C
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- Adolescent, Child, Humans, Autoantibodies, Biopsy, Immunoglobulin A, Immunoglobulin G, Transglutaminases, Celiac Disease diagnosis
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Objectives: The objective of this study was to assess the association between serological markers and changes of the intestinal mucosa in children with celiac disease (CD)., Methods: Clinical data from CD patients under 15 years old were collected from the participating centers in an on-line multicenter nationwide observational Spanish registry called REPAC-2 (2011-2017). Correlation between anti-tissue transglutaminase antibodies (t-TGA) levels and other variables, including mucosal damage and clinical findings (symptoms, age, and gender), was assessed., Results: A total of 2955 of 4838 patients had t-TGA and a small bowel biopsy (SBB) performed for CD diagnosis. A total of 1931 (66.2%) patients with normal IgA values had a Marsh 3b-c lesion and 1892 (64.9%) had t-TGA Immunoglobulin A (IgA) ≥ 10 times upper limit of normal (ULN). There is a statistically significant association between t-TGA IgA levels and the degree of mucosal damage ( P < 0.001), the higher the t-TGA IgA levels the more severe the mucosal damage. Those patients who reported symptoms had more severe mucosal damage ( P = 0.001). On the contrary, there was a negative association between age and changes of the intestinal mucosa ( P < 0.001). No association was found with gender. Regarding the IgA-deficient patients, 47.4% (18 cases) had t-TGA Immunoglobulin A (IgA) ≥ 10 times ULN and a Marsh 3b-c lesion was observed in 68.4% (26 patients). No statistical relation was found between t-TGA IgG levels and the changes of the intestinal mucosa, neither a relation with age, gender, or symptoms., Conclusions: There is a positive correlation between t-TGA IgA levels and the severity of changes of the intestinal mucosa. Such correlation was not found in IgA-deficient patients who had positive t-TGA IgG serology. The results in this group of patients support the European Society for Paediatric Gastroenterology, Hepatology, and Nutrition recommendations about the need of performing a SBB in IgA-deficient individuals despite high t-TGA IgG levels., Competing Interests: The authors report no conflicts of interest., (Copyright © 2022 by European Society for European Society for Pediatric Gastroenterology, Hepatology, and Nutrition and North American Society for Pediatric Gastroenterology, Hepatology, and Nutrition.)
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- 2022
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9. Systematic Review and Meta-analysis of Histological Gastric Biopsy Aspects According to the Updated Sydney System in Children.
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Kalach N, Misak Z, Bontems P, Kori M, Homan M, Cabral J, Casswall T, Chong S, Cilleruelo ML, Faraci S, Megraud F, Papadopoulou A, Pehlivanoglu E, Raymond J, Rea F, Maria R, Roma E, Tavares M, Ugras M, Urbonas V, Urruzuno P, Gosset P, Creusy C, Delebarre M, and Verdun S
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- Biopsy, Child, Gastric Mucosa, Gastroscopy, Humans, Metaplasia pathology, Gastritis complications, Gastritis diagnosis, Gastritis epidemiology, Helicobacter Infections complications, Helicobacter Infections diagnosis, Helicobacter Infections epidemiology, Helicobacter pylori
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Objectives: A descriptive and comparative study of gastric histological aspects according to the updated Sydney classification (USC), obtained from Helicobacter pylori-positive versus H pylori-negative children referred for upper gastrointestinal endoscopy., Methods: The Prisma method was used to perform a systematic review and meta-analysis. Selection criteria were based on following key words USC, H pylori, children, endoscopy, or biopsy. Publication biases were assessed according to the Newcastle-Ottawa Scale, and a meta-regression analysis was done. The study was registered on the PROSPERO platform., Results: Between 1994 and 2017, 1238 references were found; 97 studies were retained for the systematic review with a total number of 25,867 children; 75 studies were selected for the meta-analysis concerning 5990 H pylori-infected and 17,782 uninfected children.H pylori-positive versus H pylori-negative children, according to the USC, showed significantly higher relative risk for gastric antral and corpus chronic inflammation, presence of neutrophils, and of lymphoid follicles, and gastric mucosa atrophy, whereas, intestinal metaplasia showed a significantly higher RR only in antral biopsies. The meta-regression analysis showed that H pylori-positive versus H pylori-negative children had significantly higher risk only for corpus activity according to age, recurrent abdominal pain, and geographical area of low H pylori prevalence., Conclusions: H pylori infection in children was associated with higher relative risk for gastric antral and corpus chronic inflammation, presence of neutrophils, lymphoid follicles, and rare gastric mucosa atrophy, whereas, rare intestinal metaplasia was only significantly higher in the antral area., (Copyright © 2021 by European Society for Pediatric Gastroenterology, Hepatology, and Nutrition and North American Society for Pediatric Gastroenterology, Hepatology, and Nutrition.)
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- 2022
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10. STAT6 Variants Associate With Relapse of Eosinophilic Esophagitis in Patients Receiving Long-term Proton Pump Inhibitor Therapy.
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Mougey EB, Nguyen V, Gutiérrez-Junquera C, Fernández-Fernández S, Cilleruelo ML, Rayo A, Borrell B, Román E, González-Lois C, Chao M, Al-Atrash H, and Franciosi JP
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- Adolescent, Child, Child, Preschool, Humans, Longitudinal Studies, Prospective Studies, Recurrence, STAT6 Transcription Factor genetics, Eosinophilic Esophagitis drug therapy, Eosinophilic Esophagitis genetics, Proton Pump Inhibitors therapeutic use
- Abstract
Background & Aims: Based on histologic features, variants in STAT6 are associated with a poor initial response to proton pump inhibitor (PPI) therapy in pediatric patients with eosinophilic esophagitis (EoE). We investigated whether these genetic variants are associated with a poor long-term response in children with EoE who initially responded to PPI therapy., Methods: We performed a prospective longitudinal cohort study of children ages 2 to 16 years who met the diagnostic criteria for EoE (≥15 eosinophils/high-power field [eos/hpf]), responded to 8 weeks of treatment with 2 mg/kg/d PPI (<15 eos/hpf), and whose dose then was reduced to 1 mg/kg/d PPI (maintenance therapy) for 1 year, at which point biopsy specimens were collected by endoscopy. Genomic DNA was isolated from formalin-fixed paraffin-embedded biopsy tissue and was genotyped for variants of STAT6. Remission of inflammation was assessed at eos/hpf thresholds of <15 and ≤5., Results: Among 73 patients who received 1 mg/kg/d PPI maintenance therapy for 1 year, 13 patients (18%) had 6 to 14 eos/hpf, 36 patients (49%) had 5 or fewer eos/hpf, and 24 patients (33%) relapsed to EoE (≥15 eos/hpf). Carriage of any of 3 STAT6 variants in linkage disequilibrium (r
2 ≥0.8; rs324011, rs167769, or rs12368672) was associated with a 2.3- to 2.8-fold increase in the odds of EoE relapse, and with a 2.8- to 4.1-fold increase in the odds of having 6 to 14 eos/hpf. For rs324011, the odds ratio [95% CI] for relapse was 2.77 [1.11, 6.92]; P = .029, and the odds ratio [95% CI] for having 6 to 14 eos/hpf was 3.06 [1.27, 7.36]; P = .012., Conclusions: Pediatric EoE patients who initially respond to PPI therapy and carry STAT6 variants rs324011, rs167769, or rs12368672 are at increased risk of relapse after 1 year of PPI maintenance therapy., (Copyright © 2021 The Authors. Published by Elsevier Inc. All rights reserved.)- Published
- 2021
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11. Proton-pump Inhibitor Response Prediction Using Esophageal microRNAs in Children With Eosinophilic Esophagitis.
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Cañas JA, Tabares A, Barbero C, García-Sánchez D, Sastre B, Rodrigo-Muñoz JM, Mahíllo-Fernández I, Rayo A, Borrell B, Cilleruelo ML, Román E, Fernandez-Fernandez S, Gutiérrez-Junquera C, and Del Pozo V
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- Biomarkers analysis, Child, Filaggrin Proteins, Humans, Male, Prospective Studies, Eosinophilic Esophagitis drug therapy, Eosinophilic Esophagitis genetics, MicroRNAs metabolism, Proton Pump Inhibitors therapeutic use
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Objectives: Eosinophilic esophagitis (EoE) is a chronic esophageal disease characterized by eosinophilic inflammation. Proton-pump inhibitors (PPI) induce disease remission but no predictive factors of PPI-responsiveness have been identified yet. So, a biomarker must be found to differentiate between responders (PPI-R) and nonresponder patients (PPI-NR) to PPI. Aims were to identify any molecular biomarker that could predict PPI responsiveness and to study molecular remission after PPI therapy., Methods: This prospective study enrolled 39 controls and 43 pediatric children with EoE from 2 hospitals, and they were treated with esomeprazole for 8 to 12 weeks. After therapy, patients were classified as either PPI-R or PPI-NR. Biopsies were collected and RNA, microRNAs, and proteins were isolated from them, measuring levels by qPCR and Western blot (WB). Also, miRNAs were evaluated in serum., Results: We found several esophageal miRNAs with different expression values between PPI-R and PPI-NR children, which can be used to discriminate them (area under curve = 0.90). No useful serum miRNAs were, however, identified. Also, these miRNAs were dysregulated in responder patients before and after PPI therapy. Moreover, we corroborated in this child population, that PPI-R displayed a significant decrease in eotaxin-3, IL-5, IL-13, periostin, and major basic protein (P < 0.05) and a significant increase in filaggrin levels after PPI treatment (P < 0.01)., Conclusions: Esophageal miRNA levels found are able to discriminate between both PPI-R and PPI-NR at baseline, and before and after treatment in PPI-R, so they could be used as biomarkers. Furthermore, we observed clinical and esophageal molecular restoration in PPI-R patients after PPI therapy.
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- 2020
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12. Octreotide-related exocrine pancreatic insufficiency (EPI) in congenital hyperinsulinism.
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Ros-Pérez P, Golmayo L, Cilleruelo ML, Gutiérrez C, Celaya P, Lacámara N, Martínez-Badás I, Güemes M, and Argente J
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- Congenital Hyperinsulinism blood, Congenital Hyperinsulinism genetics, Diazoxide therapeutic use, Exocrine Pancreatic Insufficiency therapy, Hemorrhage diagnosis, Hemorrhage etiology, Hemorrhage therapy, Humans, Infant, Male, Octreotide therapeutic use, Sulfonylurea Receptors genetics, Congenital Hyperinsulinism drug therapy, Exocrine Pancreatic Insufficiency chemically induced, Octreotide adverse effects
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Background Congenital hyperinsulinism (CH) is the most frequent cause of persistent hypoglycemia in the newborn. Octreotide, a long-acting somatostatin receptor analog (SSRA), is a second line treatment for diazoxide unresponsive CH patients. Although it has been found to be a safe and effective treatment, long-term benefits and side effects, have not been thoroughly evaluated. Case presentation Some authors have indicated that exocrine pancreatic insufficiency (EPI) is a common but under-recognized adverse reaction in adults treated with octreotide. However, no pediatric patient with SSRA-induced EPI has been reported to date. Here we report a case of an infant with diazoxide unresponsive, diffuse CH, caused by a heterozygous pathogenic paternally inherited mutation in the ABCC8 gene (NM_000352.4:c.357del), that developed exocrine pancreatic insufficiency and secondary vitamin K deficiency associated to chronic octreotide therapy. Conclusions We point out the atypical clinical onset with a cutaneous hemorrhagic syndrome, emphasizing the clinical relevance of this potential side effect.
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- 2020
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13. Health-related quality of life using specific and generic questionnaires in Spanish coeliac children.
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Barrio J, Cilleruelo ML, Román E, and Fernández C
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- Adolescent, Celiac Disease diet therapy, Child, Diet, Gluten-Free psychology, Female, Humans, Male, Parents psychology, Spain, Celiac Disease psychology, Quality of Life, Surveys and Questionnaires standards
- Abstract
Background: We aimed to compare the perception of health-related quality of life (HRQOL) and related factors in Spanish coeliac children and their parents, using two questionnaires, the generic KIDSCREEN-52 and the specific the Celiac Disease DUX (CDDUX), and to assess the correlation between them., Methods: Coeliac children, aged 8-18, who are members of the Madrid Coeliac Association (MCA) and their parents, answered the Spanish version of the CDDUX and KIDSCREEN-52 questionnaires via e-mail. CDDUX was answered by 266 children and 428 parents and KIDSCREEN-52 by 255 children and 387 parents. Linear regression models were fitted to evaluate the association of demographic and clinical factors with HRQOL scores. CDDUX scores were compared with the subjective perception of health status assessed by the first question of KIDSCREEN-52. The correlation between the questionnaires was analysed., Results: We found that the main factors that negatively affected HRQOL were having social or economic difficulties associated with following the diet and having transgression-related symptoms. The maximum correlation between the questionnaires was 0.309 and - 0.254 in parents and children respectively., Conclusions: Although there is a poor correlation between the two questionnaires, both agreed that the main concerns of the respondents were related to the social and economic difficulties of following the diet. It would be interesting to use both types of questionnaires in order to perform a more complete assessment of HRQOL in coeliac children., Trial Registration: Not applicable.
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- 2020
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14. Influence of the 2012 European Guidelines in Diagnosis and Follow-up of Coeliac Children With Selective IgA Deficiency.
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López RV, Cid CM, García GR, Romero RG, Cilleruelo ML, and Riechmann ER
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- Autoantibodies, Biopsy, Child, Follow-Up Studies, Humans, Immunoglobulin A, Retrospective Studies, Transglutaminases, Celiac Disease diagnosis, IgA Deficiency complications, IgA Deficiency diagnosis
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Objectives: The aim of the study was to describe diagnostic criteria used in children with coeliac disease (CD) and selective IgA deficiency; to determine if the publication of the 2012 ESPGHAN criteria prompted any changes; to evaluate the evolution of serological markers., Methods: Multicenter, retrospective, descriptive study of a cohort of children under 15 years with selective IgA deficiency diagnosed with CD (January 2006 to December 2016). Demographic, clinical, genetic, histological and IgG-based antibodies were collected at diagnosis and follow-up., Results: Eighty-six children were included, 60 diagnosed after the guide. Two groups were established: G1 (n = 63) and G2 (n = 23) with or without diagnostic biopsy respectively. In G1: 87.3% were symptomatic, 87.3% had human leukocyte antigan (HLA) DQ2/DQ8 typing (all positive), all had IgG serology positive (71.5% ATG, 35% EMA, 19% DPG, 9.5% AGA), and all had villous atrophy (Marsh-Oberhuber 2-3). Follow-up data were available in 58 children, 34 after 2 years on a gluten-free diet. Fifty-two percentage remained ATG IgG-positive despite good dietary adherence and symptom remission. Regarding G2: all were diagnosed post-2012, had typical symptoms, HLA DQ2/DQ8 positive and ATG IgG × 10 ULN. Additionally, EMA IgG was performed in 14 (60%), all positive., Conclusions: In our cohort of children with selective IgA deficiency and diagnosed with CD, children without a diagnostic biopsy suggests that IgG serology was considered the equivalent as IgA isotype, even when this is not addressed in the aforementioned guidelines. Great heterogeneity was observed in the IgG serology used at diagnosis. After 2 years of a gluten-free diet, half of children remained with a positive serology.
- Published
- 2020
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15. CYP2C19 and STAT6 Variants Influence the Outcome of Proton Pump Inhibitor Therapy in Pediatric Eosinophilic Esophagitis.
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Mougey EB, Williams A, Coyne AJK, Gutiérrez-Junquera C, Fernández-Fernández S, Cilleruelo ML, Rayo A, Echeverría L, Román E, González Lois C, Chao M, Al-Atrash H, Lima JJ, and Franciosi JP
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- Adolescent, Child, Child, Preschool, Eosinophilic Esophagitis genetics, Esophageal pH Monitoring, Female, Humans, Male, Prospective Studies, Proton Pump Inhibitors administration & dosage, Treatment Outcome, Cytochrome P-450 CYP2C19 genetics, Eosinophilic Esophagitis drug therapy, Proton Pump Inhibitors therapeutic use, STAT6 Transcription Factor genetics
- Abstract
Objective: Proton pump inhibitors (PPIs) are an effective treatment for eosinophilic esophagitis (EoE); however, only 30% to 60% of patients respond. Common genetic variants in CYP2C19 and STAT6 associate with PPI plasma concentration and magnitude of inflammatory response, respectively. Our objective was to determine if genetic variation in the genes for CYP2C19 and STAT6 influence differentiation between PPI responsive esophageal eosinophilia versus PPI nonresponsive EoE (PPI-REE, PPI-nonresponsive EoE)., Methods: Genomic DNA was isolated from 92 esophageal tissue biopsies collected from participants of a prospective clinical trial of high-dose PPI therapy for esophageal eosinophilia in children., Results: Of the 92 patients examined, 57 (62%) were PPI-REE and 35 (38%) were PPI-nonresponsive EoE. Forty-six of the 92 patients were further characterized by pH probe monitoring; there was no association between reflux index and carriage of CYP2C1917 (P = 0.35). In children who received a PPI dose between ≥1.54 and ≤2.05 mg/kg/day, binary logistic regression modeling showed that carriage of CYP2C1917 associated with PPI-nonresponsive EoE (odds ratio (OR) [95% confidence interval (CI)] = 7.71 [1.21, 49.11], P = 0.031). Carriage of STAT6 allelic variant rs1059513 predicts PPI-REE (OR [95% CI] = 6.16 [1.44, 26.4], P = 0.028), whereas carriage of STAT6 rs324011 synergizes with CYP2C1917 to predict PPI-nonresponsive EoE (rs324011 OR [95% CI] = 5.56 [1.33, 20.72], P = 0.022; CYP2C1917 OR [95% CI] = 8.19[1.42, 50.57], P = 0.023)., Conclusions: Common variants in CYP2C19 and STAT6 associate with a PPI-nonresponsive EoE outcome of PPI therapy for esophageal eosinophilia suggesting that response rates may be improved by adopting a genotype-guided approach to PPI dosing.
- Published
- 2019
- Full Text
- View/download PDF
16. Prevalence of Celiac Disease in a Long-term Study of a Spanish At-genetic-risk Cohort From the General Population.
- Author
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Fernández-Fernández S, Borrell B, Cilleruelo ML, Tabares A, Jiménez-Jiménez J, Rayo AI, Perucho T, and García-García ML
- Subjects
- Celiac Disease epidemiology, Celiac Disease genetics, Child, Child, Preschool, Female, Follow-Up Studies, Genetic Testing, Genotype, HLA-DQ Antigens genetics, Haplotypes, Humans, Longitudinal Studies, Male, Prevalence, Prospective Studies, Risk Factors, Spain, Celiac Disease diagnosis, Genetic Predisposition to Disease epidemiology
- Abstract
Objectives: To perform long-term celiac disease (CD) screening in an HLA-DQ2 (+) cohort from the general population and to assess the influence of risk genotypes on its development., Methods: In 2004, an HLA-DQ2 (+) cohort was selected. After the first CD screening at age 2 to 3 years, we performed a follow-up screening 8 to 10 years later. Antitransglutaminase 2 antibodies were determined using a rapid test kit. Results were confirmed by serum IgA antitransglutaminase 2 and IgA endomysial antibody determination. CD diagnosis was carried out by intestinal biopsies. Four HLA-DQ2 genotypic groups were used: G1: DQ2.5/DQ2.5 (G1A) or DQ2.5/ DQ2.2 (G1B); G2: DQ2.2/DQ7.5 (DQ2.5 trans); G3: DQ2.5/ X; G4: DQ2.2/X., Results: CD prevalence after 10 years of follow-up was 5.8% (95% confidence interval 3.8-8.7). One of every 3 HLA-DQ2(+) children carried at least 1 haplotype DQ2.2 or DQ7. The homozygous genotype DQ2.5/DQ2.5 and the HLA-DQ2.5 trans genotype increased CD risk 4- and 3-fold, respectively. The homozygous genotype DQ2.5/ DQ2.2 did not increase the CD risk. Children carrying G1 or G2 genotypes were diagnosed with CD earlier and more frequently during the follow-up compare with those carrying G3 or G4 genotypes. Approximately 81% of children with spontaneous antibody negativization after the first screening maintained negative antibodies., Conclusions: A repeated screening of at-risk children during their follow-up allowed us to diagnose new CD cases. In our cohort, HLA- DQ2.5 trans genotype conferred a higher risk in the development of CD than HLA- DQ2.5/DQ2.2. The majority of children with potential CD and CD autoimmunity at 10 years of age remained healthy.
- Published
- 2019
- Full Text
- View/download PDF
17. Health-related quality of life in Spanish coeliac children using the generic KIDSCREEN-52 questionnaire.
- Author
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Barrio J, Cilleruelo ML, Román E, and Fernández C
- Subjects
- Adolescent, Child, Cross-Sectional Studies, Female, Humans, Language, Male, Parents, Severity of Illness Index, Social Support, Spain, Surveys and Questionnaires, Celiac Disease psychology, Quality of Life psychology
- Abstract
The aim of this study is to assess the impact of coeliac disease (CD) on health-related quality of life (HRQOL) using the generic KIDSCREEN-52 questionnaire in a group of Spanish children aged 8-18 years and their parents. For this cross-sectional study, coeliac children in the targeted age range, who are members of the Madrid Coeliac Association (MCA), were invited to participate. The Spanish version of the generic KIDSCREEN-52 questionnaire was administered via e-mail. Scores (on a scale from 1 to 100) were expressed as the mean and standard deviation (SD). Demographic and clinical variables related to HRQOL were also assessed. The questionnaire was completed by 434 children and/or their parents. Respondents gave scores of over 50 to seven quality of life domains. Mean scores were significantly higher in children than in their parents for six out of ten domains. In contrast, parents awarded significantly higher scores to the "social support and peers" domain than children did. Significantly lower QOL scores were reported by girls, children aged 16-18 years, children older than 7 years at the time of CD diagnosis, and respondents who did not adhere to the prescribed diet or had difficulty in doing so., Conclusion: Overall, the KIDSCREEN-52 questionnaire revealed that CD had no substantial negative impacts on the children's QOL. However, some concerns from the children's perspective were identified, such as issues regarding relationships with their peers. These issues will need to be addressed in order to improve QOL in children with CD. What is Known: • According to the Spanish version of the specific CDDUX, parents and children felt CD had no substantial negative impacts on their HRQOL. What is New: • According to the generic KIDSCREEN-52 questionnaire, QOL in Spanish coeliac children does not seem to be negatively affected by the disease in most general aspects of life. • Parents have a worse perception of their children's HRQOL than their children themselves.
- Published
- 2018
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- View/download PDF
18. Long-term Treatment With Proton Pump Inhibitors Is Effective in Children With Eosinophilic Esophagitis.
- Author
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Gutiérrez-Junquera C, Fernández-Fernández S, Cilleruelo ML, Rayo A, Echeverría L, Borrell B, and Román E
- Subjects
- Administration, Oral, Adolescent, Child, Child Health Services, Child, Preschool, Drug Administration Schedule, Eosinophilic Esophagitis pathology, Esomeprazole administration & dosage, Esophagoscopy, Female, Humans, Infant, Male, Prospective Studies, Proton Pump Inhibitors administration & dosage, Recurrence, Remission Induction, Spain, Treatment Outcome, Eosinophilic Esophagitis drug therapy, Esomeprazole therapeutic use, Proton Pump Inhibitors therapeutic use
- Abstract
Objectives: Proton pump inhibitor (PPI)-responsive eosinophilic esophagitis (EoE) is frequently observed in children, but data on long-term treatment are scarce. The objective of this study is to evaluate the long-term efficacy and safety of PPIs in children with EoE., Methods: This prospective study enrolled children with EoE and histological remission to an 8-week esomeprazole trial (1 mg/kg/dose, twice daily). Esomeprazole was maintained at 1 mg/kg/day for 1 year. Symptom recurrence and adverse events were monitored and a follow-up endoscopy was performed at 12 months. Complete histological remission was defined as ≤5 eosinophils/high-power field (eos/hpf), and partial histological remission as >5 and <15 eos/hpf. Patients had no concomitant dietary restrictions or topical steroid., Results: Fifty-seven children were included. Histological remission on maintenance PPI therapy was present in 40 children (70.1%; 95% CI 56.5-81.5). Long-term remission rate was higher in children with initial complete histological remission than in those with partial remission (81% vs 50%, P = 0.014). Forty-nine children (86%) remained asymptomatic. Pretreatment clinical and histological findings and median PPI dose/kg/day were similar between relapsers and nonrelapsers. Eleven out of 12 children (91.6%) receiving esomeprazole 0.5 mg · kg · day for 12 additional months remained in remission. Mild and transient side effects without requiring PPI avoidance were observed in 5 children., Conclusions: Up to 70% of children with PPI-responsive EoE remain in histological and clinical remission on a low-dose maintenance treatment at 1-year follow-up, with adequate safety profile. Complete histological remission to an 8-week PPI trial was associated with higher probability of histological remission on maintenance therapy.
- Published
- 2018
- Full Text
- View/download PDF
19. The Role of Proton Pump Inhibitors in the Management of Pediatric Eosinophilic Esophagitis.
- Author
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Gutiérrez-Junquera C, Fernández-Fernández S, Cilleruelo ML, Rayo A, and Román E
- Abstract
Eosinophilic esophagitis (EoE) is a chronic, local, immune-mediated disorder characterized by symptoms of esophageal dysfunction and the presence of a dense eosinophilic infiltrate in the esophageal mucosa. Consensus diagnostic recommendations for EoE diagnosis included absence of histological response to a proton-pump inhibitor (PPI) trial, to exclude gastro-oesophageal reflux disease (GERD)-associated esophagitis. This recommendation exposed an entity known as "proton pump inhibitor-responsive esophageal eosinophilia" (PPI-REE), which refers to patients with EoE phenotype who are PPI-responsive and do not present GERD. In recent years, there is evidence which indicates that PPI-REE is a sub-phenotype of EoE with similar clinical, endoscopic, histological and genetic characteristics, as well as Th2-related inflammatory response. As a result, PPIs should be considered another treatment for EoE and not a diagnostic tool. PPI-REE was originally described in a case series which included two children and in two retrospective pediatric series. Later, a prospective pediatric study showed a high rate of response to PPIs at high doses with long-term maintenance at lower doses. PPI monotherapy in children with esophageal eosinophilia (EE) has been observed to reduce eotaxin-3 expression in epithelial cells and to practically reverse the allergy and inflammatory transcriptome. These data reveal that PPIs are also an effective treatment for EoE in pediatric patients, although more studies are necessary in order to define the best induction and maintenance treatment regimen, the long-term safety profile and their influence on the occurrence of fibrosis and esophageal remodeling.
- Published
- 2018
- Full Text
- View/download PDF
20. High Prevalence of Response to Proton-pump Inhibitor Treatment in Children With Esophageal Eosinophilia.
- Author
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Gutiérrez-Junquera C, Fernández-Fernández S, Cilleruelo ML, Rayo A, Echeverría L, Quevedo S, Bracamonte T, and Román E
- Subjects
- Adolescent, Child, Child, Preschool, Deglutition Disorders pathology, Drug Administration Schedule, Eosinophilic Esophagitis pathology, Esomeprazole administration & dosage, Esophagoscopy, Female, Humans, Infant, Male, Prevalence, Prospective Studies, Proton Pump Inhibitors administration & dosage, Treatment Outcome, Deglutition Disorders drug therapy, Eosinophilic Esophagitis drug therapy, Esomeprazole therapeutic use, Proton Pump Inhibitors therapeutic use
- Abstract
Objectives: Proton-pump inhibitor-responsive esophageal eosinophilia is a newly recognized entity with an unclear prevalence in children, as only retrospective data are available. The aim of this study was to determine the prevalence and clinical features of proton-pump inhibitor-responsive esophageal eosinophilia in children., Methods: This prospective study enrolled patients with esophageal symptoms and esophageal eosinophilic counts as 15 or more than 15 eos/hpf (eosinophils per high-power field). Children received treatment with esomeprazole 1 mg · kg per dose twice daily for 8 weeks and the endoscopy was repeated. Complete response to proton-pump inhibitor (PPI) was defined as 5 or less than 5 eos/hpf, and a partial response as >5 and <15 eos/hpf in post-treatment biopsies., Results: Fifty-one children (74.5% boys) were included. Histological response was observed in 35 children (68.6%): 24 children (47%) had a complete response and 11 children (21.6%) had a partial response. Only 16 children (31.4%) were diagnosed with eosinophilic esophagitis (EoE). There were no differences in history of atopy, allergy tests, pH study results, and endoscopic scores. Clinical symptoms were similar, with the exception of food impaction, which was more frequent in children with EoE (56.2% vs 20%, P = 0.01). The mean pretreatment peak eosinophil count was higher in patients with EoE (74.8 ± 36.2 vs 46.3 ± 30.7, P = 0.007). Eleven of the 14 patients (78.6%) on a lower PPI treatment maintenance dose remained in clinicopathologic remission at 1-year follow-up., Conclusions: A significant proportion of children with esophageal eosinophilia responded to high dose PPI treatment. Clinical, endoscopic, and pH study results were similar, with exception of patients with EoE, who were more likely to experience food impaction and have higher esophageal eosinophil counts.
- Published
- 2016
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21. Prevalence and Natural History of Celiac Disease in a Cohort of At-risk Children.
- Author
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Cilleruelo ML, Fernández-Fernández S, Jiménez-Jiménez J, Rayo AI, and de Larramendi CH
- Subjects
- Celiac Disease genetics, Celiac Disease pathology, Child, Child, Preschool, Cohort Studies, Female, Genetic Predisposition to Disease, HLA-DQ Antigens blood, Humans, Male, Prevalence, Risk Factors, Sex Factors, Spain epidemiology, Celiac Disease epidemiology, HLA-DQ Antigens genetics
- Abstract
Objectives: To assess the prevalence and clinical presentation of celiac disease (CD) in a cohort of children with HLA-DQ2 positive and evaluate the risk factors in the development of CD., Methods: Between July 2004 and July 2005, parents of all healthy full-term newborns in our hospital were invited to participate. HLA-DQ2 was tested in blood sample of the umbilical cord. A point of contact serological test was performed on children between 2 and 3 years of age. Positive results were confirmed by serum anti-transglutaminase 2 and endomysial antibodies. Children with high autoantibody titers underwent an intestinal biopsy. Children of the cohort diagnosed with CD before the screening study were included. Sex, mode of delivery, breast-feeding duration, and age of gluten introduction were studied., Results: Of 1291 children, 362 were HLA-DQ2 positive and 262 participated in the study. CD was diagnosed in 4.1% (95% confidence interval (CI) 1.9-6.3). In the whole cohort, 60% had gastrointestinal symptoms, 7% poor weight gain, and 33% were asymptomatic. Five children with potential CD and 6 with CD autoimmunity became negative (42.3%) and are still negative after 5 to 7 years. Female sex was at-risk factor odds ratio 5.7 (95% CI 1.5-20.9), whereas breast-feeding during gluten introduction had a protective effect odds ratio 0.1 (95% CI 0.01-0.8)., Conclusions: Prevalence of CD in this cohort was 4%, half of whom had digestive symptoms. Because a high proportion of children showed a spontaneous disappearance of antibodies, prevalence studies of CD in young children should be based on intestinal damage so as not to overestimate results.
- Published
- 2016
- Full Text
- View/download PDF
22. Spanish national registry of celiac disease: incidence and clinical presentation.
- Author
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Cilleruelo ML, Roman-Riechmann E, Sanchez-Valverde F, Donat E, Manuel-Ramos J, Martín-Orte E, López MJ, García-Novo D, García S, Pavón P, Martín M, Ortigosa L, Barrio J, Gutierrez C, Espìn B, Castillejo G, Peña-Quintana L, Hualde I, Sebastián M, Calvo C, Fernández S, De Manueles J, Armas H, Urruzuno-Tellerias P, Juste M, Bousoño C, and Ribes-Koninckx C
- Subjects
- Body Weight, Celiac Disease blood, Celiac Disease complications, Celiac Disease pathology, Child, Child, Preschool, Female, HLA-DQ Antigens blood, Humans, Incidence, Male, Phenotype, Registries, Spain epidemiology, Antibodies blood, Celiac Disease epidemiology, Intestinal Mucosa immunology, Intestinal Mucosa pathology, Lymphocytes metabolism
- Abstract
Objectives: The aim of this study was to assess the incidence and clinical pattern of celiac disease (CD) presently diagnosed in Spanish children., Methods: A prospective, multicenter, nationwide registry of new cases of CD in children <15 years was conducted from June 1, 2006 to May 31, 2007. The parameters studied were age at diagnosis, sex, clinical symptoms, associated diseases, nutritional status, CD serology, histological lesions, and HLA-DQ2/-DQ8. The crude incidence rate of CD was calculated as new cases per 1000 live births and as new cases per 100,000 person-years <15 years of age., Results: A total of 974 new cases of CD were included. The median age at diagnosis was 2.3 years; 39.5% of CD diagnoses occurred in the first 2 years, 42% between 2 and 6, and 18.4% from 6 to 15. Total number of cases in each age group was 385, 409, and 180, respectively. Regarding clinical presentation 70.9% showed classical symptoms, 21.9% were nonclassical, and 7% were asymptomatic. A total of 95.7% of 931, 94.7% of 611, and 86.7% of 651 children tested positive, respectively, for immunoglobulin A (IgA) anti-transglutaminase type 2 antibodies, IgA endomysial antibodies, and IgA anti-gliadin antibodies. Villous atrophy was observed in 92.4% and increased intraepithelial lymphocytes with crypt hyperplasia in 3.3%. Of the children, 55% had normal growth, and 3.4% were overweight. The HLA phenotype was DQ2: 88.3%, DQ2/DQ8: 8.4%, and DQ8: 2.3%. The incidence rate was 7.9 cases of CD per 1000 live births and 54 cases per 100,000 person-years., Conclusions: In Spain, the most frequent clinical presentation of CD is the classical form, mainly diagnosed during the first 2 years of life. The observed incidence of CD in Spanish children is much higher than the present CD incidence rates observed in other European countries.
- Published
- 2014
- Full Text
- View/download PDF
23. Interplay between human leukocyte antigen genes and the microbial colonization process of the newborn intestine.
- Author
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De Palma G, Capilla A, Nadal I, Nova E, Pozo T, Varea V, Polanco I, Castillejo G, López A, Garrote JA, Calvo C, García-Novo MD, Cilleruelo ML, Ribes-Koninckx C, Palau F, and Sanz Y
- Subjects
- Bacteroides growth & development, Bacteroides isolation & purification, Flow Cytometry, Genotype, HLA-DQ alpha-Chains, HLA-DQ beta-Chains, Humans, In Situ Hybridization, Fluorescence, Infant, Infant, Newborn, Lactococcus growth & development, Lactococcus isolation & purification, Prevotella growth & development, Prevotella isolation & purification, Streptococcus growth & development, Streptococcus isolation & purification, HLA-DQ Antigens genetics, Intestines microbiology
- Abstract
Coeliac disease (CD) development involves genetic (HLA-DQ2/DQ8) and environmental factors. Herein, the influence of the HLA-DQ genotype on the gut colonization process of breast-fed children was determined. A cohort of 20 newborns, with at least one first-degree relative with CD, were classified according to their HLA-DQ genotype into high, intermediate and low genetic risk groups, showing 24-28%, 7-8% and less than 1% probability to develop CD, respectively. Faecal microbiota was analysed at 7 days, 1 and 4 months of children's age by fluorescence in situ hybridization. When considering all data, Gram-negative bacteria and Bacteroides-Prevotella group proportions were higher (P<0.05) in the high than in the intermediate and low genetic risk groups. E. coli, Streptococcus-Lactococcus, E. rectale-C. coccoides, sulphate-reducing bacteria, C. lituseburense and C. histolyticum group proportions were also significantly higher (P<0.05) in the high than in the low genetic risk group. Correlations between these bacterial groups and the genetic risk were also detected (P<0.05). In addition, the number and type of CD relative seemed to influence (P<0.050) these bacterial proportions in children at CD risk. At 4 months of age, similar relationships were established between the high genetic risk to develop CD and the proportions of Streptococcus-Lactococcus (P<0.05), E. rectale-C. coccoides (P<0.05), C. lituseburense (P<0.05), C. histolyticum (P<0.05), Bacteroides-Prevotella (P<0.10) groups and total Gram-negative bacteria (P<0.05). The results suggest a relationship between HLA-DQ genes and the gut microbial colonization process that could lead to a change in the way this disorder is investigated.
- Published
- 2010
24. Prevalence of CagA and VacA antibodies in children with Helicobacter pylori-associated peptic ulcer compared to prevalence in pediatric patients with active or nonactive chronic gastritis.
- Author
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Alarcón T, Martínez MJ, Urruzuno P, Cilleruelo ML, Madruga D, Sebastian M, Domingo D, Sanz JC, and López-Brea M
- Subjects
- Adolescent, Blotting, Western methods, Child, Child, Preschool, Chronic Disease, Female, Gastritis blood, Gastritis epidemiology, Helicobacter Infections blood, Helicobacter Infections epidemiology, Helicobacter pylori immunology, Humans, Male, Peptic Ulcer blood, Peptic Ulcer epidemiology, Predictive Value of Tests, Sensitivity and Specificity, Seroepidemiologic Studies, Spain epidemiology, Antibodies, Bacterial blood, Antigens, Bacterial immunology, Bacterial Proteins immunology, Gastritis immunology, Helicobacter Infections immunology, Peptic Ulcer immunology
- Abstract
VacA and CagA serological responses were detected in pediatric patients: 44 and 56%, respectively, in peptic ulcer (PU) patients, 33.3 and 44.4% in active chronic gastritis (ACG) patients, and 23.2 and 39.2% in non-ACG patients. Higher seroprevalence to CagA+VacA and to CagA+VacA+35-kDa antigen was found among PU patients. However, a low level of sensitivity and specificity was found for indirect detection of PU patients.
- Published
- 2000
- Full Text
- View/download PDF
25. [Caroli's syndrome. Report of a case beginning in childhood with favorable course].
- Author
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Rivero MJ, Román E, Cilleruelo ML, Sánchez F, and Barrio J
- Subjects
- Age of Onset, Caroli Disease complications, Child, Follow-Up Studies, Humans, Male, Remission Induction, Caroli Disease therapy
- Abstract
Caroli's disease is a rare entity that is included in the fibropolycystic abnormalities of the bile ducts. Ultrasonographic patterns consist of evident dilatation of the bile ducts. Although it is thought to be a congenital disease, it usually presents in young adults and few cases have been reported in children. We present the case of a 10-year-old boy with Caroli's syndrome (Caroli's disease, congenital hepatic fibrosis and polycystic renal disease). Evolution was favorable.
- Published
- 2000
26. [The treatment of children with Helicobarter pylory by triple therapy can be shortened without the risk of losing it eradicating efficacy. Letter].
- Author
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Cilleruelo ML, Urruzuno P, Martínez MJ, Madruga D, Sebastián M, Román E, Barrio A, Manzanares J, Medina E, Sarría J, and Sanz JC
- Subjects
- Adolescent, Amoxicillin administration & dosage, Anti-Bacterial Agents administration & dosage, Antitrichomonal Agents administration & dosage, Child, Child, Preschool, Drug Therapy, Combination, Helicobacter Infections microbiology, Helicobacter pylori isolation & purification, Humans, Metronidazole administration & dosage, Organometallic Compounds administration & dosage, Penicillins administration & dosage, Time Factors, Amoxicillin therapeutic use, Anti-Bacterial Agents therapeutic use, Antitrichomonal Agents therapeutic use, Helicobacter Infections drug therapy, Metronidazole therapeutic use, Organometallic Compounds therapeutic use, Penicillins therapeutic use
- Published
- 1996
27. [Nutrient and food consumption by ages and gender in school children from the Community of Madrid (CAENPE)].
- Author
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Vázquez C, de Cos AI, Martínez P, Jaunsolo MA, Román E, Gómez C, López T, Hernáez I, Seijas V, Ramos V, Cilleruelo ML, Sarrión D, García JJ, and López Nomdedeu C
- Subjects
- Adolescent, Child, Cross-Sectional Studies, Female, Humans, Male, Nutrition Surveys, Spain, Urban Population, Diet, Feeding Behavior
- Abstract
Unlabelled: The objective of the present work was to know the consume of foodstuffs and nutrients among schoolchildren in the Madrid Autonomous Community and also the relative contribution of each group of foodstuffs to the overall intake of macronutrients. This study had a sectional observational design involving 2,608 children (51% males and 49% females) with ages ranging from 6 to 14 years who were randomly recruited from conglomerate of schools, according to socio-economic status and rural/urban residence. The inquiry on nutrients in 4 non consecutive days was analyzed (2 "24 hour-recall" and 2 "intake recall")., Results: a) the consume of proteins, lipids, and carbohydrates represented 17%, 43% and 40% of the total caloric value, respectively; b) the intake of meats, sausages, sweets, tidbits, and processed products was very high. This fact is in origin of the excessive total and saturated lipid intake; c) there was a scarce intake of vegetables, cereals and potatoes among children of all ages and dairy products in children older than 12 years; d) the intake of legumes, eggs and fruits was appropriate; e) males consumed proportionally higher amounts than females of the following groups of foodstuffs: cereals, sweets and tidbits. The consume of meat, sausages, fish, eggs, potatoes, dry fruits and dairy products was similar in both sexes. Females consumed proportionally more fat foodstuffs, vegetables, fruits, legumes and processed products; f) females consumed a diet with a still higher lipid and protein content than males. These findings can serve as a basis to develop educational guidelines with a practical impact on family and school menus.
- Published
- 1996
28. [Efficacy and failure of phenobarbital in the prevention of febrile convulsions. Importance of familial antecedents].
- Author
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Palencia R, Cilleruelo ML, Tresierra F, and Blanco A
- Subjects
- Age Factors, Epilepsy genetics, Female, Fetal Hypoxia complications, Humans, Male, Pregnancy, Recurrence, Seizures, Febrile etiology, Seizures, Febrile genetics, Phenobarbital therapeutic use, Seizures, Febrile prevention & control
- Abstract
The authors analyze the value of phenobarbital for prophylaxis of new convulsions in a group of 370 patients who had previously had febrile seizures. Percentage of recurrences in phenobarbital treated cases (4 +/- 0,6 mg/Kg/d) was 18,67%, versus 55,89% in untreated children (p less than 0.001). The presence of family history of epileptic or febrile seizures is a risk factor (p less than 0.01) that facilitates failure of phenobarbital prophilaxis in future febrile seizures.
- Published
- 1983
29. [Current treatment and prevention of juvenile gastroenteritis].
- Author
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Alonso Franch M, Cilleruelo ML, Tornel J, Ortiz de Lejarazu R, and Sánchez Villares E
- Subjects
- Bacterial Toxins pharmacology, Child, Child, Preschool, Colon physiopathology, Diarrhea, Infantile microbiology, Diarrhea, Infantile prevention & control, Fluid Therapy, Gastroenteritis microbiology, Gastroenteritis prevention & control, Humans, Immunization, Infant, Infant, Newborn, Intestines microbiology, Diarrhea, Infantile therapy, Gastroenteritis therapy
- Abstract
The present knowledges about the damage mechanisms of germs in infectious diarrhea, on which are based the therapy procedures are reviewed. The management consist in oral rehydratation and precocius oral feeding as the main therapy. A scheme of treatment and the future perspectives in Valladolid during the last 9 months (609 fecal cultures) is reported. The participation of isolated germs in the pathogenesis of diarrhea is discussed. Since the acquisition of new methods to detect compylobacter, yersinia and rotavirus, the number of positive fecal cultures has increased in our medium.
- Published
- 1983
30. Increased serum beta2-microglobulin levels in active celiac disease.
- Author
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Blanco A, Alonso M, Cilleruelo ML, Solis P, Calvo C, and Sanchez Villares E
- Subjects
- Acute-Phase Proteins, Blood Proteins analysis, Celiac Disease immunology, Celiac Disease pathology, Child, Child, Preschool, Humans, Immunity, Cellular, Infant, Intestinal Mucosa pathology, Time Factors, Celiac Disease blood, beta 2-Microglobulin analysis
- Abstract
Sixty-eight sera from 36 children with celiac disease (CD) were collected in a 4-year study, and the levels of beta2-microglobulin (beta2M), orosomucoid, alpha1-antitrypsin, and C-reactive protein (CRP) were measured. A simultaneous intestinal biopsy was taken in all the patients and used to classify them. The values of beta2M were 1.94 +/- 0.49 mg/L in group A (inactive CD with normal intestinal biopsy) and 2.58 +/- 0.76 mg/L in group B (active CD with flat mucosa). These levels were higher than in the normal controls (1.87 +/- 0.49 mg/L, p less than 0.005) and in the other 19 patients with chronic diarrhea of another cause (1.72 +/- 0.63 mg/L, p less than 0.005). CRP values were generally normal; orosomucoid and alpha1-antitrypsin were variable and they did not correlate with beta2M levels. Beta2M was increased at the time of diagnosis in seven of eight (87.5%) of the CD children and only in three of 19 (15.7%) of nonceliac patients with chronic diarrhea of another cause (p less than 0.005). In 12 children with CD, serial studies were possible. The highest beta2M level coincided with a flat mucosa in 10 cases. The increase of beta2M could be related to the intestinal infiltration or even to the lymphocyte activation. It may also be a useful noninvasive method for evaluating the activity of CD.
- Published
- 1985
- Full Text
- View/download PDF
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