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1.
JPGN Rep ; 5(2): 135-139, 2024 May.
Artículo en Inglés | MEDLINE | ID: mdl-38756126

RESUMEN

Food insecurity is a rising concern for US households and leads to adverse child health outcomes. Pediatric gastroenterology providers are uniquely equipped to help guide families experiencing this challenge given their specialized training in nutritional support and dietary therapy for disease management. Hence, this study aimed to evaluate food insecurity screening practices from the perspectives of patient caregivers and healthcare providers in a tertiary pediatric gastroenterology practice. A survey was administered to 1279 caregivers and 121 providers. Of the 248 completed caregiver responses, 10%-15% reported being asked about food insecurity. Among the 36 healthcare provider responses, 53% expressed comfort in conducting food insecurity screening but only 14% routinely screened. The most cited barrier to screening was the lack of readily available patient resources. Further research is imperative to address these screening barriers and assess the impact of food insecurity screening and interventions on pediatric gastrointestinal health outcomes.

2.
Artículo en Inglés | MEDLINE | ID: mdl-38615728

RESUMEN

BACKGROUND & AIMS: Celiac disease (CD) mass screening remains controversial in part because of a paucity of data to support its benefit. The Autoimmunity Screening for Kids study is a mass screening study for pediatric CD and type 1 diabetes in Colorado. METHODS: This study prospectively follows up children ages 1 to 17 years who screened positive for tissue transglutaminase IgA autoantibodies in the Autoimmunity Screening for Kids study subsequently referred for diagnostic evaluation. Children diagnosed with CD by biopsy or serologic criteria were included in this study. Evaluation at baseline and 12 month follow-up evaluation included demographics, laboratory studies, symptoms, health-related quality of life, anxiety/depression, and gluten-free diet adherence. Paired Student t test, chi-square, and Wilcoxon sign rank tests compared baseline and follow-up data. For symptom scores, odds of improvement were assessed. RESULTS: Of the 52 children with CD enrolled, 42 children completed 12-month follow-up evaluation. On the symptom questionnaire completed at diagnostic evaluation, 38 of 42 children reported 1 or more symptoms. CD mean symptom severity and frequency scores improved from baseline to follow-up evaluation (P < .001). Reported health-related quality of life scores improved among caregivers (P = .002). There was no significant change in reported anxiety or depression. Iron deficiency without anemia was common at baseline (21 of 24 children; 87.5%) and normalized at follow-up evaluation (11 of 21 children; 52.3%). Twenty-six of 28 families reported good or excellent gluten-free diet adherence. CONCLUSIONS: This novel study of children with CD identified through a mass screening program demonstrated improvement in symptoms, quality of life, and iron deficiency after 1 year follow-up evaluation. This demonstrates that there may be benefit to CD mass screening.

3.
Lancet Child Adolesc Health ; 8(1): 63-74, 2024 Jan.
Artículo en Inglés | MEDLINE | ID: mdl-37972632

RESUMEN

Paediatric prospective studies of coeliac disease with longitudinal collection of biological samples and clinical data offer a unique perspective on disease risk. This Review highlights the information now available from international paediatric prospective studies on genetic and environmental risk factors for coeliac disease. In addition, recent omics studies have made it possible to study complex interactions between genetic and environmental factors and thereby further our insight into the causes of the disease. In the future, paediatric prospective studies will be able to provide more detailed risk prediction models combining genes, the environment, and biological corroboration from multiomics. Such studies could also contribute to biomarker development and an improved understanding of disease pathogenesis.


Asunto(s)
Enfermedad Celíaca , Niño , Humanos , Enfermedad Celíaca/epidemiología , Enfermedad Celíaca/genética , Estudios Prospectivos , Biomarcadores
5.
Curr Opin Gastroenterol ; 39(6): 455-462, 2023 Nov 01.
Artículo en Inglés | MEDLINE | ID: mdl-37678180

RESUMEN

PURPOSE OF REVIEW: As incidence and prevalence of celiac disease is increasing, subclinical and asymptomatic presentations are more commonly identified through celiac disease screening. However, the United States Preventive Services Task Force released a statement in 2017 maintaining that there is insufficient evidence to recommend general population screening for celiac disease for asymptomatic individuals. This review summarizes the current available evidence on celiac disease screening. RECENT FINDINGS: Literature demonstrates that by limiting screening to individuals with recognized symptoms, celiac disease diagnosis is frequently delayed or missed entirely. Most individuals with screening-identified celiac disease have previously unrecognized symptoms that improve through treatment with a gluten-free diet. Screening-identified individuals also demonstrate signs of impaired nutrition, growth, bone health, and quality of life which improve with treatment. Overall, celiac disease screening is viewed favorably by those identified through celiac disease screening programs. SUMMARY: Individuals with screening-identified celiac disease may still incur complications from untreated disease and receive benefit from treatment with a gluten-free diet. More data is needed to determine the cost effectiveness of different mass screening approaches that incorporate the societal perspective towards screening.


Asunto(s)
Enfermedad Celíaca , Humanos , Estados Unidos , Enfermedad Celíaca/diagnóstico , Enfermedad Celíaca/epidemiología , Enfermedad Celíaca/complicaciones , Calidad de Vida , Dieta Sin Gluten , Tamizaje Masivo , Prevalencia
6.
J Prim Care Community Health ; 14: 21501319231189147, 2023.
Artículo en Inglés | MEDLINE | ID: mdl-37501515

RESUMEN

There are few estimates of the seroprevalence of SARS-CoV-2 antibodies among children in the United States. We measured vaccine and infection induced seroprevalence among nearly 5000 healthy 1 to 17-year-old children in Colorado from 2020 to 2021. By December 2021, 89% of older children, ages 12 to 18, had antibodies detected. The increase was largely driven from vaccination rather than infection.


Asunto(s)
COVID-19 , SARS-CoV-2 , Niño , Humanos , Adolescente , Lactante , Preescolar , Colorado/epidemiología , Prevalencia , Estudios Seroepidemiológicos , COVID-19/epidemiología , Anticuerpos Antivirales
7.
Am J Gastroenterol ; 118(9): 1698-1700, 2023 09 01.
Artículo en Inglés | MEDLINE | ID: mdl-37159249

RESUMEN

INTRODUCTION: Recent reports suggest severe acute respiratory syndrome coronavirus-2 (SARS-CoV-2) infections may increase the risk of celiac disease autoimmunity. This study aims to evaluate potential associations between coronavirus disease 2019 infection and tissue transglutaminase autoantibodies (TGA) immunoglobulin A. METHODS: From 2020 to 2021, cross-sectional screening for SARS-CoV-2 antibodies and TGA was offered to 4,717 children in Colorado through the Autoimmunity Screening for Kids study. Multivariable logistic regression assessed association between previous SARS-CoV-2 infection and TGA positivity. RESULTS: Previous SARS-CoV-2 infection was not associated with TGA positivity (odds ratio 1.02, 95% confidence interval 0.63-1.59; P = 0.95). DISCUSSION: In this large-scale analysis, previous SARS-CoV-2 infection was not associated with celiac disease autoimmunity in Colorado children.


Asunto(s)
COVID-19 , Enfermedad Celíaca , Humanos , Niño , Adolescente , Autoinmunidad , Enfermedad Celíaca/diagnóstico , SARS-CoV-2 , Estudios Transversales , Transglutaminasas , Autoanticuerpos
8.
J Pediatr Gastroenterol Nutr ; 77(2): 184-190, 2023 08 01.
Artículo en Inglés | MEDLINE | ID: mdl-37184455

RESUMEN

OBJECTIVES: Patients with Trisomy 21 (T21) commonly have gastrointestinal symptoms and diseases that prompt evaluation with esophagogastroduodenoscopy (EGD). Our objective is to characterize duodenal histological abnormalities in these patients when undergoing EGD. A secondary aim is to explore associations of histologic findings with different therapies. METHODS: Patients 30 years old or younger with T21 who underwent EGD from 2000 to 2020 at 6 hospitals were included in this retrospective cohort study. Duodenal biopsies were categorized based on reported histopathology findings as normal or abnormal. Abnormal pathology reports were reviewed and categorized into villous atrophy (VA) and duodenitis without VA. The VA group was further categorized based on the presence or absence of celiac disease (CD). RESULTS: We identified 836 patients with T21 who underwent EGD, 419 (50.1%) of whom had duodenal histologic abnormalities. At the time of the first (index) abnormal duodenal biopsy, 290 of 419 had VA and of those, 172 of 290 met CD diagnostic criteria, while 118 of 290 did not meet CD criteria (nonspecific VA). Among the patients with an abnormal biopsy, acid suppression at the time of the index biopsy was less common in patients with VA-CD compared to patients without VA or patients with nonspecific VA (12.2% vs 45.7% vs 44.9%). CONCLUSIONS: Half of the T21 patients in this cohort had abnormal duodenal biopsies including a subgroup with nonspecific VA. In this cohort, acid suppression use was more prevalent in patients with abnormalities other than CD.


Asunto(s)
Enfermedad Celíaca , Síndrome de Down , Humanos , Adulto , Estudios Retrospectivos , Síndrome de Down/complicaciones , Duodeno/patología , Biopsia , Enfermedad Celíaca/diagnóstico , Mucosa Intestinal/patología
9.
PLoS One ; 18(2): e0275123, 2023.
Artículo en Inglés | MEDLINE | ID: mdl-36730234

RESUMEN

BACKGROUND: Celiac disease has an increasing incidence worldwide and is treated with lifelong adherence to a gluten-free diet. We aimed to describe gluten-free diet adherence rates in children with screening-identified celiac disease, determine adherence-related factors, and compare adherence to food records in a multinational prospective birth cohort study. METHODS: Children in The Environmental Determinants of Diabetes in the Young study with celiac disease were included. Subjects had at least annual measurement of adherence (parent-report) and completed 3-day food records. Descriptive statistics, t-tests, Kruskal-Wallis tests and multivariable logistic and linear regression were employed. RESULTS: Two hundred ninety (73%) and 199 (67%) of subjects were always adherent to a gluten-free diet at 2 and 5 years post celiac disease diagnosis respectively. The percentage of children with variable adherence increased from 1% at 2 years to 15% at 5 years. Children with a first-degree relative with celiac disease were more likely to be adherent to the gluten-free diet. Gluten intake on food records could not differentiate adherent from nonadherent subjects. Adherent children from the United States had more gluten intake based on food records than European children (P < .001 and P = .007 at 2 and 5 years respectively). CONCLUSION: Approximately three-quarters of children with screening-identified celiac disease remain strictly adherent to a gluten-free diet over time. There are no identifiable features associated with adherence aside from having a first-degree relative with celiac disease. Despite good parent-reported adherence, children from the United States have more gluten intake when assessed by food records. Studies on markers of gluten-free diet adherence, sources of gluten exposure (particularly in the United States), and effects of adherence on mucosal healing are needed.


Asunto(s)
Enfermedad Celíaca , Dieta Sin Gluten , Cooperación del Paciente , Niño , Humanos , Enfermedad Celíaca/terapia , Glútenes , Estudios Prospectivos
10.
Am J Gastroenterol ; 118(3): 539-545, 2023 03 01.
Artículo en Inglés | MEDLINE | ID: mdl-36219178

RESUMEN

INTRODUCTION: The Environmental Determinants of Diabetes in the Young study follows an HLA risk selected birth cohort for celiac disease (CD) development using a uniform protocol. Children under investigation come from 6 different regions within Europe and the United States. Our aim was to identify regional differences in CD autoimmunity and CD cumulative incidence for children born between 2004 and 2010. METHODS: Children (n = 6,628) with DQ2.5 and/or DQ8.1 were enrolled prospectively from birth in Georgia, Washington, Colorado, Finland, Germany, and Sweden. Children underwent periodic study screening for tissue transglutaminase antibodies and then CD evaluation per clinical care. Population-specific estimates were calculated by weighting the study-specific cumulative incidence with the population-specific haplogenotype frequencies obtained from large stem cell registries from each site. RESULTS: Individual haplogenotype risks for CD autoimmunity and CD varied by region and affected the cumulative incidence within that region. The CD incidence by age 10 years was highest in Swedish children at 3%. Within the United States, the incidence by age 10 years in Colorado was 2.4%. In the model adjusted for HLA, sex, and family history, Colorado children had a 2.5-fold higher risk of CD compared to Washington. Likewise, Swedish children had a 1.4-fold and 1.8-fold higher risk of CD compared with those in Finland and Germany, respectively. DISCUSSION: There is high regional variability in cumulative incidence of CD, which suggests differential environmental, genetic, and epigenetic influences even within the United States. The overall high incidence warrants a low threshold for screening and further research on region-specific CD triggers.


Asunto(s)
Enfermedad Celíaca , Niño , Humanos , Incidencia , Enfermedad Celíaca/epidemiología , Enfermedad Celíaca/genética , Enfermedad Celíaca/diagnóstico , Predisposición Genética a la Enfermedad , Autoanticuerpos , Autoinmunidad
11.
J Pediatr Gastroenterol Nutr ; 75(6): 737-742, 2022 12 01.
Artículo en Inglés | MEDLINE | ID: mdl-36122373

RESUMEN

OBJECTIVES: Celiac disease (CeD) autoimmunity and coexisting inflammatory bowel disease (IBD) present a diagnostic dilemma. Our aims were to describe the phenotype of children with IBD and CeD seropositivity and evaluate provider confidence for diagnosing CeD in this population. METHODS: We performed a single-center retrospective cohort study of subjects ≤18 years old with IBD and CeD seropositivity between 2006 and 2020. Subjects were considered to have IBD-CeD if they met CeD diagnosis by serology and histology per North American Society For Pediatric Gastroenterology, Hepatology and Nutrition guidelines and if providers suspected CeD as evaluated by a survey. The IBD-only cohort included seropositive participants that did not meet criteria for CeD. Demographic, histologic, gross endoscopic, and laboratory features were compared using Fisher exact test. RESULTS: Of 475 children with IBD, 8 had concomitant CeD, 5 had tissue transglutaminase (tTG) immunoglobulin A (IgA) > 10x upper limit of normal (ULN, P = 0.006), and 8 had villous atrophy (VA, P = 0.003) when compared with 17 seropositive participants with IBD-only. No children with IBD-CeD had esophageal eosinophilia, duodenal cryptitis, duodenal ulceration, or fecal calprotectin >250 µg/g. Factors that contributed to provider uncertainty for diagnosing CeD in IBD included the absence of VA and intraepithelial lymphocytes, the presence of neutrophilic and eosinophilic duodenitis, diffuse ulceration, elevated inflammatory markers, and immunosuppression therapy. CONCLUSIONS: Diagnosing CeD in children with IBD continues to be challenging. Although high titers of tTG IgA and VA increased provider confidence for diagnosing CeD in IBD, development of evidence-based guidelines are needed. They should better assess the importance of features atypical of concomitant CeD that contribute to uncertainty.


Asunto(s)
Enfermedad Celíaca , Enfermedades Inflamatorias del Intestino , Humanos , Enfermedad Celíaca/complicaciones , Enfermedad Celíaca/diagnóstico , Estudios Retrospectivos , Duodeno/patología , Enfermedades Inflamatorias del Intestino/complicaciones , Enfermedades Inflamatorias del Intestino/patología , Autoanticuerpos , Inmunoglobulina A , Transglutaminasas
12.
J Pediatr Gastroenterol Nutr ; 75(2): 181-185, 2022 08 01.
Artículo en Inglés | MEDLINE | ID: mdl-35641896

RESUMEN

Mental health is a growing concern in pediatric celiac disease (CD). This study utilized the Revised Children's Anxiety and Depression Scale (RCADS) to investigate anxiety and depression symptom rates. Participants were children ages 8 to 17 years (M = 11.7, SD = 2.7; N = 175) with biopsy-proven CD (Median = 1.1 years post-diagnosis, IQR = 0-4) categorized into groups based on the child's age, caregiver or child respondent, presence or absence of comorbidities, and gluten-free diet duration. Self-reported RCADS scores showed 39% of children having clinically significant concerns for anxiety or depression ( P < 0.0001) but only 7% of caregiver-proxy RCADS scores indicated significant concerns for the child's anxiety and 14% for the child's depression. Rates of child-reported anxiety and depression symptoms were significantly higher for those without medical comorbidities than those with ( P = 0.04). Therefore, screening for mental health concerns, particularly anxiety and depression, should be routinely performed in pediatric patients with CD.


Asunto(s)
Enfermedad Celíaca , Depresión , Adolescente , Ansiedad/diagnóstico , Ansiedad/epidemiología , Ansiedad/etiología , Enfermedad Celíaca/complicaciones , Enfermedad Celíaca/psicología , Niño , Estudios Transversales , Depresión/diagnóstico , Depresión/epidemiología , Depresión/etiología , Humanos , Escalas de Valoración Psiquiátrica
13.
J Pediatr Gastroenterol Nutr ; 75(1): 64-69, 2022 07 01.
Artículo en Inglés | MEDLINE | ID: mdl-35622080

RESUMEN

BACKGROUND: Research on the utilization and effectiveness of antitumor necrosis factor (TNF) biologics in children with very early onset inflammatory bowel disease (VEOIBD) is urgently needed. Here we describe anti-TNF use and durability in a multicenter cohort. METHODS: We performed a retrospective cohort study of patients diagnosed with VEOIBD (<6 years) between 2008 and 2013 at 25 North American centers. We performed chart abstraction at diagnosis and 1, 3, and 5 years after diagnosis. We examined the rate of initiation and durability of infliximab and adalimumab and evaluated associations between treatment durability and the following covariates with multivariate Cox proportional hazard regression: age at diagnosis, sex, disease duration, disease classification, and presence of combined immunomodulatory treatment versus monotherapy. RESULTS: Of 294 children with VEOIBD, 120 initiated treatment with anti-TNF therapy and 101 had follow-up data recorded [50% Crohn disease (CD), 31% ulcerative colitis (UC), and 19% IBD unclassified (IBD-U)]. The cumulative probability of anti-TNF treatment was 15% at 1 year, 30% at 3 years, and 45% at 5 years from diagnosis; 56 (55%) were treated between 0 and 6 years old. Anti-TNF durability was 90% at 1 year, 75% at 3 years, and 55% at 5 years. The most common reason for discontinuation of anti-TNF were loss of response in 24 (57%) children. Children with UC/IBD-U had lower durability than those with CD (hazard ratio [HR] 0.17; 95% confidence interval [CI], 0.06-0.51; P = 0.001). CONCLUSIONS: Utilization and durability of anti-TNF in VEOIBD is relatively high and comparable with older children. Having Crohn disease (compared with UC/IBD-U) is associated with greater durability.


Asunto(s)
Productos Biológicos , Colitis Ulcerosa , Enfermedad de Crohn , Enfermedades Inflamatorias del Intestino , Adalimumab/uso terapéutico , Adolescente , Productos Biológicos/uso terapéutico , Niño , Preescolar , Estudios de Cohortes , Colitis Ulcerosa/tratamiento farmacológico , Enfermedad de Crohn/tratamiento farmacológico , Humanos , Lactante , Recién Nacido , Enfermedades Inflamatorias del Intestino/tratamiento farmacológico , Infliximab/uso terapéutico , Necrosis , Estudios Retrospectivos , Inhibidores del Factor de Necrosis Tumoral , Factor de Necrosis Tumoral alfa
14.
Pediatrics ; 149(3)2022 03 01.
Artículo en Inglés | MEDLINE | ID: mdl-35190811

RESUMEN

BACKGROUND: Delays in advancing to biologic therapies are associated with adverse outcomes in inflammatory bowel disease (IBD). Insurer-mandated prior authorizations have been linked to prolonged medication initiation times. We hypothesized that prior authorizations are associated with prolonged biologic initiation time and increased IBD-related healthcare utilization among children with IBD. METHODS: We performed a retrospective cohort study of 190 pediatric patients with IBD initiating biologics at a tertiary care hospital to measure the association between prior authorization, biologic initiation time (physician recommendation to first dose), and healthcare utilization (hospitalization, surgery, or emergency department visit). Demographic, insurance, and disease severity-related covariables were collected. Multivariable linear regression was used to measure the association between prior authorization and biologic initiation time. Propensity score methods were used to measure the associations between prior authorization and IBD-related healthcare utilization within 180 days and corticosteroid dependence at 90 days, with adjustment for insurance type, demographics, and disease severity-related characteristics. RESULTS: Median biologic initiation time was 21 days. Prior authorization and complicated prior authorizations (requiring appeal, step therapy, or peer-to-peer review) were associated with 10.2-day (95% confidence interval [CI] 8.2 to 12.3) and 24.6-day (95% CI 16.4 to 32.8) increases in biologic initiation time, respectively. Prior authorizations increased the likelihood of IBD-related healthcare utilization within 180 days by 12.9% (95% CI 2.5 to 23.4) and corticosteroid dependence at 90 days by 14.1% (95% CI 3.3 to 24.8). CONCLUSIONS: Prior authorizations are associated with prolonged biologic initiation time and increased IBD-related healthcare utilization. Minimizing prior authorization-related delays may expedite biologic delivery and reduce the risk of IBD-related healthcare utilization.


Asunto(s)
Enfermedades Inflamatorias del Intestino , Autorización Previa , Niño , Enfermedad Crónica , Hospitalización , Humanos , Enfermedades Inflamatorias del Intestino/tratamiento farmacológico , Aceptación de la Atención de Salud , Estudios Retrospectivos
15.
Qual Life Res ; 31(7): 2107-2118, 2022 Jul.
Artículo en Inglés | MEDLINE | ID: mdl-34984588

RESUMEN

PURPOSE: To evaluate the impact of celiac disease (CD) and the gluten-free diet (GFD) on the health-related quality of life (HRQoL) in children with CD in the United States using validated measures. We hypothesize that CD negatively impacts the child and caregivers' HRQoL. METHODS: Participants included children with a confirmed diagnosis of CD and their caregivers (n = 246) seen in a CD multidisciplinary clinic. Caregivers completed the Pediatric Quality of Life (PedsQL) parent-proxy scale to report on their child's HRQoL and the Family Impact Module (FIM), which assesses the impact of caring for a child with a chronic illness. Their children completed the age-appropriate PedsQL. PedsQL and FIM results were compared to published data for children with gastroenterological conditions and a healthy cohort using non-parametric tests. RESULTS: Children with CD reported significantly lower HRQoL than reports from healthy controls across all PedsQL domains (P < 0.001, Cohen d = 0.8), and lower compared to children with other organic gastrointestinal conditions in Social Functioning (P < 0.001, Cohen d = 0.5) and overall Psychosocial Functioning (P < 0.001, Cohen d = 0.3) domains. Results from the caregiver's report on their own HRQoL were significantly worse than that reported by historical controls in the domains of Communication (P < 0.001, Cohen d = 0.3) and Worry (P < 0.001, Cohen d = 0.8), yet similar on all other domains. CONCLUSIONS: In our population, CD is associated with low HRQoL scores for both children and their caregivers. Screening children and families for HRQoL can identify patients and families in need of additional support in this higher-risk population.


Asunto(s)
Cuidadores , Enfermedad Celíaca , Cuidadores/psicología , Niño , Dieta Sin Gluten , Humanos , Padres/psicología , Calidad de Vida/psicología , Encuestas y Cuestionarios
17.
Am J Gastroenterol ; 116(1): 180-187, 2021 01 01.
Artículo en Inglés | MEDLINE | ID: mdl-32701732

RESUMEN

INTRODUCTION: The Autoimmunity Screening for Kids (ASK) study is a large scale pediatric screening study in Colorado for celiac disease (CD) and type 1 diabetes. This is a report of the CD outcomes for the first 9,973 children screened through ASK. METHODS: ASK screens children aged 1-17 years for CD using 2 highly sensitive assays for tissue transglutaminase autoantibodies (TGA): a radiobinding (RBA) assay for IgA TGA and an electrochemiluminescence (ECL) assay that detects all TGA isotypes. Children who test positive on either assay are asked to return for confirmatory testing. Those with a confirmed RBA TGA level ≥ 0.1 (twice the upper limit of normal) are referred to the Colorado Center for Celiac Disease for further evaluation; all others are referred to primary care. RESULTS: Of the initial 9,973 children screened, 242 children were TGA+ by any assay. Of those initially positive, 185 children (76.4%) have completed a confirmation blood draw with 149 children (80.5%) confirming positive by RBA TGA. Confirmed RBA TGA+ was associated with a family history of CD (odds ratio [OR] = 1.83; 95% confidence interval 1.06-3.16), non-Hispanic white ethnicity (OR = 3.34; 2.32-4.79), and female sex (OR = 1.43; 1.03-1.98). Gastrointestinal symptoms of CD, assessed at the initial screening, were reported equally often among the RBA TGA+ vs TGA- children (32.1% vs 30.5%, P = 0.65). DISCUSSION: The initial results of this ongoing mass-screening program confirm a high prevalence of undiagnosed CD autoimmunity in a screened US population. Symptoms at initial screening were not associated with TGA status (see Visual abstract, Supplementary Digital Content 5, http://links.lww.com/AJG/B587).


Asunto(s)
Autoanticuerpos/inmunología , Enfermedad Celíaca/diagnóstico , Proteínas de Unión al GTP/inmunología , Transglutaminasas/inmunología , Adolescente , Enfermedades Asintomáticas , Enfermedad Celíaca/inmunología , Niño , Preescolar , Técnicas de Diagnóstico por Radioisótopo , Técnicas Electroquímicas , Femenino , Humanos , Inmunoglobulina A/inmunología , Inmunoglobulina D/inmunología , Inmunoglobulina E/inmunología , Inmunoglobulina G/inmunología , Inmunoglobulina M/inmunología , Lactante , Masculino , Tamizaje Masivo , Proteína Glutamina Gamma Glutamiltransferasa 2 , Pruebas Serológicas
18.
J Pediatr Gastroenterol Nutr ; 72(2): 194-201, 2021 02 01.
Artículo en Inglés | MEDLINE | ID: mdl-32925550

RESUMEN

OBJECTIVES: Hepatitis-associated aplastic anemia (HAAA) is a potentially life-threatening diagnosis without clear treatment guidelines. The goal of the study was to characterize the presentation, evaluation, histopathology, and outcomes of therapy in children with HAAA to guide future research and to develop standardized care guidelines for this rare disease. METHODS: Retrospective chart review of 4 patients with HAAA who presented to Children's Hospital Colorado between 2016 and 2019 was conducted. Patient presentation, evaluation, bone marrow and liver pathology, interventions, and clinical course were collected. Immunohistochemistry of liver biopsies was performed. RESULTS: We treated 4 patients with HAAA without liver failure. All had evidence of systemic hyperinflammation and CD8+ T cell predominant liver tissue infiltration. One had a genetic mutation predisposing him to immune-mediated disease, but all other genetic testing was negative. In 3 of the 4 patients, hepatitis was poorly responsive to standard therapy with steroids, azathioprine, or tacrolimus; however, sustained biochemical remission of hepatitis was induced after more aggressive immunosuppressive therapies including Anti-Thymocyte Globulin (ATG) at standard immunosuppressive therapy (IST) dosing for severe Aplastic Anemia (sAA). Two patients underwent hematopoietic stem cell transplant (HSCT); 1 as first line therapy and 1 for refractory sAA. CONCLUSIONS: We found that ATG-based IST induced remission of hepatitis in patients with steroid-refractory HAAA. This is also an appropriate initial treatment for severe Aplastic Anemia, though may not prevent the need for HSCT. We propose that equine ATG based IST at standard dosing regimen for sAA is a therapy that in select cases can be considered early on in the treatment course and could lead to a sustained remission of both hepatitis and sAA. This should be considered in collaboration with a pediatric hematologist.


Asunto(s)
Anemia Aplásica , Hepatitis , Anemia Aplásica/complicaciones , Anemia Aplásica/terapia , Animales , Niño , Colorado , Hepatitis/complicaciones , Hepatitis/diagnóstico , Caballos , Humanos , Inmunosupresores/uso terapéutico , Masculino , Estudios Retrospectivos , Tacrolimus , Resultado del Tratamiento
19.
Inflamm Bowel Dis ; 27(3): 295-302, 2021 02 16.
Artículo en Inglés | MEDLINE | ID: mdl-32386060

RESUMEN

BACKGROUND: The incidence of very early onset inflammatory bowel disease (VEOIBD) is increasing, yet the phenotype and natural history of VEOIBD are not well described. METHODS: We performed a retrospective cohort study of patients diagnosed with VEOIBD (6 years of age and younger) between 2008 and 2013 at 25 North American centers. Eligible patients at each center were randomly selected for chart review. We abstracted data at diagnosis and at 1, 3, and 5 years after diagnosis. We compared the clinical features and outcomes with VEOIBD diagnosed younger than 3 years of age with children diagnosed with VEOIBD at age 3 to 6 years. RESULTS: The study population included 269 children (105 [39%] Crohn's disease, 106 [39%] ulcerative colitis, and 58 [22%] IBD unclassified). The median age of diagnosis was 4.2 years (interquartile range 2.9-5.2). Most (94%) Crohn's disease patients had inflammatory disease behavior (B1). Isolated colitis (L2) was the most common disease location (70% of children diagnosed younger than 3 years vs 43% of children diagnosed 3 years and older; P = 0.10). By the end of follow-up, stricturing/penetrating occurred in 7 (6.6%) children. The risk of any bowel surgery in Crohn's disease was 3% by 1 year, 12% by 3 years, and 15% by 5 years and did not differ by age at diagnosis. Most ulcerative colitis patients had pancolitis (57% of children diagnosed younger than 3 years vs 45% of children diagnosed 3 years and older; P = 0.18). The risk of colectomy in ulcerative colitis/IBD unclassified was 0% by 1 year, 3% by 3 years, and 14% by 5 years and did not differ by age of diagnosis. CONCLUSIONS: Very early onset inflammatory bowel disease has a distinct phenotype with predominantly colonic involvement and infrequent stricturing/penetrating disease. The cumulative risk of bowel surgery in children with VEOIBD was approximately 14%-15% by 5 years. These data can be used to provide anticipatory guidance in this emerging patient population.


Asunto(s)
Colitis Ulcerosa , Enfermedad de Crohn , Niño , Preescolar , Enfermedad Crónica , Colectomía , Colitis Ulcerosa/epidemiología , Constricción Patológica , Enfermedad de Crohn/epidemiología , Humanos , América del Norte/epidemiología , Estudios Retrospectivos
20.
Scand J Gastroenterol ; 55(11): 1284-1290, 2020 Nov.
Artículo en Inglés | MEDLINE | ID: mdl-32941083

RESUMEN

OBJECTIVES: To determine the association between childhood growth prior to the development of celiac disease (CD) and CD autoimmunity (CDA) identified by periodic serological screening. STUDY DESIGN: The Diabetes Autoimmunity Study in the Young cohort includes 1979 genetically at-risk children from Denver, Colorado, with annual growth measurements from age nine months until ten years. Between 1993 and February 2019, 120 children developed CDA defined by persistent positive tissue transglutaminase autoantibodies (TGA); among these, 71 met our criteria for CD based on histopathological findings or high TGA levels. Age- and sex-specific z-scores of weight, body mass index (BMI), and height prior to seroconversion were derived using US reference charts as standards. Joint modeling of serial growth measurements was used to estimate adjusted hazard ratios (aHRs) accounting for celiac-associated human leukocyte antigens, early-life feeding practices, and socio-demographics. RESULTS: In the first 10 years of life, there were no significant associations between the child's current weight, BMI and height and the risk of screening-detected CDA or CD, neither was the weight nor BMI velocity associated with CDA or CD as identified by screening (all aHRs approximated 1). Increased height velocity was associated with later CD, but not CDA, development (aHR per 0.01-z score/year, 1.28; 95% confidence interval [CI] 1.18-1.38 and 1.03; 0.97-1.09, respectively). CONCLUSIONS: In the first 10 years of life, from prospectively collected serial growth measurements, we found no evidence of impaired childhood growth before CD and CDA development as identified through early and periodic screening.


Asunto(s)
Enfermedad Celíaca , Autoanticuerpos , Autoinmunidad , Índice de Masa Corporal , Enfermedad Celíaca/diagnóstico , Niño , Femenino , Estudios de Seguimiento , Humanos , Lactante , Masculino , Estudios Prospectivos
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