RESUMO
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.
Assuntos
Doença Celíaca , Doenças Inflamatórias Intestinais , Humanos , Doença Celíaca/complicações , Doença Celíaca/diagnóstico , Estudos Retrospectivos , Duodeno/patologia , Doenças Inflamatórias Intestinais/complicações , Doenças Inflamatórias Intestinais/patologia , Autoanticorpos , Imunoglobulina A , TransglutaminasesRESUMO
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.
Assuntos
Doença Celíaca , Depressão , Adolescente , Ansiedade/diagnóstico , Ansiedade/epidemiologia , Ansiedade/etiologia , Doença Celíaca/complicações , Doença Celíaca/psicologia , Criança , Estudos Transversais , Depressão/diagnóstico , Depressão/epidemiologia , Depressão/etiologia , Humanos , Escalas de Graduação PsiquiátricaRESUMO
Feeding difficulties due to functional gastrointestinal (GI) symptoms (i.e., nausea, pain, and bloating) are well described in patients with hypermobile-type Ehlers-Danlos Syndrome. These symptoms are particularly difficult to treat when there is comorbid dysautonomia, usually manifesting as postural orthostatic tachycardia syndrome. Here, we describe a successful trial of multidisciplinary rehabilitative interventions to avoid placement of a surgical feeding tube in such a patient. Main components of intervention were intensive pelvic floor physiotherapy and biofeedback, occupational therapy focused on coping with feeding-related symptoms, psychology support, and medications targeting histamine blockade and enhancing intestinal motility.
Assuntos
Doença Celíaca/dietoterapia , Dieta Livre de Glúten , Terapia Comportamental , Criança , Glutens , HumanosRESUMO
OBJECTIVE: To assess the evidence regarding the effect of time of gluten introduction and breastfeeding on the risk of developing celiac disease (CD). STUDY DESIGN: We included randomized controlled trials and observational studies evaluating the proper timing for introducing gluten to the infant diet, the appropriate quantity of gluten consumption at weaning, and the effect of breastfeeding on CD risk. Studies were located through the electronic databases Cochrane Central Register of Controlled Trials (CENTRAL), MEDLINE (Ovid), EMBASE (Ovid), and System for Information on Grey Literature in Europe (SIGLE). Two independent authors collected the data. RESULTS: A total of 1982 studies were identified, 15 of which were eligible for data extraction. A meta-analysis was performed on 2 randomized controlled trials, 10 cohort studies, and 1 case-control study. There was a 25% increase in CD risk with late (>6 months) vs recommended (4-6 months) gluten introduction (risk ratio [RR], 1.25; 95% CI, 1.08-1.45). There was no significant effect of breastfeeding vs no breastfeeding on CD risk (OR, 0.55; 95% CI, 0.28-1.10), with substantial heterogeneity (I(2) = 92%) among studies. CONCLUSION: There is currently no evidence to support that early introduction of gluten to the infant diet increases the risk of CD; however, late introduction of gluten may be associated with increased risk of CD. More studies are needed that control for potential confounders and that evaluate environmental factors in low-risk families.
Assuntos
Aleitamento Materno , Doença Celíaca/epidemiologia , Comportamento Alimentar , Glutens/administração & dosagem , Doença Celíaca/etiologia , Humanos , Lactente , Estudos Observacionais como Assunto , Ensaios Clínicos Controlados Aleatórios como Assunto , Medição de RiscoRESUMO
OBJECTIVE: To evaluate the association between bone mineral density (BMD), glycemic control (hemoglobin A1c [HbA1c]), and celiac autoimmunity in children with type 1 diabetes mellitus (T1D) and in an appropriate control population. STUDY DESIGN: BMD was assessed cross-sectionally in 252 children with T1D (123 positive for anti-tissue transglutaminase antibody [tTGA] and 129 matched children who were negative for tTGA). In addition, BMD was assessed in 141 children without diabetes who carried T1D-associated HLD-DR, DQ genotypes (71 positive for tTGA and 70 negative). RESULTS: Children with T1D who were positive for tTGA had significantly worse BMD L1-L4 z-score compared with children with T1D who were negative for tTGA (-0.45 ± 1.22 vs 0.09 ± 1.10, P = .0003). No differences in growth measures, urine N-telopeptides, 25-hydroxyvitamin D, ferritin, thyroid stimulating hormone, or HbA1c were found. However, both higher HbA1c (ß = -1.25 ± 0.85, P = .0016) and tTGA (ß = -0.13 ± 0.05, P = .0056) were significant and independent predictors of lower BMD in multivariate analyses. No differences in BMD or other variables measured were found between children without diabetes who were positive vs negative for tTGA. CONCLUSIONS: The results suggest a synergistic effect of hyperglycemia and celiac autoimmunity on low BMD.
Assuntos
Autoimunidade , Densidade Óssea , Doença Celíaca/complicações , Doença Celíaca/fisiopatologia , Diabetes Mellitus Tipo 1/complicações , Adolescente , Doença Celíaca/sangue , Doença Celíaca/imunologia , Criança , Pré-Escolar , Estudos Transversais , Diabetes Mellitus Tipo 1/sangue , Feminino , Hemoglobinas Glicadas/análise , Humanos , Lactente , Recém-Nascido , MasculinoRESUMO
OBJECTIVE: To determine the benefits of screening for celiac autoimmunity via immunoglobulin A transglutaminase autoantibodies (TG) in children with type 1 diabetes (T1D). STUDY DESIGN: We followed up 79 screening-identified TG+ and 56 matched TG- children with T1D for 2 years to evaluate growth, bone mineral density, nutritional status, and diabetes control. TG+ subjects self-selected to gluten-free or gluten-containing diet. RESULTS: Of the initial cohort, 80% were available for reexamination after 2 years. TG+ subjects had consistently lower weight z-scores and higher urine N-telopeptides than TG- subjects, but similar measures of bone density and diabetes outcomes. TG+ children who remained on a gluten-containing diet had lower insulin-like growth factor binding protein 3 z-scores compared with TG+ subjects who reported following a gluten-free diet. Children who continued with high TG index throughout the study had lower bone mineral density z-scores, ferritin, and vitamin D 25OH levels, compared with the TG- group. CONCLUSIONS: No significant adverse outcomes were identified in children with T1D with screening-identified TG+ who delay therapy with a gluten-free diet for 2 years. Children with persistently high levels of TG may be at greater risk. The optimal timing of screening and treatment for celiac disease in children with T1D requires further investigation.
Assuntos
Autoimunidade/imunologia , Doença Celíaca/epidemiologia , Doença Celíaca/imunologia , Diabetes Mellitus Tipo 1/epidemiologia , Diabetes Mellitus Tipo 1/imunologia , Adolescente , Distribuição por Idade , Biópsia por Agulha , Estudos de Casos e Controles , Doença Celíaca/dietoterapia , Doença Celíaca/patologia , Criança , Diabetes Mellitus Tipo 1/diagnóstico , Dieta Livre de Glúten , Feminino , Seguimentos , Humanos , Imunoglobulina A/análise , Imunoglobulina A/imunologia , Imuno-Histoquímica , Incidência , Masculino , Programas de Rastreamento/métodos , Valores de Referência , Medição de Risco , Distribuição por Sexo , Fatores de Tempo , Transglutaminases/análise , Transglutaminases/imunologiaRESUMO
OBJECTIVE: Children with type 1 diabetes (T1DM) are at increased risk for celiac disease (CD); however, the benefits of screening for IgA tissue transglutaminase autoantibodies (TG), a marker for CD, are unclear. STUDY DESIGN: We compared 71 screening-identified TG+ with 63 matched TG- children with TIDM. Growth, bone density, and diabetes control measures were obtained. RESULTS: The group was 10 +/- 3 years of age, 46% male, with TIDM for 4 +/- 3 years. Z scores for weight (0.3 +/- 1 vs 0.7 +/- 0.8, P = .024), body mass index (BMI) (0.3 +/- 0.9 vs 0.8 +/- -0.8, P = .005), and midarm circumference (0.3 +/- 1.1 vs 0.6 +/- 0.9, P = .031) were lower in the TG+ group. Bone mineral density and diabetes control measures were similar. When limiting the analysis to the 35 TG+ subjects with biopsy changes of CD, the BMI Z score was lower than the control group (0.4 +/- 0.9 vs 0.7 +/- 0.7, P = .05). CONCLUSIONS: In children with TIDM, screening-identified evidence of CD is associated with altered body composition, but not bone mineral density or diabetes control. Further study is needed to determine the benefit of early diagnosis and treatment of CD in TIDM children.
Assuntos
Autoanticorpos/sangue , Doença Celíaca/sangue , Doença Celíaca/imunologia , Diabetes Mellitus Tipo 1/sangue , Diabetes Mellitus Tipo 1/complicações , Autoimunidade , Doença Celíaca/complicações , Doença Celíaca/diagnóstico , Criança , Feminino , Humanos , Masculino , Transglutaminases/imunologiaRESUMO
OBJECTIVES: To assess several transglutaminase autoantibody (TGAA) assays in their ability to distinguish celiac disease (CD) in screening-identified children with abnormal intestine biopsy specimens from those with normal biopsy specimens. STUDY DESIGN: Children at risk for CD (n = 54) composed of type 1 diabetics, first-degree relatives of type 1 diabetics or CD, and HLA-DQ2+ individuals followed from birth received intestine biopsy. Sera obtained at the time of biopsy were tested for TGAA, using the radioimmunoassay and 5 other commercially available enzyme-linked immunosorbent assays. RESULTS: False-positive rates ranged from 28% to 80%. The positive predictive value (PPV) of the tests ranged from 63% to 84% (lower than reported for symptomatic children). Setting a higher cutoff for each assay maximized PPV. CONCLUSIONS: There are significant quantitative differences among all TGAA assays that could affect interpretation of a positive test for CD. The overall false-positive rate for all assays was high in this population. Using the assay as a quantitative rather than qualitative tool by increasing the cutoff of positivity to indicate biopsy increases PPV. Multicenter workshops are needed to identify critical differences and to standardize TGAA assays among laboratories.