Case report: Late-onset hypertrophic pyloric stenosis in a 3-year-old boy: It is never too late.

Hypertrophic Pyloric Stenosis (HPS) represents a relatively rare occurrence beyond infancy. Here, we present the case of a barely 3-year-old boy diagnosed with late-onset HPS and successfully treated with extra-mucosal pyloromyotomy. We review the literature, challenging the principle that more aggressive surgical approaches should be preferred over less invasive ones.

Esophageal Eosinophilia and Eosinophilic Esophagitis in Celiac Children: A Ten Year Prospective Observational Study.

The association between eosinophilic esophagitis and celiac disease is still controversial and its prevalence is highly variable. We aimed to investigate the prevalence of esophageal eosinophilia and eosinophilic esophagitis in a large group of children with celiac disease, prospectively followed over 11 years. METHODS: Prospective observational study performed between 2008 and 2019. Celiac disease diagnosis was based on ESPGHAN criteria. At least four esophageal biopsies were sampled in patients who underwent endoscopy. The presence of at least 15 eosinophils/HPF on esophageal biopsies was considered suggestive of esophageal eosinophilia; at the same time, eosinophilic esophagitis was diagnosed according to the International Consensus Diagnostic Criteria for Eosinophilic Esophagitis. RESULTS: A total of 465 children (M 42% mean age 7.1 years (range: 1-16)) were diagnosed with celiac disease. Three hundred and seventy patients underwent endoscopy, and esophageal biopsies were available in 313. The prevalence of esophageal eosinophilia in children with celiac disease was 1.6% (95% CI: 0.54-2.9%). Only one child was diagnosed as eosinophilic esophagitis; we calculated a prevalence of 0.3% (95% CI: 0.2-0.5%). The odds ratio for an association between eosinophilic esophagitis and celiac disease was at least 6.5 times higher (95% CI: 0.89-47.7%; p = 0.06) than in the general population. CONCLUSION: The finding of an increased number of eosinophils (>15/HPF) in celiac patients does not have a clinical implication or warrant intervention, and therefore we do not recommend routine esophageal biopsies unless clinically indicated.

Long-Term Outcome of Potential Celiac Disease in Genetically at-Risk Children: The Prospective CELIPREV Cohort Study.

BACKGROUND: The long-term outcome of potential celiac disease (CD) is still a debated issue. We aimed to evaluate the progression of potential CD versus overt CD after 10-years of follow-up in a cohort of children genetically predisposed to CD. METHODS: The CELIPREV study is prospectively following from birth 553 children with CD-predisposing HLA genes. Children with a diagnosis of potential CD continued to receive a normal diet and repeated the serological screening for CD every year. An intestinal biopsy was taken in presence of persistent positive serology. RESULTS: Overall, 26 (4.7%) children received a diagnosis of potential CD (50% females, median age 24 months). All children were symptom-free. Twenty-three children continued a gluten-containing diet; at 10 years from the first biopsy, three children developed overt CD (13%), 19 (83%) became antibodies negative at 1 year from the first biopsy and remained negative up to 10 years of follow-up and one subject (4%) had fluctuating antibody course with transiently negative values and persistently negative biopsy. CONCLUSIONS: In children genetically predisposed to CD with a diagnosis of potential CD the risk of progression to overt CD while on a gluten-containing diet is very low in the long-term.

Mode of Delivery and Risk of Celiac Disease: Risk of Celiac Disease and Age at Gluten Introduction Cohort Study.

OBJECTIVE: To determine whether the mode of delivery is associated with the risk of celiac disease (CD) in a cohort of children genetically predisposed to CD prospectively followed from birth. STUDY DESIGN: By telephone interview, we recorded information on the mode of delivery of children participating in the Risk of Celiac Disease and Age at Gluten Introduction study, a multicenter, prospective intervention trial that compared early and delayed introduction of gluten in infants with at least 1 first-degree relative affected with CD. The human leukocyte antigen genotype was determined at 15 months of age, and serologic screening for CD was performed at 15, 24, and 36 months of age and at 5, 8, and 10 years of age. Patients with positive serologic findings underwent intestinal biopsy. The primary outcome of the current study was the prevalence of CD autoimmunity and overt CD at 5 years of age, according to the mode of delivery. RESULTS: The study-group included 553 children at CD risk because of positivity for human leukocyte antigen-DQ2, -DQ8, or both. We obtained data on the mode of delivery from 431 of 553 children; 233 of 431 children were born by vaginal delivery (54%). At 5 years of age, the prevalence of CD autoimmunity or overt CD was not different between children born by cesarean or vaginal delivery (24% and 19%, P = .2; 19% and 14%, P = .2 respectively, by the log-rank test). CONCLUSIONS: In this cohort of children genetically predisposed to CD, the mode of delivery did not influence the risk of developing CD.

Increased prevalence of celiac disease among pediatric patients with irritable bowel syndrome: a 6-year prospective cohort study.

IMPORTANCE: Recurrent abdominal pain is a prevalent health issue in childhood. Clinical criteria (ie, the Rome criteria) have been established to aid diagnosis. Studies of adults have shown an increased prevalence of celiac disease among patients with irritable bowel syndrome (IBS); few data are available with regard to children. OBJECTIVE: To assess the prevalence of celiac disease among children with abdominal pain-related functional gastrointestinal disorders classified according to the Rome criteria. DESIGN, SETTING, PARTICIPANTS: Six-year (2006-2012) prospective cohort study conducted in a tertiary referral center for the diagnosis and follow-up of gastrointestinal disorders in southern Italy (ie, Bari, Italy). A total of 992 children (42.8% male; median age, 6.8 years) consecutively referred for recurrent abdominal pain by their primary care physicians without previous investigation were evaluated. EXPOSURE: Patients were classified according to Rome III criteria as having IBS, functional dyspepsia, functional abdominal pain, or abdominal migraine. MAIN OUTCOMES AND MEASURES: Prevalence of celiac disease in each category of abdominal pain-related functional gastrointestinal disorder. Concentrations of IgA, IgA antitissue transglutaminase, and endomysial antibodies were measured, and a duodenal biopsy was performed in case of antibody positivity. RESULTS: A total of 992 children were evaluated: 270 were classified as having IBS, 201 as having functional dyspepsia, and 311 as having functional abdominal pain, and 210 children were excluded from the study because they had an organic disorder or some other functional gastrointestinal disorder (not related to abdominal pain). Serologic testing was performed for all 782 children included in the study, and 15 patients tested positive for celiac disease (12 of 270 patients with IBS [4.4%], 2 of 201 patients with functional dyspepsia [1%], and 1 of 311 patients with functional abdominal pain [0.3%]). Children presenting with IBS have a 4 times higher risk of having celiac disease than children without IBS (odds ratio, 4.19 [95% CI, 2.03-8.49]; P < .001). CONCLUSIONS AND RELEVANCE: The prevalence of celiac disease among children with IBS is 4 times higher than among the general pediatric population. Rome III classification of abdominal pain-related functional gastrointestinal disorders might help to select children who deserve screening for celiac disease.

Clinical, serologic, and histologic features of gluten sensitivity in children.

OBJECTIVE: To describe the clinical, serologic, and histologic characteristics of children with gluten sensitivity (GS). STUDY DESIGN: We studied 15 children (10 males and 5 females; mean age, 9.6 ± 3.9 years) with GS who were diagnosed based on a clear-cut relationship between wheat consumption and development of symptoms, after excluding celiac disease (CD) and wheat allergy, along with 15 children with active CD (5 males and 10 females; mean age, 9.1 ± 3.1 years) and 15 controls with a functional gastrointestinal disorder (6 males and 9 females; mean age, 8.6 ± 2.7 years). All children underwent CD panel testing (native antigliadin antibodies IgG and IgA, anti-tissue transglutaminase antibody IgA and IgG, and anti-endomysial antibody IgA), hematologic assessment (hemoglobin, iron, ferritin, aspartate aminotransferase, erythrocyte sedimentation rate), HLA typing, and small intestinal biopsy (on a voluntary basis in the children with GS). RESULTS: Abdominal pain was the most prevalent symptom in the children with GS (80%), followed by chronic diarrhea in (73%), tiredness (33%), bloating (26%), limb pain, vomiting, constipation, headache (20%), and failure to thrive (13%). Native antigliadin antibodies IgG was positive in 66% of the children with GS. No differences in nutritional, biochemical, or inflammatory markers were found between the children with GS and controls. HLA-DQ2 was found in 7 children with GS. Histology revealed normal to mildly inflamed mucosa (Marsh stage 0-1) in the children with GS. CONCLUSION: Our findings support the existence of GS in children across all ages with clinical, serologic, genetic, and histologic features similar to those of adults.

Prevalence and natural history of potential celiac disease in at-family-risk infants prospectively investigated from birth.

OBJECTIVE: To evaluate the frequency and the natural history of potential (serology positive/Marsh 0-1 histology) celiac disease (CD) in children with a family risk of CD and factors associated with potential instead of overt (serology positive/Marsh 2-3 histology) CD expression. STUDY DESIGN: Two-year follow-up study of 96 children (57 females; mean age: 29 ± 12 months) prospectively investigated from birth with: (1) a CD-affected first-degree relative; (2) positivity of serum IgA anti-tissue transglutaminase (tTG) or IgG antigliadin and IgA deficiency; and (3) the results of small intestinal biopsy. Children with potential CD were advised to remain on a gluten containing diet, repeat the celiac antibodies every 6 months, and to have an intestinal biopsy performed in case of persistently high anti-tTG level. Factors discriminating between potential and overt CD were analyzed by decision tree analysis based on the C4.5 algorithm. RESULTS: Twenty-four children had potential and 72 overt CD. The stronger predictors of potential CD were lack of symptoms, anti-tTG level lower than 11-fold the upper normal limit, age lower than 24 months, and breastfeeding longer than 8 months. Eighteen out of 21 (86%) patients with potential CD continuing a gluten-containing diet became antibody negative, 1/21 (5%) developed overt CD, and 2/21 (9%) had fluctuating antibodies levels after 2 years. CONCLUSIONS: The prevalence of potential CD and the percentage of short-term loss of CD-related-antibodies are high in infants at-family-risk for CD. In symptomless children with a positive celiac serology, the decision of performing an intestinal biopsy should be preceded by a period of repeated serological testing.

Clarithromycin-resistant genotypes and eradication of Helicobacter pylori.

OBJECTIVE: To compare the eradication rates among the different point mutations and the efficacy of triple therapy and a sequential regimen according to genotypic resistance. STUDY DESIGN: Post hoc retrospective cohort study in a tertiary referral center for pediatric gastroenterology in southern Italy. All 168 children who were positive for Helicobacter pylori were enrolled. Patients had received clarithromycin-based 7-day triple therapy (73 children) or 10-day sequential therapy regimen (95 children). Real-time polymerase chain reaction for assessing clarithromycin resistance was performed on sections of paraffin-embedded gastric biopsy samples. RESULTS: H pylori eradication was achieved in 16 of 32 (50%) children with the A2143G mutation, in 8 of 10 patients with either A2142G or A2142C strains (80%), and in 112 of 116 children with susceptible strains (88.9%). The presence of A2143G mutation was associated with a lower cure rate compared with the rate in the absence of this mutation (50% vs. 89%; P = .001). The sequential regimen achieved a higher cure rate than triple therapy in patients with A2143G mutant strains (80% vs nil; P < .001). CONCLUSIONS: The A2143G mutation confers higher risk of treatment failure. Sequential regimen has higher efficacy than standard therapy, even in children with A2143G mutatant strains.

Helicobacter pylori status and symptom assessment two years after eradication in pediatric patients from a high prevalence area.

OBJECTIVES: To establish the rate of Helicobacter pylori reinfection in children from an H. pylori high prevalence area, possible clinical features predictive of reinfection and the usefulness of re-treatment. METHODS: 65 consecutive children attending the authors' department between 1998 and 2000 who had proven successful H. pylori eradication were enrolled; 52 took part. Patients and family members were invited to undergo C-urea breath testing and to complete a simple questionnaire regarding symptoms and socioeconomic status. Patients with H. pylori reinfection were offered treatment; eradication was assessed by C-urea breath test 8 weeks after completion of treatment. RESULTS: Of 52 children, 15 (28.8%) were H. pylori positive. Variables predictive of reinfection were age at primary infection and presence of an infected sibling. Although reinfected children were more frequently symptomatic than non-reinfected patients, no specific symptom was associated with reinfection. Of the nine re-treated patients who returned 8 weeks after completing therapy, the bacterium was eradicated in five (56%). CONCLUSIONS: The 12.8% per year reinfection rate in childhood at 2 years that we observed should prompt a re-evaluation of H. pylori status even after a successful eradication. Living in an H. pylori high prevalence area increases the annual risk of reinfection by approximately fourfold over the annual risk in H. pylori low prevalence areas.