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[PubMed] [Google Scholar] 39. tested positive for autoantibodies (40%) or human leukocyte antigen DQ2/DQ8 (53%). No associated autoimmune comorbidities were observed. The serum levels of calprotectin and amyloid A were increased in 10/15 (67%) and 5/15 (33%) of the patients, respectively, whereas plasma C-reactive protein levels were normal in all, but 1 patient. DISCUSSION: The results indicate that childhood-onset CG is rare and has a chronic disease course. Although signs of autoimmune predisposition are frequent, early development of autoimmune comorbidities seems seldom. Serum calprotectin and amyloid A represent novel candidate biomarkers of inflammatory activity in LY335979 (Zosuquidar 3HCl) CG (see Visual Abstract, Supplementary Digital Content 4, http://links.lww.com/CTG/A349). INTRODUCTION Collagenous gastritis (CG) is a rare gastrointestinal disorder with fewer than 300 cases reported in the English-language literature (1C24). Of these cases, about one-third have been childhood-onset CG (15C45). The condition is characterized histologically by an increased subepithelial layer of collagen (conventionally defined as being 10 m in thickness) in the gastric mucosa, together with an inflammatory cell infiltrate in the lamina propria (46,47). In most pediatric cases of CG, the collagenous mucosal inflammation is restricted to the stomach (22C24,41), whereas in adult-onset disease, concurrent involvement of the small bowel and/or colon is more common (1,23,24,46,48). Pediatric cases of CG generally present with severe iron deficiency anemia and/or recurrent abdominal pain (22C24,41), whereas diarrhea and malabsorption are the predominant symptoms in adults, presumably linked to the frequently observed concurrent intestinal involvement (32,46,49). Associated immune-mediated comorbidities, such as celiac disease and type 1 diabetes, have been reported in both pediatric (22C24,26,31) and adult (3C5,23,24,45,47,50) patients. CG is believed to be a chronic disease, and there is currently no effective treatment (46). The current literature on childhood-onset CG consists mainly of case reports (15C20,27C37,42,43,51), apart from 6 small case series (21,22,38C41) and 3 histopathologic studies with limited clinical information and follow-up data (23,24,45). Consequently, the knowledge regarding the evolution of the clinical, endoscopic, and histologic features of the disease over time is sparse. Furthermore, although immune-mediated/autoimmune disease mechanisms have been hypothesized (47), convincing supporting evidence is lacking. In collagenous colitis, the human leukocyte antigen (HLA) DQ2.5 haplotype, encoded by the HLA DQ2/DQ8 genes, seems to be associated with increased disease susceptibility (52). However, no studies of the prevalence of the HLA DQ2/DQ8 haplotype in CG have been published. Moreover, the methodologies used in the previous studies of CG have not allowed estimations of the disease incidence or prevalence, although a possible female predominance has been noted (1,41). The aims of this population-based cohort study, which combines Rabbit Polyclonal to HOXA11/D11 longitudinal and cross-sectional approaches and involves 15 LY335979 (Zosuquidar 3HCl) patients with childhood-onset CG, were to LY335979 (Zosuquidar 3HCl) investigate: (i) the incidence and prevalence of CG in a pediatric population in western Sweden; (ii) the clinical, endoscopic, and histologic characteristics of childhood-onset CG through the course of the disease; and (iii) the frequencies of autoimmune comorbidities and heredity, as well as the prevalences of autoantibody development, increased blood inflammatory biomarkers, and the HLA DQ2/DQ8 haplotypes in patients with childhood-onset CG. METHODS Study design The study was designed as a population-based cohort study that comprised 15 persons of White ethnicity with childhood-onset CG (12 female and 3 male patients) and age range of 8.7C23 years (median age, 15 years) recruited from western Sweden. All cases of CG diagnosed before the age of 18 years in the counties of Halland, J?nk?ping, V?rmland, and V?stra G?taland in western Sweden during the period of January 2008 through June 2019 were identified. These 4 counties comprise 26% of the Swedish population and in 2019 had a pediatric population (aged 18 years) of approximately 568,000 (53). The Department of Pediatric Gastroenterology, Hepatology, and Nutrition at Queen Silvia Children’s Hospital, Gothenburg, Sweden, which serves as a tertiary referral center for these counties, was involved in the diagnosis of all the cases. Furthermore, an established practice in Sweden is that all pediatric endoscopies are performed under general anesthesia in county hospitals or higher-level medical institutions within the public healthcare system. This enables the identification of all.