Non-responsive celiac disease in children on a gluten free diet

查看全文 作　　者：Gopal [1,2,3]Veeraraghavan;Amelie [1,2,3]Therrien;Maya [1]Degroote;Allison [1]McKeown;Paul D [4]Mitchell;Jocelyn A [1,2,3]Silvester;Daniel A [2,3,5]Leffler;Alan M [1,3]Leichtner;Ciaran P [2,3]Kelly;Dascha C [1,3]Weir 机构地区：[1]Division of Gastroenterology,Hepatology and Nutrition,Boston Children's Hospital,Boston,MA 02115,United States;[2]The Celiac Center,Department of Medicine and Division of Gastroenterology,Beth Israel Deaconess Medical Center,Boston,MA 02215,United States;[3]Celiac Research Program,Harvard Medical School,Boston,MA 02115,United States;[4]Institutional Centers for Clinical and Translational Research,Boston Children's Hospital,Boston,MA 02115,United States;[5]Gastrointestinal Therapeutics,Takeda Pharmaceutical International Co,Cambridge,MA 02139,United States 出　　处：《World Journal of Gastroenterology》索引2021年第27卷第13期,共10页 基　　金：Supported by Boston Children’s Hospital and the National Institute of Diabetes and Digestive and Kidney Diseases of the National Institutes of Health,No. P30DK034854, No. K23DK119584 and No. T32DK07760;DG Kinnear Award from the Association Quebecoise des。 摘　　要：BACKGROUND Non-responsive celiac disease(NRCD) is defined as the persistence of symptoms in individuals with celiac disease(CeD) despite being on a gluten-free diet(GFD). There is scant literature about NRCD in the pediatric population.AIM To determine the incidence, clinical characteristics and underlying causes of NRCD in children.METHODS Retrospective cohort study performed at Boston Children’s Hospital(BCH). Children < 18 years diagnosed with CeD by positive serology and duodenal biopsies compatible with Marsh Ⅲ histology between 2008 and 2012 were identified in the BCH’s Celiac Disease Program database. Medical records were longitudinally reviewed from the time of diagnosis through September 2015. NRCD was defined as persistent symptoms at 6 mo after the initiation of a GFD and causes of NRCD as well as symptom evolution were detailed. The children without symptoms at 6 mo(responders) were compared with the NRCD group. Additionally, presenting signs and symptoms at the time of diagnosis of CeD among the responders and NRCD patients were collected and compared to identify any potential predictors for NRCD at 6 mo of GFD therapy.RESULTS Six hundred and sixteen children were included. Ninety-one(15%) met criteria for NRCD. Most were female(77%). Abdominal pain [odds ratio(OR) 1.8 95% confidence interval(CI) 1.1-2.9], constipation(OR 3.1 95%CI 1.9-4.9) and absence of abdominal distension(OR for abdominal distension 0.4 95%CI 0.1-0.98) at diagnosis were associated with NRCD. NRCD was attributed to a wide variety of diagnoses with gluten exposure(30%) and constipation(20%) being the most common causes. Other causes for NRCD included lactose intolerance(9%), gastroesophageal reflux(8%), functional abdominal pain(7%), irritable bowel syndrome(3%), depression/anxiety(3%), eosinophilic esophagitis(2%), food allergy(1%), eating disorder(1%), gastric ulcer with Helicobacter pylori(1%), lymphocytic colitis(1%), aerophagia(1%) and undetermined(13%). 64% of children with NRCD improved on follow-up.CONCLUSION NRCD after ≥ 6 mo GFD is frequent among children, especially females, and is associated with initial presenting symptoms of constipation and/or abdominal pain. Gluten exposure is the most frequent cause. 关 键 词：Celiac disease Non-responsive celiac disease CHILDREN Gluten-free diet CONSTIPATION Abdominal pain