Celiac disease is a gluten enteropathy that is treated with dietary elimination of gluten. Exposure to nondietary sources of gluten, which are used in the manufacture of products such as plastics, dental equipment, and cosmetics, can also trigger or exacerbate disease. We report the case of a 9-year-old child who presented with nonspecific abdominal discomfort with abnormal serology for celiac disease. She underwent duodenal biopsies that revealed Marsh 3B histopathology. Despite strict dietary elimination of gluten, she continued to be symptomatic and demonstrate positive serum markers for active disease. It was then discovered that the child was exposed to gluten from her orthodontic retainer that contained a plasticized methacrylate polymer. Gluten is a common additive in plastics. She discontinued its use and demonstrated symptom resolution and complete normalization of serology. All possible sources of gluten, including nondietary, must be considered when managing a child with celiac disease.
A 16-year-old boy experienced chronic vague abdominal pain, dark urine, acholic stools, and jaundice. Laboratory studies were consistent with pancreatitis and hepatitis (elevated lipase, aspartate aminotransferase, alanine aminotransferase, g-glutamyl transferase, direct bilirubin, positive anti-nuclear antibody, and antismooth muscle antibody). An endoscopic retrograde cholangiopancreatography was normal. Abdominal magnetic resonance imaging showed biliary duct dilatation with normal pancreas. He had elevated immunoglobulin (Ig) G of 4080 mg/dL. He underwent a cholecystectomy and a liver biopsy. The liver biopsy was consistent with autoimmune hepatitis. The gallbladder histology, pictured above, showed diffuse lymphoplasmacytic acalculous cholecystitis. There was portal inflammation with relative sparing of intrahepatic bile ducts and periductal inflammation but no overt cholangitis. No cholestasis or periductal fibrosis was seen. Immunohistochemistry staining for IgG4 was positive for plasma cells in liver and gallbladder specimens, diagnostic of autoimmune cholecystitis. He was treated with corticosteroids and 6-mercaptopurine. His transaminases, lipase, and amylase returned to normal.Autoimmune cholangitis is the biliary manifestation of a multisystem fibroinflammatory disorder. IgG4-associated autoimmune cholangiopathy may occur in the presence of autoimmune pancreatitis and hepatitis. Pathologically, there is a characteristic lymphoplasmacytic infiltrate and abundance in IgG4-positive cells. Although responsive to steroids, relapse is common after early withdrawal of treatment.
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