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Celiac.com 06/08/2016 - Sometimes, certain cases can stand out and grab the attention of clinicians or researchers. Such is the case of a 62-year-old woman who was suffering from severe malabsorption, and diagnosed with celiac disease based on the findings of flat, small intestinal mucosa and HLA-DQ2 positivity, although celiac blood tests were negative. A team of researchers questioned the diagnosis, because the woman showed no clinical or histological improvement after a long period of strict gluten-free diet. The research team included U Volta, MG Mumolo, G Caio, E Boschetti, R Latorre, F Giancola, P Paterini, and R De Giorgio. They variously are affiliated with the Department of Medical and Surgical Sciences at the University of Bologna, and with the Gastroenterology Unit in the Department of Gastroenterology at the University of Pisa in Italy. Based on the detection of enterocyte autoantibodies, the team found that the correct diagnosis for the woman was autoimmune enteropathy. After appropriate immunosuppressive treatment, the woman experienced the disappearance of all symptoms, and a complete recovery. Based on this case, the team notes that doctors should consider autoimmune enteropathy in the differential diagnosis of malabsorption with severe villous atrophy, including those cases with negative celiac-related serology. Source: Gastroenterol Hepatol Bed Bench. 2016 Spring;9(2):140-5.
Am J Clin Pathol. 2004 Apr;121(4):546-50 Celiac.com 04/20/2004 – According to researchers at the Department of Anatomic Pathology, William Beaumont Hospital, Royal Oak, MI, the cause of flattened villi is not always celiac disease. The researchers studied seven patients who experienced several weeks of gluten-sensitivity and the same type of villi injury—"increased lymphoplasmacytic lamina propria inflammation, moderate to complete villous flattening, numerous crypt mitoses, and markedly increased villous intraepithelial lymphocytes (IELs)." All patients were diagnosed with gluten sensitivity, and all returned 9 to 38 weeks later questioning their diagnosis, as their symptoms had substantially or completely disappeared, and clinical improvement in these patients seemed unrelated to their ingestion of gluten. A follow up endoscopy and colonoscopy was performed on these patients 4 to 16 months later, and the results of each showed a normal mucosa. According to the researchers: "Diseases other than GS can cause marked villous flattening and increased villous IELs in adults. The cause of small bowel mucosal injury is unknown. A similar non-GS-associated clinicopathologic complex, assumed to be due to a protracted viral enteritis or slow regression of a virus-induced immune reaction, occurs in children. The temporal aspects of symptom improvement and mucosal restitution in these 7 patients are similar to acute self-limited colitis. An overly exuberant immune response to an infectious agent is possible."