Definition of protracted and intractable diarrhea of infancy

Originally, the syndrome of IDI was described by Avery et al in 1968, based on the following features: diarrhea occurring in a newborn younger than 3 months of age, lasting more than 2 weeks, with three or more negative stool cultures for bacterial pathogens.1 Most cases were managed in hospital, using intravenous fluids while the diarrhea was persistent and intractable, with a high mortality rate from infection or malnutrition.2 Recently, the term 'severe diarrhea requiring parenteral nutrition' was proposed.3,4 Within this group of pathologies, two major subtypes can be differentiated. The first group is made up of patients with 'protracted diarrhea of infancy' (PDI), which subsides despite its initial severity. PDI can result from a specific immune deficiency, a sensi-tization to a common food protein (e.g. cow's milk or gluten), or it can be secondary to a severe infection of the digestive tract (post-enteritis syndrome). The second group is characterized by an 'intractable diarrhea of infancy', with onset within the first 2 years of life. In this second group, diarrhea persists sometimes for years, despite prolonged bowel resting and various therapeutic trials. In most cases, such as constitutive entero-cyte disorders5 or autoimmune enteropathy,6 the situation becomes rapidly life threatening, and these patients depend on long-term parenteral nutrition (PN). Some of them are candidates for intestinal transplantation. Table 1.1 shows the diagnostic heterogeneity of 65 cases with severe diarrhea requiring PN for more than 1 month, as recently analyzed by a French multicenter study.7 The so-called 'intractable ulcerating enterocolitis' of early onset is difficult to classify.8 It is very important to distinguish between IDI and PDI, since children with PDI always recover, sometimes after only several weeks or months of parenteral and/or enteral nutrition. In contrast, patients with IDI never recover, and are dependent on life-long parenteral nutrition or - in the case of autoimmune enteropathy - life-long massive immuno-suppressive medication.

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