Severe atopic dermatitis complicated with food protein-induced gastrointestinal syndrome, case report of 5 infants

Objective:SPLAD (Severe Protein Loss in Atopic Dermatitis), is an extreme form of atopic dermatitis in infants and their serum protein is lost through inflamed skin (Pediatr Allergy Immunol 2002, 13(4):287-94). Food Protein-Induced Gastrointestinal Syndrome (FPIGS) is cell-mediated food allergy mainly involves gastrointestinal tract (Curr Opin Allergy Clin Immunol. 2009;9(4):371-7). The prevalence of both SPLAD and FPIGS has been increasing since the 1990's in Japan. Co-existence of these two conditions in an infant may become a serious impact on health and growth. As we experienced 5 patients, we would like to clarify clinical features of these patients.

Method:A diagnosis of SPLAD was made if the patient showed; (1) severe atopic dermatitis, (2) total serum protein less than -2 SD of the normal value. FPIGS was suspected if their gastrointestinal symptoms were stopped by the avoidance of offending food and confirmed by food challenge test. Five patients were enrolled and evaluated clinical features and biological markers (serum total protein, peripheral eosinophil count, total IgE, specific IgE antibodies, and stool cytology).

Result:Two girls and 3 boys aged 4~6 months old showed severe AD (100%), serous skin discharge (100%), diarrhea (100%), bloody stool (100%) and vomiting (20%). Body weight was -3.2 ~ -1.6SD at the diagnosis. Their serum total protein was 3.7 ~ 4.9g/dl. Peripheral blood eosinophil count was 6~29 %. Total IgE was 32 ~ 1163kU/l. Dermatitis was treated with proper skin care and topical steroid therapy and responded well. Serum total protein was recovered promptly. But diarrhea was persisted and food elimination was performed. Food challenge test revealed that wheat, rice and soy were the offending food. Their body weight was recovered after treatment of both conditions.

Conclusion:SPLAD and FPIGS, both rare forms of allergic diseases, coexisted in 5 infants. This leads to an idea that there may be common factors concerning pathogenesis of those 2 conditions.