Abstract
We present here the unusual case of a male newborn infant who showed progressive severe cholestasis. The infant's gestational age was 37 weeks, and his birth weight was 2134 g. His serum level of direct bilirubin gradually increased from the 6th day of life and reached 257.5 micromol/L on the 22nd day of life. We could not find any cause for his cholestasis, but his serum level of ferritin was extremely elevated at 9211.0 ng/mL. Because we felt that his clinical condition might be related to hypercytokinemia caused by an immunologic reaction, steroid pulse therapy and cyclosporine were administered. His condition improved, and his direct bilirubin and ferritin levels declined. From the investigation of his cytokine profile, we found a preferentially elevated level of serum interleukin 17 (IL-17) (96.1 pg/mL) and high level of chemokines IL-8 and macrophage inflammatory protein 1beta. The IL-17 level gradually decreased to 7.5 pg/mL by the 124th day of life. The infant was successfully discharged from the children's hospital but later developed epilepsy at 11 months and asthma at 1 year, 2 months of age. Although we have not yet reached a definitive diagnosis, this case may be the first to show a relationship between cholestasis and an elevated serum IL-17 level in the neonatal period.
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