Massive pulmonary hemorrhage (MPH) in newborn infants is a catastrophic event with a fatal result. The aim of this study was to assess the efficacy of high frequency oscillatory ventilation (HFOV) as a rescue therapy for MPH in newborn infants. Eighteen newborn infants with MPH refractory to conventional mechanical ventilation were treated with HFOV. Changes in oxygenation were assessed using arterial-alveolar oxygen tension ratio (a/APO2) and oxygenation index (OI) during HFOV. The most common underlying disorder of MPH was preterm patent ductus arteriosus (PDA). Thirteen out of 18 (72%) newborn infants with MPH responded to HFOV and survived. Five out of 18 (28%) did not respond to HFOV and died. There were no differences between responders and nonresponders in gestational age, birth weight, pre-HFOV OI, and age of MPH onset. In responders, there was a rapid increase in a/APO2 from 0.18+/-0.04 to 0.40+/-0.08 at 30 minutes after HFOV. There was also significant decrease in OI from 14.9+/-4.7 to 8.1+/-1.5 at 1 hour after HFOV. We conclude that HFOV shows rapid and dramatic improvements and has ultimately life-saving effects in MPH of newborn infants.
We report two cases of microvillus inclusion disease and these are the first cases in Korea. The two babies (one baby had a sibling who died of diarrhea in the neonatal period) had excreted their stools up to 200 ml/kg per day since several days after birth. Workup's included extensive infectious, immunologic, hormonal and rheumatologic studies, all of which were negative or normal. Diagnosis rested on the ultrastructural finding of intracytoplasmic inclusions that contained intact microvilli on electron microscopy. We tried somatostatin analogue (octreotide, 4 micrograms/kg/day), cholestyramine (up to 4g t.i.d.), steroid (prednisone, 2 mg/kg/day) and intravenous epidermal growth factor (100 ng/kg/hr for 2 weeks), but there was mild improvement with cholestyramine (decrease stool volume) and epidermal growth factor (increase the number of microvilli per cell) but no improvement was noted with the other treatments. Although it is a rare disorder and the prognosis of microvillus inclusion disease is poor, it must be considered if an infant has chronic secretory diarrhea.
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