Effectiveness of IFN-gamma for liver abscesses in chronic granulomatous disease.
In chronic granulomatous disease, interferon-gamma (IFN-gamma) significantly reduces the incidence and severity of recurrent infections, but its effectiveness administered ex novo during acute infection has been reported in only one case. In this report, we describe two adult brothers with chronic granulomatous disease treated successfully with IFN-gamma for acute liver abscesses. Two brothers with severe recurrent infections of unknown origin were hospitalized for septic fever, malnutrition, and ultrasonographic evidence of liver abscess. Autosomal recessive chronic granulomatous disease was diagnosed based on lack of superoxide anion production by phagocytes and absence of p47-phox protein. An antibiotic regimen specifically directed against Staphylococcus aureus was ineffective, whereas treatment with 50 microg/m2 IFN-gamma s.c. thrice weekly induced complete healing with scarring within 3 months. No septic recurrence was observed during a 4-year follow-up period. In chronic granulomatous disease, IFN-gamma is effective not only in preventing but also in healing life-threatening acute infections.
Conte
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D
Cattedra di Gastroenterologia, IRCCS Ospedale Maggiore, Milano, Italy. dconte@imiucca.csi.unimi.it
Fraquelli
M
M
Capsoni
F
F
Giacca
M
M
Zentilin
L
L
Bardella
M T
MT
D000293:Adolescent; D000328:Adult; D005808:Genes, Recessive; D006105:Granulomatous Disease, Chronic / Q000188:drug therapy* / Q000235:genetics; D006801:Humans; D007371:Interferon-gamma / Q000627:therapeutic use*; D008100:Liver Abscess / Q000150:complications / Q000188:drug therapy*; D008163:Luminescent Measurements; D008297:Male
D007371:Interferon-gamma