Chronic granulomatous disease : long-term follow-up and prognosis in Serbia = Kronična granulomatozna bolezen: dolgoročno sledenje in napoved izida v Srbiji : dolgoročno sledenje in napoved izida v Srbiji
Pašić, Srdjan ...
We report on the long-term follow-up of 16 paediatric patients with chronic granulomatous disease diagnosed between 1975 and 2004. Their mean age at the onset of symptoms was 5 months, while the ...median delay in diagnosis was 2.5 years. Bacillus Calmette-Guerin lymphadenitis was the most common presenting infection (7) followed by suppurative lymphadenitis (5), lung infections (1), skin infection (1), liver abscess (1) and Salmonella sepsis (1). Prophylaxis with co-trimoxazole was recommended for all patients, and after 1996 itraconazole was used as antifungal prophylaxis in 8 patients. During the meanfollow-up of 13 years (range, 3 - 30 years) pneumonitis was the most prevalent infection, followed by BCG or suppurative lymphadenitis, skin infections, aphthous stomatitis and liver abscesses. Six (37%) patients died, and in 5 patients lethal outcomes occurred during the second decade of life (4Aspergillus infection, 2 chronic lung disease). The probability of survival into the third decade was estimated to be only 19%. However, in eight surviving patients with documented strict use of antibacterial and antifungal prophylaxis, better survival into adulthood may be expected. In view of the poor long-term prognosis, early haematopoetic stem cell transplantation shouldbe considered, especially in patients with a suitable HLA-identical donor.
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