Isolated cutaneous response to granulocyte-monocyte colony stimulating factor in fatal idiopathic disseminated Bacillus-Calmette-Guerin infection

Severe disseminated Bacillus-Calmette-Guerin (BCG) infection is very rare a nd has been regarded as idiopathic when no immunodeficiency is present. Thi s entity seems to be due to several new types of inherited abnormalities in the pathways important in defence against Mycobacteria. Although improveme nt with interferon-gamma (IFN-gamma) has been reported in some patients, to our knowledge there are no reports on the effect of other cytokines in the treatment of these patients. We report here the clinical response to IFN-g amma and granulocyte-monocyte colony stimulating factor (GM-CSF) treatment in a patient with idiopathic disseminated BCG infection who failed to respo nd to multiple antimycobacterial agents. The patient showed partial and tra nsitory response to IFN-gamma, however, GM-CSF treatment led to rapid impro vement of skin lesions within 2 weeks without any effect on the progression of the disease in the other organ systems. Conclusion The response of idiopathic disseminated Bacillus-Calmette-Guerin infection to granulocyte-monocyte colony stimulating factor treatment was limited to cutaneous lesions. Granulocyte-monocyte colony stimulating facto r may have acted to promote wound healing or the levels of this factor achi eved in other affected organs may have been inadequate.