A 35-year-old woman with severe fistulizing Crohn's disease presented with 
pyostomatitis vegetans affecting both the mouth and the vulva. The coalescing 
pustules transformed within several days into vegetating lesions on areas of 
inflammation. Microbial assessments revealed no pathogenic agent. Histology 
showed neutrophilic microabscesses, but no granulomas. Three injections of 
infliximab and maintenance therapy with methotrexate resulted in rapid and 
complete regression of both the pyostomatitis vegetans and the Crohn's disease. 
Infliximab and methotrexate may be a promising treatment for the rare cases of 
pyostomatitis vegetans associated with Crohn's disease.