Sinus histiocytosis with massive lymphadenopathy is an uncommon disease affecting individuals of either sex and of all ages and ethnic backgrounds. We present a case of sinus histiocytosis confined to the skin in a 65-year-old woman whose diagnosis was coincident with herpes simplex virus infection and polybacterial urinary infection, both asymptomatic. The polymerase chain reaction for herpes simplex virus and human herpesvirus-6 from involved skin was negative. We review previously published cases of sinus histiocytosis limited to the skin.