This report delineates a new clinical presentation of multiple benign
spiradenoma (BS), a diffuse linear nevoid pattern. A 16-year-old girl,
in otherwise good health, had many papulonodular lesions, varying in
color from blue-gray to bright red, disposed linearly on the right hal
f of her body, involving the periorbital region, cheek, lateral side o
f the neck, trunk, linea alba, right thigh, and leg. Only a few nodule
s were slightly painful. Lesions were reported to be present at birth;
no family members had similar lesions, either in solitary or multiple
form. Excisional biopsies of three lesions from the neck, abdomen, an
d leg, respectively, were performed; histologically, all three samples
showed a picture of BS. In some areas, the tumor was directly connect
ed to a pilosebaceous unit. Furthermore, a focal area with pilar diffe
rentiation at the periphery of the lobules was observed, which might s
uggest that BS can differentiate toward various cutaneous adnexal stru
ctures. Our patient probably is the first one reported with diffuse li
near nevoid lesions of BS, present at birth and involving the right ha
lf of the body.