Congenital self-healing reticulohistiocytosis (CSHR) represents the benign
end of the spectrum of Langerhans cell histiocytoses, with spontaneous reso
lution of lesions within the first year of life. However, Involvement of or
gan systems other than the skin has been described occasionally and recurre
nce of disease at sites distant from the skin has been documented. We repor
t a case of CSHR with eye involvement that spontaneously resolved concurren
t with resolution of skin lesions, Because multiple organ systems can be in
volved and recurrences are possible, long-term follow-up of these patients
is indicated.