Lentigo maligna (LM) is treated to prevent progression to lentigo maligna melanoma (LMM). Surgery is the gold standard but an alternative treatment is off-label topical imiquimod. The aim of this study was to evaluate the effectiveness of 5% topical imiquimod treatment for lentigo maligna. In the period 2007-2017 57 patients with lentigo maligna were treated with off-label topical imiquimod once daily for 12 weeks. Complete clinical clearance was observed in 48 patients (84.2%) and partial clearance in 3 patients (5.3%). Three patients (5.3%) showed no response and another 3 patients (5.3%) stopped treatment due to side-effects. After 4.5 years during follow-up one patient developed a lentigo maligna melanoma which was subsequently excised. Treatment with topical imiquimod resulted in complete clearance of lentigo maligna in 48 out of 57 patients (84.2%). Topical imiquimod is an acceptable treatment option for patients with lentigo maligna who prefer topical treatment to surgery or radiotherapy.